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Music therapy for autistic people (Review)
Geretsegger M, Fusar-Poli L, Elefant C, Mössler KA, Vitale G, Gold C
Geretsegger M, Fusar-Poli L, Elefant C, Mössler KA, Vitale G, Gold C.

Music therapy for autistic people.
Cochrane Database of Systematic Reviews 2022, Issue 5. Art. No.: CD004381.
DOI: 10.1002/14651858.CD004381.pub4.
www.cochranelibrary.com

Copyright © 2022 The Cochrane Collaboration. Published by John Wiley & Sons, Ltd.
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TABLE OF CONTENTS
ABSTRACT..................................................................................................................................................................................................... 1
PLAIN LANGUAGE SUMMARY....................................................................................................................................................................... 2
SUMMARY OF FINDINGS.............................................................................................................................................................................. 4
BACKGROUND.............................................................................................................................................................................................. 8
OBJECTIVES.................................................................................................................................................................................................. 10
METHODS..................................................................................................................................................................................................... 10
RESULTS........................................................................................................................................................................................................ 14
Figure 1.................................................................................................................................................................................................. 15
Figure 2.................................................................................................................................................................................................. 17
Figure 3.................................................................................................................................................................................................. 18
Figure 4.................................................................................................................................................................................................. 19
Figure 5.................................................................................................................................................................................................. 26
Figure 6.................................................................................................................................................................................................. 27
DISCUSSION.................................................................................................................................................................................................. 32
AUTHORS' CONCLUSIONS........................................................................................................................................................................... 34
ACKNOWLEDGEMENTS................................................................................................................................................................................ 35
REFERENCES................................................................................................................................................................................................ 37
CHARACTERISTICS OF STUDIES.................................................................................................................................................................. 48
DATA AND ANALYSES.................................................................................................................................................................................... 86
Analysis 1.1. Comparison 1: Music therapy vs placebo therapy or standard care, Outcome 1: Global improvement..................... 89
Analysis 1.2. Comparison 1: Music therapy vs placebo therapy or standard care, Outcome 2: Social interaction.......................... 90
Analysis 1.3. Comparison 1: Music therapy vs placebo therapy or standard care, Outcome 3: Non-verbal communication.......... 91
Analysis 1.4. Comparison 1: Music therapy vs placebo therapy or standard care, Outcome 4: Verbal communication.................. 92
Analysis 1.5. Comparison 1: Music therapy vs placebo therapy or standard care, Outcome 5: Quality of life................................. 92
Analysis 1.6. Comparison 1: Music therapy vs placebo therapy or standard care, Outcome 6: Total autism symptom severity...... 93
Analysis 1.7. Comparison 1: Music therapy vs placebo therapy or standard care, Outcome 7: Adverse events.............................. 93
Analysis 1.8. Comparison 1: Music therapy vs placebo therapy or standard care, Outcome 8: Adaptive behaviour....................... 94
Analysis 1.9. Comparison 1: Music therapy vs placebo therapy or standard care, Outcome 9: Quality of family relationships....... 94
Analysis 1.10. Comparison 1: Music therapy vs placebo therapy or standard care, Outcome 10: Identity formation..................... 95
Analysis 1.11. Comparison 1: Music therapy vs placebo therapy or standard care, Outcome 11: Depression................................. 95
ADDITIONAL TABLES.................................................................................................................................................................................... 95
APPENDICES................................................................................................................................................................................................. 98
WHAT'S NEW................................................................................................................................................................................................. 107
HISTORY........................................................................................................................................................................................................ 107
CONTRIBUTIONS OF AUTHORS................................................................................................................................................................... 107
DECLARATIONS OF INTEREST..................................................................................................................................................................... 108
SOURCES OF SUPPORT............................................................................................................................................................................... 108
DIFFERENCES BETWEEN PROTOCOL AND REVIEW.................................................................................................................................... 108
INDEX TERMS............................................................................................................................................................................................... 109
Music therapy for autistic people (Review) i

Informed decisions.
[Intervention Review]
Music therapy for autistic people
Monika Geretsegger1, Laura Fusar-Poli2, Cochavit Elefant3, Karin A Mössler1, Giovanni Vitale4, Christian Gold1,5
1GAMUT - The Grieg Academy Music Therapy Research Centre, NORCE Norwegian Research Centre, Bergen, Norway. 2Department
of Clinical and Experimental Medicine, Psychiatry Unit, University of Catania, Catania, Italy. 3Department of Creative Arts Therapies,
University of Haifa, Haifa, Israel. 4Child and Adolescent Neuropsychiatry, University of Naples Federico II, Naples, Italy. 5Department of
Clinical and Health Psychology, University of Vienna, Vienna, Austria
Contact: Christian Gold, chgo@norceresearch.no.
Editorial group: Cochrane Developmental, Psychosocial and Learning Problems Group.

Publication status and date: New search for studies and content updated (conclusions changed), published in Issue 5, 2022.
Citation: Geretsegger M, Fusar-Poli L, Elefant C, Mössler KA, Vitale G, Gold C.Music therapy for autistic people. Cochrane Database of
Systematic Reviews 2022, Issue 5. Art. No.: CD004381. DOI: 10.1002/14651858.CD004381.pub4.
ABSTRACT
Background
Social interaction and social communication are among the central areas of difficulty for autistic people. Music therapy uses music
experiences and the relationships that develop through them to enable communication and expression, thus attempting to address some
of the core problems of autistic people. Music therapy has been applied in autism since the early 1950s, but its availability to autistic
individuals varies across countries and settings. The application of music therapy requires specialised academic and clinical training which
enables therapists to tailor the intervention to the specific needs of the individual. The present version of this review on music therapy for
autistic people is an update of the previous Cochrane review update published in 2014 (following the original Cochrane review published
in 2006).
Objectives
To review the effects of music therapy, or music therapy added to standard care, for autistic people.
Search methods
In August 2021, we searched CENTRAL, MEDLINE, Embase, eleven other databases and two trials registers. We also ran citation searches,
checked reference lists, and contacted study authors to identify additional studies.
Selection criteria
All randomised controlled trials (RCTs), quasi-randomised trials and controlled clinical trials comparing music therapy (or music therapy
alongside standard care) to 'placebo' therapy, no treatment, or standard care for people with a diagnosis of autism spectrum disorder
were considered for inclusion.
Data collection and analysis

We used standard Cochrane methodological procedures. Four authors independently selected studies and extracted data from all included
studies. We synthesised the results of included studies in meta-analyses. Four authors independently assessed risk of bias (RoB) of each
included study using the original RoB tool as well as the certainty of evidence using GRADE.
Main results
We included 16 new studies in this update which brought the total number of included studies to 26 (1165 participants). These studies
examined the short- and medium-term effect of music therapy (intervention duration: three days to eight months) for autistic people in
individual or group settings. More than half of the studies were conducted in North America or Asia. Twenty-one studies included children
Music therapy for autistic people (Review) 1

Informed decisions.
aged from two to 12 years. Five studies included children and adolescents, and/or young adults. Severity levels, language skills, and
cognition were widely variable across studies.
Measured immediately post-intervention, music therapy compared with 'placebo' therapy or standard care was more likely to positively
effect global improvement (risk ratio (RR) 1.22, 95% confidence interval (CI) 1.06 to 1.40; 8 studies, 583 participants; moderate-certainty
evidence; number needed to treat for an additional beneficial outcome (NNTB) = 11 for low-risk population, 95% CI 6 to 39; NNTB = 6
for high-risk population, 95% CI 3 to 21) and to slightly increase quality of life (SMD 0.28, 95% CI 0.06 to 0.49; 3 RCTs, 340 participants;
moderate-certainty evidence, small to medium effect size). In addition, music therapy probably results in a large reduction in total autism
symptom severity (SMD −0.83, 95% CI −1.41 to −0.24; 9 studies, 575 participants; moderate-certainty evidence). No clear evidence of a
difference between music therapy and comparison groups at immediately post-intervention was found for social interaction (SMD 0.26,
95% CI −0.05 to 0.57, 12 studies, 603 participants; low-certainty evidence); non-verbal communication (SMD 0.26, 95% CI −0.03 to 0.55; 7
RCTs, 192 participants; low-certainty evidence); and verbal communication (SMD 0.30, 95% CI −0.18 to 0.78; 8 studies, 276 participants;
very low-certainty evidence). Two studies investigated adverse events with one (36 participants) reporting no adverse events; the other
study found no differences between music therapy and standard care immediately post-intervention (RR 1.52, 95% CI 0.39 to 5.94; 1 study,
290 participants; moderate-certainty evidence).
Authors' conclusions
The findings of this updated review provide evidence that music therapy is probably associated with an increased chance of global
improvement for autistic people, likely helps them to improve total autism severity and quality of life, and probably does not increase
adverse events immediately after the intervention. The certainty of the evidence was rated as 'moderate' for these four outcomes,
meaning that we are moderately confident in the effect estimate. No clear evidence of a difference was found for social interaction, non-
verbal communication, and verbal communication measured immediately post-intervention. For these outcomes, the certainty of the
evidence was rated as 'low' or 'very low', meaning that the true effect may be substantially different from these results. Compared with
earlier versions of this review, the new studies included in this update helped to increase the certainty and applicability of this review's
findings through larger sample sizes, extended age groups, longer periods of intervention and inclusion of follow-up assessments, and by
predominantly using validated scales measuring generalised behaviour (i.e. behaviour outside of the therapy context). This new evidence is
important for autistic individuals and their families as well as for policymakers, service providers and clinicians, to help in decisions around
the types and amount of intervention that should be provided and in the planning of resources. The applicability of the findings is still
limited to the age groups included in the studies, and no direct conclusions can be drawn about music therapy in autistic individuals above
the young adult age. More research using rigorous designs, relevant outcome measures, and longer-term follow-up periods is needed to
corroborate these findings and to examine whether the effects of music therapy are enduring.
PLAIN LANGUAGE SUMMARY
Music therapy for autistic people
Review question
We reviewed the evidence about the effect of music therapy for autistic people. We compared results from people receiving music therapy
(or music therapy added to standard care) with results from people receiving a similar therapy without music ('placebo' therapy), standard
care or no therapy at all.
Background
Autism is a lifelong neurodevelopmental condition that affects how people perceive the world around them, and how they communicate
with and relate to others. Thus, social interaction and social communication are among the central areas of difficulty for autistic
people. Music therapy uses music experiences and the relationships that develop through them to enable people to relate to others, to
communicate, and to share their feelings. In this way, music therapy addresses some of the core problems of autistic people. Music therapy
has been applied in autism since the early 1950s. Its availability to autistic people varies across countries and settings. The application of
music therapy requires specialised academic and clinical training. This helps therapists in tailoring the intervention to the specific needs
of the person. We wanted to investigate whether music therapy helps autistic people compared with other options.
Search date
The evidence is current to August 2021.
Study characteristics
We included 16 new studies in this update, so the evidence in this review now rests on 26 studies with a total number of 1165 participants.
The studies examined the short- and medium-term effect of music therapy interventions (three days to eight months) for autistic children,
youth, and young adults in one-to-one or group settings. None of the studies reported funding by an agency with a commercial interest in
the result of the studies; reported sources of support included governmental, university and foundation funding; in three studies, support
was provided by a music therapy association.

Informed decisions.
Key results
Music therapy compared with 'placebo' therapy or standard care probably increases the chance of overall improvement by the end of
therapy, likely improves quality of life and total autism symptom severity immediately after therapy, and probably does not increase
adverse events. From the available evidence, we cannot tell whether music therapy has any effects on social interaction, and verbal and
non-verbal communication at the end of therapy.
Quality of the evidence
The evidence we found in this review is of very low to moderate certainty. This means that future research may change these findings
and our confidence in them. We found that music therapy is probably effective regarding global improvement, quality of life, total autism
symptom severity and adverse events measured at the end of therapy based on the moderate certainty of the evidence in these domains.
It remains unclear whether music therapy has an effect on social interaction, non-verbal communication and verbal communication at the
end of therapy since the certainty of evidence was low to very low. Reasons for limited certainty of the evidence were issues with study
design and blinding (i.e. those who applied outcome measures often knew whether or not participants had received music therapy, which
may have influenced their assessments).
Authors' conclusions
Music therapy compared with 'placebo' therapy or standard care probably increases the chance of overall improvement by the end of
therapy. It also probably helps to enhance quality of life, and lessen symptom severity. Music therapy probably does not increase adverse
events. We cannot tell whether music therapy may help with social interaction, non-verbal communication and verbal communication
at the end of therapy. Most of the included studies featured interventions that correspond well with music therapy in clinical practice
concerning methods and settings. This new evidence is important for autistic people and their families as well as for policymakers, service
providers and clinicians, to help in decisions around what intervention to choose, and in the planning of resources. More research with
adequate design (i.e. producing reliable evidence) looking at areas that matter to autistic people is needed. Because long-term outcomes
of therapy matter to autistic people and their families, it is important to specifically examine how long the effects of music therapy last.

SUMMARY OF FINDINGS
Summary of findings 1. Music therapy compared with placebo therapy or standard care for autistic people
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Music therapy compared with placebo therapy or standard care for autistic people
Population: individuals with a diagnosis of autism spectrum disorder

Settings: outpatient therapy centre, hospital, school, summer camp or home; individual and group setting
Intervention: music therapy
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Trusted evidence.
Comparison: placebo therapy or standard care
Outcomes Illustrative comparative risks* (95% CI) Relative effect Number of par- Certainty of Comments
(95% CI) ticipants the evidence
(studies) (GRADE)
Assumed risk Corresponding risk Music therapy
versus place-
Risk with Risk with music therapy bo therapy or
placebo or standard care
standard care
Global improvement Low-risk populationa RR 1.22 (1.06 to 583 ⊕⊕⊕⊝ Higher scores represent
Follow-up: immediately 1.40) (8 studies) Moderateb greater improvement.
post-intervention (M = 3.4
months, SD = 2.4) 430 per 1000 525 per 1000
(456 to 602)
High-risk populationa
800 per 1000 976 per 1000

(848 to 1000)
Social interaction - The mean social interaction score - 603 ⊕⊕⊝⊝ Higher scores represent
Follow-up: immediate- at immediately post-intervention (12 studies) Lowc higher social interaction
ly post-intervention (M = in the intervention groups was 0.26 capabilities.
3.5 months, SD = 2.4) standard deviations higher (0.05
lower to 0.57 higher)
4
Small to medium effect
size according to Cohen
1988
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Non-verbal communica- - The mean non-verbal communi- - 192 ⊕⊕⊝⊝ Higher scores represent
tion cation score at immediately post- (7 studies) Lowd higher non-verbal com-
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Informed decisions.
Trusted evidence.
Follow-up: immediately intervention in the intervention munication capabilities.
post-intervention (M = 4.2 groups was 0.26 standard de-
months, SD = 2.4) viations higher (0.03 lower to
0.55 higher)
1988
Verbal communication - The mean verbal communication - 276 ⊕⊝⊝⊝ Higher scores represent
score at immediately post-inter- (8 studies) Very lowe higher verbal communica-
Follow-up: immediately vention in the intervention groups tion capabilities.
post-intervention (M = 3.2 was 0.30 standard deviations
months, SD = 2.8) higher (0.18 lower to 0.78 higher)
1988

Quality of life - The mean quality of life score - 340 ⊕⊕⊕⊝ Higher scores represent
at immediately post-intervention- (3 studies) Moderatef higher quality of life.
Follow-up: immediately in the intervention groups was
post-intervention (M = 3.3 0.28 standard deviations high-
months, SD = 1.5) er (0.06 to 0.49 higher)
1988
Total autism symptom - The mean total autism symptom - 575 ⊕⊕⊕⊝ Higher scores represent
severity severity score at immediately post- (9 studies) Moderateb higher symptom severity.
intervention in the intervention
5
Follow-up: immediately groups was 0.83 standard devia- Large effect size according
post-intervention (M = 3.6 tions lower (1.41 to 0.24 lower) to Cohen 1988
months, SD = 2.1)
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Adverse events Low-risk populationa RR 1.52 (0.39 to 326 ⊕⊕⊕⊝ Higher scores represent
5.94) (2 studies) Moderatef higher numbers of ad-
Any serious or non-serious verse events.
0 per 1000 0 per 1000
adverse event
Better health.
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Trusted evidence.
Follow-up: immediately (0 to 0)
post-intervention (M = 4.0
months, SD = 1.4) Adverse events reported
High-risk populationa are hospitalisation peri-
ods, typically planned and
24 per 1000 37 per 1000 short-term.
(9 to 150)
One study with 36 par-

ticipants reported no ad-
verse events and was not
included in the RR analy-
sis.
*The basis for the assumed risk is provided in footnotes. The corresponding risk (and its 95% CI) is based on the assumed risk in the inter-
vention group and the relative effect of the intervention (and its 95% CI).
CI: Confidence interval; M: Mean; RR: Risk ratio; SD: Standard deviation.
GRADE Working Group grades of evidence

High certainty: We are very confident that the true effect lies close to that of the estimate of the effect.
Moderate certainty: We are moderately confident in the effect estimate; the true effect is likely to be close to the estimate of the effect, but
there is a possibility that it is substantially different.

Low certainty: Our confidence in the effect estimate is limited; the true effect may be substantially different from the estimate of the effect.
Very low certainty: We have very little confidence in the effect estimate; the true effect is likely to be substantially different from the estimate
of the effect.
aTypical risks are not known, so we chose the risk from included studies providing the second highest (Kim 2008) for a high-risk population and the second lowest (Porter 2017)
for a low-risk population for the outcome 'Global improvement' (Schünemann 2021). For the outcome of 'Adverse events', where only two studies were included, we based the
risk of the high-risk population on Bieleninik 2017 and that of the low-risk population on Porter 2017.
bWe downgraded the certainty of the evidence by one level for risk of bias (limitations in the designs such as poorly reported randomisation, blinding of outcomes, incomplete
outcome data).
cWe downgraded the certainty of the evidence by one level for risk of bias and one level for imprecision (wide CI: 95% CI included no effect and the upper confidence limit crossed
an effect size of 0.5; GRADEpro GDT).
6
dWe downgraded the certainty of the evidence by two levels for imprecision (wide CIs) and because the total number of participants in this outcome was lower than 400.
eWe downgraded the certainty of the evidence by one level for risk of bias and two levels for imprecision (wide CIs), and because the total number of participants in this outcome
was lower than 400.
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fWe downgraded the certainty of the evidence by one level for imprecision because the total number of participants in this outcome was lower than 400.
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Trusted evidence.
7
Informed decisions.
BACKGROUND changes from the environment rather than solely from the
individual. Accordingly, the enabling and disabling impact any
Description of the condition given interaction, context or society can hold for autistic people
needs to be considered when defining autism or interacting with
Autism is a complex neurodevelopmental condition that usually
autistic people (Milton 2019).
manifests in early childhood and persists throughout life. When
following a medical paradigm, and according to the criteria of Regarding the terminology used in autism research, there is an
the International Classification of Diseases and Related Health ongoing debate on the type of language that is most appropriate
Problems, 11th edition (ICD-11) (WHO 2021), and the Diagnostic and most respectful to people with a diagnosis of ASD, their
and Statistical Manual of Mental Disorders, fifth edition (DSM-5) families and caregivers. A growing body of literature documents
(APA 2013), autism spectrum disorder (ASD) is characterised by that person-first language (e.g. 'people with ASD') may actually
'persistent deficits in social communication and social interaction increase effects of stigmatisation for autistic people (Bottema-
across multiple contexts', and by the presence of 'restricted, Beutel 2021; Gernsbacher 2017), and that people with a diagnosis
repetitive patterns of behavior, interests, or activities'. For a of ASD themselves often prefer using identity-first language (e.g.
diagnosis of ASD, children must show symptoms of ASD since early "autistic individuals") as a means of showing that autism is a central
childhood (i.e. before the age of three) (APA 2013; WHO 2021). part of their identity rather than something that needs to be fixed
In some instances, these symptoms may only be detectable later or cured (Bury 2020; Kenny 2016). This preference has also been
when social demands become intractable, or may continue to be expressed by autistic people and their families who have been
masked through learned strategies (APA 2013) in an attempt to consulted by the authors while conducting this review. Considering
mimic neurotypical behaviours. these contexts and perspectives, we chose to use identity-first
language in this review.
The prevalence of ASD has considerably risen over the last decades.
While the first epidemiological study estimated a prevalence of the Depending on the way autism is conceptualised - either as
condition as lower than 0.5% in young children (Lotter 1966), the a set of cognitive or behavioural deficits (APA 2013) or as a
latest estimates of the Centers for Disease Control and Prevention social construction and as a description of a culturally filtered
reported that one in 54 children in the United States may be on experience (Milton 2019) - therapeutic aims and approaches
the autism spectrum (Maenner 2020). The increased prevalence will differ. Following a medical paradigm, psychosocial and
rates are attributable to the broadening of the diagnostic criteria, behavioural therapies are considered the first-line evidence-based
diagnostic switching from other developmental disabilities, service treatments for people with a diagnosis of ASD. These therapy
availability, and awareness of the condition among the community approaches traditionally aim at achieving changes regarding the
and professionals (Elsabbagh 2012; Lyall 2017). Of note, ASD is more way autistic people communicate and interact with others and
commonly diagnosed among males than females, with a ratio of 4:1 often follow a normalising agenda which tries to lessen or remove
(Maenner 2020). outward signs of autism. Contrary to this, some of the same
therapies may follow a maximising agenda (Winter 2012) where
The clinical picture is widely variable in presentation, severity,
the aims of any intervention are to maximise an individual's
and hence levels of support needed. Additionally, ASD
capabilities as an autistic person.
may be accompanied by co-occurring conditions, such as
intellectual disability (ID), language impairments, as well as other A variety of music therapy approaches have been developed for
neurodevelopmental, mental, and behavioural disorders (APA working with autistic people, many of them defined as relational
2013). The most frequent co-occurring mental health conditions are or child-led (Carpente 2009; Geretsegger 2015; Schumacher 1994),
attention-deficit hyperactivity disorders (ADHD), anxiety disorders, following the individual's strengths and resources and allowing for
sleep-wake disorders, depression, disruptive, impulse-control, and participatory processes in the development of social interaction
conduct disorders (Lai 2019). Autistic people might be more and understanding, thus more aligned with a maximising agenda
vulnerable to negative life experiences (Griffiths 2019) and to the and a cultural or social model of autism.
development of post-traumatic stress symptoms (Rumball 2020).
As a consequence, outcome domains beyond the core symptoms of As symptom change is often assessed as a primary outcome in
ASD, such as depression, anxiety, or quality of life, are increasingly scientific research, a normalisation agenda might be considered
receiving more attention in autism research. to form the epistemological background of music therapy research
as well. However, music therapy research also combines these
As autistic ways of communicating and being social deviate from two agendas (Pickard 2020) by applying music therapy approaches
neurotypical socialising, approaches following a medical model striving for maximisation, while concurrently using outcomes
tend to seek to change this deviation. Within such a model, measuring neurotypical social behaviour and communication and
challenges emerging from being autistic are situated within the general domains such as quality of life (see, for example, Bieleninik
autistic individuals rather than the environment, culture or society 2017). Thus, music therapy can be seen as relating to the coexisting
surrounding them. The medical model has been criticised by "dual nature of autism" (Lai 2020), being categorised as medical
scholars as well as by the autistic community (De Jaegher 2013; condition leading to developmental disability and at the same time
Milton 2012 ). Instead, a social or cultural model of autism has being an example of neurodivergent development forming identity
been suggested (Sinclair 2010; Sinclair 2012). A social model of and culture.
understanding autism looks at autistic characteristics as part
of human diversity and understands social interaction as a Description of the intervention
shared responsibility and participatory practice (De Jaegher 2007).
Hence, challenges causing dysfunction in social interaction can Music therapy for autistic people is often provided as individual
also be located outside the autistic person and might require therapy, although there are also reports of group-based and peer-

Informed decisions.
mediated interventions (e.g. Boso 2007; Ghasemtabar 2015; Kern people to access sensory experiences and for people without
2006; Kern 2007; LaGasse 2014; Mateos-Moreno 2013). Family- spoken language to interact communicatively without words. It
centred approaches, where parents or other family members enables all to engage on a more emotional, relationship-oriented
are included in therapy sessions (Oldfield 2012; Pasiali 2004; level than may be accessible through verbal language (Alvin
Thompson 2014; Thompson 2012) or trained in relevant music- 1991). Behaviouristic and educational approaches typically use
based techniques for social engagement (Gottfried 2016), have music activities to motivate the child and to reinforce targeted
increasingly become an important part of music therapy for autistic behaviour. Developmental approaches often use music to focus on
children, especially to help generalise skills acquired in therapy to the sensory, motor-coordination and affective aspects of music-
everyday contexts, that is, to transfer these skills from the therapy making, e.g. through intra- and inter-personal synchronisation
context to new and different settings. experiences (Berger 2002; Schumacher 2019). In improvisational
approaches, therapists attune to the child's intrinsic way of sound-
Music therapy has been defined as "a systematic process of making and moving, using the shared history of musical interaction
intervention wherein the therapist helps the client to promote and jointly developed musical activities to motivate and engage
health, using music experiences and the relationships that the child in interactive processes (Geretsegger 2015; Holck 2004).
develop through them as dynamic forces of change" (Bruscia Listening to music within music therapy also involves an interactive
1998, p. 20). Music therapy approaches for autistic people process that often includes selecting music that is meaningful for
are based on sensory-perceptional, developmental, creative, the person (e.g. relating to an issue that the person is occupied
behavioural, and educational conceptualisations (Bergmann with) and, where possible, reflecting on personal issues related to
2016). Accordingly, aims in music therapy are wide including the the music or associations brought up by the music. For those with
work on communication and interaction, sensory processing and verbal abilities, verbal reflection on the musical processes is often
integration, affect regulation, identity formation as well as creative an important part of music therapy (Wigram 2002).
and recreational needs that can lead to an increased quality of life.
Active music-making with a variety of instruments that are easy There are several psychological theories and neurobiological
to play is widely used, involving the client and the therapist in models that aim to explain the mechanisms through which music
joint musical play. Central music therapy techniques include free therapy helps autistic individuals (Fusar-Poli in press). One area of
and structured improvisation, recreating songs and vocalisation, research underpinning the potential of music therapy in autism is
or songwriting. Listening to pre-recorded or live music played based on findings suggesting that motor timing and sensorimotor
by the therapist can be used for e.g. relaxation purposes or, in integration are disrupted in autistic people (De Jaegher 2013;
the context of behavioural approaches of music therapy, focusing Sharda 2018), which may contribute to broader challenges in
on training of specific skills. Some music therapy approaches interacting with others (Mössler 2019). Functional neuroimaging
also include movement activities or story-telling. The delivery studies with autistic individuals showed an overconnectivity
of music therapy varies in its degree of structuredness: while between sensory brain networks which is related to the sensory
behavioural approaches often make use of fixed manuals specifying processing differences and multisensory integration difficulties
training phases and materials (e.g. Lim 2011), developmental (Chen 2020). Thus, sensorimotor integration facilitated by musical
or improvisational approaches are usually less pre-structured. interaction may lead to modulation of atypical sensory processing,
However, there are also some flexible yet systematic treatment which may in turn enhance social communication (Thye 2018).
guidelines for improvisational music therapy in autism which Another, related rationale for the use of music therapy for
specify core therapeutic principles and techniques (Geretsegger individuals with communication disorders is based on the findings
2015; Kim 2006; Thompson 2014; Wigram 2006). of infancy researchers such as Stern and Trevarthen who describe
sound dialogues between mothers and infants using 'musical'
Music therapy has been applied in autism since the early 1950s terms (Stern 1985; Stern 1989; Stern 2010; Trevarthen 1999b).
(Fusar-Poli in press; Reschke-Hernández 2011), but its availability When describing tonal qualities, researchers use the terms pitch,
to autistic individuals varies across countries, depending on timbre, and tonal movement and, when describing temporal
other factors such as age or educational setting (Kern 2017). qualities, they speak of pulse, tempo, rhythm, and timing
The application of music therapy requires specialised academic (Wigram 2002). Trevarthen 1999a describes the sensitivity of
and clinical training, typically achieved through Bachelor and very young infants to the rhythmic and melodic dimensions of
Master's level degree courses in music therapy which usually lead maternal speech, and to its emotional tone, as demonstrating
to accreditation with professional associations or governmental that we are born ready to engage with the 'communicative
registries, or both. Training courses in music therapy not only teach musicality' of conversation. The experience of attunement through
clinical music therapy techniques, but also aim at developing the synchronisation in timing, tonality or affective dynamics shapes
therapist's personality and clinical sensitivity, which is necessary the attachment between infant and caregivers and has been
to apply music therapy responsibly. Thus, this specialised training suggested as influencing the development of social understanding
enables music therapists to tailor their methods and techniques to (Greenspan 2007; Stern 1985; Trevarthen 2011). These premises
meet individual therapeutic goals and needs (Fusar-Poli in press). allow music to act as an effective medium for engaging in
non-verbal social exchange for autistic children and adults.
How the intervention might work Communicative behaviours, such as joint attention, eye contact,
The processes that occur within musical interaction may help and turn-taking, are characteristic events in shared, active music-
autistic people to develop communication skills and the capacity making and, therefore, inherent components of music therapy
for social interaction. Through engaging in musical interaction, processes. Recent research has shown that musical and emotional
participants in music therapy can shift between verbal, non- attunement within music therapy processes can support social
verbal and pre-verbal modes of communication. Thus, musical responsiveness in autistic children (Mössler 2019; Mössler 2020).
interaction can be understood and described as a means for verbal In addition to music's potential to stimulate communication (as

Informed decisions.
described for vocal communication in Salomon-Gimmon 2019), METHODS

music therapists use music, especially improvisational music-
making, to provide autistic people with opportunities to experience Criteria for considering studies for this review
structure combined with measured flexibility, thus helping them to
Types of studies
find ways of coping in less predictable situations that will typically
pose challenges for them (Wigram 2009). All relevant randomised controlled trials (RCTs), quasi-randomised
trials and controlled clinical trials (CCTs), including cluster-
The potential for predictability and anticipation brought about trials were considered for inclusion. Studies using single-case
by musical structures is an element also used in behavioural experimental designs were included if they also met the definition
approaches where music is utilised as a stimulus facilitating the of RCTs or CCTs, that is, if the different interventions were provided
perception and production of speech and language and enhancing in a different order to different participants (i.e. cross-over RCTs/
communication skills (Lim 2010; Lim 2011). Another rationale for CCTs). Studies in which all participants received interventions in the
using music in this way is the increased attention and enjoyment same order (i.e. case series) were excluded.
observed in autistic individuals when presented with musical as
opposed to verbal stimuli (Buday 1995; Lim 2010; Lim 2011). Types of participants
Why it is important to do this review Individuals of any age who were diagnosed with ASD as defined
in DSM-5 (APA 2013) or ICD-11 (WHO 2021) criteria, whether
This is an update of a Cochrane review first published in 2006 identified by a psychological assessment or a psychiatric diagnosis,
(Gold 2006) and previously updated in 2014 (Geretsegger 2014). were considered for inclusion. Moreover, we included individuals
The first version of this review concluded that music therapy may diagnosed with pervasive developmental disorders, as defined in
help autistic children to improve their communicative skills, but ICD-10 criteria (WHO 1994) or in previous versions of the DSM,
also noted that more research was needed to investigate the effects including childhood autism, atypical autism, Asperger's syndrome,
of music therapy in typical clinical practice and within longer and pervasive developmental disorder not otherwise specified, as
periods of observation (Gold 2006). In the 2014 update of this these previous diagnostic labels are now included in the category
review, we found that music therapy may help autistic children to of ASD in DSM-5 and ICD-11. Individuals with Rett's disorder or
improve their skills in social interaction, verbal communication, childhood disintegrative disorder were not included as they have
initiating behaviour, and social-emotional reciprocity; we also been excluded from the ASD diagnostic category in the current
concluded that more research with larger samples addressing classifications, given their significantly different clinical course.
relevant outcomes through standardised scales was needed to
corroborate these findings and to examine long-term effects of Types of interventions
music therapy as well as effects of music therapy for adolescents
Interventions included music therapy (i.e. regular sessions of music
and adults (Geretsegger 2014).
therapy involving music experiences and relationships developing
More recently, further systematic reviews have appeared, often through them as defined above, delivered by a professional music
with limited scope (e.g. Shi 2016 focusing on only Chinese therapist).
data), methodological flaws (e.g. Whipple 2012 where designs
Comparators
of included studies lacked homogeneity and included sample
sizes of only one), or providing only narrative summaries (e.g. De Interventions were compared with either 'placebo' therapy (i.e.
Vries 2015), thus highlighting the continued need for an updated, a similar intervention without the elements specific to music
comprehensive review. Furthermore, considerable changes have therapy, e.g. play therapy without music, or music listening
occurred in the knowledge about ASD in recent years, and a number without interaction with a music therapist; the concept of attention
of new studies of music therapy for autism were published since placebo in psychotherapy research is discussed in Kendall 2004),
the 2014 version of this review, which necessitated an update no treatment, or standard care control; or music therapy added
of the previous review. We conducted the current update to to standard care compared with standard care (with or without
summarise and evaluate these new studies in order to provide 'placebo' therapy).
comprehensive and up-to-date conclusions, as well as implications
for practice and research that are based on the most recent Types of outcome measures
findings. This information is highly relevant for autistic individuals To ensure that all user-important outcomes were addressed
and their families as well as for policymakers, service providers (McKenzie 2021), and to update our approach in correspondence
and clinicians, to help in decisions around the types and amount with changes that occurred in the knowledge and nosological
of intervention and support that should be provided, and in the classification of the condition in recent years (see Differences
planning of resources. between protocol and review), we adapted the outcome categories
used in the previous version of the review, as described below.
OBJECTIVES In our adaptations, we also sought to broaden outcome areas in
To review the effects of music therapy, or music therapy added to order to not only address specific skills (e.g. social adaptation;
standard care, for autistic people. communicative skills such as eye contact, imitating gestures or
words), but also wider areas of capacity (e.g. adaptive behaviour
in more than just the social domain; communication including
all domains of verbal or non-verbal communication, pragmatics,
language structure, and communication behaviours such as
withdrawal within a group).

Informed decisions.
We considered the broad-based measures 'global Secondary outcomes

improvement' (binary) and 'total autism symptom
Secondary outcomes included the following.
severity' (continuous) as primary outcomes. Although not
endorsed when applying a social-model approach to autism, 8. Adaptive behaviour: continuous; this could be measured as
measures relating to these overall categories are still considered positive adaptive behaviours (enabling a person to get along in
important in a medical-model perspective on autism which is their environment with greatest success and least conflict with
likely to be relevant for many policymakers, service providers others) or as maladaptive, dysfunctional behaviours (which stop a
and clinicians. As in the 2014 version of this review, we also person from adapting to new or difficult circumstances, including
regarded outcome measures in all areas of social communication 'restricted and repetitive behaviours');
as primary outcomes as they refer to the core characteristics 9. Quality of family relationships: continuous;
defining ASD (social interaction, non-verbal communication, verbal 10. Identity formation: continuous; including self-esteem and
communication). In addition, we moved the category of 'quality related concepts;
of life' (secondary outcome 'quality of life in school, home, and 11. Depression: continuous;
other environments' in the 2014 version of this review) into primary 12. Cognitive ability: continuous; including attention and
outcomes due to the increased relevance of this outcome to autistic concentration.
individuals and their families, as demonstrated in recent studies
and reports (e.g. McConachie 2015; Provenzani 2020). To keep this Changes in generalised skills that are measured outside of the
review focused and manageable for users (McKenzie 2021), we immediate therapy context pose the biggest challenge for any
merged previously separate outcomes that concern specific sub- interventions for autism (Warren 2011). Generalised outcomes
skills of social interaction ('initiating behaviour', 'social-emotional refer to changes that generalise to other behaviours and to other
reciprocity', 'joy'), with the wider category of 'social interaction'. contexts across settings, people, or materials. In the Summary of
Finally, we retained 'adverse events' as a primary outcome findings 1, we report the results of seven generalised outcomes
category. (all listed under Primary outcomes) measured immediately post-
intervention.
We regarded other commonly examined outcome measures in
areas not specific to defining ASD characteristics as secondary We grouped outcome time points as follows: during
outcomes. The outcome 'social adaptation skills' was re-labelled the intervention (previously labelled "within sessions/non-
as 'adaptive behaviour'. In order to address outcomes that generalised"); immediately post-intervention; one to five months
are regarded as highly relevant by autistic people, their family post-intervention; six to 11 months post-intervention; 12 to 23
members and professionals (Lipinski 2019; McConachie 2015) and months post-intervention; and 24 to 35 months post-intervention.
that were evaluated in included studies, we newly added 'identity Where outcomes were measured at multiple time points during the
formation' (including self-esteem) and 'depression' as secondary course of therapy, we used mean values of all data from the second
outcome categories. therapy session onwards.
Finally, we removed the outcome category 'hyperacusis Search methods for identification of studies
(hypersensitivity to sound)', as we did not find it measured in any
study, or mentioned in any review. We ran the searches for this update in July 2020 and again in
August 2021. We revised the original search strategy by removing
Data sources could have included non-standardised or redundant search terms, and by adding relevant database sources
standardised instruments (for a review of relevant standardised which were either not available at the time of search for the
instruments, see Ozonoff 2005; McConachie 2015; Provenzani previous update (e.g. MEDLINE Epub Ahead of Print) or not
2020), parent or teacher report, or school records. Data from rating routinely included previously (e.g. trial registers). Where possible,
scales were only included if the instrument was either a self-report searches were limited to the period since the last update (2013
or completed by an independent rater or relative (i.e. not the onwards). For newly added databases, searches were conducted
therapist, unless reconfirmed by an independent rater). since their inception.
Primary outcomes Electronic searches

Primary outcomes included the following. The Cochrane Developmental, Psychosocial and Learning
Problems Group Information Specialist, Margaret Anderson,
1. Global improvement: binary (improved versus not improved or conducted systematic searches in the following databases for
unknown, on a scale measuring clinical global impressions or on randomised controlled trials and controlled clinical trials without
a global measure used as primary outcome in a study); language or publication status restrictions.
2. Social interaction: continuous;
1. Cochrane Central Register of Controlled Trials (CENTRAL; 2021,
3. Non-verbal communication: continuous;
Issue 8), part of the Cochrane Library (searched 4 August 2021).
4. Verbal communication: continuous;
2. MEDLINE Ovid (1946 to July week 4 2021).
5. Quality of life: continuous; could be measured in various
3. MEDLINE In-Process & Other Non-indexed Ciations Ovid (1946
contexts (school, home, other) and with varying scope
to 3 August 2021).
(individual, family);
4. MEDLINE EPub Ahead of Print Ovid (3 August 2021).
6. Total autism symptom severity: continuous;
5. Embase Ovid (1974 to 3 August 2021).
7. Adverse events: binary (any adverse event/no adverse event), as
defined by study authors. 6. LILACS (lilacs.bvsalud.org/en/; searched 4 August 2021).

Informed decisions.
7. APA PsycINFO Ovid (1806 to July week 4 2021). Information Specialist’s portal: https://community.cochrane.org/
8. CINAHL EBSCOhost (1937 to 4 August 2021). organizational-info/resources/resourcesgroups/ information-
9. ERIC EBSCOhost (1966 to 4 August 2021). specialists-portal. In addition, more detailed information
regarding evaluations of the Screen4Me components can be found
10.Sociological Abstracts Proquest (1952 to 4 August 2021).
in the following publications: Marshall 2018; Noel-Storr 2020; Noel-
11.Proquest Global Dissertations & Theses (searched 4 August Storr 2021; Thomas 2020.
2021).
12.Proquest Music Periodicals Database (1996 to 4 August 2021). Four authors (CE, LFP, MG, GV) independently inspected all titles
13.Proquest Performing Arts Periodicals Database (1864 to 4 August and abstracts identified from the search in such a way that each
2021). record was screened by two authors. We obtained potentially
relevant papers and resolved any disagreement about eligibility
14.RILM Abstracts of Music Literature Online (1967 to 4 August
through discussion or consultation with the other authors. For
2021).
non-English study reports, we provided for their translation. We
15.Cochrane Database of Systematic Reviews (CDSR; 2021, Issue 8), recorded the reasons for excluding trials.
part of the Cochrane Library (searched 4 August 2021).
16.Epistemonikos (www.epistemonikos.org/en/; searched 4 August We recorded the selection process in sufficient detail to produce a
2021). PRISMA flow diagram (Liberati 2009).
17.ClinicalTrials.gov (clinicaltrials.gov/ct2/home; searched 5
Data extraction and management
August 2021).
18.WHO International Clinical Trials Registry Platform Four reviewers (CE, LFP, MG, GV) independently performed data
(apps.who.int/trialsearch/; searched 5 August 2021). extraction using a data collection form so that data from each
study were extracted by two reviewers. We made sure that studies
Detailed search strategies for this update are reported in Appendix in which any of the reviewers were involved with were dealt
1. Details of the previous search strategies are available with by two other reviewers not involved in these studies. The
in Geretsegger 2014. data collection form was initially piloted to ensure feasibility
and included details on study design, participants, interventions,
Searching other resources outcomes including measurement time points and allocation to
Adverse events outcome categories, and funding sources. Any disagreements were
resolved by discussion, or consultation with the other reviewers, or
We did not perform a separate search for adverse events. We both. When necessary, we contacted the study authors to provide
considered adverse events described in included studies only. missing data.
Searching reference lists Assessment of risk of bias in included studies
We checked the bibliographies of included studies and relevant Four authors (KM, MG, LFP, GV) independently assessed the risk
reviews (Accordino 2007; Ball 2004; Brondino 2015; De Vries 2015; of bias of each included study using the Cochrane risk of bias
Pater 2017; Reschke-Hernández 2011; Shi 2016; Simpson 2011; tool (Higgins 2011). We made sure that studies where any of the
Weitlauf 2014; Whipple 2004; Whipple 2012) for further references reviewers were involved were dealt with by other reviewers not
to relevant trials. involved in these studies. Any disagreements were resolved by
discussion, or consultation with the other reviewers, or both.
Searching by contacting individuals or organisations
We contacted experts and organisations in the field through For each included study, we presented the risk of bias assessments
correspondence in researcher networks, conferences and social in a table where the judgement of the review authors (low, high or
media to gather information on ongoing trials and any relevant unclear risk of bias) was followed by a text box providing details on
material not captured by our searches. Where necessary, we the available information that led to each judgement.
contacted authors of key papers and abstracts to request further
We assessed the following items:
information about their trials.
1. random sequence generation;
2. allocation concealment;
Selection of studies 3. blinding of participants and personnel;
We used Cochrane’s Screen4Me workflow to help assess the 4. blinding of outcome assessment;
search results. Screen4Me comprises three components: known 5. completeness of outcome data;
assessments — a service that matches records in the search 6. selective reporting; and
results to records that have already been screened in Cochrane
7. other sources of bias.
Crowd and been labelled as an RCT or as Not an RCT; the
RCT classifier — a machine learning model that distinguishes The criteria for assigning judgements of high, low and unclear risk
RCTs from non-RCTs and, if appropriate, Cochrane Crowd — of bias are provided in Appendix 2.
Cochrane’s citizen science platform where the Crowd help to
identify and describe health evidence. For more information Measures of treatment effect
about Screen4Me and the evaluations that have been done,
Where available, we used individual participant data (IPD) in order
please go to the Screen4Me webpage on the Cochrane
to calculate measures of treatment effect consistently.

Informed decisions.
Binary data method in RevMan and possibly a change score or a positive

correlation estimate) might show no effect in this update.
We calculated the risk ratio (RR) and corresponding 95% confidence
interval (95% CI) for binary outcomes. The number needed to treat Unit of analysis issues
for one beneficial outcome was calculated, where appropriate.
Cluster-RCTs
Continuous data
For cluster-RCTs, we adjusted the sample size according to the
We preferred endpoints over change scores. If IPD were available, design effect, based on an intraclass coefficient calculated from
the distributions of values were visually checked for skewness. IPD, if available.
Where skewness was found, we attempted to remove it by
log-transformation. We then examined how log-transformation Cross-over trials
influenced the effect size estimate and used the more conservative The appropriateness of cross-over designs is difficult to assess.
estimate. In general, autism as a lifelong condition lends itself well to such
designs. However, it is less clear how lasting any effects of music
We calculated the standardised mean difference (SMD) and
therapy may be. In general, we judged cross-over designs as
corresponding 95% CI for all continuous outcomes. When
appropriate unless there was clear evidence to the contrary (e.g. a
combining different scales for the same outcome, it was necessary
clearly irreversible outcome). We therefore combined the results of
to standardise the effects in order to make them comparable. When
cross-over trials with the results of parallel-group trials, and used
combining results for the same scale, either the mean difference
data from all periods in order to retain a maximum of information
(MD) or SMD could have been used. We decided to use SMD in
provided by those studies. Data from washout periods in cross-over
order to facilitate the interpretation of effect sizes as small (up
studies were excluded from the analysis.
to 0.2), medium (around 0.5) or large (0.8 and above) based on
guidelines that are commonly used in the behavioural sciences Multiple treatment arms
(Cohen 1988; Schünemann 2021a ). In the absence of any anchor-
based minimally clinical important differences (MCIDs) for the For studies including more than one relevant music therapy group
outcomes in this review, the general guidelines for the behavioural or more than one relevant control group, we combined the data of
sciences developed by Cohen 1988 state that an effect size needs to the relevant groups by calculating a weighted mean and pooled SD.
reach at least a level of 0.2 to be regarded as potentially meaningful;
effects smaller than 0.2 may be negligible. It is noted that the choice Dealing with missing data
of SMD or MD does not usually affect the significance level of the We assessed loss to follow-up and dropouts in the included studies
results and the authors cautiously assessed whether this was the as reported in the risk of bias tables. Where unclear, we contacted
case. the study authors to confirm any loss to follow-up and dropouts
in their studies. We applied an intention-to-treat analysis (except
All SMDs, regardless of whether the study was a parallel or a cross- for adverse events) for available cases and did not impute missing
over design, were standardised by the pooled standard deviation values for continuous outcomes. We are aware that this may
between participants, rather than the standard deviation of the introduce bias if being lost to follow-up is related to a participants’
difference within participants. This is the standard procedure, response to intervention (Moher 2010). Therefore, we examined
which enables comparisons of different scales and facilitates the impact of studies with high risk of bias due to dropout using
interpretation of the magnitude of effects (Cohen 1988; Gold 2004). sensitivity analyses, where these studies were excluded.
The calculation of the standard error then depends on the study
design. For parallel designs, the standard error was calculated using Assessment of heterogeneity
the standard formulae for SMDs as implemented in RevMan Web
(RevMan Web 2020). For cross-over studies, outcomes are usually Because statistical tests of heterogeneity have low power,
positively correlated within participants; we assumed a correlation particularly when the number of studies is low, we relied primarily
of 0 as a conservative estimate; this avoided giving too high weight on descriptive analyses of heterogeneity. We visually inspected
to small studies and also enabled use of standard methods for SMDs forest plots for consistency of results and calculated the I2 statistic
within RevMan (Elbourne 2002; Higgins 2021). (Higgins 2002), which describes the proportion of variation in
point estimates that is due to heterogeneity rather than sampling
For studies where outcomes were measured on several occasions error, and followed suggested threshold bands for interpreting the
during each treatment intervention, we used the mean of all I2 statistic which define 0% to 40% as "might not be important";
measurements from the second occasion onwards. Where the same 30% to 60% as "may represent moderate heterogeneity"; 50%
outcome was measured on multiple occasions using the same to 90% as "may represent substantial heterogeneity"; and 75%
scale, we calculated the mean and the pooled SD and entered these to 100% as "considerable heterogeneity" (Deeks 2021). We
into RevMan. Where the same outcome was measured on multiple supplemented this by calculating the Chi2 statistic to determine
occasions using different scales, we calculated a mean effect size the strength of evidence that the heterogeneity was genuine.
for that study outcome and entered that into RevMan (along with We investigated possible sources of heterogeneity when it was
SD 1 and mean in control 0). detected.
In comparison to the previous review update, these procedures Assessment of reporting biases
ensure better consistency and transparency, but also tend to
We used funnel plots to investigate any relationship between effect
show more conservative results. Thus, a study that in Geretsegger
size and study precision in cases where 10 or more studies were
2014 showed a significant effect (using the generic inverse variance
pooled for an outcome. With other design aspects equal, a funnel

Informed decisions.
plot would be symmetric within chance variation in the absence of 2013). We made sure that studies in which any of the reviewers
publication bias; a noticeable asymmetry may therefore indicate were involved with were dealt with by two other reviewers not
a strong publication bias. However, because the method may involved in these studies. Any disagreements were resolved by
not work well when larger studies differ in other design aspects, discussion, or consultation with the other review authors, or both.
as well as because of its subjective interpretation, we did not The certainty of the evidence for each outcome was graded as
interpret a lack of an apparent asymmetry as evidence of absence high, moderate, low, or very low, according to the presence of
of publication bias. the following five criteria: risk of bias, inconsistency, indirectness,
imprecision and publication bias. Downgrading the certainty of
Data synthesis evidence for the included study outcomes was related to issues
Using RevMan Web (RevMan Web 2020), we conducted meta- concerning the risk of bias (e.g. reported randomisation; blinding
analyses utilising RRs for dichotomous outcomes and SMDs for of outcomes; incomplete outcome data) as well as imprecision
continuous outcomes. A fixed-effect model was initially used (e.g. wide CI, total number of participants lower than 400). We
for all analyses. If a common effect size was not tenable downgraded up to a maximum of three levels. We presented these
because a substantial amount of heterogeneity (i.e. 50% or ratings in the summary of findings table and provided our reasons
higher; Deeks 2021) was identified that could not be explained for downgrading the certainty of the evidence in the explanations.
by clinical subgroups in the outcome domain immediately
post-intervention (see Subgroup analysis and investigation of
RESULTS
heterogeneity), we chose a random-effects model. Where we
Description of studies
conducted fixed-effect analyses, we also examined whether
random-effects analyses would have altered the results by Results of the search
conducting sensitivity analyses, and reported any differences in
The electronic searches for this update identified a total of
the Effects of interventions section. We used the inverse variance
1356 records (see Figure 1). These were imported in EndNote where
method, which is most commonly used, in random-effects analyses
355 duplicates were identified, leaving 1001 records from electronic
of dichotomous outcomes and in all analyses of continuous
searches. Seven additional records were identified through other
outcomes. In fixed-effect meta-analyses of dichotomous outcomes,
sources, so that 1008 records needed to be screened. We used
we used the Mantel-Haenszel method, which is the default method
Cochrane’s Screen4Me workflow to help screen the 1001 records
in RevMan Web and is commonly preferred because it has better
from the electronic searches. First, we identified 28 database
statistical properties when there are few events (Deeks 2021).
records of reviews or systematic reviews which we separated
Subgroup analysis and investigation of heterogeneity from the rest of the records. The remaining 973 records from
electronic searches were classified using Cochrane’s Screen4Me
When substantial heterogeneity was identified (I2 ≥ 50%), we workflow to help identify potential reports of randomised trials.
examined the impact of clients' age (children versus adolescents or The results of the Screen4Me assessment process can be seen
adults), intensity of therapy (i.e. number and frequency of music in Figure 2 (July 2020 search) and Figure 3 (August 2021 search).
therapy sessions), and treatment quality (i.e. adequate music We excluded 321 records as they were ineligible regarding study
therapy methods; adequate training of therapists; see definitions type (267 records when applying the Sceen4Me workflow on the
specified in Appendix 2, 'Other bias') in subgroup analyses. results of the July 2020 search, and 45 records following the
August 2021 search). Based on title and abstract assessment, we
Sensitivity analysis then screened the remaining 652 records left in after Screen4Me
We conducted sensitivity analyses to determine the impact of and the seven records identified through other sources, and
attrition bias risk by removing studies at high risk of attrition excluded 624 (July 2020: 509; August 2021: 115). We examined
bias. We also investigated the impact of the choice of model by the remaining 35 records in full text, and excluded nine (Bringas
conducting a random-effects analysis where fixed analysis was 2015; Cowan 2016; Dezfoolian 2013; Gooding 2011; Iseri 2014; Kim
chosen and comparing the findings. 2000; Mendelson 2016; Sanglakh Goochan Atigh 2017; Yoo 2018;
see Characteristics of excluded studies). Six of these were excluded
Summary of findings and assessment of the certainty of the because they were not RCTs or CCTs; one because participants were
evidence not diagnosed with ASD; one because the intervention was not
music therapy; and one because no relevant comparison condition
We created a summary of findings table for our main comparison:
was included. Additionally, four relevant ongoing studies were
music therapy compared with placebo therapy or standard care. We
identified, and another ongoing study is still awaiting classification.
included the following primary outcomes, assessed immediately
Thus, we included 16 new studies from 20 reports, along with
post-intervention: global improvement; social interaction; non-
10 studies from the previous update (including a new report of
verbal communication; verbal communication; quality of life; total
a previously included study, Thompson 2014), which brought the
autism symptom severity; adverse events.
total number of included studies to 26 (see Characteristics of
Four review authors (KM, MG, LFP, GV) assessed the overall certainty included studies). 25 of these studies were included in the meta-
of the body of evidence using the GRADE approach (Schünemann analysis. Figure 4 (a) shows the accumulation of studies over time.

Informed decisions.
Figure 1. Study flow diagram

Informed decisions.
Figure 1. (Continued)

Informed decisions.
Figure 2. Sreen4Me summary diagram - July 2020 search

Informed decisions.
Figure 3. Screen4Me summary diagram - August 2021 search

Informed decisions.
Figure 4. Accumulation of evidence from 1995 to 2020. Key: black circles = parallel design; red circles = cross-over
design. Bubble sizes in panels (c) and (d) reflect number of participants randomised.
Included studies (Bharathi 2019), and one in Iran (Ghasemtabar 2015). Four studies
were conducted in Europe, i.e. France (Rabeyron 2020), Spain
Twenty-six studies met the criteria for the review
(Mateos-Moreno 2013), Turkey (Yurteri 2019), and the UK (Porter
(see Characteristics of included studies). Of these, three studies
2017). One study was conducted in Brazil (Gattino 2011) and one
were included in the first version of this review in 2006, seven
in Australia (Thompson 2014). Finally, one study (Bieleninik 2017)
studies were added for the update of 2014, and 16 new studies
was a multinational trial that recruited participants in nine different
(from 20 reports) were added for the present update (see Table
countries across the world (Australia, Austria, Brazil, Korea, Israel,
1 for details on this and on further summarised characteristics of
Italy, Norway, UK, USA).
included studies).
IPD were available for 14 studies, either published or from
Most studies (n = 12) were conducted in North America, of which
correspondence with authors.
11 were in the USA and one, Sharda 2018, in Canada. Seven studies
were conducted in Asia, specifically three in China (Chen 2010; Chen
2013; Huang 2015), two in Korea (Kim 2008; Moon 2010), one in India
Informed decisions.
Length of trials participants in the music therapy group had associated language
impairments.
The mean duration of follow-up was 3.0 months (SD = 2.87;
median 2.5; range 3 days to 12 months). The mean duration of the Intelligence quotient (IQ) was reported only in four studies, and was
intervention was 2.5 months (SD = 2.05; median 2 months; range evaluated using different instruments. Bieleninik 2017 used either
3 days to 8 months). Figure 4 (c) and (d) shows the duration of the Kaufman Assessment Battery for Children (KABC; Kaufman
interventions and follow-up, respectively. It can be seen that most 1987), other instruments, or clinical judgement, with 45% of the
studies lasted up to about six months (cross-over trials up to three sample having an IQ < 70. Gattino 2011 adopted the Raven's
months). Coloured Progressive Matrices as a cognitive measure in 22
participants (Pasquali 2002), with six having ID. Two trials used
Participants
the Wechsler scales in line with participants' chronological
Age age: Sharda 2018 used the Wechsler Abbreviated Scale of
Intelligence (WASI; Wechsler 1999) or the Wechsler Intelligence
Most studies (n = 21) included only children aged between two
Scale for Children (WISC; Wechsler 1949), while Rabeyron
and 12 years. One study, Porter 2017, included children and
2020 used the Wechsler Preschool and Primary Scale of Intelligence
adolescents, with ages ranging between eight and 16 years. Another
(WPPSI; Wechsler 1967). Finally, Buday 1995 reported participants
study, Sa 2020, recruited students aged 10 to 14, but this study's
to be ranging from mildly to severely mentally retarded (according
data were not used in the meta-analyses. Two studies recruited
to DSM III-R), but did not systematically evaluate the IQ of
both children and adults who were between nine and 21 years
participants.
old (Schwartzberg 2013; Schwartzberg 2016). Finally, the study
by Mateos-Moreno 2013 included only adults, with a mean age of Autism severity
25 years. The majority of the participants were males (range 50 to
100%). Severity levels were reported in 14 studies, ranging from mild to
severe autism, and were mostly evaluated using the CARS (Bharathi
Diagnosis 2019; Buday 1995; Chen 2010; Gattino 2011; Ghasemtabar 2015;
Kim 2008; LaGasse 2014; Lim 2010; Mateos-Moreno 2013;
All participants had received a diagnosis of ASD according to
Rabeyron 2020; Sa 2020; Sharda 2018). Levels of functioning
current or past classification systems (ICD and DSM), whether
and adaptive abilities at baseline were systematically assessed
identified by a psychological assessment or a psychiatric diagnosis.
only in four studies: Sharda 2018 and Thompson 2014 used
The study by Porter 2017 included participants with different
different versions of the Vineland Scales (Sparrow 1998; Sparrow
diagnoses (i.e. anxiety, depression, or ASD); however, only
1984); Chen 2013 and Kim 2008 used the Psychoeducational Profile
participants with an ASD diagnosis were included in the meta-
(PEP; Schopler 1979).
analyses.
Setting
Standardised tools for diagnosis were used in eight studies
(Bieleninik 2017; Ghasemtabar 2015; Gattino 2011; Kim 2008; The participants received therapy either at home (Thompson
Mateos-Moreno 2013; Rabeyron 2020; Sa 2020; Sharda 2018). 2014), at school (Brownell 2002; Buday 1995; Sa 2020), in hospital
Specifically, the Autism Diagnostic Observation Schedule (Chen 2010; Chen 2013; Gattino 2011; Huang 2015; Moon 2010),
(ADOS; Lord 1999) was used in three studies (Bieleninik 2017; at outpatient therapy centres (Bieleninik 2017; Ghasemtabar 2015;
Kim 2008; Sharda 2018) for diagnostic confirmation. Of these, Kim 2008; Mateos-Moreno 2013; Porter 2017; Rabeyron 2020),
two studies (Bieleninik 2017; Sharda 2018) used the Autism or a combination thereof (Farmer 2003; Lim 2010). Two studies
Diagnostic Interview-Revised (ADI-R; Lord 1994) in addition to the were conducted during summer camps (Schwartzberg 2013;
ADOS. The Childhood Autism Rating Scale (CARS; Schopler 1980) Schwartzberg 2016). For the remaining seven studies, the therapy
was adopted in five studies as a diagnostic tool (Gattino 2011; setting was not reported.
Ghasemtabar 2015; Kim 2008; Rabeyron 2020; Sharda 2018). The
High-Functioning Version of the Childhood Autism Rating Scale Study size and design
(CARS2-HF; The present systematic review involved a total of 1165 participants,
Schopler 2010) was used in Sa 2020. In the Mateos-Moreno with sample size ranging from 4 (Brownell 2002) to 364 (Bieleninik
2013 study, the diagnosis of ASD was confirmed using 2017). The median sample size was 24 participants (M = 45, SD = 70).
the Structured Clinical Interview for DSM IV Axis I Disorders (SCID-
I; First 2004). Three studies (Buday 1995; Lim 2010; Lim 2011) Twenty trials adopted a parallel design, of which two were cluster-
reported that the ASD diagnoses were performed by healthcare randomised (Schwartzberg 2013; Schwartzberg 2016). Six studies
providers of participants. LaGasse 2014 included participants with had a cross-over design (Arezina 2011; Brownell 2002; Buday
'a formal documentation of ASD'. 1995; Kim 2008; Lim 2011; Thomas 2003). The high proportion
of parallel designs is in contrast to the previous update, where
With a few exceptions (Brownell 2002; Mateos-Moreno 2013; the majority of included trials used cross-over designs. The cross-
Rabeyron 2020; Sharda 2018), the studies included both non-verbal over trials included in this update were designed to compensate
and verbal children with varied cognitive and adaptive abilities, for small sample sizes: the cross-over trials ranged from four to
ranging from mild to severe autism. Brownell 2002 recruited 22 participants, whereas the parallel trials ranged from 10 to 364
four verbal children with 'at least prereading skills'. The Mateos- participants. From Figure 4 (b) it can be seen that the sample size of
Moreno 2013 study included only young adults with severe studies tended to increase over time, especially in parallel trials.
autism. Rabeyron 2020 reported that all participants had an IQ
below 70. Conversely, Sharda 2018 included only participants
without intellectual disability (ID), although it was reported that 13
Informed decisions.
Interventions offering opportunities for the individual to make choices and

to initiate contact. Generally, the therapist's interventions were
Music therapy
depicted as drawing on the individual person's skills, interests,
The majority of studies included in this review examined music preferences, and motivations as well as on their immediate
therapy in an individual (i.e. one-to-one) setting (n = 13). In eight expression and behaviour. By attuning to the individual musically
trials, music therapy was delivered in a group setting (Bharathi and emotionally, the therapists create moments of synchronisation
2019; Ghasemtabar 2015; LaGasse 2014; Mateos-Moreno 2013; that help the individual to experience and recognise core elements
Rabeyron 2020; Sa 2020; Schwartzberg 2013; Schwartzberg 2016). of reciprocal communication (Kim 2008; Schumacher 1999a;
One study reported that music therapy was delivered either Schumacher 1999b; Stephens 2008; Thompson 2014; Wigram
individually or in small groups of up to three people (Yurteri 2009). Mateos-Moreno 2013 combined music therapy with dance/
2019). Thompson 2014 applied a family-based setting where movement therapy activities, such as massages with small balls,
parents or other family members were also involved in therapy simulation situations, imitation, role-playing, and dancing.
sessions. In four studies, it was unclear whether music therapy
sessions were conducted in an individual or group setting (Chen Several of the studies employed specifically developed treatment
2010; Chen 2013; Huang 2015; Moon 2010). guidelines in the form of a treatment contingency plan (Thompson
2014), or a treatment manual (Bieleninik 2017; Ghasemtabar 2015;
The frequency of music therapy sessions ranged from daily to Kim 2008; LaGasse 2014; Porter 2017; Sa 2020; Sharda 2018).
weekly. In seven studies music therapy was provided daily, all In these protocols, principles and procedures of therapy are
with a very short duration of one or two weeks. Of the studies specified whilst allowing the therapist to adapt interventions
that provided music therapy over a longer time period, it was flexibly according to the child's needs and the specific requirements
provided weekly in nine studies, twice weekly in six studies, and of the situation.
in the remaining studies three (Bharathi 2019), four (Chen 2010),
or six times (Huang 2015) per week. One study (Bieleninik 2017) Comparators
randomised to either one or three sessions per week. The duration 'Placebo' therapy
of sessions ranged from 10 (Arezina 2011; Lim 2010) to 60 minutes
(Ghasemtabar 2015; Mateos-Moreno 2013) with a median of 30 A total of 15 studies used a 'placebo' therapy to control for the non-
minutes. specific elements of the therapy. Thirteen of these used a 'placebo'
activity to control for the non-specific effects of therapeutic
In two studies, we combined the data from the two music attention. Since, in all of these studies, music was considered as the
therapy groups (i.e. low-intensity and high-intensity music therapy specific ingredient of music therapy, the placebo conditions were
in Bieleninik 2017; social stories music therapy and music therapy constructed to closely match the music therapy condition, only that
without lyrics in Chen 2013). music was not used. Specifically, a social story was read instead of
sung to the participants (Brownell 2002; Moon 2010; Schwartzberg
Content of the intervention 2013; Schwartzberg 2016); rhythmic or normal speech was used
Twelve studies utilised a highly structured approach to music instead of singing (Buday 1995; Lim 2010; Lim 2011); play activities
therapy using receptive techniques (i.e. listening to live or, in the were offered without using songs or musical instruments (Farmer
case of Lim 2010 and Rabeyron 2020, pre-recorded music presented 2003); the therapist engaged in interaction with the child by
by the therapist) or a mix of receptive elements and active music- responding to the child's behaviour non-musically and using non-
making. Songs sung by the music therapist were composed or music toys (Arezina 2011; Kim 2008; Sharda 2018; Thomas 2003);
chosen individually for the participants and were usually used or the participants were involved in a social skills group (LaGasse
with specific aims. For example, songs were based on a social 2014). Two studies (Bharathi 2019; Rabeyron 2020) used another
story addressing a central problem behaviour of the particular type of 'placebo' therapy consisting of passive music listening. In
individual in treatment (Brownell 2002) or autistic individuals in both studies participants passively listened to songs played using
general (Schwartzberg 2013; Schwartzberg 2016); they contained a CD player, without any interaction with the therapists. Thus, not
signs and words to be learned (Buday 1995; Lim 2010; Lim 2011); the music, but the therapist's attention was seen as the specific
or they were used to build a relationship and to provide a safe ingredient in these studies.
and understandable structure for the participants in the study Standard care
(Chen 2010, Chen 2013, Farmer 2003). Active music-making by the
participants, which is often typical for music therapy in clinical Eleven studies compared music therapy with standard care.
practice (Wigram 2006), was reported in five of those studies (Chen In Bieleninik 2017, the control group received enhanced standard
2010; Chen 2013; Farmer 2003; Moon 2010; Sa 2020). Participants care, which consisted of the routine care available at the site,
were invited to play guitar, pitched or unpitched percussion plus three 60-minute sessions of parent counselling over the
instruments, and sing songs. Playing instruments was partly used five months of intervention (at 0, two, and 5 months). Three
to reinforce adjusted behaviour. Moon 2010 used a music drama studies (Chen 2010; Chen 2013; Huang 2015) compared music
based on a theory of mind approach, including narration, singing, therapy with a comprehensive/integrated treatment including
and musical instrument playing. several activities, such as auditory integration training, sensory
integration, special education, language therapy, speech therapy,
In the other fourteen studies, particular emphasis was put on and play therapy. Gattino 2011 reported that participants received
the interactive and relational aspects of music therapy. Music routine clinical services, including medical examinations and
therapy techniques included improvisation, songs, and structured consultations. Sa 2020, where a waiting-list control design
musical games. Interventions followed a non-directive approach was applied, and Ghasemtabar 2015 described no intervention.
and focused on engaging the individual in musical interaction, In Mateos-Moreno 2013, participants in the control group

Informed decisions.
were attending their regular therapies as well as receiving ii. The 'social communication' domain of the Childhood Autism
pharmacological treatment. Analogously, in the Porter 2017 study, Rating Scale (CARS; Magyar 2007; Brazilian version: Pereira 2008;
participants were following psychiatric counselling and/or Rapin 2008) was used in three studies (Chen 2013; Gattino
medication. In the Thompson 2014 study, participants received 2011; Rabeyron 2020). The CARS (Schopler 1980) is a 15-
varying forms of services and support from early childhood item observation-based behavioural rating scale administered by
intervention centres. Finally, in Yurteri 2019, participants in the health professionals for the diagnosis of children with autism
control condition received no treatment except monthly routine and pervasive developmental disorders. Total scores can range
child psychiatric follow-up and special education. between 15 and 60, with higher scores indicating higher severity.
The 'social communication' domain has been derived from
Multiple-armed trials the factor analysis of the CARS (see Magyar 2007; McConachie
Some studies included other conditions whose data were not 2015) and is composed of five items of the original tool, all
included in this review. Brownell 2002 reported observations related to social communication skills (i.e. imitation, verbal and
during a baseline period and a washout period with no nonverbal communication, consistency of intellectual responses
intervention. Arezina 2011 also observed behaviour in an and general impressions). Similarly to the full scale, this domain
'independent play' group, which we considered was neither was administered by investigators blind to group allocation
'placebo' therapy nor 'standard care'. Therefore, data from this (unclear for Chen 2013). As in Chen 2013 SD were missing, we
group were not included in this review. Lim 2010 and Lim imputed SD = 3 in line with other studies using the same scale.
2011 compared music training with both a speech training
(included) and a 'no training' group (excluded). iii. The 'social affect' (SA) subscale of the Autism Diagnostic
Obervation Schedule (ADOS; Lord 1999) was used in Bieleninik
Outcome measures 2017. The ADOS is a semi-structured, interactive observation
by trained health professionals. It has been designed to assess
Both generalised and non-generalised outcomes were used in the aspects of communication, social reciprocal interaction, play, and
included studies. Non-generalised outcomes refer to changes in stereotyped behaviours and restricted interests. It consists of four
the child's non-generalised behaviour in the same setting where modules, appropriate for individuals with different developmental
the intervention takes place, as opposed to generalised outcomes and language levels. ADOS-SA is composed of two subdomains, i.e.
which are observed in other settings (Warren 2011). 'language and Communication' and 'reciprocal Social interaction'.
The ADOS-SA score can range from 0 to 24 (module 1 and 2)
Primary outcomes
or 0 to 27 (module 3), with higher scores indicating greater
1) Global improvement symptom severity. In the study by Bieleninik 2017, it was rated by
Global improvement was defined as a binary outcome (improved independent, blinded health professionals.
versus not improved or unknown, on a scale measuring clinical
iv. The total score of the Social Responsiveness Scale
global impressions or on a global measure used as primary
(SRS; Constantino 2005) was used in four studies (Bieleninik 2017;
outcome in a study). The negative outcome was imputed for
LaGasse 2014; Sharda 2018; Thompson 2014). The SRS is a 65-
missing values, enabling a full intention-to-treat analysis. Global
item scale measuring the severity of autism symptoms as they
improvement was measured using the Clinical Global Impression
occur in natural social settings. The total score can range from 0
scale (CGI; Busner 2007) or if this was not available, the primary
to 195. Higher scores are indicative of greater symptom severity.
outcome chosen by study authors.
The SRS is rated by parents or teachers and it is appropriate for use
Rabeyron 2020 used the Clinical Global Impression-Severity with children from four to 18 years of age. In Thompson 2014, the
scale (CGI-S; Busner 2007). The CGI-S is a 7-point clinician- Preschool Version of the SRS was used. Sharda 2018 used the SRS-2,
rated scale used to rate the severity of a disorder, with higher scores a revised and more recent version of the SRS (Constantino 2012).
indicating greater severity. The scores range between 1 ('normal') For Sharda 2018, where the SD was not reported, we imputed SD =
and 7 ('among the most extremely ill patients'). 30 based on other studies using the same scale.
Seven studies (Bieleninik 2017; Bharathi 2019; Kim 2008; LaGasse
v. The Social Skills Rating System scale (SSRS; Gresham
2014; Porter 2017; Schwartzberg 2013; Thompson 2014) had a
1990), elementary form, was completed by participants' parents
clearly defined primary outcome (other than CGI) and provided IPD
in Ghasemtabar 2015. The total score can range between 0 and
from which to calculate global improvement.
80. Higher scores indicate higher social skills and thus favourable
2) Social interaction outcome.
Social interaction was examined in 14 studies. The following scales vi. The 'social approach behaviours' subscale of the Pervasive
were used: Developmental Disorder Behavior Inventory, Korean version
(PDDBI; Cohen 1999) was used in Kim 2008. The scale was filled
i. The TRIAD Social Skills Assessment (TSSA; Stone 2010) is out by professionals (i.e. a teacher or a therapist of the child) who
a 'criterion-based tool' which provides specific assessment were blind to experimental condition. Higher scores are indicative
considering parent, teacher, observation, and direct interaction of better social skills.
with the children aged six to 12 years. It consists of three
components: Problem Behavior Rating Scale, Social Skills Survey, vii. The total score of the Social Skills Improvement System (SSIS)
and Social Skills Rating Form. In Bharathi 2019, one of the rating scales (Gresham 2008) was used in one study (Porter
three components of the TSSA (i.e. the Social Skills Rating Form) 2017). The scale was rated by parents and self-rated by youth.
was used. Each item is rated on a 4-point Likert scale, with higher The SSIS is a scale with 75 (self) to 79 (parent) items across
scores indicating more favourable behaviours. 3 subdomains (i.e. social skills, competing problem behaviours,
Informed decisions.
academic competence). The total score can range from 0 to 225 coding supplemented by a trained research assistant who was blind
(self) or 237 (parent), with higher scores representing favourable to session order.
outcomes.
3) Non-verbal communication
viii. The Autism Social Skills Profile (ASSP; Bellini 2007) was used Non-verbal (i.e. gaze-related and gestural) communication was
in Schwartzberg 2013. The ASSP is a 49-item tool divided into three examined in 11 studies. Six studies used validated outcome
sub-categories: social reciprocity (SR), composed of 23 items; social measures, as follows:
participation (SP), composed of 12 items; and detrimental social
behaviours (DSB), composed of 10 items. Each item is rated on a 4- i. The 'non-verbal communication' domain of the CARS was used in
point Likert scale. Even though Schwartzberg 2013 calculated the three studies (Chen 2013; Gattino 2011; Rabeyron 2020).
ASSP score for each sub-category, the ASSP total score was used
for the outcome 'social interaction'. The scale was completed by ii. The Early Social Communication Scales (ESCS; Mundy 2003) is
participants’ legal guardians. a videotaped structured play-based assessment measuring non-
verbal social communication skills in children aged between six
ix. The Vineland Social-Emotional Early Childhood and 30 months. In Kim 2008, the shortened version of the ESCS
Scales (SEEC; Sparrow 1998) were used in one study (Thompson was used. The ESCS provides frequencies of scores for 'initiation
2014). The Vineland SEEC is a 88-item measure used to assess the of joint attention' and 'responding to joint attention'. The scoring
social and emotional functioning of children from birth through was administered by the researcher and by two trained research
5.11 years. In Thompson 2014, It was administered through a assistants who were blind to group assignment.
semi-structured interview with the child's parent participating
in the study. Only two out of three subscales (i.e. interpersonal iii. The Children’s Communication Checklist (CCC-2; Bishop 1998),
relationships; play and leisure time) were used in the study. used in Sharda 2018, is a parent/caregiver-administered 70-item
rating scale to measure children’s social communication skills
Some studies used more than one score to measure social across 10 domains. This tool is focused on the assessment of
interaction at the same time point. Bieleninik 2017 used both non-verbal communication, pragmatics, as well as aspects of
ADOS-SA and SRS; only ADOS assessors were blinded, however language structure and discourse. Sharda 2018 used the standard
both perspectives of parents (SRS) and professionals (ADOS-SA) general communication composite standard score as a measure of
were important so we merged both. Porter 2017 used parent the child's general pragmatics and communication ability. Higher
and self-reports of the same scale; both were considered equally scores indicate better social communication skills.
valid; we merged both to represent both perspectives. Thompson
2014 used both the SRS and Vineland SEEC; again, we merged both iv. The MacArthur-Bates Communicative Development Inventories
because both were equally valid. – Words and Gestures (MBCDI-W&G; Fenson 2007) are a set of
parent-rated measures designed to evaluate the verbal and non-
For the meta-analysis, given that some scales in this domain were verbal communicative skills of young children. The section 'action
'negative' (ADOS, SRS, CARS) and others 'positive' (ASSP, SSIS, and gestures' of the MBCDI-W&G was used as a measure of
SSRS), we reversed the 'negative' scales in the analysis so that non-verbal communication in Thompson 2014. Higher scores are
the positive sign of the analysis matches the positive meaning indicative of higher levels of non-verbal communication.
conveyed by 'social interaction' (i.e. positive effects represent a
favourable outcome). Four studies assessed non-verbal communication skills using
non-validated outcome measures, through the observation of
Three studies assessed social interaction skills using non-validated participants' behaviour within therapy sessions (Buday 1995;
outcome measures, through the observation of participants' Farmer 2003; Kim 2008; LaGasse 2014). Measures of non-verbal
behaviour within therapy sessions: communication skills in these studies are reported below:
i. In Arezina 2011, the researcher coded videotaped sessions for i. In Buday 1995, the outcome consisted simply of the number
'requesting (initiating joint attention)' behaviours such as pointing, of signs correctly imitated within a session.
giving an object to the therapist, or touching the therapist while
making eye contact; an independent observer additionally coded a ii. In Farmer 2003, a completed gesture was given a score of two, and
third of the session material. In Thomas 2003, 'requesting behavior' an attempt a score of one, and the outcome consisted of the sum
was defined in a similar way. Video tapes were coded by a music of these scores for all attempted and completed gestures within a
therapy intern and rated for two outcomes, task behaviour and session.
requesting.
iii. In Kim 2008, frequency and duration of eye contact (i.e. the child
ii. One study (Kim 2008) also investigated observed behaviours looking at the therapist) was coded by microanalytic analysis of the
related to social interaction in the intervention setting. These session material.
measures included frequency and duration of the child's turn-
taking, frequency of imitation behaviours, frequency and duration iv. In LaGasse 2014, video recordings of children in both groups
of both 'emotional synchronicity' and 'musical synchronicity', and were analysed for instances of group communication and
behaviours associated with the frequency and duration of joy (i.e. social interaction attempts. Two trained music therapy
smiling and laughing) on the part of the child. The coding procedure research assistants completed the coding of predefined behaviours
was conducted by the lead investigator by microanalytically (i.e., eye gaze, joint attention, initiation of communication,
(second by second) observing DVD recordings, with subsequent response to communication, withdrawal behaviours). Five-minute

Informed decisions.
clips were randomly selected from each session for each child. The 6) Total autism symptom severity
session order was concealed from the coders. Total autism symptom severity was measured in nine studies.
4) Verbal communication Outcome measures included the following:
Communicative skills in verbal communication were addressed in i. The CARS (Schopler 1980) was used in three studies (Bharathi
11 studies. The authors used the following outcome measures: 2019; Chen 2013; Rabeyron 2020), although in Chen 2013 CARS
scores were not reported or made available to us.
i. The 'verbal communication' domain of the CARS was used in three
studies (Chen 2013; Gattino 2011; Rabeyron 2020). ii. The total score of the ADOS (Lord 1999) was used by Bieleninik
2017. The ADOS total score is calculated summing up the raw scores
ii. Thompson 2014 used the subscales 'phrases understood', 'words of the ADOS-SA and 'restricted and repetitive behaviour' (ADOS-
understood' and 'words produced' of the MBCDI-W&G (Fenson RRB) scores.
2007).
iii. the Autism Treatment Evaluation Checklist (ATEC; Rimland
iii. Comprehension Checks (CCs) were used by Schwartzberg 1999) was used by LaGasse 2014. The ATEC is a 77-item checklist
2013 and Schwartzberg 2016. They consisted of a series of that includes four areas (speech and communication, sociability,
five close-ended questions (yes or no) to evaluate participants' sensory/cognitive awareness, health/physical behaviour). It is
comprehension of social stories. completed by parents, teachers and/or primary caretakers of
autistic children. Total ATEC scores range from 0 to 180. Lower
iv. The Peabody Picture Vocabulary Test (PPVT-4; Dunn 1981), a
scores on ATEC demonstrate higher functioning.
short, standardised measure of one-word receptive vocabulary,
was used in one study (Sharda 2018). The test requires the iii. The total score of the Autism Behavior Checklist (AuBC; Krug
participant to choose one of four colour pictures on a page. Higher 1980) was adopted as a measure of total symptom severity in
scores indicate better receptive vocabulary. four studies (Chen 2010; Chen 2013; Huang 2015; Yurteri 2019). It
consists of 57 items with higher scores indicating higher severity.
v. For Buday 1995, Farmer 2003, Lim 2010, and Lim 2011,
For Huang 2015, where no SD was reported, we imputed SD = 12
independent observers rated in-session behaviour by counting the
from other studies that used the AuBC (Chen 2010; Chen 2013;
frequency of appropriate verbal responses in a manner similar to
Yurteri 2019).
the previous outcome. The outcome measures used in these four
studies were unpublished. v. The Revised Clinical Scale for the Evaluation of Autistic Behavior
(ECA-R; Barthélémy 2003) was used by one study (Mateos-Moreno
5) Quality of life
2013). It is composed of 29 items with lower scores corresponding
Quality of life (QoL) was measured in three studies, using three to favourable outcomes.
different scales:
7) Adverse events
i. Bieleninik 2017 evaluated the QoL of both the child and the family
Two studies collected adverse event data. In Bieleninik 2017,
as a whole using a Visual Analogue Scale (VAS) ranging from 0 to
hospitalisation or other institutional stay (including pre-planned
100, where 0 corresponded to the worst and 100 to the best possible
stays) were included as adverse events; these and any other serious
QoL.
or non-serious adverse events were reported by parents. Porter
ii. Sharda 2018 used the Beach Center Family Quality of Life Scale 2017 collected serious adverse events and non-serious adverse
(FQoL; Park 2003) to assess satisfaction with different aspects of events related to study procedures. None of the other studies
family quality of life. FQoL is a 25-item questionnaire containing reported information on adverse events.
five subscales: family interaction, parenting, emotional well- Secondary outcomes
being, physical/material well-being, and disability-related support.
Higher scores correspond to better satisfaction in QoL. 8) Adaptive behaviour
Nine studies evaluated adaptive behaviours. Validated scales were

iii. Yurteri 2019 evaluated participants' QoL using the Pediatric
used in five studies, along with other non-validated measures:
Quality of Life Inventory (PedsQL; Varni 1999). It consists of a 23-
item scale designed to measure the core dimensions of health i. The Psychoeducational Profile (PEP; Schopler 1979; Muris 1997)
as delineated by the World Health Organization, as well as role was used by Chen 2010. The PEP consists of a series of toys, objects,
(school) functioning. In Yurteri 2019, the scale was completed and games which are offered to the child. It provides information
by parents according to participants' age. The PedsQL is a on developmental items and pathology items. Higher scores are
multidimensional scale composed of four dimensions (i.e. physical favourable. In the Chen 2010 study, a total score as well as the
functioning, emotional functioning, social functioning, school scores of three domains (i.e. relationship and emotions, interest
functioning). Moreover, three summary scores can be calculated in games and objects, sensory response) were provided. The total
(i.e. total scale score, physical health summary score, psychosocial scores were used as measure of adaptive behaviour.
health summary score). The Yurteri 2019 paper reported both the
total scale and the psychosocial health summary scores. Higher ii. The Child Behavior Checklist (CBC; Achenbach 2001), used
scores correspond to better quality of life. by Porter 2017, is a parent-rated tool consisting of 113 questions,
scored on a three-point Likert scale (0: absent, 1: occurs sometimes,
2: occurs often). Thus, lower scores are favourable. CBC scores were

Informed decisions.
not reported in the publication, but available in IPD from the study rated on a 4-level scale ranging from 'strongly agree' to 'strongly
authors. disagree'. Higher scores are indicative of positive parenting.
iii. The Aberrant Behavior Checklist (ABC; Aman 1985) is a 58- 10) Identity formation
item caregiver-report checklist designed to assess maladaptive Identity formation includes all the processes that allow autistic
behaviours in people with developmental disabilities. Higher people to develop a clear and unique view of themselves and
scores correspond to greater maladaptive behaviours. The ABC of their identity. Domains related to identity formation were
Total Score was used in Rabeyron 2020. evaluated in two studies.
iv. The maladaptive behaviours subdomain of the Vineland i. the Bandura self-efficacy scale (Bandura 1978) was used to
Adaptive Behavior Scales (VABS; Sparrow 1984) was used measure self-efficacy in the Moon 2010 study. The scale is
by Sharda 2018 to identify the presence of behavioural problems, composed of nine items. Higher scores indicate higher self-efficacy
such as challenging internalising and externalising behaviours. The levels.
scale is administered as a semi-structured interview to a parent or
caregiver. Lower scores are favourable. ii. the Fenigstein self-awareness scale (Fenigstein 1979) was used
to measure self-awareness in the Moon 2010 study. It is composed
v. Three studies investigated adaptive behaviours within the of 20 items, with higher scores indicating greater levels of self-
interventions setting (Arezina 2011; Kim 2008; Thomas 2003). awareness.
In Arezina 2011 and Thomas 2003, videotaped sessions were coded
for 'interaction (engaging in joint attention)' and 'on-task behavior', iii. the Rosenberg self-esteem scale (Rosenberg 1965) was adopted
respectively; this included activities such as following a direction, as a measure of self-esteem in both Moon 2010 and Porter 2017.
physically manipulating a toy in a functional manner, and imitating It is a 10-item self-report scale that measures global self-worth by
a movement or vocal sound. In Kim 2008, sessions were scored by measuring both positive and negative feelings about oneself. All
frequencies of 'compliant response', 'non-compliant response', and items are answered using a 4-point Likert scale format ranging from
'no response'. 'strongly agree' to 'strongly disagree'. The total score can range
between 10 and 40. Higher scores indicate higher self-esteem.
vi. Restricted and repetitive behaviours were measured
in Bieleninik 2017 using the ADOS-RRB domain (Lord 1999). Higher 11) Depression
scores indicate more severe repetitive behaviours.
Depression was evaluated in one study (Porter 2017), using
vii. Brownell 2002 addressed occurrence of individually targeted the Center for Epidemiological Studies Depression Scale for
repetitive behaviours outside therapy sessions. Independent Children (CES-DC; Faulstich 1986; Weissman 1980). This is a 20-
observers (i.e. teachers) counted how often the targeted behaviour item self-report questionnaire for young people between the ages
occurred in the classroom. The frequency count was used as the of six and 17. It asks young people to rate how many depressive
outcome measure. No published scale was used in the Brownell symptoms they have experienced in the last week. Higher scores
2002 study. represent higher levels of symptoms.
Where necessary, we reversed scores so that a high score on 12) Cognitive ability
adaptive behaviour indicated a favourable outcome. Cognitive ability was evaluated in one study (Sa 2020), using the
Test of Everyday Attention for Children 2 (TEA-Ch2; Manly 2016). The
9) Quality of family relationships
TEA-Ch2 is a tool for young people between the ages of five and
Family relationships were evaluated in three studies, with different 15 that assesses three areas of attention skills (selective attention,
tools: sustained attention, and attentional control/switching attention)
using eight tasks. However, data from this study were not included
i. Kim 2008 used the Mother Play Intervention Profile (MPIP), as the outcome measure was not applied by an independent rater,
a measure specifically developed for the study to describe but by the researcher who also administered the intervention
characteristics of interactions between mothers and autistic protocol (i.e. the therapist), thus violating this review's eligibility
children during a casual play situation at their home. Scores criteria for outcome measures.
were based on video observations conducted by the researcher,
supplemented by an independent observer's coding for a third of Funding sources
the sessions.
The American Music Therapy Association (AMTA) provided funding
ii. In Porter 2017, the McMaster Family Assessment Device support for two studies (LaGasse 2014: Arthur Flagler Fultz
(FAD; Epstein 1983) was completed by parents. The FAD is a 60-item Research Fund; Thomas 2003: Mid-Atlantic Region of the AMTA).
questionnaire that measures an individual’s perceptions of his/her University funding was available for two studies (Kim 2008:
family. Each item is scored on a 4-point scale. The higher the score, Aalborg University, Denmark; Thompson 2014: University of
the more problematic the family member perceives the family's Melbourne, Australia). The Thompson 2014 study was also
overall functioning supported by the Victorian Department of Education and Early
Childhood Development. Further funding sources included the
iii. Thompson 2014 used the Parent-Child Relationship Inventory Science and Engineering Research Board, Government of India,
(PCRI; Gerard 1994), a self-report questionnaire for parents New Delhi (Bharathi 2019); the Chongquing Natural Science
to assess the parent-child relationship and parents' attitudes Foundation (Chen 2010; Chen 2013); the Chongqing Medical
towards parenting. The full instrument consists of 78 items, Specialty Construction (Chen 2013); the Fund of Incentive to

Informed decisions.
Research of Porto Alegre Clinical Hospital and the Brazilian therapy sessions. The estimated sample size of children aged six
Research Council (Gattino 2011); the Big Lottery Fund (Porter 2017); to 12 years was 80. Primary outcomes included the CCC-2 and
Entreprendre pour Aider and the Academie Francaise (Rabeyron measures of brain connectivity of frontotemporal regions.
2020); the Canadian Institutes of Health Research and Quebec
Bioimaging Network (Sharda 2018). Bieleninik 2017 was supported Studies awaiting classification
by the Research Council of Norway, the University of Bergen, One potentially relevant study is awaiting assessment since the
Norway, POLYFON Knowledge Cluster for Music Therapy, and a information available in the trial registration was not sufficient to
range of further governmental and university funding sources and assess eligibility (NCT03267095); recruitment has not started. It is
foundations across participating countries (see Characteristics of planned to be a randomised, unblinded study, conducted in Egypt,
included studies for details). For the remaining 14 studies, no comparing the effects of a music therapy intervention to parent
funding sources were reported, or sources of support were reported counselling over a 12-month period. The researchers planned to
as 'nil' (Ghasemtabar 2015). recruit 60 children between three and seven years old with an
IQ > 75. The outcome was focused on verbal communication,
Ongoing studies
through the administration of an Arabic Language test evaluating
Four relevant studies were still ongoing at the time of assessment semantics, expressive morphology, syntax, and pragmatics.
(see Ongoing studies). Conducted in the USA, NCT03560297 used
a cross-over design and applied a parent-child music class Excluded studies
programme including parent training, peer inclusion, and musical Nine studies identified through the update search were excluded
play for 12 weeks, compared with a waiting-list programme. for the following reasons: six studies did not have an RCT or CCT
The estimated sample size of children aged 20 to 72 months design (six case series, i.e. studies comparing different treatments
was 68. Primary outcomes included a standardised motor that all participants received in the same order); one study
imitation assessment, and parent questionnaires on non-verbal because the intervention was not music therapy, but movement
communication, parenting stress, and parenting efficacy/quality. activities with music; one study because participants were not
diagnosed with ASD, but with severe neurological disorders; and
Conducted in South Korea, ISRCTN18340173 used a parallel
one study because it did not include a relevant comparison
design involving propensity score matching and applied weekly
condition (both groups were music therapy). See Characteristics of
improvisational music therapy sessions for one year in addition to
excluded studies, where in addition to the nine studies excluded
standard care, compared with standard care alone. The estimated
with reasons in this update, we also report seven studies that were
sample size of children aged 24 to 72 months was 50. Primary
excluded in previous versions of this review. From the fifty-nine
outcomes were the ADOS and the CARS-2.
studies excluded with reasons in the two previous versions of this
Conducted in Hong Kong, NCT04557488 used a parallel design review, these seven were selected in a process of reassessment
and applied a 12-week social skill intervention using group as the most relevant that one might expect to see in this review.
music therapy, compared with a 12-week non-musical intervention Six of them were excluded because they did not have an RCT or
(i.e. behavioural-based social skill group training). The estimated CCT design; one study because it was not an intervention, but an
sample size of children aged six to 13 years was 80. Primary assessment study.
outcomes included the CARS-2, the SRS-2, and in-session social
behaviour. Risk of bias in included studies
A visual representation of the included studies' risk of bias for each
Conducted in Austria and Norway, NCT04936048 uses a cross- domain, as specified below, is shown in Figure 5. Figure 6 provides
over design and applied 12 weekly sessions of one-on-one music a summary of the risk of bias results for each included study.
therapy with an equal number of non-musical one-on-one play
Figure 5. Risk of bias graph: review authors' judgements about each risk of bias item presented as percentages
across all included studies
Random sequence generation (selection bias)

Allocation concealment (selection bias)
Blinding of participants and personnel (performance bias): All outcomes
Blinding of outcome assessment (detection bias): All outcomes
Incomplete outcome data (attrition bias): All outcomes
Selective reporting (reporting bias)
Other bias
0% 25% 50% 75% 100%
Low risk of bias Unclear risk of bias High risk of bias

Informed decisions.
Figure 6. Risk of bias summary: review authors' judgements about each risk of bias item for each included study
Blinding of participants and personnel (performance bias): All outcomes

Blinding of outcome assessment (detection bias): All outcomes
Incomplete outcome data (attrition bias): All outcomes
Random sequence generation (selection bias)
Allocation concealment (selection bias)
Selective reporting (reporting bias)

Other bias
Arezina 2011 + ? ? ? + + +
Bharathi 2019 - - ? - + + +
Bieleninik 2017 + + ? + + + +
Brownell 2002 - ? ? ? + + +
Buday 1995 ? ? ? + + + ?
Chen 2010 ? ? ? ? + + +
Chen 2013 ? ? ? ? + + +
Farmer 2003 ? ? ? - + + +
Gattino 2011 + + ? + + + +
Ghasemtabar 2015 - - ? ? + + +
Huang 2015 ? ? ? ? + + ?
Kim 2008 + ? ? ? - + +
LaGasse 2014 + ? ? ? + + +
Lim 2010 ? ? ? + + + +
Lim 2011 + ? ? + + + +
Mateos-Moreno 2013 - - ? ? + + +
Moon 2010 - - ? - + + ?
Porter 2017 + + ? - + + +
Rabeyron 2020 + ? ? + + + +
Sa 2020 ? ? ? - + + +
Schwartzberg 2013 ? ? ? - - + +
Schwartzberg 2016 ? ? ? - - + +
Sharda 2018 + + ? + + + +

Informed decisions.
Figure 6. (Continued)
Schwartzberg 2016 ? ? ? - - + +
Sharda 2018 + + ? + + + +
Thomas 2003 ? ? ? ? + + +
Thompson 2014 + + ? ? + + +
Yurteri 2019 ? ? ? ? + + +
Allocation reports as outcomes were judged as being at unclear risk of bias

(Ghasemtabar 2015; LaGasse 2014; Yurteri 2019). In Thompson
Twenty of the included studies stated explicitly that randomisation
2014, measures were based on parent reports; however, they
was used to assign participants to treatment groups. Methods
contained internal safeguards to address bias as evidenced by
of randomisation included using computer-generated random
high correlations with non-parent-rated measures and high test-
sequences for determining allocation to an experimental condition
retest correlations (e.g. Pearson's r = 0.70, P = 0.01, for the SRS's
(Bieleninik 2017; Gattino 2011; LaGasse 2014; Rabeyron 2020;
one-month test-retest reliability). Nevertheless, we judged this
Sharda 2018; Thompson 2014), manually generated random
study to be at unclear risk. We also judged studies as being at
sequences by, for example, coin tossing (Kim 2008; Lim 2011;
unclear risk of bias where detailed information about assessor
Sharda 2018), and a Latin Square for determining session order
blinding was missing (Arezina 2011; Brownell 2002; Chen 2010;
(Arezina 2011). We judged these studies as being at low risk of
Chen 2013; Huang 2015; Mateos-Moreno 2013; Thomas 2003). In
bias. In eleven of the 26 studies (Buday 1995; Chen 2010; Chen
four studies, outcomes were assessed by the participants through
2013; Farmer 2003; Huang 2015; Lim 2010; Thomas 2003; Sa 2020;
self-report questionnaires (Moon 2010; Porter 2017; Schwartzberg
Schwartzberg 2013; Schwartzberg 2016; Yurteri 2019), methods
2013; Schwartzberg 2016). In two studies, outcome assessors were
of randomisation were not specified and the risk of bias was
not blinded (Bharathi 2019; Farmer 2003), and in one study, Sa 2020,
judged as unclear. In the remaining five studies, no information
the outcome measure was applied by the therapist administering
about randomisation was provided or the described methods of
the intervention (thus yielding the data ineligible for our meta-
randomisation did not ensure a random allocation of participants;
analysis). For these remaining seven studies, we judged the risk of
these were rated as having high risk of bias.
bias to be high.
The description of allocation concealment was adequately
Six studies used more than one rater to independently assess
described in two studies (Bieleninik 2017; Thompson 2014)
outcomes. Five of those studies reported a high inter-rater
and partly clarified in three studies (Gattino 2011; Porter 2017;
reliability for the assessment of outcomes (Arezina 2011: inter-
Sharda 2018); we judged these to be at low risk of bias. For
observer agreement ranging from 85.7% to 98.9%; Brownell 2002:
three studies, allocation was not concealed and these were judged
inter-rater reliability 0.86 to 0.94; Buday 1995: agreement rate
as being at high risk of bias (Bharathi 2019; Ghasemtabar 2015;
98%; Farmer 2003: agreement rate 91%; Kim 2008: inter-rater
Mateos-Moreno 2013). The remaining eighteen studies did not
reliability 0.70 to 0.98, as reported above). In Mateos-Moreno
provide specific information on allocation concealment and the
2013, measures were taken independently by two assessors. Any
risk of bias was rated as unclear.
possible disagreement was discussed until agreement was reached
Blinding on a final score to be used for analysis.
Due to the nature of the intervention, it was not possible to Incomplete outcome data
blind those who delivered music therapy or those who received
Twenty-three studies reported no or low attrition rates, leading
it. Consequently, neither participants nor therapists of the studies
to a low risk of bias judgement. Out of these studies, very low to
under review could be declared as blinded. However, although
acceptable dropout rates, ranging from 2% to 28% until the post-
autistic individuals were not blinded, this was unlikely to introduce
intervention assessment, were reported for five studies (Bieleninik
bias as they were usually not fully aware of available treatment
2017; Porter 2017; Rabeyron 2020; Sharda 2018; Thompson
options or study design (Cheuk 2011). The possible risk of
2014). LaGasse 2014 excluded a participant with available data
bias introduced by therapists administering the intervention was
from the published analysis; however, the IPD-based analyses
unknown. Therefore, we judged the risk of performance bias as
presented here included all participants. Three studies had dropout
unclear in all studies in the review.
rates above 30% and were judged as entailing a high risk of bias
In four of the included studies, assessors were blinded to the due to attrition (Kim 2008: 5/15, 33%; Schwartzberg 2013: 77/107,
treatment condition (Bieleninik 2017; Gattino 2011; Rabeyron 72%; Schwartzberg 2016: 64/93, 69%).
2020; Sharda 2018). In three further studies (Buday 1995; Lim
Selective reporting
2010; Lim 2011), assessors were blinded to the purpose of the
research. We judged all these seven studies as being at low risk There was no evidence of selective reporting of outcomes in the
of bias. In Kim 2008, non-generalised outcome measures and included studies, leading to a low risk of bias judgement.
two of the measures assessing generalised skills (ESCS, MPIP)
were rated by the researcher and complemented by independent Other potential sources of bias
coders (inter-rater reliability ranging from 0.70 to 0.98). We judged We considered inadequate music therapy methods and inadequate
the risk of bias as being unclear. Studies primarily using parent music therapy training of therapists as additional potential sources
Informed decisions.
of bias. For the majority of studies, we detected none of these Social interaction
sources of bias. For Chen 2013 and Huang 2015, it was unclear
Post-intervention
whether the music therapy was provided by a trained music
therapist. Moon 2010 described a music drama approach which Immediately post-intervention, average endpoint scores of social
might closely link to music therapy, but it was unclear whether this interaction were available from 12 studies (Bharathi 2019;
approach was provided by a trained music therapist. For Buday Bieleninik 2017; Chen 2013; Gattino 2011; Ghasemtabar 2015; Kim
1995, we found both the music therapy methods and the training of 2008; LaGasse 2014; Porter 2017; Rabeyron 2020; Schwartzberg
the person delivering the intervention to be of unclear adequacy. 2013; Sharda 2018; Thompson 2014). As heterogeneity was
substantial (Chi2 = 29.51, P = 0.002, I2 = 63%) and could not be
Effects of interventions explained clinically via subgroup analyses (results not shown), we
See: Summary of findings 1 Music therapy compared with placebo accordingly conducted a random-effects analysis for this outcome.
therapy or standard care for autistic people The SMD effect estimate was in the small to medium range,
but the CI ranged from no effect to a medium effect (SMD 0.26,
Twenty-five of the included studies were included in the meta- 95% CI −0.05 to 0.57, P = 0.11; 12 studies, 603 participants; low-
analyses; in one study (Sa 2020), outcomes were measured by the certainty evidence; Analysis 1.2), thus indicating no clear evidence
therapist and therefore not eligible to be included. We used fixed- of a difference between music therapy and comparison groups.
effect analyses for all outcomes, but changed to a random-effects Investigating the related funnel plot did not yield any asymmetry,
model when a substantial amount of heterogeneity (i.e. 50% or thus there was no clear indication of a risk of non-reporting bias.
higher; Deeks 2021) was identified immediately post-intervention
During intervention
that could not be explained by clinical subgroups.
Average endpoint scores of social interaction during the
Primary outcomes intervention were available from three studies (Arezina 2011; Kim
Global improvement 2008; Thomas 2003) and showed a large effect (SMD 1.15, 95% CI
0.49 to 1.80, P < 0.001; 3 studies, 44 participants; Analysis 1.2),
Post-intervention favouring music therapy over comparison groups. The results were
In eight studies, global improvement was assessed immediately homogeneous (Chi2 = 1.50, P = 0.47, I2 = 0%). We conducted a
post-intervention (Bharathi 2019; Bieleninik 2017; Kim 2008; sensitivity analysis excluding data from the high-attrition study
LaGasse 2014; Porter 2017; Rabeyron 2020; Schwartzberg 2013; (Kim 2008), and found that the effect for social interaction remained
Thompson 2014). The RR for global improvement between music statistically significant (P = 0.05). No heterogeneity was detected for
therapy and comparison groups was 1.22 (95% confidence interval this analysis (Chi2 = 0.64, P = 0.42, I2 = 0%).
(CI) 1.06 to 1.40, P = 0.006; number needed to treat for an additional
beneficial outcome NNTB = 11 for low-risk population, 95% CI One to five months follow-up
6 to 39; NNTB = 6 for high-risk population, 95% CI 3 to 21; 8 Effect estimates in the period one to five months post-intervention
studies, 583 participants; moderate-certainty evidence; Analysis (SMD 0.54, 95% CI −0.11 to 1.19, P = 0.10; 2 studies, 59
1.1), suggesting that global improvement is more likely to occur participants) showed little to no difference between the conditions.
with music therapy than with 'placebo therapy' or standard care
alone. There was no heterogeneity (Chi2 = 5.53, P = 0.60, I2 = 0%) and Six to 11 months follow-up
therefore we did not examine potential moderators; we retained Effect estimates in the period six to 11 months post-intervention
the fixed-effect model for this outcome. Changing to a random- (SMD −0.06, 95% CI −0.30 to 0.18, P = 0.63; 1 study, 258 participants)
effects model yielded similar results (P = 0.002). In a sensitivity indicated no clear evidence of a difference between music therapy
analysis excluding data from two high-attrition studies (Kim 2008; and comparison groups.
Schwartzberg 2013), the effect for global improvement showed no
substantial changes (P = 0.004). Non-verbal communication
One to five months follow-up Post-intervention
Two studies (Bharathi 2019; Porter 2017) also evaluated global Seven studies assessed non-verbal communication immediately
improvement in the period one to five months post-intervention. post-intervention (Chen 2013; Gattino 2011; Kim 2008; LaGasse
The RR for global improvement between music therapy and 2014; Rabeyron 2020; Sharda 2018; Thompson 2014). The
comparison groups for this period was 1.19 (95% CI 0.90 to 1.57; P heterogeneity found for this comparison was only moderate (Chi2 =
= 0.22; 2 studies, 99 participants), indicating no clear evidence of a 9.81, P = 0.13, I2 = 39%), and therefore we did not examine potential
difference between music therapy and comparison groups. moderators; we kept a fixed-effect SMD model for this outcome.
The effect size for difference between music therapy and control
Six to 11 months follow-up was in the small to medium range, but the CI ranged from no
One study (Bieleninik 2017) measured global improvement in effect to a medium effect (SMD 0.26, 95% CI −0.03 to 0.55, P = 0.08;
the period six to 11 months post-intervention. The RR for global 7 studies, 192 participants; low-certainty evidence; Analysis 1.3),
improvement between music therapy and comparison groups for suggesting little to no difference between the conditions. Changing
this period was 1.14 (95% CI 0.91 to 1.41, P = 0.25; 1 study, 364 to a random-effects model yielded similar results (P = 0.14). A
participants), again indicating no clear evidence of a difference sensitivity analysis excluding the study with a high dropout rate
between the groups. (Kim 2008) also did not lead to substantial changes in the results for
generalised non-verbal communication (P = 0.15).

Informed decisions.
During intervention = 0.30, I2 = 17%) and therefore we did not examine potential
Average endpoint scores for non-verbal communication during moderators; we retained the fixed-effects model for this outcome.
the intervention were available from three studies (Buday 1995; Changing to a random-effects model did not lead to substantial
Farmer 2003; Kim 2008) and indicated a large effect favouring music changes of the results (P = 0.02).
therapy (SMD 1.06, 95% CI 0.44 to 1.69, P < 0.001; 3 studies, 50 Six to 11 months follow-up
participants; Analysis 1.3). The results showed heterogeneity (Chi2
= 4.71, P = 0.09, I2 = 58%), which may be related to the relatively high One of the studies also measured QoL seven months after the
attrition rate in Kim 2008, or the unclear quality of music therapy end of the intervention, i.e. in the period six to 11 months post-
methods and therapist's training in Buday 1995. When excluding intervention (Bieleninik 2017). The CI of the effect estimate for
data from both studies, the overall effect did not show substantial difference in quality of life in this period ranged from a small
changes (SMD 1.64, 95% CI 0.10 to 3.19, P = 0.04). harmful to a small to medium beneficial effect (SMD 0.04, 95% CI
−0.21 to 0.29, P = 0.73; 1 study, 249 participants), indicating no clear
Verbal communication evidence of a difference between music therapy and comparison
groups.
Post-intervention
Eight studies assessed verbal communication immediately post- Total autism symptom severity
intervention (Chen 2013; Gattino 2011; Lim 2010; Lim 2011; Post-intervention
Rabeyron 2020; Schwartzberg 2013; Sharda 2018; Thompson
2014). The results showed substantial heterogeneity (Chi2 = 25.30, P Nine studies assessed total autism symptom severity immediately
< 0.001, I2 = 72%) that could not be explained clinically via subgroup post-intervention (Bharathi 2019; Bieleninik 2017: Chen 2010;
analyses (results not shown), resulting in a random-effects model Chen 2013; Huang 2015; LaGasse 2014; Mateos-Moreno 2013;
being used for this outcome. The effect size for difference in verbal Rabeyron 2020; Yurteri 2019). The results showed substantial
communication immediately post-intervention was in the small to heterogeneity (Chi2 = 61.33, P < 0.001, I2 = 87%) that could
medium range, but the CI ranged from no effect to a medium effect not be explained clinically via subgroup analyses (results not
(SMD 0.30, 95% CI −0.18 to 0.78; P = 0.21; 8 studies, 276 participants; shown), so we chose a random-effects model. The effect size for
very low-certainty evidence; Analysis 1.4), suggesting little to no difference in total autism symptom severity immediately post-
difference between the conditions. intervention was large (SMD −0.83, 95% CI −1.41 to −0.24, P = 0.005;
9 studies, 575 participants; moderate-certainty evidence; Analysis
During intervention 1.6), suggesting that music therapy probably decreases total autism
Four studies investigated verbal communication during the symptom severity compared to 'placebo therapy' or standard care
intervention (Buday 1995; Farmer 2003; Schwartzberg 2013; alone.
Schwartzberg 2016). The CI of the effect estimate for difference During the intervention
in verbal communication during the intervention ranged from a
medium harmful effect to a small to medium beneficial effect Total autism symptom severity during the intervention was
(SMD −0.06, 95 % CI −0.41 to 0.28, P = 0.71; 4 studies, 129 measured in one study (Mateos-Moreno 2013). The effect estimate
participants; Analysis 1.4), indicating no clear evidence of a was small, with a wide CI (SMD 0.15, 95% CI -0.83 to 1.14, P = 0.76; 1
difference between the groups. There was no heterogeneity (Chi2 = study, 16 participants; Analysis 1.6), indicating no clear evidence of
1.95, P = 0.58, I2 = 0%). a difference between music therapy and comparison groups.
One to five months follow-up

Data for verbal communication, measured in the period of one Average endpoint scores of total autism symptom severity
to five months post-intervention using a standardised scale, were measured in the period of one to five months post-intervention
available from one study (Bharathi 2019). The SMD effect size for were available from two studies (Bharathi 2019; LaGasse 2014) and
this follow-up period was small, but the CI ranged from a small showed a large effect in favour of music therapy (SMD −0.93, 95% CI
to medium harmful to a large beneficial effect (SMD 0.22, 95% −1.81 to −0.06, P = 0.04; 2 studies, 69 participants).
CI −0.33 to 0.76, P = 0.44; 1 study, 52 participants; Analysis 1.4), Six to 11 months follow-up
indicating no clear evidence of a difference between music therapy
and comparison groups, a similar finding to the other time points One study also assessed total autism symptom severity seven
for this outcome. months after the end of the intervention, i.e. in the period six to 11
months post-intervention (Bieleninik 2017). The SMD effect size for
Quality of life this time point was small, but the CI ranged from no effect to a small
to medium effect (SMD 0.18, 95% CI −0.05 to 0.41, P = 0.13; 1 study,
Post-intervention
289 participants), indicating no certain differences between music
Three studies investigated quality of life (QoL) of participants and/ therapy and comparison groups.
or their families immediately post-intervention (Bieleninik 2017;
Sharda 2018; Yurteri 2019). The SMD effect size across studies was Adverse events
0.28 (95% CI 0.06 to 0.49, P = 0.01; 3 studies, 340 participants; Data for adverse events immediately post-intervention and in
moderate-certainty evidence; Analysis 1.5), indicating a small to the period six to 11 months post-intervention were available
medium effect favouring music therapy, which suggests that music from two studies (Bieleninik 2017; Porter 2017). However, as no
therapy probably increases QoL compared with 'placebo therapy' events occurred in Porter 2017, only Bieleninik 2017 contributed
or standard care alone. Heterogeneity was low (Chi2 = 2.41, P an effect estimate (Analysis 1.7). Adverse events were rare,

Informed decisions.
and no differences were observed between music therapy or therapy and control groups was in the small to medium range,
standard care in either time period (RR 1.52, 95% CI 0.39 to but the CI ranged from a small harmful to a large beneficial
5.94, P = 0.55 immediately post-intervention, 1 study, 290 effect (SMD 0.29, 95% CI −0.24 to 0.83, P = 0.28; 3 studies,
participants; RR 0.88, 95% CI 0.23 to 3.46, P = 0.86 at 6-11 months 56 participants; Analysis 1.9), indicating no clear evidence of a
post-intervention, 1 RCT, 290 participants; moderate-certainty difference between music therapy and comparison groups. There
evidence), indicating similar frequencies of adverse events in both was no indication of heterogeneity between studies (Chi2 = 0.37, P
trial arms. Bieleninik 2017 reported that adverse events included = 0.83, I2 = 0%), therefore we did not examine potential moderators
hospitalisation and institutional stay, as reported by parents, and retained the fixed-effects model for this outcome. Changing to
and mainly referred to planned and short-term institutional a random-effects model yielded similar results (P = 0.28).
stays. Porter 2017 reported that no serious adverse events or
non-serious adverse events attributable to either arm of the trial One to five months follow-up
occurred (personal communication, 25 January 2021). No other For follow-up in the period of one to five months post-intervention,
adverse events were reported in any of the other included studies. the CI of the effect estimate ranging from a large harmful to a large
Secondary outcomes beneficial effect indicated uncertain differences between music
therapy and standard care (Porter 2017; SMD −0.04, 95% CI −1.07 to
Adaptive behaviour 0.99, P = 0.94; 1 study, 15 participants).
Post-intervention
Identity formation
Immediately post-intervention, average endpoint scores of Post-intervention
adaptive behaviour were available from five studies (Bieleninik
2017; Chen 2010; Porter 2017; Rabeyron 2020; Sharda 2018). The CI Two studies assessed aspects of identity formation (including
of the effect estimate for difference in adaptive behaviour at this self-esteem, self-awareness, and self-efficacy) immediately post-
time point ranged from no effect to a small effect (SMD −0.02, 95% intervention (Moon 2010; Porter 2017). The results showed
CI −0.20 to 0.16, P = 0.84; 5 studies, 462 participants; Analysis 1.8), substantial heterogeneity (Chi2 = 7.82, P = 0.005, I2 = 87%) that
indicating no differences between music therapy and comparison could not be explained clinically via subgroup analyses (results not
groups. No heterogeneity was detected for this comparison (Chi2 = shown), so we used a random-effects model. The SMD effect size
0.62, P = 0.96, I2 = 0%), so we did not examine potential moderators for difference in identity formation immediately post-intervention
and retained the fixed-effects model for this outcome. Changing to was large, but the CI ranged from a medium harmful to a large
a random-effects model did not lead to substantial changes of the beneficial effect (SMD 1.35, 95% CI −0.58 to 3.28, P = 0.17; 2 studies,
results (P = 0.84). 55 participants; Analysis 1.10), indicating no clear evidence of a
difference between music therapy and comparison groups.
During the intervention
Four studies investigated adaptive behaviour during the
intervention (Arezina 2011; Brownell 2002; Kim 2008; Thomas For the period of one to five months post-intervention, results from
2003). The SMD effect size for difference between music therapy one study (Porter 2017) for self-esteem indicated a large effect in
and 'placebo' therapy groups was 1.19 (95 % CI 0.56 to 1.82, P < favour of music therapy (SMD 0.86, 95% CI 0.16 to 1.55, P = 0.02; 1
0.001; 4 studies, 52 participants; Analysis 1.8), indicating a large study, 35 participants).
effect in favour of music therapy. Heterogeneity was low (Chi2 = Depression
4.16, P = 0.24, I2 = 28%). The effect on adaptive behaviour during
the intervention remained large and homogeneous in a sensitivity Post-intervention
analysis excluding two studies with high risk of bias (Brownell 2002; Depression was assessed in one study (Porter 2017). Results
Kim 2008). showed no clear evidence of a difference between music therapy
and treatment-as-usual (SMD −0.34, 95% CI −1.01 to 0.34, P = 0.33;
1 study, 34 participants; Analysis 1.11).
Effects in the period one to five months post-intervention (Porter
2017; SMD 0.56, 95% CI −0.12 to 1.24, P = 0.11; 1 study, 35 One to five months follow-up
participants) indicated no clear evidence of a difference between There was little to no difference between the conditions at the
music therapy and comparison groups. period of one to five months post-intervention (SMD −0.60, 95% CI
−1.27 to 0.07, P = 0.08; 1 study, 36 participants).
Six to 11 months follow-up
Similarly, effects in the period six to 11 months post-intervention Cognitive ability

(Bieleninik 2017; SMD −0.12, 95% CI −0.36 to 0.11, P = 0.29; 1 Post-intervention
study, 290 participants) indicated no clear evidence of a difference
between music therapy and comparison groups. One study assessed aspects of cognitive ability by measuring
attention skills immediately post-intervention (Sa 2020). However,
Quality of family relationships data from this study were not included as the outcome measure was
applied by the therapist.
Post-intervention
Three studies assessed the quality of family relationships

immediately following the intervention (Kim 2008; Porter 2017;
Thompson 2014). The effect size for difference between music
Informed decisions.
DISCUSSION measures outside of the therapy environment and after completion

of therapy in four outcome areas (global improvement, quality of
Summary of main results life, total autism symptom severity, and identity formation).
We found 26 trials that evaluated the effects of music therapy Overall completeness and applicability of evidence
for autistic individuals aged two years to young adult age.
Outcomes were assessed during the intervention, immediately Music therapy conditions
post-intervention, and within two periods of follow-up post- An important improvement regarding the applicability of the
intervention (one to five months; six to 11 months after the end of evidence in this update is that it includes more clinical techniques
therapy). Music therapy was compared with standard care, or with and components of music therapy that are in line with those used
a 'placebo' therapy which attempted to control for all non-specific in clinical practice. The early studies that were included in the first
elements of music therapy, such as the use of music or the attention version of this review (Gold 2006) were of limited generalisability
of a therapist. to clinical practice (Brownell 2002; Buday 1995; Farmer 2003).
The results show evidence of a large effect in favour of music These studies only used a limited subset of the music therapy
therapy on social interaction during the intervention. However, the techniques described in the clinical literature in the experimental
certainty of the evidence using the GRADE system (Schünemann treatment conditions. Receptive music therapy techniques with
2013) was rated as 'low' meaning that our confidence in the a high level of structuring predominated in those interventions;
effect estimate is limited. There was also a large effect in improvisational techniques were not utilised. However, active
favour of music therapy on non-verbal communication during the and improvisational techniques are widely used in many parts
intervention, again with a 'low' certainty of the evidence meaning of the world (Edgerton 1994; Gattino 2011; Geretsegger 2015;
that results should be considered with caution. In addition, a Holck 2004; Kim 2006; Schumacher 1999a; Schumacher 1999b;
large effect in favour of music therapy was found for total autism Thompson 2014; Thompson 2012; Wigram 2006; Wigram 2009).
symptom severity both immediately and one to five months post- In addition to five of the seven studies added in the previous
intervention; we rated the certainty as 'moderate' for this outcome, review update (Geretsegger 2014), 20 newly included studies
meaning that we are moderately confident in the effect estimate. added in this review update reflect active techniques. Most of
them emphasise relational and/or improvisational approaches to
Large effects in favour of music therapy were also found music therapy, thus considerably increasing the applicability of
for the secondary outcomes adaptive behaviour during the findings to clinical practice and hence the external validity of this
intervention, and for identity formation one to five months post- review.
intervention. Small to moderate effect sizes resulted for the primary
outcomes global improvement and quality of life immediately post- In terms of therapy setting, it is noteworthy that in about half of the
intervention. The certainty of the evidence was rated as 'moderate' newly added studies in this update, music therapy was provided in
for these two outcomes, meaning that the true effect is likely to be a group setting, while group music therapy was not applied in any
close to the estimate of the effect, but there is a possibility that it is of the studies in the previous version of this review (Geretsegger
substantially different. 2014; except for the family-based setting in Thompson 2014). We
did not plan to analyse the comparative effects of these settings.
No evidence of effect was found for the primary outcome verbal Clinically, both settings may be relevant for different individuals,
communication (rated as 'very low' certainty of the evidence or for the same individuals at different times. Although a group
immediately post-intervention), and for secondary outcomes setting may be overwhelming for some autistic individuals, music
quality of family relationships and depression. For adverse events, therapy groups with a tailored mix of structured and more flexible
no differences were found between music therapy and standard elements may also provide valuable opportunities for engaging
care ('moderate' certainty of the evidence, which means that we are in predictable and pleasurable social interactions with a variety
moderately confident about this result). of persons. This aspect of our review may also help in applying
this review's findings to contemporary healthcare settings where
It is interesting to note that social interaction and non-verbal individual therapy is often difficult to obtain due to economical
communication skills, which may be more closely related to non- reasons.
verbal interaction occurring within music therapy, showed change
compared with no change detected for verbal communication. Generally speaking, music therapy for autistic individuals should
However, it may also be that social interaction and non-verbal be backed by research evidence from both music therapy and
communication skills are relatively easier to address than verbal related fields, aiming at cooperation with others involved in
communication skills, especially in minimally verbal children and treatment and care of clients, active engagement of clients,
through short- to medium-term interventions. It is also interesting and establishing structure, predictability, and routines. It is
to see that both social interaction and non-verbal communication important to note that providing structure does not equal rigidity
showed change during the intervention, but not following the within interventions. Music contains rhythmic, melodic, harmonic,
intervention. Generalising skills acquired within the intervention and dynamic structure which, when applied systematically and
context to novel contexts and across interaction partners is a skilfully, can be effective in engaging autistic children. Intervention
known difficulty in autism (Green 2018), and it may be that strategies employing music improvisation are usually not pre-
helping individuals in generalising their skills requires longer structured in the sense of a fixed manual. In recent years, flexible
periods of intervention, and/or different approaches such as but systematic treatment protocols for music therapy have been
including the individual's everyday/family system in interventions. developed in clinical practice and research investigations in autism
Considering this challenge of skill generalisation across contexts, (Geretsegger 2015; Kim 2006; Thompson 2014; Wigram 2006) as well
it is remarkable to see the benefits of music therapy based on as in other fields (Baker 2019; Millstein 2021; Rolvsjord 2005). As

Informed decisions.
described above (see Included studies), several of the studies in possible detrimental effects of approaches aiming at reducing
this review have successfully applied such guidelines. More studies autism severity, particularly in the areas of social interaction
employing therapy approaches which are close to those applied and communication. Such approaches might support or even
in clinical practice will be needed in order to further improve the provoke the masking of autistic traits, which has been reported
clinical applicability of research findings. to be associated with negative consequences for mental health
including an increase in the risk of lifetime suicidality (Cassidy
Control conditions 2020). Additionally, the concept of autism severity and functioning-
Thirteen of the included studies used a 'dismantling' strategy to level descriptors such as 'high-functioning' are highly contentious
isolate the effect of the specific 'ingredients' of music therapy by and have recently shown to be an imprecise understanding
setting up comparison conditions which were very similar to the of autistic peoples’ specific needs; it has been suggested to
music therapy interventions, excluding only the music component instead acknowledge that the level of support needs of autistic
(Arezina 2011; Brownell 2002; Buday 1995; Farmer 2003; Kim 2008; people likely varies across domains, so that describing support
LaGasse 2014; Lim 2010; Lim 2011; Moon 2010; Schwartzberg 2013; needs in different domains (e.g. unstructured recreation activities,
Schwartzberg 2016; Sharda 2018; Thomas 2003). Any conclusion academic work) would be more appropriate (Bottema-Beutel
from such comparisons will therefore address the effects of specific 2021). However, as described above, many of the included studies
music therapy techniques, rather than the absolute effects of employed music therapy approaches where therapists follow the
music therapy in general. This type of design is most justified in individual's strengths and resources in an effort to maximise the
explanatory trials (Thorpe 2009) or when exploring music therapy individual's capabilities rather than to simply decrease autism
intervention strategies. However, such comparison conditions are symptoms or teaching specific skills for neurotypical interaction
less appropriate in pragmatic trials designed to inform practice and communication. This and an emerging focus on outcomes such
(Thorpe 2009) as they may introduce some artificiality into the as quality of life, depression, and identity formation enhance the
studies through selecting out and applying a single intervention relevance of this review's findings for autistic individuals.
strategy. This is not typically undertaken in clinical treatment,
although it does isolate specific components of music therapy. In
Quality of the evidence
the broader field of psychotherapy research, similar constructions Using the GRADE system (Schünemann 2013), we rated the
of 'placebo' therapy to control for the therapist's attention and the certainty of the evidence as 'moderate' for four outcomes, 'low'
non-specific elements have been broadly used (Kendall 2004, pp. for two outcomes, and 'very low' for one outcome included in
20-1). However, research on common factors in psychotherapy raise the Summary of findings 1, which means that further research
the question of how adequate it is conceptually, and also whether is likely to change the effect estimates and our confidence that
it is technically possible to separate the active from the non-active they are precise; results should therefore be considered with
elements of therapy (Lambert 2004, pp. 150-2). caution. Our assessments of the certainty of the evidence mainly
reflect concerns about risk of bias and imprecision due to wide
Duration, population, and outcomes CIs and small sample sizes. Limitations to the methodological
Autism as a pervasive developmental disorder is a chronic strength of the evidence are due to poor reporting of randomisation
condition, which requires sustained therapeutic intervention and allocation procedures or lack of randomisation and/or
starting as early as possible. In clinical reports for autism, music concealment in some studies. When interpreting the results, it is
therapy is usually described as a longer-term intervention and, important to note that, due to the nature of the intervention, it was
given the typical emergence of entrenched and deteriorating not possible to blind those who delivered music therapy or those
behaviour, therapeutic intervention relies on consolidating who received it. However, although participants were not blinded,
progress over time. With the therapy duration of included studies this was unlikely to introduce bias as they are usually not fully
ranging up to eight months, we consider this review's findings as aware of available therapy options or study design (Cheuk 2011).
sufficiently applicable to clinical contexts. Additionally, blinding of assessors was not assured in the majority
of studies as some of the measures in the included studies relied on
With regards to the population addressed, it is noteworthy that, reports from parents or participants themselves who were aware of
different from the previous version of this review (Geretsegger the respective group allocation. However, change in participants'
2014) which only included studies with children up to nine skills as assessed by parents or self-report may reflect effects of
years, this update included studies with adolescents and young interventions that are meaningful and relevant to clients and their
adults. The applicability of the findings is still limited to the families and is therefore considered important to include.
age groups included in the studies (two years to young adult
age). No direct conclusions can be drawn about music therapy Overall, we also observed several positive trends in this update
in autistic individuals above the young adult age. As with most that improve the certainty of the evidence: Most notably, both the
autism research, the majority of the participants in this review were median number of participants per study and the total number of
males from Western countries. It is positive that some included participants included have considerably increased (from a median
studies have been conducted in non-Western countries. To improve of 10 and a total of 165 participants in the previous review to
generalisability to the broader population, it will be important to 24 and 1165 participants, respectively, in the present update).
further diversify the populations studied in future trials to include Studies also employed longer periods of intervention on average,
non-male, non-Western participants. and a fifth of the studies in this review also included follow-up
assessments ranging from three weeks until seven months after
The outcomes addressed in the included studies cover areas that the end of the intervention, thus providing important information
form the core of the condition and relevant related areas that regarding the question of whether the effects of music therapy are
we consider as highly relevant to autistic individuals and their enduring. It is also noteworthy that the number of studies in this
families. Having said that, it is also important to consider
Informed decisions.
review that used validated scales (usually measuring generalised The findings of the present meta-analysis add considerably to the
behaviour) has substantially increased; thus, the findings are external validity of older and more recently published systematic
both more relevant and more reliable, and more comparable across reviews. First, the methodology was more rigorous, with clear
interventions. predefined inclusion/exclusion criteria, especially concerning the
population under study, the type of intervention, and the study
Potential biases in the review process design. Second, our systematic review was more inclusive in terms
of timeframe, age of participants, and outcomes examined; of note,
One can never be completely sure that all relevant trials have been
electronic searches were combined with a consultation of the grey
identified. However, our searches included not only exhaustive
literature and experts in the field. Most importantly, we performed
electronic and handsearches, but relied additionally on an existing
not only a qualitative, but also a quantitative synthesis, which
international network of leading researchers in the field. Therefore,
may allow a clearer and more objective interpretation of findings,
it seems unlikely that an important trial exists that did not come
especially in light of the scattered outcome measures adopted in
to our attention. Furthermore, this field does not seem to be
the included trials. The evaluation of outcomes with immediate
characterised by strongly selective publication. The trials that were
relevance for autistic individuals, such as quality of life, identity
unpublished or published only in the grey literature tended to have
formation, and depression may add a considerable value to the
positive results and were unpublished for reasons unrelated to
results of the present review. Notwithstanding, in agreement with
study results (Arezina 2011; Thomas 2003).
the most recent systematic reviews on the topic (Marquez-Garcia
The potential bias regarding the inclusion of studies in which one 2021; Mayer-Benarous 2021), we have underlined the urgent need
or more review authors were involved (Bieleninik 2017; Kim 2008; for improving the methodology of trials evaluating the efficacy of
Thomas 2003) was mitigated by ensuring that eligibility, risk of music therapy for autistic people.
bias and certainty of evidence assessment and data extraction were
performed by two independent reviewers not involved in these AUTHORS' CONCLUSIONS
studies.
Implications for practice
We found five ongoing studies (one of which is awaiting The evidence compiled in this review suggests that music
classification due to incomplete information regarding eligibility); therapy is probably associated with an increased chance of
incorporating these studies in a future update may alter the global improvement, and likely results in a small improvement
conclusions of this review. in quality of life and a large improvement in total autism
symptom severity immediately post-intervention. It may also
Agreements and disagreements with other studies or improve social interaction and non-verbal communication during
reviews the intervention but not after the intervention. The evidence
The findings of the present systematic review add substantial and for verbal communication is uncertain. The evidence in our
relevant information to previous works about the effectiveness of review also suggests that music therapy may improve adaptive
music therapy for autistic people (Gold 2006; James 2015; Marquez- behaviour in autistic children during the intervention but not
Garcia 2021; Mayer-Benarous 2021; Wheeler 2008; Whipple 2004; after the intervention, and identity formation in autistic children
Whipple 2012). and adolescents measured in the period of one to five months
after the end of the intervention, but not immediately after the
Focusing on the most recent reviews on the topic, Marquez- intervention. Music therapy has been shown to be superior to
Garcia (Marquez-Garcia 2021) summarized 36 longitudinal and standard care and to similar forms of therapy where music was not
retrospective peer-reviewed studies published between 2008 and used, which may be indicative of a specificity of the effect of music
2018. The review examined family interaction, communication, within music therapy.
psychological, and physiological changes. The authors concluded
that the poor methodology of the included studies (e.g. Certain behaviours of autistic children, adolescents and adults such
experimental designs, sample sizes, outcome measures) prevented as self-injurious or aggressive behaviour may be a challenge to their
them from recommending music therapy in this population. parents and other family members (Oono 2013). Therefore, the
They also encouraged the integration of behavioural evaluations increases in adaptive behaviour and in quality of life through music
with neuroscience (e.g. neuroimaging) and a more detailed therapy as found in this review may be highly relevant findings for
characterisation of study participants (e.g. severity level, presence families affected by autism.
of intellectual disability).
The possible positive effect of music therapy for social interaction
Mayer-Benarous 2021 evaluated the efficacy of educational and non-verbal communication measured during, but not after
and improvisational music therapy in children with the intervention might be related to the known challenge of
neurodevelopmental disorders such as ASD, attention deficit- generalising skills acquired within the intervention context to novel
hyperactivity disorder (ADHD), and learning and intellectual contexts and across interaction partners. It may be conducive for
disabilities. The authors principally analysed outcomes related skill generalisation across contexts if family members are included
to socio-communication. Evidence on the efficacy of educational in therapy sessions (as done in Thompson 2014) and/or informed
music therapy was based on 12 studies and supported a about and trained in relevant music-based techniques and
positive but small effect of educational music therapy for autistic approaches that help in creating opportunities for mutual social
children. According to the nine studies evaluating improvisational engagement (Gottfried 2016).
music therapy, efficacy appeared limited, but promising. Similarly
As only short- to medium-term effects up to 12 months have been
to Marquez-Garcia 2021, Mayer-Benarous 2021 highlighted the
examined, it remains unknown how enduring the effects of music
methodological issues of the included studies.
Informed decisions.
therapy are in the longer term. However, we found some evidence in this review, this modality has not been applied. Due to the
that positive effects of music therapy can be maintained after the specific benefits and limitations of online service delivery, it will be
intervention has ended. Effects on outcomes measured at follow- important for future studies to also examine the effects of online
up in the period of one to five months post-intervention showed a music therapy for autistic individuals.
possible positive effect of music therapy for total autism symptom
severity and self-esteem. For other outcomes and other follow- (3) Relevant outcome measures: There is currently no consensus
up time points, no clear evidence of differences between music about the most pertinent outcome measures to be used in autism
therapy and comparison groups was found. intervention research (McConachie 2015; Provenzani 2020; Warren
2011; Wheeler 2008). However, in line with recommendation
This review suggests that music therapy probably does not (1) above, future trials should include outcomes that address
increase adverse events. However, when applying the results of the core problems of ASD in a generalised setting utilising
this review to practice, it is important to note that the application standardised scales. They should also apply outcomes that are
of music therapy requires academic and clinical training in music regarded as important by autistic people and their family members
therapy. Trained music therapists and academic training courses (McConachie 2015). Participatory approaches to research that
are available in many countries, and information is usually incorporate the views of autistic people and those who support
accessible through professional associations. Training courses in them in all stages of the research process are an important
music therapy teach not only the clinical music therapy techniques avenue to ensure that research yields relevant benefits and
as described in the background of this review, but also aim improved outcomes for autistic people (Fletcher-Watson 2019).
at developing the therapist's personality and clinical sensitivity, When viewing social interaction as a shared responsibility and
which is necessary to apply music therapy responsibly. participatory practice situated within a historical, cultural, and
social ecology (De Jaegher 2007; Milton 2019), measuring social
Implications for research skills on neurotypical premises is likely to fail in capturing
progress meaningfully for autistic people. Hence, future research
The evidence included in this review centres on children and young
would benefit from incorporating embodied and enactive social
adults, meaning that the findings are not generalisable to autistic
cognitive perspectives, taking into account the disabling impact
adults. Research is needed examining effects of music therapy for
any given interaction, context or environment can hold for autistic
autistic individuals above the young adult age.
people, when designing studies and choosing or developing
We recommend that future trials on music therapy in this area outcomes. Outcome domains outside of core symptom areas such
should be: (1) pragmatic; (2) conscious of types of music therapy; as psychiatric disorders which are highly prevalent in autistic
(3) conscious of relevant outcome measures; and (4) include long- adults (Lipinski 2019) should also be considered, particularly
term follow-up assessments. as music therapy has also shown to be beneficial for mental
health conditions such as depression and anxiety in neurotypical
(1) Pragmatic trials of effectiveness: The earliest trials included populations (Aalbers 2017). Finally, combining biological markers
in this review tended to be designed as efficacy or explanatory with behavioural measures as done in one study in this review
trials. Such trials are designed with internal validity in mind and (Sharda 2018) may yield important findings about underlying
are limited in their generalisability. According to Thorpe 2009, neurobiological mechanisms in music therapy for autism (Sharda
explanatory trials tend to use inflexible experimental interventions, 2019).
inflexible comparison interventions, and outcomes that are not
directly relevant to autistic people, but rather an indicator of a (4) Long-term follow-up assessments: Although an increasing
direct intervention effect. Their relevance to informing practice number of studies in this update have addressed extended time
may be limited. Many of the more recent trials included in periods compared with earlier studies, only one study to date
this review (see Included studies) have used more flexible has examined outcomes up to 12 months from randomisation.
interventions, standard care comparisons, and downstream With the increasing prevalence of parallel trials, long-term follow-
outcomes. Further pragmatic trials should use rigorous designs in up assessments are becoming feasible and should be considered.
order to reliably address the question of effectiveness (i.e. whether Examples of other psychosocial interventions for autism that failed
music therapy works 'under usual conditions', Thorpe 2009). For to show effects at 12 months but showed effects after five years
increasing the methodological quality of trials and reducing risk (Pickles 2016) should be encouraging.
of bias, standards on randomisation, allocation, and blinding
procedures should be followed and reported more strictly. ACKNOWLEDGEMENTS
(2) Types of music therapy: As discussed in this review, various We thank Margaret Anderson, Information Specialist of the
types of music therapy have been proposed. Future trials should Cochrane Developmental, Psychosocial and Learning Problems
continue to be conscious of the quality, clinical applicability and Group (CDPLPG), for undertaking the searches for this and the
link to usual practice, and type of music therapy examined, and previous update, and Joanne Duffield and Sarah Davies, Managing
also investigate heterogeneity in populations (i.e. what works for Editors of the CDPLPG, for their kind guidance and assistance
whom; for example, regarding levels of support, verbal skills, throughout the review process.
socioeconomic status, or cultural background). Future trials might
We thank Xi-Jing Chen, Jinah Kim, and Eajin Yoo for their kind
entail comparisons between types and settings of music therapy,
help in retrieving Chinese and Korean studies, respectively, and for
but should also continue to investigate music therapy compared
providing support in translating them. Thanks also go to Asli Doğrul
with other interventions or standard care. As online delivery of
and Ayşegül Yay Pençe for translating information from Turkish
music therapy services is currently an emerging area of practice
studies, to Xueli Tan and Yaning Wu for translating information from
(Gaddy 2020), it is important to note that, in the studies included

Informed decisions.
Chinese studies, and to Eun Jin Nausner for translating information and Community Psychiatry, Queen Mary University of London, UK;
from Korean studies. Michelle Hintz, PsyD, MT-BC, Cadenza Center for Psychotherapy
& the Arts, Hollywood, FL; Ming Yuan Low, PhD, MT-BC, Berklee
We also thank all study authors who kindly provided additional College of Music, Boston, MA; and Vanessa M Young, MS, School of
data from their respective studies. Social and Behavioral Sciences, Arizona State University.
The CRG Editorial Team are grateful to the following peer reviewers The CRG Editorial Team are grateful to Anne Lethaby for copyediting
for their time and comments: Dr Catherine E Carr, Unit for Social this review.

Informed decisions.
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CHARACTERISTICS OF STUDIES
Characteristics of included studies [ordered by study ID]
Arezina 2011
Methods Allocation: session order randomised using Latin Square

Blindness: unclear; random sub-sample (33.33% of sessions) assessed by independent observer
Duration: 5 weeks
Design: cross-over
Participants Diagnosis: ASD

N: 6
Age range: 36 to 64 months
Sex: 5 boys, 1 girl
Setting: unclear
Location: Midwestern, USA
Interventions 1.Experimental (n = 6): interactive MT; musical instrument play, songs, music books, sung and verbal
responses to verbalisations; 6 x 10-minute individual sessions
Informed decisions.
Arezina 2011 (Continued)

2. Control (n = 6): non-music interactive play; non-music toys and books, verbal responses to verbali-
sations; 6 x 10-minute individual sessions
3. Control (n = 6): independent play; 6 x 10-minute individual sessions
Outcomes Behaviour observation based on videotaped sessions, coded by researcher (33.3% of sessions also cod-
ed by another observer)
1. Social interaction: requesting or initiating joint attention (number of requests during a given time
period), measured during the intervention
2. Adaptive behaviour: interaction or engaging in joint attention (percent of 15-second intervals en-
gaged in interaction), measured during the intervention
Notes Funding source(s): not reported
Risk of bias
Bias Authors' judgement Support for judgement
Random sequence genera- Low risk Order of sessions (including different therapeutic approaches) was ran-
tion (selection bias) domised for each child using a Latin Square.
Allocation concealment Unclear risk No details given

(selection bias)
Blinding of participants Unclear risk The fact that children with ASD participating in the study were not blinded was
and personnel (perfor- considered unlikely to introduce bias.
mance bias)
All outcomes The possible risk of bias introduced by therapists administering the interven-
tion was unknown.
Blinding of outcome as- Unclear risk No details about blinding reported; however, a random subsample (33.33%)
sessment (detection bias) was assessed by an independent observer (inter-observer agreement ranged
All outcomes from 85.7% to 98.9%).
Incomplete outcome data Low risk No dropouts

(attrition bias)
All outcomes No missing data reported
Selective reporting (re- Low risk All outcome measures of interest were considered in the analysis.
porting bias)
Other bias Low risk No financial bias could be found. The researcher is a music therapist.
Adequate music therapy method: yes
Adequate music therapy training: yes
Bharathi 2019
Methods Allocation: allocated by researchers

Blindness: no blinding
Duration: 6 months (3 months intervention + 3 months follow-up)
Design: quasi-experimental, parallel group, with control and a pre-post and follow-up test; single cen-
tre, recruited through a convenience sample method

Informed decisions.
Bharathi 2019 (Continued)
Participants Diagnosis: ASD according to DSM-5 criteria; mild, moderate or severe as per Childhood Autism Rating
Scales (CARS) scores
N: 52
Age range: 6 to 12 years (mean 9.5)
Sex: 26 boys, 26 girls
Setting: unclear
Location: Coimbatore, South India
Interventions 1.Experimental (n = 26): active group MT; singing, dancing, playing musical instruments while listen-
ing to music; 3 x 35-minute sessions each week for 3 months
2. Control (n = 26): passive group MT; no interaction, only music listening; 3 x 35-minute sessions each
week for 3 months
Outcomes 1. Social interaction: TRIAD Social Skills Assessment (TSSA) Total, measuring emotion understand-
ing/perspective taking ability, initiating interaction, responding to initiations, maintaining interac-
tions; higher scores are favourable; measured at pre- and post-intervention and at follow-up at 6
months; carried out by researcher considering parent, teacher, and direct interaction with children
2. Total autism symptom severity: CARS; lower scores are favourable; measured at pre- and post-in-
tervention; administered by researcher
Notes Funding source(s): Science and Engineering Research Board (SERB) (ECR/2016/001688), Government
of India, New Delhi
Risk of bias
Random sequence genera- High risk "Sixty children (30 boys and 30 girls) from ages 6–12 years were chosen-
tion (selection bias) through a convenience sampling method. [...] The study group was stratified
further into two groups as active MT and passive MT intervention group."
Allocation concealment High risk As the groups were selected by the researchers, it is likely that the allocation
(selection bias) was not concealed.
and personnel (perfor- considered unlikely to introduce bias. The possible risk of bias introduced by
mance bias) therapists administering the intervention was unknown.
All outcomes
Blinding of outcome as- High risk Non-blinded outcome assessment: "For each child, pre- and post-therapy
sessment (detection bias) CARS score was taken by the researcher."
All outcomes
Incomplete outcome data Low risk No dropouts reported

(attrition bias)
Selective reporting (re- Low risk No reference to a protocol or trial registration provided. However, as the re-
porting bias) search received both ethical approval and governmental funding, it is likely
that the researchers followed a protocol and reported according to that.
Other bias Low risk No financial bias could be found. The researchers might be music therapists.
Adequate music therapy training: unclear as no information was provided

Informed decisions.
Bieleninik 2017
Methods Allocation: randomised using computer-generated randomisation list

Blindness: assessors blind to treatment condition
Duration: 12 months (5 months intervention + 7 months follow-up)
Design: parallel-group, multicentre (9 countries, 10 centres)

N: 364
Age range: 4.0 to 6.11 years
Setting: outpatient
Location: Australia, Austria, Brazil, Israel, Italy, Korea, Norway, UK, USA
Interventions 1. Experimental (n = 90): individual improvisational music therapy; 30-minute sessions for 5 months,
3 sessions per week
2. Experimental (n = 92): individual improvisational music therapy; 30-minute sessions for 5 months,
1 session per week
3. Control (n = 182): enhanced standard care
Outcomes 1. Social interaction:

a. Autism Diagnostic Observation Schedule-Social Affect (ADOS-SA), administered by blinded asses-
sors; lower scores are favourable; evaluated before and during intervention (2 months), post-inter-
vention, and at 7-month follow-up
b. Social Responsiveness Scale (SRS) Total, parent-rated (not blinded); lower scores are favourable;
evaluated before and during intervention (2 months), post-intervention, and at 7-month follow-up
2. Quality of life: 100 mm visual analog scales for parent-reported quality of life of the child (QoL-child)
and of the family as a whole (QoL-family); higher scores are favourable; evaluated pre-post interven-
tion, and at 7-month follow-up; completed by participants' parents or guardians
3. Total autism symptom severity: ADOS Total, administered by blinded assessors; lower scores are
favourable; evaluated before and during intervention (2 months), post-intervention, and at 7-month
follow-up
4. Adverse events: any adverse events (parent reports)
5. Adaptive behaviour: ADOS-Restricted and Repetitive Behaviors (ADOS-RRB), administered by blind-
ed assessors; lower scores are favourable; evaluated before and during intervention (2 months), post-
intervention, and at 7-month follow-up
Notes Funding source(s):
Research Council of Norway (grant 213844, the Clinical Research and Mental Health Pro-
grammes); POLYFON Knowledge Cluster for Music Therapy, The Grieg Academy Department of Music,
University of Bergen, Norway;
Australia: Melbourne Conservatorium of Music, the University of Melbourne;
Austria: Danish Council for Independent Research/Humanities (FKK), Aalborg University, and Faculty of
Psychology, University of Vienna;
Brazil: Research Incentive Fund (FIPE) of the Hospital de Clínicas de Porto Alegre (HCPA);
Italy: IRCCS Stella Maris Foundation, Pisa, and University of Pisa;
Korea: Research Institute of Health and Science, Jeonju University;
Norway: Health Authority of Western Norway (Helse Vest grant 911800), Bergen municipality (Bergen
Culture School), Fjell municipality (Fjell Culture School);
UK: National Institute for Health Research (Health Technology Assessment Programme, National Insti-
tute for Health Research grant 12/167/95), Cambridge and Peterborough Foundation National Health

Informed decisions.
Bieleninik 2017 (Continued)

Service Trust, The Evelyn Trust, Cambridgeshire Music, Anglia Ruskin University; USA: Molloy College
Faculty research scholarship and a collaborative research grant from the Mid-Atlantic Region of the
American Music Therapy Association
Risk of bias
Random sequence genera- Low risk Computer-generated sequence

tion (selection bias)
Allocation concealment Low risk Randomisation list stored centrally, allocation via an electronic system after
(selection bias) decision on inclusion
All outcomes
Blinding of outcome as- Low risk Assessors of the primary outcome were blinded and success of blinding was
sessment (detection bias) verified.
All outcomes
SRS was administered by parents which were not blinded to intervention.
Incomplete outcome data Low risk Low attrition rate

(attrition bias)
All outcomes
Selective reporting (re- Low risk Trial was prospectively registered; trial protocol published; original trial proto-
porting bias) col included with trial report
Other bias Low risk No personal or financial bias could be found. The majority of researchers in-
volved are trained music therapists.
Brownell 2002
Methods Allocation: quasi-randomised, possibly randomised ('counterbalanced')

Blindness: independent assessor (teacher), blinding not reported
Duration: 4 weeks
Design: cross-over
Participants Diagnosis: autism

N: 4
Age range: 6 to 9 years
Sex: 4 boys, no girls
Setting: elementary school
Location: Eastern Iowa, USA
Interventions 1. Experimental (n = 4): structured receptive MT; songs with social stories; 5 individual, daily sessions
2. Experimental (n = 4): structured receptive 'story therapy'; reading of social stories; 5 individual, daily
sessions

Informed decisions.
Brownell 2002 (Continued)

3. Control (n = 4): no intervention; 2 x 5 days
Outcomes 1. Adaptive behaviour: frequency of repetitive behaviours outside therapy sessions (in classroom);
occurrence of behaviour was assessed by independent observers (i.e. teachers; inter-rater reliability
0.86-0.94) during intervention
Risk of bias
Random sequence genera- High risk Assignment to a counterbalanced treatment order (either ABAC or ACAB); un-
tion (selection bias) clear whether participants were randomly assigned to the two different treat-
ment orders

(selection bias)
All outcomes
Blinding of outcome as- Unclear risk Outcomes were assessed by a teacher or instructional associate assigned to
sessment (detection bias) the participant.
All outcomes
No details given on blinding of assessors

(attrition bias)
Selective reporting (re- Low risk All outcomes (targeted behaviours) of interest were considered in the analysis.
porting bias)
Buday 1995
Methods Allocation: randomised

Blindness: assessor blind to the nature of the hypothesis and to treatment condition
Duration: 2 weeks
Design: cross-over

N: 10
Setting: public school
Location: Chicago, Illinois, USA

Informed decisions.
Buday 1995 (Continued)
Interventions 1. Experimental (n = 10): structured receptive MT; songs used to teach signs; 5 individual sessions
2. Control (n =10): 'Rhythm therapy'; rhythmic speech used to teach signs; 5 individual sessions
Outcomes Imitating behaviour in sessions (rating of a video recording with sound turned off to ensure blinding
of rater; rater blind to nature of hypothesis; inter-rater agreement 98% for 25% of each participant's
scores)
1. Non-verbal communication: sign imitation, assessed during intervention

2. Verbal communication: speech imitation, assessed during intervention
Risk of bias
Random sequence genera- Unclear risk Counterbalancing of target signs for each treatment condition. "Five of the
tion (selection bias) subjects were randomly assigned to be tested on the music condition during
the first week, while the other five were tested on the rhythm condition."
Allocation concealment Unclear risk No details given, probably no

(selection bias)
All outcomes
Blinding of outcome as- Low risk Assessments were conducted by a person blinded to the nature of the hypoth-
sessment (detection bias) esis. Unlikely that the researcher was blinded to the treatment condition as as-
All outcomes sessments were based on video analysis of the treatment sessions.

(attrition bias)
porting bias)
Other bias Unclear risk No personal or financial bias could be found.
Adequate music therapy method: unclear
Adequate music therapy training: unclear
Chen 2010
Methods Allocation: randomly divided into experimental group and control group (no further specification)
Blindness: not reported
Duration: 3 months
Design: parallel group
Participants Diagnosis: childhood autism according to DSM-IV

N: 30

Informed decisions.
Chen 2010 (Continued)

Setting: Ninth People's Children's Hospital
Location: Chongqing, China
Interventions 1. Experimental (n = 15): comprehensive treatment (medicine and education, including auditory in-
tegration training, sensory integration training, special education, language therapy, play therapy,
etc.) plus MT; 4 times a week for 30 minutes each, for 3 months, including receptive and active methods
2. Control (n = 15): comprehensive treatment alone; 3 months. The process, specifically: the therapist
plays the opening song, the children listen, rhythm, experience the rhythm, perform; sing for a total
of 5 minutes; the therapist will improvise to guide the children to sing, recreate and knock; play for
10 minutes; rest for 5 minutes; the therapist plays musical stories and guides the children to act, sing
and make corresponding expressions and actions for 5 mins; the therapist plays the goodbye song,
children listen, move, experience rhythm, and children sing for a total of 5 minutes.
Outcomes 1. Total autism symptom severity: Autism Child Behavior Scale (AuBC) Total; lower scores are
favourable; completed by parents pre- and post-intervention
2. Adaptive behaviour: Psychoeducational Profile (PEP); higher scores are favourable; rated by re-
searchers pre- and post-intervention
Notes Funding source(s): Chongqing Natural Science Foundation (CSTC, 2009BB5129)
Risk of bias
Random sequence genera- Unclear risk Randomly divided into experimental group and control group (no further spec-
tion (selection bias) ification)
Allocation concealment Unclear risk No details provided

(selection bias)
mance bias)
tion was unknown.
Blinding of outcome as- Unclear risk No details given on blinding of assessors

sessment (detection bias)
All outcomes

(attrition bias)
All outcomes No missing data reported.
Selective reporting (re- Low risk No trial registration/study protocol. It was likely that all intended measures
porting bias) were included in the analysis.
Other bias Low risk No personal or financial bias could be found.
Adequate music therapy training: yes (⾳乐治疗由专门的⾳乐治疗师进⾏训

练" translates as "music therapy was provided by a professional music thera-
pist")

Informed decisions.
Chen 2013
Methods Allocation: randomised (no further specification)

Duration: 3 months
Participants Diagnosis: childhood autism based on DSM-IV criteria

N: 27
Setting: Ninth People's Hospital
Location: Chongqing, China
Interventions 1. Experimental (n = 9): social stories MT (including learning to sing social story songs, performing
social story content, etc.), in addition to standard care; sessions twice a week, for 50 minutes each,
over 3 months
2. Experimental (n = 9): MT without lyrics (music group, including learning the tune of the song learned
by the social story group, the lyrics replaced by meaningless pronunciation such as "ah.."), in addition
to standard care; sessions twice a week, for 50 minutes each, over 3 months
3. Control (n = 9): standard care; "medicine and education", including auditory integration training,
sensory integration training, special education training, speech therapy, play therapy, etc.
Outcomes 1. Social interaction: Childhood Autism Rating Scale (CARS) social communication domain; before
treatment and after 3 months (end of treatment); lower scores are favourable; assessed by specially
trained clinicians pre- and post-intervention
2. Non-verbal communication: CARS non-verbal communication, assessed by clinicians pre- and post-
intervention
3. Verbal communication: CARS verbal communication, assessed by clinicians pre- and post-interven-
tion
4. Total autism symptom severity: Autism Child Behavior Scale (AuBC) Total; lower scores are
favourable; before treatment and after 3 months (end of treatment); assessed by caregiver pre- and
post-intervention
Notes Funding source(s): Chongqing Natural Science Foundation (CSTC, 2009BB5129); Chongqing Medical
Specialty Construction (Chongqing Health Science and Education 2009, 71)
Risk of bias
Random sequence genera- Unclear risk Randomised (without further specification)


(selection bias)
mance bias)
tion was unknown.
Blinding of outcome as- Unclear risk The scale was assessed by specially trained clinicians.
All outcomes No details given on blinding of assessors

Informed decisions.
Chen 2013 (Continued)

(attrition bias)
Selective reporting (re- Low risk No trial registration/study protocol. It was likely that all intended measures
porting bias) were included in the analysis.
Adequate music therapy training: unclear, however it is likely that it was also
a professional music therapist, as described in Chen 2010, who provided the
music therapy.
Farmer 2003

Blindness: not known
Duration: 5 days

N: 10
Sex: 9 boys, 1 girl
Setting: homes and therapy centres
Location: Atlanta, Georgia, USA
Interventions 1. Experimental (n = 5): MT sessions; combined active and receptive: guitar playing, songs, mostly in-
dividual sessions of 20 minutes
2. Control (n = 5): placebo; no music sessions
Outcomes Responses within sessions (inter-rater agreement 91% for independent observer who rated 10% of ses-
sions)
1. Non-verbal communication: gestural responses, assessed during intervention

2. Verbal communication: verbal responses, assessed during intervention
Risk of bias
Random sequence genera- Unclear risk Randomised, no further details given


(selection bias)
mance bias)

Informed decisions.
Farmer 2003 (Continued)

tion was unknown.
Blinding of outcome as- High risk Unlikely that assessors were masked to the randomisation result as the assess-
sessment (detection bias) ments were based on video analysis of the sessions
All outcomes

(attrition bias)
porting bias)
Gattino 2011
Methods Allocation: balanced randomisation using a table of random numbers

Blindness: assessors blind
Duration: 7 months

N: 24
Age range: 7 to 12 years (mean 9.75 years)
Setting: hospital
Location: Porto Alegre, Brazil
Interventions 1. Experimental (n = 12): relational MT; improvisation not using a structured protocol; 3 assessment
sessions, 16 intervention sessions, 1 final assessment session, in addition to standard treatment, 20
x 30-minute individual sessions, scheduled weekly
2. Control (n = 12): standard treatment; clinical routine activities, including medical examinations and
consultations
Outcomes 1. Social interaction: Childhood Autism Rating Scale, Brazilian version (CARS-BR), social communica-
tion domain; lower scores are favourable; carried out by assessor blind to group allocation pre- and
post-intervention
2. Non-verbal communication: CARS-BR non-verbal communication domain; lower scores are
favourable; carried out by assessor blind to group allocation pre- and post-intervention
3. Verbal communication: CARS-BR verbal communication domain; lower scores are favourable; car-
ried out by assessor blind to group allocation pre- and post-intervention
Notes Funding source(s): Fund of Incentive to Research of Porto Alegre Clinical Hospital (project no. 08006),
Brazilian Research Council (CNPq)
Risk of bias

Informed decisions.
Gattino 2011 (Continued)
Random sequence genera- Low risk Randomised (computer-generated random sequence)

Allocation concealment Low risk Allocation was conducted by an external investigator.

(selection bias)
mance bias)
tion was unknown.
Blinding of outcome as- Low risk Assessors were blinded to the randomisation result.
All outcomes

(attrition bias)
porting bias)
Ghasemtabar 2015
Methods Allocation: not randomised (allocated by researcher, matched by age and gender)
Duration: 3.5 months (45 days intervention + 2 months follow-up)
Design: parallel group, pretest/post-test/follow‑up
Participants Diagnosis: mild to moderate autism based on Childhood Autism Rating Scale (CARS) scores
N: 27
Setting: three child and adolescent psychiatry centres
Location: Teheran, Iran
Interventions 1. Experimental (n = 13): MT Orff-Schulwerk; 12 sessions (2 sessions of 1 h/week) in 45 days, delivered

by two music therapists, including music listening, singing songs and chants, clapping, movement
and dancing, special musical drama, playing of instruments, etc.
2. Control (n = 14): without intervention
Outcomes 1. Social interaction: Social Skills Rating System Scale-Parent form (SSRS-P); high score means high
social skills; parent-rated, pre- and post-intervention and 2 months after the intervention
Notes Funding source(s): none
Risk of bias

Informed decisions.
Ghasemtabar 2015 (Continued)
Random sequence genera- High risk "Regarding the fact that the present research’s design is pretest/post-
tion (selection bias) test‑follow-up with control group, in order to eliminate the possible in-
tervening variables, we have tried to match the children of both groups by age
and gender variables. Therefore, 6 girls and 7 boys were replaced in the exper-
iment group (n = 13), and 7 girls and 7 boys were replaced in the control group
(n = 14)."
Allocation concealment High risk No randomisation procedures followed

(selection bias)
mance bias)
tion was unknown.
Blinding of outcome as- Unclear risk The SSRS is a parent-administered outcome. It was unlikely that parents were
sessment (detection bias) blinded to intervention.
All outcomes
Incomplete outcome data Low risk No missing data

(attrition bias)
All outcomes
Selective reporting (re- Low risk No published study protocol/trial registration. It was likely that all intended
porting bias) measures were included in the analysis.
Huang 2015
Methods Allocation: randomised (no further specification)

Duration: 2 months

N: 60
Setting: Xihua County People's Hospital
Location: Zhoukou, China
Interventions 1. Control (n = 30): integrated therapy; mainly including auditory integration training, speech therapy,
sensory integration training, play therapy, special education training, etc.
2. Experimental (n = 30): integrated therapy plus MT; as described above and including listening, active
participation, improvisation, suggestive relaxation, etc.

Informed decisions.
Huang 2015 (Continued)

6 sessions per week for 3 weeks (18 sessions), 30-35 minutes each, then 10 days rest, followed by anoth-
er period of 3 weeks with 6 sessions each (18 more sessions), followed by another period of rest until
day 60
Outcomes 1. Total autism symptom severity: Autism Child Behavior Scale (AuBC) Total, including emotional, so-
cial, behavioural, and perceptual aspects; low scores are favourable; rated by parents/caregivers pre-
and post-intervention
Risk of bias
Random sequence genera- Unclear risk Randomised (without further specification)


(selection bias)
mance bias)
tion was unknown.
Blinding of outcome as- Unclear risk No details given on blinding of assessors

All outcomes
Incomplete outcome data Low risk No missing data

(attrition bias)
All outcomes
Selective reporting (re- Low risk No published study protocol/trial registration. It was likely that all intended
porting bias) measures were included in the analysis.
Other bias Unclear risk No financial or personal bias could be found.
Adequate music therapy training: not clear whether the intervention was con-
ducted by a music therapist. It only reported "therapist" (治疗师) in the pa-
per.
Kim 2008

Blindness: assessors blind to the treatment condition, except for parent-based measures conducted
by mothers
Duration: 8 months (approximately 4 months per intervention)
Design: cross-over

N: 15 at entry; 10 for analysis

Informed decisions.
Kim 2008 (Continued)

Age range: 39 to 71 months (mean 51 months)
Sex: 13 boys, 2 girls at entry; 10 boys; no girls for analysis
Setting: private practice clinic
Location: Seoul, Korea
Interventions 1. Experimental (n = 10): improvisational MT; 12 x 30-minute individual sessions, scheduled weekly
2. Control (n = 10): play sessions with toys; 12 x 30-minute sessions, scheduled weekly

a. Social Approach Subscale (Pervasive Developmental Disorder Behavior Inventory, PDDBI); high
scores are favourable; completed by parents (not blind) and independent observers (blinded) be-
fore, during, and immediately after the intervention
b. behavioural observations in sessions (DVD recordings), assessed by researchers during interven-
tion (inter-rater reliability, with rater blind to session order for 30% of recordings, between 0.59 and
0.98):
i. turn-taking frequency and duration
ii. initiation of engagement frequency
iii. imitation frequency
iv. emotional synchronicity frequency and duration
v. musical synchronicity frequency and duration
vi. joy frequency and duration
2. Non-verbal communication:
a. Early Social Communication Scale (ESCS), abridged version; high scores are favourable; adminis-
tered by assessors blind to group allocation pre- and post-intervention
b. behavioural observations in sessions: eye contact frequency and duration
3. Adaptive behaviour: behavioural observations in sessions:
a. compliant response frequency
b. non-compliant response frequency
c. no response frequency
4. Quality of family relationships: Mother Play Intervention Profile (MPIP); video observations of play
situations at home scored by researcher, combined with independent observer's scores for a third of
the sessions; assessed pre- and post-intervention
Notes Funding source(s): Aalborg University, Denmark
Risk of bias
Random sequence genera- Low risk Randomised (picking the randomisation result from an opaque box)

(selection bias)
mance bias)
tion was unknown.
Blinding of outcome as- Unclear risk Assessors were blinded to the randomisation result, except for non-gener-
sessment (detection bias) alised measures, ESCS, and MPIP, where a random subsample (30%) was addi-
All outcomes tionally assessed by independent observers (inter-rater reliability ranging from
0.70 to 0.98)

Informed decisions.
Kim 2008 (Continued)
Incomplete outcome data High risk High dropout rate (5 of 15 participants dropped out)
(attrition bias)
All outcomes Data from dropouts were excluded.
porting bias)
Other bias Low risk No financial bias could be found. The researchers are a music therapists.
LaGasse 2014
Methods Allocation: randomised using computerised random numbers table; placed in a group in the order that
they consented to the study
Duration: 8 weeks (5 weeks intervention + 3 weeks follow-up)
Design: parallel group, single centre
Participants Diagnosis: ASD; negative report of dual disability diagnosis

N: 17
Setting: Colorado State University
Location: Fort Collins, USA
Interventions 1. Experimental (n = 9): MT group; twice a week for 50 minutes, over 5 weeks; small groups (3-4
children/group) led by board-certified music therapist (the Transformational Design Model (Thaut
2000) was used to create music experiences that were functionally similar to the non-musical experi-
ences, with the addition of music experiences and cues to facilitate the desired social skills. The pri-
mary role of the music was to provide anticipatory cues to aid in follow-through with all tasks and
to use engagement in music-making to practice the social skills. Rhythmic cues and music structure
were used to help the child plan their response, anticipate the timing for the response, and follow
through with the response).
2. Control (n = 8): social skills group; twice a week for 50 minutes, over 5 weeks; included cooperative
play experiences that involved taking turns, passing cards/game pieces, and interacting with their
peers. During these activities, the lead therapist provided cues and prompts to facilitate peer-to-peer
interaction and joint attention to manipulatives/peers.
Outcomes 1. Social interaction: Social Responsiveness Scale (SRS) Total; lower scores are favourable; completed
by child's parent/caregiver before and after intervention
2. Non-verbal communication: behavioural observations at 3rd and 10th session, coded by trained mu-
sic therapy research assistants:
a. eye gaze
b. joint attention with child
c. withdrawal behaviours
d. joint attention with adult
e. initiation of communication with child
f. initiation of communication with adult
g. response to communication
3. Total autism symptom severity: Autism Treatment Evaluation Checklist (ATEC) Total, investigated
four areas: speech and communication, sociability, sensory/cognitive awareness, health/physical be-
Informed decisions.
LaGasse 2014 (Continued)

haviour; lower scores indicate higher functioning (i.e. lower scores are favourable); completed by par-
ents (before study, after sessions 2, 4, and 6, and 3 days following the final session, and at follow-up 3
weeks after session completion), and by the lead therapist (after sessions 2, 4, 8, and 10)
Notes Funding source(s): Arthur Flagler Fultz Research Fund of the American Music Therapy Association
Risk of bias
Random sequence genera- Low risk Randomised using computerised random numbers table; placed in a group in
tion (selection bias) the order that they consented to the study

(selection bias)
mance bias)
tion was unknown.
Blinding of outcome as- Unclear risk Non-blinded. The SRS and the ATEC are parent-administered. Parents were
sessment (detection bias) not blinded to the treatment allocation. Assessors were blinded to the order of
All outcomes sessions for the rating of behaviour via video observation.
Incomplete outcome data Low risk Not all the data from randomised participants were included in the published
(attrition bias) report. However, in our analysis they were included.
All outcomes
Selective reporting (re- Low risk No trial registration/study protocol, however ethical approval was provided. It
porting bias) was likely that the researcher followed the procedures that were ethically ap-
proved.
Lim 2010

Blindness: assessors blind to the purpose of the study
Duration: 5 days

N: 50
Setting: recruiting site (schools, therapy centres, etc.)
Location: USA

Informed decisions.
Lim 2010 (Continued)
Interventions 1. Experimental (n = 18): music training; 'Developmental Speech and Language Training through Mu-
sic'; videotaped songs with target words; 6 individual sessions within 3 days
2. Control (n = 18): speech training; videotaped spoken stories with target words; 6 individual sessions
within 3 days
3. Control (n = 14): no training
Outcomes 1. Verbal communication: Verbal Production Evaluation Scale (VPES); behaviour observation based
on videotaped post-test sessions, coded by two trained speech/language pathologists specialised in
treating young children with language impairment who were blind to the purpose of the study (in-
ter-rater reliability 0.999)
Risk of bias


(selection bias)
mance bias)
tion was unknown.
Blinding of outcome as- Low risk Assessors were blind to the purpose of the study.
All outcomes

(attrition bias)
porting bias)
Lim 2011
Methods Allocation: training order randomised

Blindness: assessors blind to the purpose of the study
Duration: 2 weeks
Design: cross-over

Informed decisions.
Lim 2011 (Continued)

N: 22
Sex: 17 males, 5 females
Setting: no details given
Location: USA
Interventions 1. Experimental (n = 22): music training ("music incorporated Applied Behavior Analysis Verbal Behav-
ior"; sung instructions, songs with target words); 6 individual sessions within 2 weeks
2. Control (n = 22): speech training; applied behaviour analysis verbal behaviour; spoken instructions,
sentences with target words; 6 individual sessions within 2 weeks
3. Control (n = 22): no training
Outcomes 1. Verbal communication: Verbal Production Evaluation Scale (VPES); behaviour observation of video-
taped post-test sessions; coded by two trained speech/language pathologists specialised in treating
young children with language impairment who were blind to the purpose of the study
Risk of bias
Random sequence genera- Low risk Order of sessions (including different therapeutic approaches) was ran-
tion (selection bias) domised for each child using a random number chart. Participants were also
randomly assigned which order to receive the training to avoid order effects.

(selection bias)
mance bias)
tion was unknown.
Blinding of outcome as- Low risk Assessors were blind to the purpose of the study.
All outcomes

(attrition bias)
porting bias)
Other bias Low risk No financial bias could be found. The researchers are music therapists.
Mateos-Moreno 2013
Methods Allocation: group allocation conducted incidentally by care-centre staff

Informed decisions.
Mateos-Moreno 2013 (Continued)

Duration: 17 weeks
Participants Diagnosis: severe autism (DSM-IV); Childhood Autism Rating Scale (CARS) ≥ 37
N: 16
Age range: not reported (mean 25 years)
Sex: 15 males, 1 female
Setting: specialised care centre
Location: Spain
Interventions 1. Experimental (n = 8): combined dance/movement and MT; total of 36 ~ 60-minute group sessions
(2 per week), led by both a music and dance accredited therapist. Plurisensory approach, with musical
activities related to the Orff method and instruments: patients sitting in a circle beat a tempo with
percussive instruments imitatively and creatively with or without background music; song tunes in
a limited tessitura; corporal percussion and dancing while singing; and ‘gesturalised’ song and lyric
meanings/feelings. Background classical music always present while patients were entering, sitting
and leaving the room. Activities: massage the classmate with a small ball; imagine and simulate situ-
ations; imitate or guess emotions showed in pictures; move on the ground in different positions; role-
play; dancing; drawing; moving as a ‘flamenco’ dancer; playing with objects
2. Control (n = 8): TAU; no alternative therapies apart from regular activities
Outcomes 1. Total autism symptom severity: Revised Clinical Scale for the Evaluation of Autistic Behavior (ECA-
R) global score; lower scores are favourable; rated by two independent experienced clinician psychol-
ogists every three weeks during intervention and immediately after the intervention
Risk of bias
Random sequence genera- High risk Group allocation conducted incidentally by care centre staff
Allocation concealment High risk Not concealed

(selection bias)
Blinding of participants Unclear risk The fact that individuals with ASD participating in the study were not blinded
and personnel (perfor- was considered unlikely to introduce bias.
mance bias)
tion was unknown.
Blinding of outcome as- Unclear risk No details provided; "participants were monitored by two independent psy-
sessment (detection bias) chologists."
All outcomes

(attrition bias)
porting bias)
Other bias Low risk No personal and financial bias could be found.

Informed decisions.
Mateos-Moreno 2013 (Continued)

Moon 2010
Methods Allocation: unclear

Blindness: no blinding (self-report by participants)
Duration: 9 weeks
Participants Diagnosis: Asperger's syndrome based on DSM-IV criteria

N: 20
Setting: hospital
Location: Seoul, Korea
Interventions 1. Experimental (n = 10): music drama; based on Theory of Mind approach, including narration, song,
and musical instrument play (the content of a story/fairy tale and the actions and emotions of the
characters are enacted using music as the main medium); 2 sessions per week, 18 sessions in total,
40 minutes each
2. Control (n = 10): story-sharing/story-telling activity through talking/listening and discussing the con-
tents; story/fairy tale is told/enacted in an interactive way, using nonverbal elements such as emo-
tion-related facial expressions and gestures; 2 sessions per week, 18 sessions in total, 40 minutes each
Outcomes 1. Identity formation

a. Self-Awareness Scale of Fenigstein (Fenigstein 1979), standardised in Korean by Hyukki Eun (Eun
2000); high scores mean high degree of self-awareness; self-report by participants pre- and post-
intervention
b. Self-Efficacy Scale by Bandura (Bandura 1978), applied to Korean by Keumjoo Kwak (Kwak 1998);
high score means high degree of self-efficacy; self-report by participants pre- and post-intervention
c. Self-Esteem Scale by Rosenberg (Rosenberg 1965), revised and supplemented by Heewha Kim (Kim
1998) and re-constructed by Youngsook Cho (Cho 2004); high score means high self-esteem; self-
report by participants pre- and post-intervention
Risk of bias
Random sequence genera- High risk No information given

Allocation concealment High risk No information given

(selection bias)
mance bias)
tion was unknown.
Blinding of outcome as- High risk The chosen outcomes on self-esteem/awareness/efficacy were administered
sessment (detection bias) by the participants.

Informed decisions.
Moon 2010 (Continued)

All outcomes
Incomplete outcome data Low risk Likely to be no dropouts or missing data

(attrition bias)
All outcomes
porting bias)
Other bias Unclear risk No personal and financial bias could be found.
Adequate music therapy method: unclear (music drama)
Adequate music therapy training: unclear
Porter 2017

Blindness: single blind
Duration: 6 months (12 weeks intervention + follow-up at 26 weeks after randomisation)
Design: parallel group, multicentre
Participants Diagnosis: ASD based on ICD-10 criteria

N: 47
Setting: 6 community care Child and Adolescent Mental Health Services (CAMHS) within the Belfast
Health and Social Care Trust
Location: Belfast, Northern Ireland
Interventions 1. Experimental (n = 24): MT; Alvin model of ‘Free Improvisation’ plus TAU; 12 weekly individual sessions
of 30 minutes
2. Control (n = 23): TAU; psychiatric counselling or medication, or both
Outcomes 1. Social interaction: Social Skills Improvement System Rating Scales (SSIS) measuring communicative
and interactional skills; high scores are favourable; parental report and self-report pre-post interven-
tion and 13 weeks after the intervention
2. Quality of life: EQ5D Health State Scale; completed by a parent pre- and post-intervention; not suffi-
ciently completed for analyses due to participant fatigue
3. Adverse events: All serious adverse events (SAEs) were to be recorded in the case report form during
the study and reported to the Principal Investigator within 24 hours. They were then to be reviewed
by the Trial Steering Committee (TSC) at regular intervals throughout the trial. The Principal Investi-
gator together with the trial sponsor collated and reported annual safety reports to the Trial Steering
Committee. The trial sponsor and the chair of the ethics committee were also informed about severe
adverse events by the TSC chair if considered appropriate.
4. Adaptive behaviour: Child Behavior Checklist (CBC) measuring social functioning; low scores are
favourable; completed by a parent pre- and post-intervention and 13 weeks after the intervention
5. Quality of family relationships: Family Assessment Device (FAD) measuring family functioning; low
scores are favourable; completed by a parent pre- and post-intervention and 13 weeks after the inter-
vention
6. Identity formation: Rosenberg Self-Esteem Scale; high scores are favourable; completed by the
young person pre- and post-intervention and at 13-week follow-up
7. Depression: Centre for Epidemiological Studies Depression Scale for Children (CES-DC); low scores
are favourable; completed by the young person pre- and post-intervention and at 13-week follow-up

Informed decisions.
Porter 2017 (Continued)
Notes Funding source(s): Big Lottery Fund
Risk of bias
Random sequence genera- Low risk The randomisation list was computer-generated, using nQuery Advisor v.
tion (selection bias) 16.01, stratified by centre with a 1:1 allocation using random variable block
lengths of 2, 4, 6 and 8.
Allocation concealment Low risk Randomisation was carried out independently.

(selection bias)
mance bias)
tion was unknown.
Blinding of outcome as- High risk Non-blinded. Outcomes were measured via parental- and self-reports.
All outcomes
Incomplete outcome data Low risk "In line with the intention-to-treat principle, patients who attended fewer ses-
(attrition bias) sions were not excluded from data analysis."
All outcomes
Selective reporting (re- Low risk Published study protocol. All outcome measures of interest were considered in
porting bias) the analysis.
Rabeyron 2020
Methods Allocation: randomised (using a generated randomisation list)

Blindness: assessors blind to treatment condition
Duration: 8 months
Participants Diagnosis: ASD based on Childhood Autism Rating Scale (CARS)

N: 37
Setting: 5 psychiatric day-care centres
Location: Nantes, France
Interventions 1. Experimental (n = 19): MT; 25 weekly, structured, 30-minute sessions (5 minutes opening ritual of
music listening, 20 minutes instrumental and vocal improvisation, 5 minutes closing ritual of music
listening) performed by a music therapist and a co-therapist (nurse or educator)

Informed decisions.
Rabeyron 2020 (Continued)

2. Control (n = 18): music listening; 25 weekly, 30-minute sessions performed by a nurse or educator,
listening to playlist of commercial music, including French and foreign songs
Outcomes 1. Global improvement: Clinical Global Impression (CGI); higher scores indicating greater severity; as-
sessed by two trained clinical psychologists who were blind to group allocation pre- and post-inter-
vention
2. Social interaction: CARS, social communication domain; lower scores are favourable; assessed by
two trained clinical psychologists who were blind to group allocation pre- and post-intervention
3. Non-verbal communication: CARS non-verbal communication domain; lower scores are
favourable; assessed by two trained clinical psychologists who were blind to group allocation pre- and
post-intervention
4. Verbal communication: CARS verbal communication domain; lower scores are favourable; assessed
by two trained clinical psychologists who were blind to group allocation pre- and post-intervention
5. Total autism symptom severity: CARS Total; lower scores are favourable; assessed by two trained
clinical psychologists who were blind to group allocation pre- and post-intervention
6. Adaptive behaviour: Aberrant Behavior Checklist (ABC); low scores are favourable; assessed by two
trained clinical psychologists who were blind to group allocation pre- and post-intervention
Notes Funding source(s): Entreprendre pour Aider; Academie Francaise
Risk of bias
Random sequence genera- Low risk "Children were randomly assigned to one of these two groups using a generat-
tion (selection bias) ed randomization list for each group at t0."
Allocation concealment Unclear risk No information given

(selection bias)
mance bias)
tion was unknown.
Blinding of outcome as- Low risk "[A]ll participants were assessed by two trained clinical psychologists working
sessment (detection bias) at the Nantes Hospital. They were totally blind, both at t0 and t1, to the groups
All outcomes the children belonged to".
Incomplete outcome data Low risk All available data were included.
(attrition bias)
All outcomes
Selective reporting (re- Low risk Outcome measures matched the evaluation aims in the trial registration.
porting bias)
Sa 2020

Informed decisions.
Sa 2020 (Continued)
Methods Allocation: randomised - no further information provided; participants stratified by severity level
(mild, moderate, severe)
Blindness: not blinded (outcome measures and treatment protocol applied by the same person)
Duration: 7-11 weeks
Participants Diagnosis: ASD; severity check at baseline administered by the PI using the CARS2-HF
N: 23
Sex: 16 males, 7 females
Setting: school
Location: Central Valley, California, USA
Interventions 1. Experimental (n = 11): Music attention control training (MACT); 45-minute group sessions includ-
ing 5 to 6 participants led by a board-certified music therapist. MACT (Thaut 2014) includes structured
active or receptive musical exercises involving precomposed performance or improvisation in which
musical elements (pitch, rhythm, dynamics, etc.) cue different musical responses to address attention
skills (selective, sustained, and alternating functions).
2. Control (n = 12): waiting-list control group
Outcomes 1. Cognitive ability: Test of Everyday Attention for Children 2 (TEA-Ch 2): selective attention, sustained
attention, switching attention; administered by the therapist pre- and post-intervention, therefore not
used in meta-analysis
Notes Funding sources(s): not reported
Risk of bias
Random sequence genera- Unclear risk Randomised

Allocation concealment Unclear risk Not reported

(selection bias)
mance bias)
tion was unknown.
Blinding of outcome as- High risk Non-blinded. Outcome assessment was performed by the therapist.
All outcomes

(attrition bias)
porting bias)

Informed decisions.
Sa 2020 (Continued)
Other bias Low risk No personal and financial bias could be found.
Schwartzberg 2013
Methods Allocation: randomised by the principal investigator (PI)

Duration: 3 days
Design: parallel, 6-group, randomised design with cluster-randomisation

N: 30 for analysis
Sex: 29 boys, 1 girl
Setting: 3 separate 1-week summer camps for ASD
Location: Minneapolis, Minnesota, USA
Interventions 1. Experimental (n = 16): MT structured plus music-based social story; 50-minute sessions for 3 con-
secutive days. Consisted of 6 campers and 8 staff. MT sessions followed a similar routine (hello song,
movement and music intervention, social story intervention, instrument playing intervention, relax-
ation and music, and goodbye song)
2. Control (n = 14): social story; sessions for 3 consecutive days
Outcomes 1. Social interaction: Autism Social Skills Profile (ASSP; Bellini 2007); high scores are favourable; com-
pleted by parents post-intervention (one week after the end of the camp)
2. Verbal communication: comprehension checks; administered by camp counsellors, completed by
participants before and one week after intervention and at the end of each day of intervention
Risk of bias
Random sequence genera- Unclear risk "The PI cluster-randomised participants". No further details about randomisa-
tion (selection bias) tion method given

(selection bias)
mance bias)
tion was unknown.
Blinding of outcome as- High risk Non-blinded. Outcomes were measured via parental- and self-reports.
All outcomes

Informed decisions.
Schwartzberg 2013 (Continued)
Incomplete outcome data High risk Only data from those also completing the follow-up assessment were includ-
(attrition bias) ed.
All outcomes
Selective reporting (re- Low risk No trial registration/study protocol; however ethical approval. It was likely
porting bias) that all outcome measures of interest were considered in the analysis.
Schwartzberg 2016
Methods Allocation: cluster-randomisation performed by the principal investigator (PI)

Duration: 3 days
Design: parallel group, cluster-randomised

N: 29 for analysis
Setting: 3 separate 1-week summer camps
Location: Minnesota, USA
Interventions 1. Experimental (n = 13): MT; sing short stories (music-based social story session). Procedure: During
each day of the summer camp, the PI provided 50-minute music sessions to all campers attending the
camp. Each music session consisted of six campers and eight staff.
2. Control (n = 16): read aloud short stories
Outcomes Verbal communication: comprehension checks; administered by camp counsellors, completed by

participants each day of intervention at the conclusion of the MT session (short-term) and approxi-
mately seven hours later (long-term)
Risk of bias
Random sequence genera- Unclear risk "Participants were cluster-randomised". No further details about randomisa-
tion (selection bias) tion method given

(selection bias)
mance bias)
tion was unknown.

Informed decisions.
Schwartzberg 2016 (Continued)
Blinding of outcome as- High risk Non-blinded. Outcomes self-administered by participants

All outcomes
Incomplete outcome data High risk Only data from those also completing the follow-up assessment were includ-
(attrition bias) ed.
All outcomes
Selective reporting (re- Low risk No trial registration/study protocol. It was likely that there were no deviations
porting bias) from the intended intervention that arose because of the trial context.
Sharda 2018

Blindness: assessors blind
Duration: 8-12 weeks
Participants Diagnosis: ASD according to the DSM-IV criteria

N: 51
Setting: unclear
Location: Montreal, Canada
Interventions 1. Experimental (n = 26): MT; use of musical instruments, songs and rhythmic cues while targeting
communication, turn-taking, sensorimotor integration, social appropriateness and musical interac-
tion; 45-minute weekly sessions over 8-12 weeks
2. Control (n = 25): play-based intervention; to control for non-specific factors, such as positive
treatment expectancies, intervention support, therapist attention and emotional engagement; 45-
minute weekly sessions over 8-12 weeks

a. Social Responsiveness Scale (SRS-2); lower scores are favourable; rated by parents pre- and post-
intervention
b. Joint engagement, measured using a coding scheme for various engagement states observed from
session videos; these were not used as group means were only reported in graphs, with no individ-
ual participant data available.
2. Non-verbal communication: Children's Communication Checklist (CCC-2), measuring pragmatic
communication; higher scores are favourable; rated by parents pre- and post-intervention
3. Verbal communication: Peabody Picture Vocabulary Test-4 (PPVT-4), measuring language; higher
scores are favourable; rated by blinded assessors pre- and post-intervention
4. Quality of life: Beach Family Quality of Life (FQoL); higher scores are favourable; rated by parents pre-
5. Adaptive behaviour:
a. Vineland Adaptive Behaviour Scales (VABS), subscale maladaptive behaviour; lower scores are
favourable; rated by parents pre- and post-intervention

Informed decisions.
Sharda 2018 (Continued)

b. Neuroimaging outcomes (rsfMRI: intrinsic brain connectivity of fronto-temporal brain networks);
not used in meta-analysis
6. Movement: measured using a video-based optical flow analysis method, with whole-body movement
amplitude of child and therapist calculated separately; not used since outside of predefined outcome
categories
Notes Funding source(s): Canadian Institutes of Health Research; Quebec Bioimaging Network
Risk of bias
Random sequence genera- Low risk "Participants were randomized to MT (n = 26) or NM (n = 25) using the covari-
tion (selection bias) ate-adaptive method where the first 20 participants were randomized us-
ing simple coin toss and remaining 31 by the MinimPy software
(http://minimpy.sourceforge.net/) by the first author."
Allocation concealment Low risk Likely to be concealed. Randomisation was conducted by the first author, who
(selection bias) was not involved in assessing behavioural outcomes and was not involved in
interventions.
mance bias)
tion was unknown.
Blinding of outcome as- Low risk "All other assessors and authors were blind to group allocation informa-
sessment (detection bias) tion. Our attempt to blind parents (who assessed
All outcomes parent-rated outcomes) was only partially successful, with 31 out of the 51
parents reporting awareness of group allocation."
Incomplete outcome data Low risk Intention-to-treat analysis

(attrition bias)
All outcomes
Selective reporting (re- Low risk Registered trial. No deviations from the intended intervention that arose be-
porting bias) cause of the trial context
Thomas 2003
Methods Allocation: randomised order of treatment

Duration: 12 weeks
Design: cross-over (within each session)

N: 6

Informed decisions.
Thomas 2003 (Continued)

Sex: 5 boys, 1 girl
Setting: unclear
Location: USA
Interventions 1. Experimental (n = 6): MT; using songs, instruments, vocal sounds and movement to interact with the
child and musically or verbally respond to the child's verbal or non-verbal behaviour; 12 x 15-minute
sessions, immediately following or preceding playtime sessions
2. Control (n = 6): playtime; attempts to interact with the child using toys and verbally responding to the
child's non-verbal or verbal behaviour; 12 x 15-minute sessions, immediately following or preceding
music therapy sessions
Outcomes Behaviour observation during intervention based on videotaped sessions, coded by trained music ther-
apy intern (inter-rater reliability 0.85)
1. Social interaction: requesting behaviour (percentage of session time)

2. Adaptive behaviour: on-task behaviour (percentage of session time)
Notes Funding source(s): Mid-Atlantic Region of the American Music Therapy Association
Risk of bias


(selection bias)
mance bias)
tion was unknown.
Blinding of outcome as- Unclear risk No details given whether the assessor was blinded to the randomisation result
All outcomes

(attrition bias)
porting bias)
Thompson 2014

Informed decisions.
Thompson 2014 (Continued)

Duration: 16 weeks (plus 8 weeks of follow-up, but not used, as only for music therapy group)

N: 23
Setting: participants' homes
Location: Australia
Interventions 1. Experimental (n = 12): home-based, family-centred MT (using songs, improvisation, structured music
interactions) plus standard care; 16 sessions, scheduled weekly
2. Control (n = 11): standard care

a. Vineland Social Emotional Early Childhood Scales (Vineland SEEC); higher scores are favourable;
rated by researcher based on parent's responses at baseline and post-intervention
b. Social Responsiveness Scale-Preschool version (SRS-PS); lower scores are favourable; rated by par-
ents pre- and post-intervention
2. Non-verbal communication: MacArthur-Bates Communicative Development Inventories-Words and
Gestures (MBCDI-W&G), subscale 2 (actions & gestures used); higher scores are favourable; rated by
parents pre- and post-intervention
3. Verbal communication: MBCDI-W&G, subscales 1B (phrases understood) and 1D (words understood
+ words produced) assessed pre- and post-intervention
4. Quality of family relationships:
a. Parent-Child Relationship Inventory (PCRI); higher scores are favourable; rated by parents pre- and
post-intervention
b. Music Therapy Diagnostic Assessment (MTDA), rated by researcher; not used since rated for music
therapy group only
Notes Funding source(s): University of Melbourne; Victorian Department of Education and Early Childhood
Development, Australia
Risk of bias
Random sequence genera- Low risk Randomised (computer-generated random sequence)

Allocation concealment Low risk An independent statistician prepared opaque, numbered allocation en-
(selection bias) velopes.
mance bias)
tion is unknown.
Blinding of outcome as- Unclear risk Parent-report based measures were used.
All outcomes However, measures contained internal safeguards to address bias as evi-
denced by high correlations with non-parent rated measures or high test-
retest correlations.
Incomplete outcome data Low risk Low dropout rate

(attrition bias)
Intention-to-treat analysis
Informed decisions.
Thompson 2014 (Continued)

All outcomes
porting bias)
Yurteri 2019
Methods Allocation: unclear

Blindness: not known
Duration: 8 weeks

N: 24
Age range: 4.8 to 9.3 years
Setting: unclear
Location: Düzce, Turkey
Interventions 1. Experimental (n = 12): improvisational MT; led by a music therapist and "mostly one-on-one, some-
times groups of three"; twice-weekly, 40-minute sessions for 8 weeks
2. Control (n = 12): TAU; no treatment except monthly routine child psychiatric follow-up and special
education
Outcomes 1. Quality of life: Pediatric Quality of Life Inventory (PedsQL) Total score; high scores are favourable;
measured pre- and post-intervention
2. Total autism symptom severity: Autism Behavior Checklist (AuBC) Total; low scores are favourable;
measured pre- and post-intervention
Risk of bias
Random sequence genera- Unclear risk No details given. Probably randomised


(selection bias)
mance bias)
tion is unknown.
Blinding of outcome as- Unclear risk Outcome assessments were administered by parents.
Informed decisions.
Yurteri 2019 (Continued)

All outcomes
Incomplete outcome data Low risk No dropouts.

(attrition bias)
Selective reporting (re- Low risk No trial registration/study protocol. It was likely that there were no deviations
porting bias) from the intended intervention that arose because of the trial context.
ABC: Aberrant Behavior Checklist

ADOS(-RRB, -SA): Autism Diagostic Observation Schedule (-Restricted and Repetitive Behaviors, -Social Affect)
ASD: autism spectrum disorder
ASSP: Autism Social Skills Profile
ATEC: Autism Treatment Evaluation Checklist
AuBC: Autism Behavior Checklist
CAMHS: Child and Adolescent Mental Health Services
CARS(-2-HF, -BR): Childhood Autism Rating Scale (-Second Edition-High Functioning, -Brazilian Version)
CBC: Child Behavior Checklist
CCC-2: Children's Communication Checklist-Second Edition
CES-DC: Center for Epidemiological Studies Depression Scale for Children
CGI: Clinical Global Impression scale
DSM: Diagnostic and Statistical Manual of Mental Disorders
DVD: digital video/versatile disc
ECA-R: Revised Clinical Scale for the Evaluation of Autistic Behavior
EQ5D: a term (not an abbreviation) to describe a generic measure of health-related quality of life developed by the EuroQol Group
ESCS: Early Social Communication Scales
FAD: McMaster Family Assessment Device
FQoL: Beach Center Family Quality of Life Scale
ICD: International Classification of Diseases
MACT: music attention control training
MBCDI-W&G: MacArthur-Bates Communicative Development Inventories-Words and Gestures
MPIP: Mother Play Intervention Profile
MT: music therapy
MTDA: Music Therapy Diagnostic Assessment
NM: non-music
PCRI: Parent-Child Relationship Inventory
PDDBI: Pervasive Developmental Disorder Behavior Inventory
PedsQL: Pediatric Quality of Life Inventory
PEP: Psychoeducational Profile
PI: principal investigator
PPVT-4: Peabody Picture Vocabulary Test-4th Edition
QoL: quality of life
rsfMRI: resting-state functional magnetic resonance imaging
SAE: serious adverse effects
SEEC: Vineland Social-Emotional Early Childhood Scales
SRS(-2, -PS): Social Responsiveness Scale (-Second Edition, -Preschool Version)
SSIS: Social Skills Improvement System
SSRS(-P): Social Skills Rating System Scale (-Parent form)
T(0, 1): time
TAU: treatment-as-usual
TEA-Ch 2: Test of Everyday Attention for Children 2
TRIAD: Treatment and Research Institute for Autism Spectrum Disorders
TSC: Trial Steering Committee
TSSA: TRIAD Social Skills Assessment

Informed decisions.
VABS: Vineland Adaptive Behavior Scales

VPES: Verbal Production Evaluation Scale
Characteristics of excluded studies [ordered by study ID]
Study Reason for exclusion
Bringas 2015 Not ASD (severe neurological disorders)
Cowan 2016 Not an RCT or CCT (uncontrolled design)
Dezfoolian 2013 Not an RCT or CCT (uncontrolled design)
Edgerton 1994 Not an RCT or CCT (case series)
Finnigan 2010 Not an RCT or CCT (case study)
Gooding 2011 Not an RCT or CCT (uncontrolled design)
Hairston 1990 Not an RCT or CCT (case series)
Iseri 2014 Not an RCT or CCT (uncontrolled design)
Kern 2006 Not an RCT or CCT (case series)
Kern 2007 Not an RCT or CCT (case study)
Kim 2000 Not an RCT or CCT (uncontrolled design)
Mendelson 2016 No relevant comparison condition (both groups MT)
Sanglakh Goochan Atigh 2017 Not MT (movement activities with music)
Saperston 1973 Not an RCT or CCT (case study)
Thaut 1988 Not an intervention study (assessment)
Yoo 2018 Not an RCT or CCT (uncontrolled design)

CCT: controlled clinical trial
MT: music therapy
RCT: randomised controlled trial
Characteristics of studies awaiting classification [ordered by study ID]
NCT03267095
Blindness: none
Duration: 12 months
Participants Diagnosis: autism spectrum disorder; IQ ≥ 75

Estimated N: 60
Ages eligible: 3 to 7 years
Sexes eligible: all

Informed decisions.
NCT03267095 (Continued)
Setting: unknown
Location: Egypt
Interventions 1. Experimental: music therapy sessions for 12 months; songs composed by the therapist and target
words (selected from functional vocabularies that typically developing 3-year-old children can
use effectively in everyday interactions) will be used for the study
2. Control:counselling (setting, duration, and number of sessions unclear)
Outcomes 1. Arabic language test, pre- and post intervention
Notes Comment: awaiting classification as the trial registration does not contain sufficient information
on some aspects of the design (randomisation, diagnosis, types of interventions) to ensure all eligi-
bility criteria are met; recruitment has not started
Funding source: Assiut University
IQ: Intelligence quotient

N: number
Characteristics of ongoing studies [ordered by study ID]
ISRCTN18340173
Study name Public title: Improvisational music therapy for children with autism spectrum disorder assessed
using brain imaging
Scientific title: Music therapy outcome study for children with Autism Spectrum Disorder through
integration of child neuroimaging and neuropsychology: an exploratory study
Methods Allocation: non-randomised (propensity score matching)

Blindness: single (outcomes assessor)
Duration: 1 year
Design: parallel
Participants Diagnosis: children with ASD (confirmed by DSM-5 and ADOS)

Exclusion criteria: hearing/visual impairment, congenital genetic disease (cerebral palsy); seizure
and other neurological condition; other psychiatric disorder such as ADHD, schizophrenia, depres-
sion, bi-polar; previous and on-going experiences of music therapy; medication such as risperi-
done, aripirazole, SSRI, etc.
Estimated N: 50
Ages eligible: 24-72 months
Sexes eligible: all
Setting: Jeonju University, Seoul National University Hospital, and four music therapy centres
Location: Seoul, Jeonju, and Namyangju-si, South Korea
Interventions 1. Experimental: improvisational music therapy, once weekly, for one year
2. Control: standard care
Outcomes Primary outcome measures
1. Severity of autism measured using the Autism Diagnostic Observation Schedule (ADOS), pre-, mid-
(6 months), and post-intervention
2. Severity of autism measured using the Childhood Autism Rating Scale-2 (CARS-2),pre-, mid-(6
months), and post-intervention
Secondary outcome measures
Collected pre-, mid-(6 months), and post-intervention:
1. Social Responsiveness Scale (SRS)

Informed decisions.
ISRCTN18340173 (Continued)
2. Aberrant Behavior Checklist (ABC)
3. Social Communication Questionnaire (SCQ)
4. Social Maturity Scale (SMS)
5. Child Behavior Checklist 1.5-5 (CBCL 1.5-5)
6. Beck Depression Inventory (BDI)
7. State-Trait Anxiety Inventory (STAI-T/S)
8. Maternal Behavior Research Instrument (MBRI)
9. Korean Parenting Stress Index (PSI)
10.Mental Health Continuum-Short Form (MHC-SF)
11.Carer-QoL-7D/Child QoL (VAS)
12.Concomitant treatment form
Collected pre- and post-intervention:
1. Neuroimaging: aMRI/DTI/rsfMRI
2. Psychoeducational Profile-Revised (PEP-R)
Baseline test only: Blood and Urine for detecting DNA and harmful environmental exposure
Post-trial in-depth interview with the mothers and the therapist (thematic analysis; qualitative
study)
Starting date 1 August 2018
Contact information Jinah Kim, Professor, Jeonju University, Jeonju, South Korea
Notes Funding source: National Research Foundation of Korea
NCT03560297
Study name Public title: SeRenade parent-child music class program
Scientific title: SeRenade parent-child music class program for families of children with and with-
out ASD

Duration: 12 weeks
Design: cross-over
Participants Diagnosis: children diagnosed with ASD or children without ASD

Exclusion criteria: currently enrolled in music therapy or parent-child music classes (for children
with ASD); significant behaviour problems (e.g. aggression toward other children); significant hear-
ing or visual impairments
Estimated N: 68
Ages eligible: 20-72 months
Sexes eligible: all
Setting: Vanderbilt University Medical Center
Location: Nashville, Tennessee, USA
Interventions 1. Experimental: SeRenade programme: parent-child music class programme (parent training,
peer inclusion, musical play)
2. Control: delayed/waiting-list programme: participants do not participate in the programme for a
time period

Informed decisions.
1. Motor Imitation Scale (standardised, elicited imitation assessment; scores range 0-32; greater
scores indicate greater imitation skills), pre- and post-intervention
2. Actions & Gestures (standardised, parent-report questionnaire of child's gestures/actions/pre-
tend play), pre- and post-intervention
3. Parenting Stress Index, 4th edition Short Form (standardised, parent-report questionnaire), pre-
4. Parenting efficacy/quality (parent-report questionnaire; range 10-40; higher scores indicate
greater efficacy), pre- and post-intervention
1. Parent-child interaction (parent-child play/music session), pre- and post-intervention

2. Social visual attention eye-tracking paradigm, pre- and post-intervention
Other outcome measures
1. Parent mood/connection (parent ratings), pre- and post-intervention

2. Programme acceptability (parent programme evaluation survey), post-intervention
Starting date 15 May 2018
Contact information Miriam Lense, Research Instructor, Vanderbilt University Medical Center, Nashville, Tennessee,
USA, 37232
Notes Funding sources: Vanderbilt University Medical Center; National Endowment for the Arts; USA
NCT04557488
Study name Public title: Effectiveness of music therapy in social skill intervention for children with ASD/ID
Scientific title: Effectiveness of music therapy in social skill intervention for children with ASD/ID:
a randomized controlled trial

Duration: 12 weeks
Design: parallel
Participants Diagnosis: children with a formal clinical diagnosis of ASD and an overall and verbal IQ of 50-84 as
assessed by certified clinician
Exclusion criteria: severe physical or sensory disabilities (e.g. deafness); other neurodevelopmen-
tal, psychiatric, or neurological comorbidities or on prescribed psychiatric medication
Estimated N: 80
Ages eligible: 6-13 years
Sexes eligible: all
Setting: The Education University of Hong Kong
Location: Hong Kong
Interventions 1. Experimental: Group music therapy: sessions follow a similar structure (hello song, musical ac-
tivities, goodbye song); musical activities will vary in each session and will be mixed in later ses-
sions to revisit and practice social skills; groups of eight, led by certified music therapist
2. Control: Behavioural-based social skill group training: sessions follow a standard structure
(opening greetings, social activities according to the theme of the session, closing activity); activi-
ties and games will vary in each session and will be mixed in later session to revisit and practice so-
cial skills; groups of eight, led by registered social worker with experience in providing social skill
training for children with ASD and ID

Informed decisions.
1. Childhood Autism Rating Scale-2; pre- and post-intervention and follow-up 4 months after inter-
vention
2. Social Responsiveness Scale second edition; pre- and post-intervention and follow-up 4 months
after intervention
3. In-session social behaviour (coding of videos); first and last session
1. EEG recordings (in three conditions: resting state, social scenes, preferred music; for 5 minutes
each); pre- and post-intervention and follow-up 4 months after intervention
Starting date 1 October 2020
Contact information Yen Na Cherry Yum, Assistant Professor, Education University of Hong Kong, Hong Kong
Notes Funding source: Education University of Hong Kong
NCT04936048
Study name Public title: Music for autism (M4A)
Scientific title: Music for autism: binational randomised controlled trial of music therapy versus
play therapy for autistic children

Duration: 12 weeks
Design: cross-over
Participants Diagnosis: children diagnosed with ASD by a licensed clinical professional using standardised di-
agnostic tools (ADOS, ADI-R)
Exclusion criteria: recent or current music therapy; metallic or electronic implants
Estimated N: 80
Ages eligible: 6-12 years
Sexes eligible: all
Setting: University of Vienna, Vienna, Austria; NORCE Norwegian Research Centre, Bergen, Norway
Locations: Vienna, Austria; Bergen, Norway
Interventions 1. Experimental: music therapy (MT) using rhythmic cues, music instruments, songs, and stories
accompanied by songs or musical instruments to target common goals (multisensory integration,
verbal and social communication, emotion regulation, turn-taking, social appropriateness, interac-
tion); 12 weekly one-on-one sessions of 45 minutes each, conducted by a licensed music therapist
2. Control: play therapy using verbal interaction, toys (Lego, finger puppets, Play Doh, puzzles),
and the same stories as in MT, but without a musical component, to target the same common goals
as MT; 12 weekly one-on-one sessions of 45 minutes each, conducted by a licensed music therapist
1. Children's Communication Checklist-2; pre- and post-intervention

2. Brain connectivity of frontotemporal regions; pre- and post-intervention
1. Child and Adolescent Scale of Participation; pre- and post-intervention

2. Beach Center Family Quality of Life Scale; pre- and post-intervention
3. Peabody Picture Vocabulary Test-4th edition; pre- and post-intervention
4. Social Responsiveness Scale; pre- and post-intervention
5. Vineland Adaptive Behavior Scales; pre- and post-intervention

Informed decisions.
6. Hair cortisol concentration; pre- and post-intervention
7. Grey and white matter volume (structural brain changes); pre- and post-intervention
Starting date 1 August 2021
Contact information Christian Gold, NORCE Norwegian Research Centre AS, Bergen, Norway
Notes Funding sources: NORCE Norwegian Research Centre AS & University of Bergen, Norway; Universi-
ty of Vienna, Austria
ABC: Aberrant Behavior Checklist

ADHD: attention deficit hyperactivity disorder
ADI-R: Autism Diagnostic Interview-Revised
ADOS: Autism Diagostic Observation Schedule
aMRI/DTI/rsfMRI: advanced magnetic resonance imaging/diffusion tensor imaging/resting-state functional magnetic resonance imaging
BDI: Beck Depression Inventory
CARS: Childhood Autism Rating Scale
CBCL: Child Behavior Checklist
DNA: deoxyribonucleic acid
DSM: Diagnostic and Statistical Manual of Mental Disorders
EEG: electroencephalogram
ID: intellectual disability
IQ: intelligence quotient
M4A: Music for Autism
MBRI: Maternal Behavior Research Instrument
MHC-SF: Mental Health Continuum-Short Form
MT: music therapy
PEP-R: Psychoeducational Profile Revised
PSI: Parenting Stress Index
QoL: quality of life
SCQ: Social Communication Questionnaire
SMS: Social Maturity Scale
SRS: Social Responsiveness Scale
SSRI: selective serotonin reuptake inhibitors
STAI-T/S: State-Trait Anxiety Inventory
VAS: visual analogue scale
DATA AND ANALYSES
Comparison 1. Music therapy vs placebo therapy or standard care
Outcome or subgroup title No. of studies No. of partici- Statistical method Effect size
pants
1.1 Global improvement 8 Risk Ratio (M-H, Fixed, 95% CI) Subtotals only
1.1.1 Immediately post-inter- 8 583 Risk Ratio (M-H, Fixed, 95% CI) 1.22 [1.06, 1.40]
vention
1.1.2 1-5 months post-inter- 2 99 Risk Ratio (M-H, Fixed, 95% CI) 1.19 [0.90, 1.57]
vention
vention

Informed decisions.
pants
1.2 Social interaction 14 Std. Mean Difference (IV, Random, Subtotals only
95% CI)
1.2.1 During intervention 3 44 Std. Mean Difference (IV, Random, 1.15 [0.49, 1.80]
95% CI)
1.2.2 Immediately post-inter- 12 603 Std. Mean Difference (IV, Random, 0.26 [-0.05, 0.57]
vention 95% CI)
1.2.3 1-5 months post-inter- 2 59 Std. Mean Difference (IV, Random, 0.54 [-0.11, 1.19]
vention 95% CI)
1.2.4 6-11 months post-inter- 1 258 Std. Mean Difference (IV, Random, -0.06 [-0.30, 0.18]
vention 95% CI)
1.3 Non-verbal communica- 9 Std. Mean Difference (IV, Fixed, 95% Subtotals only
tion CI)
1.3.1 During intervention 3 50 Std. Mean Difference (IV, Fixed, 95% 1.06 [0.44, 1.69]
CI)
1.3.2 Immediately post-inter- 7 192 Std. Mean Difference (IV, Fixed, 95% 0.26 [-0.03, 0.55]
vention CI)
1.4 Verbal communication 12 Std. Mean Difference (IV, Random, Subtotals only
95% CI)
1.4.1 During intervention 4 129 Std. Mean Difference (IV, Random, -0.06 [-0.41, 0.28]
95% CI)
1.4.2 Immediately post-inter- 8 276 Std. Mean Difference (IV, Random, 0.30 [-0.18, 0.78]
vention 95% CI)
vention 95% CI)
1.5 Quality of life 3 Std. Mean Difference (IV, Fixed, 95% Subtotals only
CI)
1.5.1 Immediately post-inter- 3 340 Std. Mean Difference (IV, Fixed, 95% 0.28 [0.06, 0.49]
vention CI)
1.5.2 6-11 months post-inter- 1 249 Std. Mean Difference (IV, Fixed, 95% 0.04 [-0.21, 0.29]
vention CI)
1.6 Total autism symptom 9 Std. Mean Difference (IV, Random, Subtotals only
severity 95% CI)
1.6.1 During intervention 1 16 Std. Mean Difference (IV, Random, 0.15 [-0.83, 1.14]
95% CI)
1.6.2 Immediately post-inter- 9 575 Std. Mean Difference (IV, Random, -0.83 [-1.41, -0.24]
vention 95% CI)

Informed decisions.
pants
1.6.3 1-5 months post-inter- 2 69 Std. Mean Difference (IV, Random, -0.93 [-1.81, -0.06]
vention 95% CI)
vention 95% CI)
1.7 Adverse events 1 Risk Ratio (M-H, Fixed, 95% CI) Subtotals only
1.7.1 Immediately post-inter- 1 290 Risk Ratio (M-H, Fixed, 95% CI) 1.52 [0.39, 5.94]
vention
vention
1.8 Adaptive behaviour 9 Std. Mean Difference (IV, Fixed, 95% Subtotals only
CI)
1.8.1 During intervention 4 52 Std. Mean Difference (IV, Fixed, 95% 1.19 [0.56, 1.82]
CI)
1.8.2 Immediately post-inter- 5 462 Std. Mean Difference (IV, Fixed, 95% -0.02 [-0.20, 0.16]
vention CI)
1.8.3 1-5 months post-inter- 1 35 Std. Mean Difference (IV, Fixed, 95% 0.56 [-0.12, 1.24]
vention CI)
1.8.4 6-11 months post-inter- 1 290 Std. Mean Difference (IV, Fixed, 95% -0.12 [-0.36, 0.11]
vention CI)
1.9 Quality of family relation- 3 Std. Mean Difference (IV, Fixed, 95% Subtotals only
ships CI)
1.9.1 Immediately post-inter- 3 56 Std. Mean Difference (IV, Fixed, 95% 0.29 [-0.24, 0.83]
vention CI)
vention CI)
1.10 Identity formation 2 Std. Mean Difference (IV, Random, Subtotals only
95% CI)
1.10.1 Immediately post-in- 2 55 Std. Mean Difference (IV, Random, 1.35 [-0.58, 3.28]
tervention 95% CI)
1.10.2 1-5 months post-inter- 1 35 Std. Mean Difference (IV, Random, 0.86 [0.16, 1.55]
vention 95% CI)
1.11 Depression 1 Std. Mean Difference (IV, Fixed, 95% Subtotals only
CI)
1.11.1 Immediately post-in- 1 34 Std. Mean Difference (IV, Fixed, 95% -0.34 [-1.01, 0.34]
tervention CI)
vention CI)

Informed decisions.
Analysis 1.1. Comparison 1: Music therapy vs placebo therapy or standard care, Outcome 1: Global improvement
Experimental Control Risk Ratio Risk Ratio

Study or Subgroup Events Total Events Total Weight M-H, Fixed, 95% CI M-H, Fixed, 95% CI
1.1.1 Immediately post-intervention

Bharathi 2019 25 26 22 26 15.1% 1.14 [0.95 , 1.36]
Bieleninik 2017 95 182 76 182 52.1% 1.25 [1.00 , 1.56]
Kim 2008 10 10 8 10 5.8% 1.24 [0.87 , 1.75]
LaGasse 2014 7 9 3 8 2.2% 2.07 [0.79 , 5.42]
Porter 2017 10 24 10 23 7.0% 0.96 [0.49 , 1.86]
Rabeyron 2020 17 19 13 18 9.2% 1.24 [0.89 , 1.72]
Schwartzberg 2013 4 12 6 11 4.3% 0.61 [0.23 , 1.60]
Thompson 2014 11 12 6 11 4.3% 1.68 [0.95 , 2.96]
Subtotal (95% CI) 294 289 100.0% 1.22 [1.06 , 1.40]
Total events: 179 144
Heterogeneity: Chi² = 5.53, df = 7 (P = 0.60); I² = 0%
Test for overall effect: Z = 2.77 (P = 0.006)
1.1.2 1-5 months post-intervention

Bharathi 2019 23 26 19 26 65.0% 1.21 [0.92 , 1.59]
Porter 2017 12 24 10 23 35.0% 1.15 [0.62 , 2.13]
Subtotal (95% CI) 50 49 100.0% 1.19 [0.90 , 1.57]
Total events: 35 29

Bieleninik 2017 92 182 81 182 100.0% 1.14 [0.91 , 1.41]
Subtotal (95% CI) 182 182 100.0% 1.14 [0.91 , 1.41]
Total events: 92 81
Heterogeneity: Not applicable
Test for subgroup differences: Chi² = 0.29, df = 2 (P = 0.86), I² = 0% 0.5 0.7 1 1.5 2
Favours control Favours music therapy

Informed decisions.
Analysis 1.2. Comparison 1: Music therapy vs placebo therapy or standard care, Outcome 2: Social interaction
MT Control Std. Mean Difference Std. Mean Difference Risk of Bias
Study or Subgroup Mean SD Total Mean SD Total Weight IV, Random, 95% CI IV, Random, 95% CI A B C D E F G
1.2.1 During intervention

Arezina 2011 10.87 2.29 6 7.2 2.99 6 26.1% 1.27 [-0.02 , 2.56] + ? ? ? + + +
Kim 2008 1.58 1 10 0 1 10 41.8% 1.51 [0.49 , 2.53] + ? ? ? - + +
Thomas 2003 0.61 1 6 0 1 6 32.1% 0.56 [-0.60 , 1.73] ? ? ? ? + + +
Subtotal (95% CI) 22 22 100.0% 1.15 [0.49 , 1.80]
Heterogeneity: Tau² = 0.00; Chi² = 1.50, df = 2 (P = 0.47); I² = 0%

Bharathi 2019 114.85 15.07 26 112.62 15.27 26 10.3% 0.14 [-0.40 , 0.69] - - ? - + + +
Bieleninik 2017 -0.02 1 142 0 1 129 13.8% -0.02 [-0.26 , 0.22] + + ? + + + +
Chen 2013 -10.67 3 18 -17.81 3 9 5.6% 2.31 [1.27 , 3.35] ? ? ? ? + + +
Gattino 2011 -12.25 1.54 12 -11.92 1.24 12 7.5% -0.23 [-1.03 , 0.58] + + ? + + + +
Ghasemtabar 2015 30.55 4 13 27.34 3.54 14 7.6% 0.83 [0.03 , 1.62] - - ? ? + + +
Kim 2008 50.5 17.54 10 47.6 18.9 10 6.9% 0.15 [-0.73 , 1.03] + ? ? ? - + +
LaGasse 2014 -93.5 17.57 9 -103.88 26.52 8 6.2% 0.44 [-0.52 , 1.41] + ? ? ? + + +
Porter 2017 81.88 12.82 16 83.42 13.34 18 8.8% -0.11 [-0.79 , 0.56] + + ? - + + +
Rabeyron 2020 -12.95 3.34 19 -11.09 4 17 8.9% -0.50 [-1.16 , 0.17] + ? ? + + + +
Schwartzberg 2013 97.56 13 12 95.79 13.06 11 7.4% 0.13 [-0.69 , 0.95] ? ? ? - - + +
Sharda 2018 -69.36 10.89 26 -70.8 9.66 25 10.3% 0.14 [-0.41 , 0.69] + + ? + + + +
Thompson 2014 1 1 11 0 1 10 6.6% 0.96 [0.05 , 1.87] + + ? ? + + +
Subtotal (95% CI) 314 289 100.0% 0.26 [-0.05 , 0.57]

Ghasemtabar 2015 30.61 4.25 13 26.85 3.82 14 45.7% 0.90 [0.11 , 1.70] - - ? ? + + +
Porter 2017 82.57 13.96 14 79.14 14.14 18 54.3% 0.24 [-0.46 , 0.94] + + ? - + + +
Subtotal (95% CI) 27 32 100.0% 0.54 [-0.11 , 1.19]

Bieleninik 2017 -0.06 1 132 0 1 126 100.0% -0.06 [-0.30 , 0.18] + + ? + + + +
Subtotal (95% CI) 132 126 100.0% -0.06 [-0.30 , 0.18]
Test for subgroup differences: Chi² = 13.36, df = 3 (P = 0.004), I² = 77.5% -4 -2 0 2 4

Risk of bias legend
(A) Random sequence generation (selection bias)
(B) Allocation concealment (selection bias)
(C) Blinding of participants and personnel (performance bias)
(D) Blinding of outcome assessment (detection bias)
(E) Incomplete outcome data (attrition bias)
(F) Selective reporting (reporting bias)
(G) Other bias

Informed decisions.
Analysis 1.3. Comparison 1: Music therapy vs placebo therapy

or standard care, Outcome 3: Non-verbal communication
Study or Subgroup Mean SD Total Mean SD Total Weight IV, Fixed, 95% CI IV, Fixed, 95% CI A B C D E F G

Buday 1995 5.1 2.89 10 4 2.83 10 49.8% 0.37 [-0.52 , 1.25] ? ? ? + + + ?
Farmer 2003 28.85 15.69 5 8.05 3.9 5 16.3% 1.64 [0.10 , 3.19] ? ? ? - + + +
Kim 2008 1.88 1 10 0 1 10 33.8% 1.80 [0.73 , 2.88] + ? ? ? - + +
Subtotal (95% CI) 25 25 100.0% 1.06 [0.44 , 1.69]

Chen 2013 -1.83 0.7 18 -2.67 0.7 9 11.2% 1.16 [0.30 , 2.03] ? ? ? ? + + +
Gattino 2011 -2.5 0.37 12 -2.33 0.54 12 13.0% -0.35 [-1.16 , 0.45] + + ? + + + +
Kim 2008 29.1 15.33 10 20.2 15.64 10 10.5% 0.55 [-0.35 , 1.45] + ? ? ? - + +
LaGasse 2014 0.27 1 9 0 1 8 9.2% 0.26 [-0.70 , 1.21] + ? ? ? + + +
Rabeyron 2020 -2.13 0.81 19 -1.91 0.91 17 19.6% -0.25 [-0.91 , 0.41] + ? ? + + + +
Sharda 2018 80.46 15.26 24 76.43 11.64 23 25.6% 0.29 [-0.28 , 0.87] + + ? + + + +
Thompson 2014 34.91 10.83 11 27.8 10.7 10 10.9% 0.63 [-0.25 , 1.52] + + ? ? + + +
Subtotal (95% CI) 103 89 100.0% 0.26 [-0.03 , 0.55]

Risk of bias legend
(G) Other bias

Informed decisions.
Analysis 1.4. Comparison 1: Music therapy vs placebo therapy or standard care, Outcome 4: Verbal communication

Buday 1995 4.2 3.36 10 3.2 2.94 10 15.5% 0.30 [-0.58 , 1.19] ? ? ? + + + ?
Farmer 2003 113.3 129.8 5 53.15 60.85 5 7.4% 0.54 [-0.74 , 1.81] ? ? ? - + + +
Schwartzberg 2013 3.22 1.56 12 3.54 1.17 11 17.9% -0.22 [-1.04 , 0.60] ? ? ? - - + +
Schwartzberg 2016 3.86 1.06 38 4.05 0.93 38 59.3% -0.19 [-0.64 , 0.26] ? ? ? - - + +
Subtotal (95% CI) 65 64 100.0% -0.06 [-0.41 , 0.28]

Chen 2013 -2.33 0.71 18 -4 0.71 9 9.7% 2.28 [1.24 , 3.32] ? ? ? ? + + +
Gattino 2011 -2.54 0.45 12 -2.58 0.44 12 11.9% 0.09 [-0.71 , 0.89] + + ? + + + +
Lim 2010 133.11 77.23 18 89.62 74.06 32 14.0% 0.57 [-0.02 , 1.16] ? ? ? + + + +
Lim 2011 5.39 75 22 0 75 22 14.0% 0.07 [-0.52 , 0.66] + ? ? + + + +
Rabeyron 2020 -2.92 0.92 19 -2.26 0.87 17 13.1% -0.72 [-1.40 , -0.04] + ? ? + + + +
Schwartzberg 2013 4.12 1.26 12 4.29 1.14 11 11.7% -0.14 [-0.96 , 0.68] ? ? ? - - + +
Sharda 2018 95.04 26.44 26 85.92 29.4 25 14.4% 0.32 [-0.23 , 0.87] + + ? + + + +
Thompson 2014 232.82 147.42 11 178.7 98.18 10 11.2% 0.41 [-0.46 , 1.28] + + ? ? + + +
Subtotal (95% CI) 138 138 100.0% 0.30 [-0.18 , 0.78]

Bharathi 2019 82.46 18.73 26 78.73 14.99 26 100.0% 0.22 [-0.33 , 0.76] - - ? - + + +
Subtotal (95% CI) 26 26 100.0% 0.22 [-0.33 , 0.76]
Test for subgroup differences: Chi² = 1.73, df = 2 (P = 0.42), I² = 0% -4 -2 0 2 4

Risk of bias legend
(G) Other bias
Analysis 1.5. Comparison 1: Music therapy vs placebo therapy or standard care, Outcome 5: Quality of life
Experimental Control Std. Mean Difference Std. Mean Difference
Study or Subgroup Mean SD Total Mean SD Total Weight IV, Fixed, 95% CI IV, Fixed, 95% CI

Bieleninik 2017 75.76 14.4 136 72.76 16.29 129 78.7% 0.19 [-0.05 , 0.44]
Sharda 2018 105.36 9.58 26 99.96 11.29 25 14.7% 0.51 [-0.05 , 1.07]
Yurteri 2019 58.88 9.33 12 52.4 6.86 12 6.6% 0.76 [-0.07 , 1.60]
Subtotal (95% CI) 174 166 100.0% 0.28 [0.06 , 0.49]

Bieleninik 2017 75.16 16.08 125 74.48 15.27 124 100.0% 0.04 [-0.21 , 0.29]
Subtotal (95% CI) 125 124 100.0% 0.04 [-0.21 , 0.29]


Informed decisions.

or standard care, Outcome 6: Total autism symptom severity
Music therapy Placebo therapy or standard care Std. Mean Difference Std. Mean Difference Risk of Bias

Mateos-Moreno 2013 19.75 11.74 8 18.04 9.18 8 100.0% 0.15 [-0.83 , 1.14] - - ? ? + + +
Subtotal (95% CI) 8 8 100.0% 0.15 [-0.83 , 1.14]

Bharathi 2019 29.88 0.78 26 31.08 1.26 26 12.1% -1.13 [-1.72 , -0.54] - - ? - + + +
Bieleninik 2017 17.09 5.65 165 16.13 5.79 148 13.5% 0.17 [-0.05 , 0.39] + + ? + + + +
Chen 2010 68.4 6.23 15 74.73 6.67 15 11.2% -0.95 [-1.72 , -0.19] ? ? ? ? + + +
Chen 2013 38.17 4.79 18 64.56 23.19 9 10.0% -1.87 [-2.83 , -0.90] ? ? ? ? + + +
Huang 2015 48.1 12 30 64.5 12 30 12.2% -1.35 [-1.91 , -0.78] ? ? ? ? + + ?
LaGasse 2014 53.22 25.34 9 59.13 15.24 8 10.0% -0.26 [-1.22 , 0.69] + ? ? ? + + +
Mateos-Moreno 2013 4.62 3.29 8 14.25 5.8 8 8.5% -1.93 [-3.18 , -0.68] - - ? ? + + +
Rabeyron 2020 35.9 8.2 19 33.8 10.8 17 11.7% 0.22 [-0.44 , 0.87] + ? ? + + + +
Yurteri 2019 59 16.46 12 71.42 11.98 12 10.7% -0.83 [-1.67 , 0.01] ? ? ? ? + + +
Subtotal (95% CI) 302 273 100.0% -0.83 [-1.41 , -0.24]
Heterogeneity: Tau² = 0.64; Chi² = 61.33, df = 8 (P < 0.00001); I² = 87%

Bharathi 2019 30.04 0.56 26 31.23 1.14 26 59.0% -1.31 [-1.91 , -0.70] - - ? - + + +
LaGasse 2014 43.57 18.5 9 50.83 15.79 8 41.0% -0.40 [-1.36 , 0.57] + ? ? ? + + +
Subtotal (95% CI) 35 34 100.0% -0.93 [-1.81 , -0.06]

Bieleninik 2017 16.25 6.23 154 15.16 5.79 135 100.0% 0.18 [-0.05 , 0.41] + + ? + + + +
Subtotal (95% CI) 154 135 100.0% 0.18 [-0.05 , 0.41]
-4 -2 0 2 4
Risk of bias legend Favours music therapy Favours control
(G) Other bias
Analysis 1.7. Comparison 1: Music therapy vs placebo therapy or standard care, Outcome 7: Adverse events
Music therapy Placebo or standard care Risk Ratio Risk Ratio

Study or Subgroup Events Total Events Total Weight M-H, Fixed, 95% CI M-H, Fixed, 95% CI

Bieleninik 2017 6 165 3 125 100.0% 1.52 [0.39 , 5.94]
Subtotal (95% CI) 165 125 100.0% 1.52 [0.39 , 5.94]
Total events: 6 3

Bieleninik 2017 4 154 4 136 100.0% 0.88 [0.23 , 3.46]
Subtotal (95% CI) 154 136 100.0% 0.88 [0.23 , 3.46]
Total events: 4 4
Test for subgroup differences: Chi² = 0.30, df = 1 (P = 0.58), I² = 0% 0.1 0.2 0.5 1 2 5 10
Favours music therapy Favours control

Informed decisions.
Analysis 1.8. Comparison 1: Music therapy vs placebo therapy or standard care, Outcome 8: Adaptive behaviour
MT Control Std. Mean Difference Std. Mean Difference

Arezina 2011 89.2 12.18 6 51.08 24.53 6 19.0% 1.82 [0.38 , 3.25]
Brownell 2002 -10.62 8.53 4 -12.5 6.99 4 20.2% 0.21 [-1.18 , 1.60]
Kim 2008 1.06 1 10 0 1 10 44.0% 1.02 [0.07 , 1.96]
Thomas 2003 632.67 115.15 6 407.17 78.35 6 16.7% 2.11 [0.58 , 3.64]
Subtotal (95% CI) 26 26 100.0% 1.19 [0.56 , 1.82]

Bieleninik 2017 -3.82 1.94 165 -3.69 1.99 148 67.8% -0.07 [-0.29 , 0.16]
Chen 2010 55.47 15.68 15 52.27 21.46 15 6.5% 0.17 [-0.55 , 0.88]
Porter 2017 -84.53 33.01 15 -87 30.97 18 7.1% 0.08 [-0.61 , 0.76]
Rabeyron 2020 -48.6 27.2 19 -49.8 28.6 17 7.8% 0.04 [-0.61 , 0.70]
Sharda 2018 -19.42 1.76 26 -19.54 2.04 24 10.8% 0.06 [-0.49 , 0.62]
Subtotal (95% CI) 240 222 100.0% -0.02 [-0.20 , 0.16]

Porter 2017 -71.19 33.43 16 -89.55 30.8 19 100.0% 0.56 [-0.12 , 1.24]
Subtotal (95% CI) 16 19 100.0% 0.56 [-0.12 , 1.24]

Bieleninik 2017 -3.66 2.11 155 -3.4 2.05 135 100.0% -0.12 [-0.36 , 0.11]
Subtotal (95% CI) 155 135 100.0% -0.12 [-0.36 , 0.11]


or standard care, Outcome 9: Quality of family relationships

Kim 2008 16.4 1.43 10 15.8 1.55 10 36.5% 0.39 [-0.50 , 1.27]
Porter 2017 -2.1 0.26 6 -2.11 0.28 11 29.0% 0.03 [-0.96 , 1.03]
Thompson 2014 201.6 24.82 10 191.33 21.93 9 34.5% 0.42 [-0.50 , 1.33]
Subtotal (95% CI) 26 30 100.0% 0.29 [-0.24 , 0.83]

Porter 2017 -2.03 0.23 6 -2.02 0.25 9 100.0% -0.04 [-1.07 , 0.99]
Subtotal (95% CI) 6 9 100.0% -0.04 [-1.07 , 0.99]


Informed decisions.
Analysis 1.10. Comparison 1: Music therapy vs placebo therapy or standard care, Outcome 10: Identity formation
Study or Subgroup Mean SD Total Mean SD Total Weight IV, Random, 95% CI IV, Random, 95% CI

Moon 2010 2.51 1 10 0 1 10 46.6% 2.40 [1.20 , 3.61]
Porter 2017 18.88 6.43 16 16.11 6.15 19 53.4% 0.43 [-0.24 , 1.10]
Subtotal (95% CI) 26 29 100.0% 1.35 [-0.58 , 3.28]

Porter 2017 20.81 5.42 16 15.89 5.77 19 100.0% 0.86 [0.16 , 1.55]
Subtotal (95% CI) 16 19 100.0% 0.86 [0.16 , 1.55]

Analysis 1.11. Comparison 1: Music therapy vs placebo therapy or standard care, Outcome 11: Depression


Porter 2017 14.44 11.38 16 18.56 12.5 18 100.0% -0.34 [-1.01 , 0.34]
Subtotal (95% CI) 16 18 100.0% -0.34 [-1.01 , 0.34]

Porter 2017 13.71 11.9 17 21.16 12.45 19 100.0% -0.60 [-1.27 , 0.07]
Subtotal (95% CI) 17 19 100.0% -0.60 [-1.27 , 0.07]

Favours music therapy Favours control
ADDITIONAL TABLES
Table 1. Summarised characteristics of included studies

Category Studies
Studies included in each version of this review
First version (2006) Brownell 2002; Buday 1995; Farmer 2003
Second version (2014) Arezina 2011; Gattino 2011; Kim 2008; Lim 2010; Lim 2011; Thomas 2003; Thompson 2014 (which is
a new report to a previously reported study)
Current update Bharathi 2019; Bieleninik 2017 (with two more reports related to this study); Chen 2010; Chen 2013;
Ghasemtabar 2015; Huang 2015; LaGasse 2014; Mateos-Moreno 2013; Moon 2010; Porter 2017 (with

Informed decisions.
Table 1. Summarised characteristics of included studies (Continued)

another report related to this study); Rabeyron 2020; Sa 2020; Schwartzberg 2013; Schwartzberg
2016; Sharda 2018 (with another report related to this study); Yurteri 2019
Location
North America Canada: Sharda 2018

USA: Arezina 2011; Brownell 2002; Buday 1995; Farmer 2003; LaGasse 2014; Lim 2010; Lim 2011; Sa
2020 Schwartzberg 2013; Schwartzberg 2016; Thomas 2003
South America Brazil: Gattino 2011
Asia China: Chen 2010; Chen 2013; Huang 2015
Korea: Kim 2008; Moon 2010

India: Bharathi 2019
Iran: Ghasemtabar 2015
Europe France: Rabeyron 2020

Spain: Mateos-Moreno 2013
Turkey: Yurteri 2019
UK: Porter 2017
Oceania Australia: Thompson 2014
Multinational Bieleninik 2017 (Australia, Austria, Brazil, Korea, Israel, Italy, Norway, UK, USA)
Design
Parallel group Bharathi 2019; Bieleninik 2017; Chen 2010; Chen 2013; Farmer 2003; Gattino 2011;
Ghasemtabar 2015; Huang 2015; LaGasse 2014; Lim 2010; Mateos-Moreno 2013; Moon 2010; Porter
2017; Rabeyron 2020; Sa 2020; Schwartzberg 2013; Schwartzberg 2016; Sharda 2018; Thompson
2014; Yurteri 2019
Cross-over Arezina 2011; Brownell 2002; Buday 1995; Kim 2008; Lim 2011; Thomas 2003
Individual participant data
Available Arezina 2011; Bharathi 2019; Bieleninik 2017; Brownell 2002; Farmer 2003; Gattino 2011; Kim 2008;
LaGasse 2014; Porter 2017; Rabeyron 2020; Schwartzberg 2013; Schwartzberg 2016; Thomas 2003;
Thompson 2014
Interventions
Music therapy setting Individual setting (one-to-one): Arezina 2011; Bieleninik 2017; Brownell 2002; Buday 1995; Farmer
2003; Gattino 2011; Kim 2008; Lim 2010; Lim 2011; Porter 2017; Sharda 2018; Thomas 2003; Yurteri
2019
Group setting: Bharathi 2019; Ghasemtabar 2015; LaGasse 2014; Mateos-Moreno 2013; Rabeyron
2020; Sa 2020; Schwartzberg 2013; Schwartzberg 2016
Either individually or in small groups: Yurteri 2019
Family-based setting: Thompson 2014
Unclear: Chen 2010; Chen 2013; Huang 2015; Moon 2010

Informed decisions.
Music therapy frequency Daily (for 1-2 weeks): Brownell 2002; Buday 1995; Farmer 2003; Lim 2010; Lim 2011; Schwartzberg
2013; Schwartzberg 2016
Weekly: Arezina 2011; Gattino 2011; Kim 2008; Porter 2017; Rabeyron 2020; Sharda 2018; Thomas
2003; Thompson 2014
Twice weekly: Chen 2013; Ghasemtabar 2015; LaGasse 2014; Mateos-Moreno 2013; Moon 2010; Yur-
teri 2019
Several times a week: Bharathi 2019 (3 times a week); Chen 2010 (4 times a week); Huang 2015 (6
times a week)
1 or 3 times a week: Bieleninik 2017
Music therapy content Highly structured: Brownell 2002; Buday 1995; Chen 2010; Chen 2013; Farmer 2003; Lim 2010; Lim
2011; Moon 2010; Rabeyron 2020; Sa 2020; Schwartzberg 2013; Schwartzberg 2016
Emphasis on interactive and relational aspects: Arezina 2011; Bharathi 2019; Bieleninik 2017; Gat-
tino 2011; Ghasemtabar 2015; Huang 2015; Kim 2008; LaGasse 2014; Mateos-Moreno 2013; Porter
2017; Sharda 2018; Thomas 2003; Thompson 2014; Yurteri 2019
Comparators
'Placebo' therapy 'Placebo' activity without music: Arezina 2011; Brownell 2002; Buday 1995; Farmer 2003; Kim 2008;
LaGasse 2014; Lim 2010; Lim 2011; Moon 2010; Schwartzberg 2013; Schwartzberg 2016; Shar-
da 2018; Thomas 2003
Passive music listening: Bharathi 2019; Rabeyron 2020
Standard care Bieleninik 2017; Chen 2010; Chen 2013; Gattino 2011; Ghasemtabar 2015; Huang 2015; Ma-
teos-Moreno 2013; Porter 2017; Sa 2020; Thompson 2014; Yurteri 2019
Outcomes
Global improvement Bieleninik 2017; Bharathi 2019; Kim 2008; LaGasse 2014; Porter 2017; Rabeyron 2020; Schwartzberg
2013; Thompson 2014
Social interaction Arezina 2011; Bharathi 2019; Bieleninik 2017; Chen 2013; Gattino 2011; Ghasemtabar 2015; Kim
2008; LaGasse 2014; Porter 2017; Rabeyron 2020; Schwartzberg 2013; Sharda 2018; Thomas 2003;
Thompson 2014
Non-verbal communication Arezina 2011; Buday 1995; Chen 2013; Farmer 2003; Gattino 2011; Kim 2008; LaGasse 2014; Rabey-
ron 2020; Sharda 2018; Thomas 2003; Thompson 2014
Verbal communication Buday 1995; Chen 2013; Farmer 2003; Gattino 2011; Lim 2010; Lim 2011; Rabeyron 2020;
Schwartzberg 2013; Schwartzberg 2016; Sharda 2018; Thompson 2014
Quality of life Bieleninik 2017; Sharda 2018; Yurteri 2019
Total autism symptom severity Bharathi 2019; Bieleninik 2017; Chen 2010; Chen 2013; Huang 2015; LaGasse 2014; Mateos-Moreno
2013; Rabeyron 2020; Yurteri 2019
Adverse events Bieleninik 2017; Porter 2017
Adaptive behaviour Arezina 2011; Bieleninik 2017; Brownell 2002; Chen 2010; Kim 2008; Porter 2017; Rabeyron 2020;
Sharda 2018; Thomas 2003

Informed decisions.
Quality of family relationships Kim 2008; Porter 2017; Thompson 2014
Identity formation Moon 2010; Porter 2017
Depression Porter 2017
Cognitive ability Sa 2020
APPENDICES
Appendix 1. Search strategies 2020 onwards

When we ran the searches for this review update, we removed the search terms listed below from the original strategy because they did
not identify any unique relevant records:
1. (speech adj3 disorder$).tw.

2. (language adj3 delay$).tw.
CENTRAL
Searched 7 July 2020. Limited to publication year 2013-2020 (217 records)
Searched 4 August 2021. Limited to records added between 7 July 2021 and 4 August 2021 (60 records)
#1 MeSH descriptor: [Music]

#2 MeSH descriptor: [Music Therapy]
#3 music*
#4 ((guided next imagery) near music)
#5 GIM
#6 vibroacoustic
#7 vibro-acoustic
#8 (sing or singing or song* or choral* or choir*)
#9 (percussion* or rhythm* or tempo* or melod*)
#10 improvis*
#11 (Nordoff-Robbin* or bonny*)
#12 ((auditory or acoustic or sound*) near/5 (stimulat* or cue*))
#13 (#1 or #2 or #3 or #4 or #5 or #6 or #7 or #8 or #9 or #10 or #11 or #12)
#14 MeSH descriptor: [Child Development Disorders, Pervasive] 1 tree(s) exploded
#15 MeSH descriptor: [Neurodevelopmental Disorders] this term only
#16 MeSH descriptor: [Developmental Disabilities] this term only
#17 asperg* or autis* or kanner* or (childhood next schizophren*)
#18 ASD or ASDs or PDD or PDDs or PDD-NOS
#19 {or #14-#18}
#20 (#13 and #19) in Trials
MEDLINE Ovid
Searched 8 July 2020 (217 records)
Searched 4 August 2021 (29 records)
1 music therapy/
2 music/
3 music$.tw,kf.
4 ((guided imagery adj3 music) or gim).tw,kf.
5 (vibro-acoustic$ or vibroacoustic$).tw,kf.
6 (sing or singing or song$ or choral$ or choir$).tw,kf.
7 (percussion$ or rhythm$ or tempo).tw,kf.
8 melod$.tw,kf.
9 improvis$.tw,kf.

Informed decisions.
10 (Nordoff-Robbin$ or bonny$).tw,kf.
11 ((auditory or acoustic or sound$) adj5 (stimulat$ or cue$)).tw,kf.
12 or/1-11
13 exp child development disorders, pervasive/
14 Developmental Disabilities/
15 Neurodevelopmental Disorders/
16 pervasive development$ disorder$.tw,kf.
17 (pervasive adj3 child$).tw,kf.
18 (PDD or PDDs or PDD-NOS or ASD or ASDs).tw,kf.
19 autis$.tw,kf.
20 asperg$.tw,kf.
21 kanner$.tw,kf.
22 childhood schizophreni$.tw,kf.
23 or/13-22
24 randomized controlled trial.pt.
25 controlled clinical trial.pt.
26 randomi#ed.ab.
27 placebo$.ab.
28 drug therapy.fs.
29 randomly.ab.
30 trial.ab.
31 groups.ab.
32 or/24-31
33 exp animals/ not humans.sh.
34 32 not 33
35 12 and 23 and 34
36 limit 35 to ed=20130701-20200626 Annotation: Final line 2020 update search
37 (20200626* or 20200627* or 20200628* or 20200629* or 20200630* or 202007* or 202008* or 202009* or 202010* or 202011* or 202012*
or 2021*).dt,ez,da.
38 35 and 37 Annotation: Final line 2021 top-up search
MEDLINE In-Process & Other Non-Indexed Citations Ovid

1 music$.tw,kf.
2 (guided imagery adj3 music).tw,kf. or gim.tw,kf.
6 melod$.tw,kf.
7 improvis$.tw,kf.
10 or/1-9
14 autis$.tw,kf.
15 asperg$.tw,kf.
16 kanner$.tw,kf.
18 or/11-17
19 10 and 18
20 (random$ or trial$ or control$ or group$ or placebo$ or blind$ or prospectiv$ or longitudinal$ or meta-analys$ or systematic review
$).tw,kf.
21 19 and 20
MEDLINE Epub Ahead of Print Ovid


Informed decisions.
1 music$.tw,kf.
2 (guided imagery adj3 music).tw,kf. or gim.tw,kf.
6 melod$.tw,kf.
7 improvis$.tw,kf.
10 or/1-9
14 autis$.tw,kf.
15 asperg$.tw,kf.
16 kanner$.tw,kf.
18 or/11-17
19 10 and 18
20 (random$ or trial$ or control$ or group$ or placebo$ or blind$ or prospectiv$ or longitudinal$ or meta-analys$ or systematic review
$).tw,kf.
21 19 and 20
Embase Ovid
1 exp music/
2 music therapy/
3 music$.tw,kw.
4 (guided imagery adj3 music).tw,kw.
5 GIM.tw,kw.
6 (vibro-acoustic therapy or vibroacoustic therapy).tw,kw.
7 (sing or singing or song$ or choral$ or choir$).tw,kw.
8 (percussion$ or rhythm$).tw,kw.
9 melod$.tw,kw.
10 improvis$.tw,kw.
11 (Nordoff-Robbin$ or bonny$).tw,kw.
12 ((auditory or acoustic or sound$) adj5 (stimulat$ or cue$)).tw,kw.
13 or/1-12
14 exp autism/
15 developmental disorder/
16 pervasive development$ disorder$.tw.
17 (PDD or PDDs or PDD_NOS or ASD or ASDs).tw,kw.
18 autis$.tw.
19 asperg$.tw.
20 kanner$.tw.
21 childhood schizophreni$.tw.
22 or/14-21
23 13 and 22
24 Randomized controlled trial/
25 Controlled clinical study/
26 random$.ti,ab.
27 randomization/
28 intermethod comparison/
29 placebo.ti,ab.
30 (compare or compared or comparison).ti.
31 ((evaluated or evaluate or evaluating or assessed or assess) and (compare or compared or comparing or comparison)).ab.
32 (open adj label).ti,ab.
33 ((double or single or doubly or singly) adj (blind or blinded or blindly)).ti,ab.
34 double blind procedure/
35 parallel group$1.ti,ab.
Informed decisions.
36 (crossover or cross over).ti,ab.

37 ((assign$ or match or matched or allocation) adj5 (alternate or group$1 or intervention$1 or patient$1 or subject$1 or participant
$1)).ti,ab.
38 (assigned or allocated).ti,ab.
39 (controlled adj7 (study or design or trial)).ti,ab.
40 (volunteer or volunteers).ti,ab.
41 human experiment/
42 trial.ti.
43 or/24-42
44 (random$ adj sampl$ adj7 ("cross section$" or questionnaire$1 or survey$ or database$1)).ti,ab. not (comparative study/ or controlled
study/ or randomi?ed controlled.ti,ab. or randomly assigned.ti,ab.)
45 Cross-sectional study/ not (randomized controlled trial/ or controlled clinical study/ or controlled study/ or randomi?ed
controlled.ti,ab. or control group$1.ti,ab.) (238532)
46 (((case adj control$) and random$) not randomi?ed controlled).ti,ab.
47 (Systematic review not (trial or study)).ti.
48 (nonrandom$ not random$).ti,ab.
49 "Random field$".ti,ab.
50 (random cluster adj3 sampl$).ti,ab.
51 (review.ab. and review.pt.) not trial.ti.
52 "we searched".ab. and (review.ti. or review.pt.)
53 "update review".ab.
54 (databases adj4 searched).ab.
55 (rat or rats or mouse or mice or swine or porcine or murine or sheep or lambs or pigs or piglets or rabbit or rabbits or cat or cats or
dog or dogs or cattle or bovine or monkey or monkeys or trout or marmoset$1).ti. and animal experiment/
56 Animal experiment/ not (human experiment/ or human/)
57 or/44-56
58 43 not 57
59 23 and 58
60 limit 59 to yr="2013 -Current"
LILACS
Searched 7 July 2020. Limited by year =2013-2020 (7 records)
Searched 4 August 2021. Limited by year =2020-2021 (0 records)
(tw:((music* OR gim OR percussion* OR rhythm* OR tempo OR improvis* OR melod* OR sing OR singing OR song* OR choral* OR choir* OR
auditory OR acoustic OR sound* OR vibro*))) AND (tw:((autis* OR asperger* OR "pervasive developmental" OR ASD OR PDD OR PDD-nos)))
Results filtered by source (LILACS ) and study type (controlled clinical trial)
APA PsycInfo Ovid

1 exp music/
2 music therapy/
3 music$.tw.
4 (guided imag$ adj3 music* or gim).tw.
5 GIM.tw.
6 (vibroacoustic$ or vibro-acoustic$).tw.
7 rhythm/ or tempo/
8 (percussion$ or rhythm$ or tempo).tw.
9 singing/
10 (sing or singing or song$ or choral$ or choir$).tw.
11 melod$.tw.
12 improvis$.tw.
13 (Bonny or Nordoff$).tw.
14 ((auditory or acoustic or sound$) adj5 (stimulat$ or cue$)).tw.
15 or/1-14
16 exp autism spectrum disorders/
17 Developmental Disabilities/
18 neurodevelopmental disorders/
Informed decisions.
19 pervasive development$ disorder$.tw.

20 (PDD or PDDs or PDD-NOS or ASD or ASDs).tw.
21 autis$.tw.
22 asperger$.tw.
23 kanner$.tw.
24 childhood schizophreni$.tw.
25 or/16-24
26 randomized controlled trials/
27 randomized clinical trials/
28 Clinical Trials/
29 exp treatment effectiveness evaluation/
30 Placebo/
31 (control$ adj3 (study or studies or trial$ or group$)).tw.
32 (allocat$ or assign$).ab.
33 (placebo or treatment as usual or tau).ab.
34 (random$ or RCT).tw.
35 ((singl$ or doubl$ or tripl$ or trebl$) adj3 (blind$ or mask$)).tw.
36 (crossover$ or cross-over$).tw.
37 ((evaluat$ or effectiveness$) adj3 (study or studies or research$)).tw.
38 or/26-37
39 15 and 25 and 38
40 limit 39 to up=20130701-20200629
42 40 or 41
43 limit 39 to up=20200629-20210726
45 43 or 44
CINAHL Plus EBSCOhost

S1 (MH "Music")
S2 (MH "Music Therapy")
S3 music*
S4 ((guided imagery N3 music) or gim)
S5 (vibro-acoustic* or vibroacoustic*)
S6 (Nordoff* or Bonny*)
S7 (percussion* or rhythm* or tempo)
S8 melod*
S9 (MH "Singing")
S10 (sing or singing or song* or choral* or choir*)
S12 ((auditory or acoustic or sound*) N5 (stimulat* or cue*))
S13 S1 OR S2 OR S3 OR S4 OR S5 OR S6 OR S7 OR S8 OR S9 OR S10 OR S11 OR S12
S14 (MH "Child Development Disorders, Pervasive+")
S15 (pervasive development* disorder* or PDD or PDDs)
S16 (autis* or ASD or ASDs)
S17 Asperger*
S18 Kanner*
S19 childhood schizophren*
S20 (MH "Developmental Disabilities")
S21 S14 OR S15 OR S16 OR S17 OR S18 OR S19 OR S20
S22 S13 AND S21
S23 MH randomized controlled trials
S24 MH double-blind studies
S25 MH single-blind studies
S26 MH random assignment
S27 MH pretest-posttest design
S28 MH cluster sample5
S29 TI (randomised OR randomized)
S30 AB (random*)
Informed decisions.
S31 TI (trial)
S32 MH (sample size) AND AB (assigned OR allocated OR control)
S33 MH (placebos)
S34 PT (randomized controlled trial)
S35 AB (control W5 group)
S36 MH (crossover design) OR MH (comparative studies)
S37 AB (cluster W3 RCT)
S38 MH animals+
S39 MH (animal studies)
S40 TI (animal model*)
S41 S38 OR S39 OR S40
S42 MH (human)
S43 S41 not S42
S44 S23 OR S24 OR S25 OR S26 OR S27 OR S28 OR S29 OR S30 OR S31 OR S32 OR S33 OR S34 OR S35 OR S36 OR S37
S45 S44 not S43
S46 S22 AND S45
S47 EM 20130701-
S48 S46 AND S47
S49 EM 20200701-
S50 S46 AND S49
ERIC EBSCOhost
Searched 6 July 2020. Limited by year =2013-2020 (58 records)
S1 DE "Developmental Disabilities"
S2 DE "Pervasive Developmental Disorders" OR DE "Asperger Syndrome" OR DE "Autism"
S3 (pervasive development* disorder* or PDD or PDDs)
S5 Asperger*
S6 Kanner*
S8 S1 OR S2 OR S3 OR S4 OR S5 OR S6 OR S7
S9 DE "Music Therapy"
S10 (DE "Music" OR DE "Music Activities")
S11 music*
S15 (percussion* or rhythm* or tempo)
S16 melod*
S17 (DE "Singing")
S19 improvis*
S21 S9 OR S10 OR S11 OR S12 OR S13 OR S14 OR S15 OR S16 OR S17 OR S18 OR S19 OR S20
S22 S8 AND S21
S23 DE "Randomized Controlled Trials" OR DE "Meta Analysis" OR DE "Evaluation Research" OR DE "Control Groups" OR DE "Experimental
Groups" OR DE "Longitudinal Studies" OR DE "Followup Studies" OR DE "Program Effectiveness" OR DE "Program Evaluation"
S24 TI (random* or trial* or experiment* or prospectiv* OR longitudinal or BLIND* or CONTROL*) OR AB (random* or trial* or experiment*
or prospectiv* OR longitudinal or BLIND* or CONTROL*)
S25 S23 OR S24
S26 S22 AND S25
Sociological Abstracts Proquest

((su.EXACT("Music" ) or NOFT(music* or guided imag* or GIM or vibro-acoustic therapy* or vibroacoustic therapy* or Bonny* or Nordoff* or
singing or song* or choral* or choir* or percussion* or rhythm* or improvis*) OR NOFT((auditory or acoustic or sound*) near/5 (stimulat*
or cue*))) and (su.EXACT("autism" ) or NOFT(autism* or asperg* or "pervasive development* disorder*" or ("childhood schizophrenia")
or Kanner*)) and NOFT(random* or placebo* or trial* or blind* or group* or control or controlled or RCT or TAU or "usual treatment" or
"treatment as usual" )
Informed decisions.
Proquest Global Dissertations & Theses

( NOFT(music* or guided imag* or GIM or vibro-acoustic therapy* or "vibroacoustic therapy*" or Bonny* or Nordoff* or singing or song*
or choral* or choir* or percussion* or rhythm* or improvis*) OR NOFT((auditory or acoustic or sound*) near/5 (stimulat* or cue*))) and
( NOFT(autism* or asperg* or "pervasive development* disorder*" or "childhood schizophrenia*" or Kanner*)) and NOFT((random* or
placebo* or trial* or blind* or group* or control or controlled or RCT or TAU or "usual treatment" or "treatment as usual" ))
Proquest Music Periodicals Database

Searched 6 July 2020 No limits (138 records)
(NOFT(MUSIC* n/3 THERAP* ) OR NOFT((guided imagery N/3 music) OR gim ) OR NOFT( percussion* OR rhythm* OR tempo OR improvis*
OR melod* OR sing OR singing OR song* OR choral* OR choir* ) OR NOFT((auditory OR acoustic OR sound*) N/5 (stimulat* OR cue*)) OR
NOFT(Nordoff* OR Bonny*) OR NOFT((vibro-acoustic* OR vibroacoustic*))) AND NOFT(autis* OR asperger* OR kanner* OR "pervasive
developmental" OR ASD OR ASDs OR PDD or PDDs OR PDD-NOS ) AND NOFT(random* OR trial* OR experiment* OR prospectiv* OR
longitudinal OR blind* OR control* OR placebo OR "treatment as usual" OR TAU OR intervention* OR treat* )
Proquest Performing Arts Periodicals Database

Searched 4 August 2021. Limited by year =2020-2021 (1 record)
( NOFT((MUSIC* n/3 THERAP* ) OR (guided imagery N/3 music) OR gim OR percussion* or rhythm* or tempo or improvis* OR melod* OR
sing OR singing OR song* OR choral* OR choir* ) OR NOFT((auditory OR acoustic OR sound*) N/5 (stimulat* or cue*)) OR NOFT(Nordoff*
OR Bonny*) OR NOFT((vibro-acoustic* OR vibroacoustic*)) OR NOFT((MUSIC* n/3 THERAP* ) OR ("guided imagery" N/3 music) OR gim OR
percussion* OR rhythm* OR tempo OR improvis* OR melod* OR sing OR singing OR song* OR choral* OR choir* )) AND NOFT(autis* OR
asperger* OR kanner* OR "pervasive developmental" OR ASD OR ASDs OR PDD OR PDDs OR PDD-NOS )
RILM Abstracts of Music Literature Online EBSCOhost

Searched 4 August 2021. Limited by year =2020-2021 (1 record)
S1 SU "therapy—music therapy --*"

S2 music* N3 therap*
Database - RILM Abstracts of Music Literature (1967 to present)
S6 (percussion* or rhythm* or tempo or improvis*)
S7 melod*
S10 S1 OR S2 OR S3 OR S4 OR S5 OR S6 OR S7 OR S8 OR S9
S12 Asperger*
S13 Kanner*
S15 (pervasive development* disorder* or PDD or PDDs or PDD-NOS)

Informed decisions.
S16 S11 OR S12 OR S13 OR S14 OR S15

S17 S10 AND S16
S18 (random* or trial* or experiment* or prospectiv* OR longitudinal or blind* or control* or placebo or "treatment as usual" or TAU)
S19 S17 AND S18

#1 MeSH descriptor: [Music] this term only

#2 MeSH descriptor: [Music Therapy] this term only
#3 music*:ti,ab,kw
#4 (guided next imagery):ti,ab,kw
#5 GIM:ti,ab,kw
#6 vibroacoustic:ti,ab,kw
#7 vibro-acoustic:ti,ab,kw
#8 (sing or singing or song* or choral* or choir*):ti,ab,kw
#9 ((percussion* or rhythm* or tempo* or melod*) and music*):ti,ab,kw
#10 (improvis* near music*):ti,ab,kw
#11 (Nordoff-Robbin* or bonny*):ti,ab,kw
#12 ((auditory or acoustic or sound*) near/5 (stimulat* or cue*)):ti,ab,kw
#13 (#1 or #2 or #3 or #4 or #5 or #6 or #7 or #8 or #9 or #10 or #11 or #12)
#14 MeSH descriptor: [Child Development Disorders, Pervasive] this term only
#15 MeSH descriptor: [Neurodevelopmental Disorders] this term only
#16 MeSH descriptor: [Developmental Disabilities] this term only
#17 asperg* or autis* or kanner* or (childhood next schizophren*):ti,ab,kw
#18 (ASD or ASDs or PDD or PDDs or PDD-NOS):ti,ab,kw
#19 {or #14-#18}
#20 (#13 and #19) with Cochrane Library publication date Between Jul 2013 and Jul 2020, in Cochrane Reviews, Cochrane Protocols
#21 (#13 and #19) with Cochrane Library publication date Between Jul 2020 and Aug 2021, in Cochrane Reviews.Cochrane Protocols
Epistemonikos
Searched 4 August 2021. Limited to records added from 7 July 2020 to 4 August 2021 (2 records)
title:((autis* OR asperger* OR "pervasive developmental" OR ASD OR PDD OR PDD-nos)) AND (title:(((cue* OR signal*) AND (auditor* OR
acoustic* OR sound ))) OR abstract:(((cue* OR signal*) AND (auditor* OR acoustic* OR sound ))))
title:((autis* OR asperger* OR "pervasive developmental" OR ASD OR PDD OR PDD-nos)) AND (title:(music*) OR abstract:(music*))
title:((autis* OR asperger* OR "pervasive developmental" OR ASD OR PDD OR PDD-nos)) AND (title:(sing OR singing OR choral OR choir OR
tempo OR improvis* OR rhythm) OR abstract:(sing OR singing OR choral OR choir OR tempo OR improvis* OR rhythm))
ClinicalTrials.gov
Searched 5 August 2021. Limited to records first posted between 7 July 2020 and 5 August 2021 (7 records)
Advanced search: Interventional Studies | Autism OR autistic OR asperger OR ASD OR pervasive developmental disorder OR PDD OR PDD-
NOS | music OR tempo OR rhythm OR GIM or guided imagery OR acoustic OR auditory OR sound
WHO ICTRP
Search attempted 7 July 2020, but due to heavy traffic generated by the COVID-19 outbreak, the ICTRP Search Portal was not responding
from outside WHO.
Searched 5 August 2021 No limits (21 records)
Standard search: (music AND autism) OR (music AND ASD) OR (music AND asperger)

Informed decisions.
Appendix 2. Criteria for assigning risk of bias judgements

Random sequence generation
We judged the risk of bias for random sequence generation as follows.
1. Studies were judged to be at low risk of bias if participants were allocated to treatment interventions using randomisation, such as
computer-generated random numbers, a random numbers table, or coin-tossing.
2. Studies were judged to be at unclear risk of bias if the randomisation method was not clearly stated or was unknown.
3. Studies were judged to be at high risk of bias if the method sequence generation was non-random.
Randomised as well as controlled clinical trials were included in the review, as noted above (see Types of studies).
Allocation concealment
We judged the risk of bias for allocation concealment as follows.
1. Studies were judged to be at low risk of bias if the allocation concealment was adequate; participants and researchers were unaware of
participants' future allocation to an intervention until after decisions about eligibility were made, and informed consent was obtained.
2. Studies were judged to be at unclear risk of bias if the methods used for allocation concealment were not described in detail.
3. Studies were judged to be at high risk of bias if the allocation concealment was inadequate; allocation was not concealed, either
from participants before informed consent or from researchers before decisions about inclusion were made (this will always be the
case for quasi-randomised studies).
Both randomised and controlled trials were judged using the same criteria for gaining a descriptive measure of study quality.
Blindness of participants and personnel

Due to the nature of the intervention, it was not possible to blind those who delivered music therapy or those who received it. Consequently,
neither participants nor therapists of the studies under review could be declared to be blinded. However, although autistic individuals
were not blinded, this was unlikely to introduce bias as usually they are not fully aware of available treatment options or study design
(Cheuk 2011). The possible risk of bias introduced by therapists administering the intervention was unknown. Therefore, we judged the
risk of performance bias as unclear in all studies included in this review.
Blinding of outcome assessors

We determined whether those who assessed and coded the outcome measures were blind to treatment assignment using the following
categories.
1. Studies were judged to be at low risk of bias if the assessor was blind to treatment assignment.
2. Studies were judged to be at unclear risk of bias if the blinding of the assessor was not reported and information was not available from
the researchers.
3. Studies were judged to be at high risk of bias if the assessor was not blind to treatment assignment.
Completeness of outcome data

We assessed whether study authors adequately dealt with missing data as follows.
• Studies were judged to be at low risk of bias if the number of participants randomised to groups was clear and it was clear that all
participants completed the trials in all participant groups. Studies were also judged to be at low risk of bias if outcome data were missing
in both intervention groups, but reasons for these were both reported and balanced across groups.
• Studies were judged to be at unclear risk of bias if information about which participants completed the study could not be acquired
by contacting the study authors.
• Studies were judged to be at high risk of bias if there was clear evidence of attrition or exclusion from analysis in at least one participant
group that was likely to be related to the true outcome.
Selective reporting
We judged the risk of selective outcome reporting as follows.
1. Studies were judged to be at low risk of bias if all collected data seemed to be reported and all expected outcomes were reported.
2. Studies were judged to be at unclear risk of bias if it was not clear whether other data were collected and not reported.
3. Studies were judged to be at high risk of bias if data for one or more expected outcomes were missing.

Informed decisions.
Other bias
Through assessment, we determined whether any other bias was present in the trials, including inadequate music therapy methods (not
corresponding to the definition of music therapy used for this review) or inadequate music therapy training of therapists delivering the
intervention (without formal training based on the particular country’s regulations for music therapy).
WHAT'S NEW
Date Event Description
16 November 2021 New search has been performed A top-up search for new studies was conducted, resulting in the
inclusion of one new study in the qualitative synthesis and three
new ongoing studies.
9 April 2021 New search has been performed A search for new studies was conducted, resulting in the inclu-
sion of fifteen new studies; the categories of outcome measures
were adapted; based on the added studies' findings, new meta-
analyses were performed, and pre-existing results and conclu-
sions were modified.
16 October 2020 New citation required and conclusions Updated review with one new author.
have changed
HISTORY
Protocol first published: Issue 3, 2003
Review first published: Issue 2, 2006
Date Event Description
18 March 2016 Amended Abstract, main results - adding the word 'therapy' in the second
sentence
2 December 2013 New search has been performed A search for new studies was conducted, resulting in the inclu-
sion of seven new studies; based on the added studies' findings,
the categories of outcome measures were revised, new meta-
analyses were performed, and pre-existing results and conclu-
sions were modified.
31 March 2013 New citation required and conclusions Updated review with two new authors.
have changed
5 November 2009 Amended Minor edit in background.
10 November 2008 Amended Converted to new review format.
21 February 2006 Amended Minor update
29 January 2006 New citation required and conclusions Substantive amendment

have changed
CONTRIBUTIONS OF AUTHORS
CG is the guarantor, conceived the review, designed the protocol and co-ordinated the reviewing. MG co-ordinated this review's update.
CE, LFP, and MG searched for studies. CE, LFP, MG, and GV screened search results. LFP, MG, KM, and GV assessed risk of bias and assessed
Informed decisions.
the certainty in the body of evidence. CE, LFP, MG, CG, KM, and GV extracted data, analysed data, interpreted data, wrote the report, and
approved the full review.
Contribution of previous authors: Tony Wigram, co-author of the 2006 version of this review, contributed to the development of the protocol,
extracted and analysed data, and helped with writing the original report.
DECLARATIONS OF INTEREST
Cochavit Elefant (CE), Monika Geretsegger (MG), Christian Gold (CG), and Karin Mössler (KM) are clinically trained music therapists. CE,
MG and KM report having been involved in publications from one study included in this review (Bieleninik 2017), without it supporting
or influencing their work on this review; the study was funded by the Research Council of Norway (grant 213844, the Clinical Research
and Mental Health Programmes); POLYFON Knowledge Cluster for Music Therapy; The Grieg Academy Department of Music, University of
Bergen, Norway; and a range of further governmental and university funding sources and foundations across participating countries (see
Characteristics of included studies for details). Assessment of eligibility, extraction of data, and assessment of risk of bias and the certainty
of the evidence of this study was performed by two independent review authors who were not involved in the study.
Cochavit Elefant (CE) has declared that she has no other conflicts of interest.
Monika Geretsegger (MG) has declared that she has no other conflicts of interest.
Christian Gold (CG) is an Associate Editor of the Cochrane Developmental, Psychosocial and Learning Problems Review Group, without
it supporting or influencing his work on this review. He is entirely excluded from the editorial decisions and related activities concerning
this review. CG is a member of the Austrian Professional Association of Music Therapists, and until 2020, worked as a health professional.
CG reports a grant from the Kavli Trust for the project 'Music for Autism' (M4A), paid to NORCE Norwegian Research Centre. CG reports
being involved in publications from three studies included in this review (Bieleninik 2017; Kim 2008; and Thompson 2014). He also reports
publishing an invited commentary in the Lancet Child and Adolescent Health in 2019. CG reports being the PI of two studies (Bieleninik 2017,
funded by the Research Council of Norway, National Institute of Health Research; and NCT04936048, funded by the Kavli Trust), as well as
a statistical advisor for one study (Kim 2008, funded by Aalborg University) eligible for inclusion in this review; none of which supported
or influenced his work on this review. Assessment of eligibility, extraction of data, and assessment of risk of bias and the certainty of the
evidence of these three studies were performed by two independent review authors who were not involved in the studies.
Laura Fusar-Poli is a Clinical Research Fellow (RTD-A) and psychiatrist in training at the University of Catania, Italy. She has declared that
she has no conflicts of interest.
Giovanni Vitale is a child psychiatrist in training. He has declared that he has no conflicts of interest.
Karin Mössler has declared that she has no other conflicts of interest.
SOURCES OF SUPPORT
Internal sources
• NORCE Norwegian Research Centre, Bergen, Norway
Monika Geretsegger, Christian Gold, and Karin A Mössler received salary support from NORCE during this update.
• University of Vienna, Austria
Christian Gold received salary support from the University of Vienna during this update.
• University of Haifa, Israel
Cochavit Elefant received salary support from the University of Haifa during this update.
External sources
• Kavli Trust, Norway
Kavli Trust funded the Project 'Music for Autism (M4A)'. This review update is included among the expected publications from M4A. Kavli
Trust had no role in the design, conduct or publication of this review update.
DIFFERENCES BETWEEN PROTOCOL AND REVIEW
Differences between protocol and original review

In compliance with the developments in systematic review methods since publication of the first version of this review (Gold 2006), a
distinction was made between primary and secondary outcome measures, and risk of bias and summary of findings tables were included
in the 2014 update (Geretsegger 2014).

Informed decisions.
Differences between previous versions of the review and this update

Title
We have updated the term 'people with autism spectrum disorder' to 'autistic people' throughout the text to meet the preferences generally
expressed by autistic people.
Types of outcome measures

The knowledge of the condition itself and its nosological classification have changed a lot since the protocol was published in 2003. Back
then, there was still the conception that ASD was mostly a paediatric condition; the concept of ASD was introduced in 2013 with DSM-5 (APA
2013), and now we know that ASD is a life-long condition which results in different aspects being relevant (i.e. mental health problems or
self-esteem). To ensure that all user-important outcomes are addressed (McKenzie 2021), and to update our approach in correspondence
with changes that occurred in the knowledge about ASD in recent years, we adapted the outcome categories used in the previous versions
of the review as described in the Methods section (see Types of outcome measures). We also merged some previously separate outcome
measures to broader outcome categories to keep the review focused and manageable for users.
Electronic searches
We made the following changes to the databases listed in the protocol (Gold 2003) and used in the in the previous versions of the review
(Geretsegger 2014; Gold 2006), due to changes to standard search methods at the Cochrane Developmental, Psychosocial and Learning
Problems Review Group. For this update, we searched the Cochrane Database of Systematic Reviews (CDSR), two trials registers, and two
daily updated segments of MEDLINE (MEDLINE In-Process and Other Non-Indexed Citations, and MEDLINE EPub Ahead of Print). We also
added Epistemonikos as a source of reviews.
Two subscription databases used in previous versions of the review (Dissertation Abstracts and ASSIA ) were no longer available for the
updated review, and were replaced by Proquest Global Dissertations & Theses, and three specialist music databases (Proquest Music
Periodical Database, Proquest Performing Arts Periodicals Database and RILM Abstracts of Music Literature Online).
Three other resources used in previous versions of the review were not used for this update as they were no longer maintained
(musictherapyworld.net website) or no longer updated (Music Therapy Research CD ROM, AMTA 1999; Music Therapy World Info-CD ROM
IV, Aldridge 2002).

In this update, we applied the Cochrane’s Screen4Me workflow to help assess the search results. We also added a new section, 'Summary
of findings and assessment of the certainty of the evidence', in line with changes to standard methods in Cochrane.
Appendix 1
We removed search terms that identified irrelevant studies in the original search.
INDEX TERMS
Medical Subject Headings (MeSH)

*Autistic Disorder [therapy]; Bias; *Music Therapy; Odds Ratio; Quality of Life
MeSH check words

Adolescent; Child; Humans


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Cochrane

Music therapy for autistic people (Review)

Geretsegger M, Fusar-Poli L, Elefant C, Mössler KA, Vitale G, Gold C

Geretsegger M, Fusar-Poli L, Elefant C, Mössler KA, Vitale G, Gold C.

Music therapy for autistic people (Review)

Music therapy for autistic people (Review) i

Music therapy for autistic people

Contact: Christian Gold, chgo@norceresearch.no.

Editorial group: Cochrane Developmental, Psychosocial and Learning Problems Group.

Data collection and analysis

Music therapy for autistic people (Review) 1

PLAIN LANGUAGE SUMMARY

Music therapy for autistic people

The evidence is current to August 2021.

Music therapy for autistic people (Review) 2

Quality of the evidence

Music therapy for autistic people (Review) 3

Population: individuals with a diagnosis of autism spectrum disorder

800 per 1000 976 per 1000

Cochrane Database of Systematic Reviews

Cochrane Database of Systematic Reviews

One study with 36 par-

GRADE Working Group grades of evidence

Cochrane Database of Systematic Reviews

Music therapy for autistic people (Review) 8

Music therapy for autistic people (Review) 9

described for vocal communication in Salomon-Gimmon 2019), METHODS

Music therapy for autistic people (Review) 10

We considered the broad-based measures 'global Secondary outcomes

Primary outcomes Electronic searches

Music therapy for autistic people (Review) 11

Music therapy for autistic people (Review) 12

Binary data method in RevMan and possibly a change score or a positive

Music therapy for autistic people (Review) 13

Music therapy for autistic people (Review) 14

Figure 1. Study flow diagram

Music therapy for autistic people (Review) 15

Music therapy for autistic people (Review) 16

Figure 2. Sreen4Me summary diagram - July 2020 search

Music therapy for autistic people (Review) 17

Figure 3. Screen4Me summary diagram - August 2021 search

Music therapy for autistic people (Review) 18

Interventions offering opportunities for the individual to make choices and

Music therapy for autistic people (Review) 21

Music therapy for autistic people (Review) 23

Nine studies evaluated adaptive behaviours. Validated scales were

Music therapy for autistic people (Review) 24

Music therapy for autistic people (Review) 25

Random sequence generation (selection bias)

0% 25% 50% 75% 100%

Low risk of bias Unclear risk of bias High risk of bias

Music therapy for autistic people (Review) 26

Blinding of participants and personnel (performance bias): All outcomes

Selective reporting (reporting bias)

Music therapy for autistic people (Review) 27

Allocation reports as outcomes were judged as being at unclear risk of bias

Music therapy for autistic people (Review) 29

One to five months follow-up

Music therapy for autistic people (Review) 30

Similarly, effects in the period six to 11 months post-intervention Cognitive ability