Frenotomy For Tongue Tie in Newborn Infants

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Cochrane Database of Systematic Reviews
Frenotomy for tongue-tie in newborn infants (Review)
O'Shea JE, Foster JP, O'Donnell CPF, Breathnach D, Jacobs SE, Todd DA, Davis PG
O'Shea JE, Foster JP, O'Donnell CPF, Breathnach D, Jacobs SE, Todd DA, Davis PG.
Frenotomy for tongue-tie in newborn infants.
Cochrane Database of Systematic Reviews 2017, Issue 3. Art. No.: CD011065.
DOI: 10.1002/14651858.CD011065.pub2.
www.cochranelibrary.com

Copyright © 2017 The Cochrane Collaboration. Published by John Wiley & Sons, Ltd.
Cochrane Trusted evidence.
Informed decisions.
Library Better health. Cochrane Database of Systematic Reviews
TABLE OF CONTENTS
ABSTRACT..................................................................................................................................................................................................... 1
PLAIN LANGUAGE SUMMARY....................................................................................................................................................................... 2
SUMMARY OF FINDINGS.............................................................................................................................................................................. 3
BACKGROUND.............................................................................................................................................................................................. 5
OBJECTIVES.................................................................................................................................................................................................. 5
METHODS..................................................................................................................................................................................................... 6
RESULTS........................................................................................................................................................................................................ 8
Figure 1.................................................................................................................................................................................................. 9
Figure 2.................................................................................................................................................................................................. 10
Figure 3.................................................................................................................................................................................................. 11
Figure 4.................................................................................................................................................................................................. 13
Figure 5.................................................................................................................................................................................................. 14
DISCUSSION.................................................................................................................................................................................................. 14
AUTHORS' CONCLUSIONS........................................................................................................................................................................... 15
ACKNOWLEDGEMENTS................................................................................................................................................................................ 15
REFERENCES................................................................................................................................................................................................ 16
CHARACTERISTICS OF STUDIES.................................................................................................................................................................. 18
DATA AND ANALYSES.................................................................................................................................................................................... 23
Analysis 1.1. Comparison 1: Frenotomy versus no frenotomy or sham procedure, Outcome 1: Infant breastfeeding assessed by 24
a validated scale...................................................................................................................................................................................
Analysis 1.2. Comparison 1: Frenotomy versus no frenotomy or sham procedure, Outcome 2: Infant breastfeeding assessed by 24
a validated scale 2 to 7 days following procedure.............................................................................................................................
Analysis 1.3. Comparison 1: Frenotomy versus no frenotomy or sham procedure, Outcome 3: Maternal nipple pain assessed by 25
a validated pain scale...........................................................................................................................................................................
Analysis 1.4. Comparison 1: Frenotomy versus no frenotomy or sham procedure, Outcome 4: Qualitative assessment of infant 25
feeding by parental survey performed within 48 hours of procedure...............................................................................................
Analysis 1.5. Comparison 1: Frenotomy versus no frenotomy or sham procedure, Outcome 5: Excessive bleeding at the time or 25
within 24 hours of frenotomy (as determined by study investigators).............................................................................................
Analysis 1.6. Comparison 1: Frenotomy versus no frenotomy or sham procedure, Outcome 6: Infection at the site of frenotomy 26
requiring treatment with antibiotics within 7 days of procedure......................................................................................................
Analysis 1.7. Comparison 1: Frenotomy versus no frenotomy or sham procedure, Outcome 7: Damage to the tongue and/or 26
submandibular ducts noted within 7 days of procedure (as determined by study investigators)...................................................
WHAT'S NEW................................................................................................................................................................................................. 26
HISTORY........................................................................................................................................................................................................ 27
CONTRIBUTIONS OF AUTHORS................................................................................................................................................................... 27
DECLARATIONS OF INTEREST..................................................................................................................................................................... 27
SOURCES OF SUPPORT............................................................................................................................................................................... 27
DIFFERENCES BETWEEN PROTOCOL AND REVIEW.................................................................................................................................... 27
INDEX TERMS............................................................................................................................................................................................... 27
Frenotomy for tongue-tie in newborn infants (Review) i

Informed decisions.
[Intervention Review]
Frenotomy for tongue-tie in newborn infants
Joyce E O'Shea1, Jann P Foster2, Colm PF O'Donnell3, Deirdre Breathnach4, Susan E Jacobs5,6,7, David A Todd8, Peter G Davis9,10,11
1Royal Hospital for Children, Glasgow, UK. 2School of Nursing and Midwifery, Western Sydney University, Penrith DC, Australia.
3Department of Neonatology, National Maternity Hospital, Dublin 2, Ireland. 4Campaspe Family Practice, Kyneton, Australia. 5Neonatal
Services, The Royal Women's Hospital, Parkville, Melbourne, Australia. 6The University of Melbourne, Melbourne, Australia. 7Murdoch
Childrens Research Institute, Parkville, Australia. 8Neonatal Unit, The Canberra Hospital, Canberra, Australia. 9Newborn Research
Centre and Neonatal Services, The Royal Women’s Hospital, Melbourne, Australia. 10Murdoch Children's Research Institute, Melbourne,
Australia. 11Department of Obstetrics and Gynecology, University of Melbourne, Melbourne, Australia
Contact: Joyce E O'Shea, joyce.o'shea@nhs.net, jem.oshea@gmail.com.
Editorial group: Cochrane Neonatal Group.

Publication status and date: Edited (no change to conclusions), published in Issue 6, 2021.
Citation: O'Shea JE, Foster JP, O'Donnell CPF, Breathnach D, Jacobs SE, Todd DA, Davis PG. Frenotomy for tongue-tie in newborn infants.
Cochrane Database of Systematic Reviews 2017, Issue 3. Art. No.: CD011065. DOI: 10.1002/14651858.CD011065.pub2.
ABSTRACT
Background
Tongue-tie, or ankyloglossia, is a condition whereby the lingual frenulum attaches near the tip of the tongue and may be short, tight and
thick. Tongue-tie is present in 4% to 11% of newborns. Tongue-tie has been cited as a cause of poor breastfeeding and maternal nipple
pain. Frenotomy, which is commonly performed, may correct the restriction to tongue movement and allow more effective breastfeeding
with less maternal nipple pain.
Objectives
To determine whether frenotomy is safe and effective in improving ability to feed orally among infants younger than three months of age
with tongue-tie (and problems feeding).
Also, to perform subgroup analysis to determine the following.
• Severity of tongue-tie before frenotomy as measured by a validated tool (e.g. Hazelbaker Assessment Tool for Lingual Frenulum Function
(ATLFF) scores < 11; scores ≥ 11) (Hazelbaker 1993).
• Gestational age at birth (< 37 weeks' gestation; 37 weeks' gestation and above).
• Method of feeding (breast or bottle).
• Age at frenotomy (≤ 10 days of age; > 10 days to three months of age).
• Severity of feeding difficulty (infants with feeding difficulty affecting weight gain (as assessed by infant's not regaining birth weight by day
14 or falling off centiles); infants with symptomatic feeding difficulty but thriving (greater than birth weight by day 14 and tracking centiles).
Search methods
We searched the Cochrane Central Register of Controlled Trials (CENTRAL), MEDLINE, Embase and CINAHL up to January 2017, as well
as previous reviews including cross-references, expert informants and journal handsearching. We searched clinical trials databases for
ongoing and recently completed trials. We applied no language restrictions.
Frenotomy for tongue-tie in newborn infants (Review) 1

Informed decisions.
Selection criteria
Randomised, quasi-randomised controlled trials or cluster-randomised trials that compared frenotomy versus no frenotomy or frenotomy
versus sham procedure in newborn infants.
Data collection and analysis

Review authors extracted from the reports of clinical trials data regarding clinical outcomes including infant feeding, maternal nipple pain,
duration of breastfeeding, cessation of breastfeeding, infant pain, excessive bleeding, infection at the site of frenotomy, ulceration at the
site of frenotomy, damage to the tongue and/or submandibular ducts and recurrence of tongue-tie. We used the GRADE approach to assess
the quality of evidence.
Main results
Five randomised trials met our inclusion criteria (n = 302). Three studies objectively measured infant breastfeeding using standardised
assessment tools. Pooled analysis of two studies (n = 155) showed no change on a 10-point feeding scale following frenotomy (mean
difference (MD) -0.1, 95% confidence interval (CI) -0.6 to 0.5 units on a 10-point feeding scale). A third study (n = 58) showed objective
improvement on a 12-point feeding scale (MD 3.5, 95% CI 3.1 to 4.0 units of a 12-point feeding scale). Four studies objectively assessed
maternal pain. Pooled analysis of three studies (n = 212) based on a 10-point pain scale showed a reduction in maternal pain scores
following frenotomy (MD -0.7, 95% CI -1.4 to -0.1 units on a 10-point pain scale). A fourth study (n = 58) also showed a reduction in pain
scores on a 50-point pain scale (MD -8.6, 95% CI -9.4 to -7.8 units on a 50-point pain scale). All studies reported no adverse effects following
frenotomy. These studies had serious methodological shortcomings. They included small sample sizes, and only two studies blinded both
mothers and assessors; one did not attempt blinding for mothers nor for assessors. All studies offered frenotomy to controls, and most
controls underwent the procedure, suggesting lack of equipoise. No study was able to report whether frenotomy led to long-term successful
breastfeeding.
Authors' conclusions
Frenotomy reduced breastfeeding mothers’ nipple pain in the short term. Investigators did not find a consistent positive effect on infant
breastfeeding. Researchers reported no serious complications, but the total number of infants studied was small. The small number of trials
along with methodological shortcomings limits the certainty of these findings. Further randomised controlled trials of high methodological
quality are necessary to determine the effects of frenotomy.
PLAIN LANGUAGE SUMMARY
Surgical release of tongue-tie for the treatment of tongue-tie in young babies
Review question: Tongue-tie is a potentially treatable cause of breastfeeding problems - if a baby is tongue-tied and is having feeding
difficulties, does releasing the tongue-tie help?
Background: Tongue-tie is a condition whereby the membrane between the tongue and the floor of the mouth is too tight or too short.
This may cause feeding problems for the baby and/or nipple pain for a breastfeeding mother.
Study characteristics: Five randomised controlled trials enrolling 302 infants met the inclusion criteria.
Key results: In an infant with tongue-tie and feeding difficulties, surgical release of the tongue-tie does not consistently improve infant
feeding but is likely to improve maternal nipple pain. Further research is needed to clarify and confirm this effect.
Quality of evidence: The quality of the evidence is very low to moderate because overall only a small number of studies have looked at
this condition, the total number of babies included in these studies was low and some studies could have been better designed.

SUMMARY OF FINDINGS
Summary of findings 1. Frenotomy compared with no frenotomy or sham procedure in infants with tongue-tie and feeding difficulties
Library
Cochrane
Undefined
Patient or population: tongue-tie in newborn infants

Setting: maternity hospitals
Intervention: frenotomy
Better health.
Informed decisions.
Trusted evidence.
Comparison: no frenotomy or sham procedure
Outcomes Illustrative comparative risks (mean and SD) Relative effect Number of par- Quality of the Comments
(95% CI) ticipants evidence
Risk with no frenotomy or Risk with frenotomy (studies) (GRADE)
sham procedure
Infant breastfeeding as- Mean IBFAT scores following Mean IBFAT scores following Mean difference 58 ⊕⊕⊝⊝ IBFAT score is
sessed by validated scale procedure in the control group procedure in the frenotomy is 3.50 (3.06 to (1 RCT) LOWa,b based on a 12-
- IBFAT scores following was 8.1 (SD 0.9) group was 11.6 (SD 0.8) 3.94) point scale
procedure
Infant breastfeeding as- Mean LATCH scores following Mean LATCH scores following Mean difference 155 ⊕⊕⊝⊝ LATCH score is
sessed by validated scale procedure in the control group procedure in the frenotomy is -0.07 (-0.63 to (2 RCTs) LOWa,b based on a 10-
- LATCH scores following was 6.8 to 8.5 (SD < 1.9) group was 6.8 to 8.4 (SD < 2) 0.48) point scale
procedure
Maternal nipple pain as- Mean visual analogue pain Mean IBFAT scores following Mean difference 183 ⊕⊕⊝⊝ Visual analogue
sessed by a validated pain scale scores following proce- procedure in the frenotomy is -0.74 (-1.35 to (3 RCTs) LOWa,b pain scale score
scale - visual analogue dure in the control group was group was 1.6 to 5.3 (SD < 2.4) -0.13) is based on a
pain scale 2.9 to 5.5 (SD < 2.6) 10-point scale
Maternal nipple pain as- Mean SF-MPQ scores following Mean IBFAT scores following Mean difference 58 ⊕⊕⊝⊝ SF-MPQ score is
sessed by a validated pain procedure in the control group procedure in the frenotomy is -8.60 (-9.37 to (1 RCT) LOWa,b based on a 50-
Cochrane Database of Systematic Reviews

scale - SF-MPQ pain scale was 13.5 (SD 1.5) group was 4.9 (SD 1.5) -7.83) point scale
*The risk in the intervention group (and its 95% confidence interval) is based on assumed risk in the comparison group and the relative effect of the intervention (and its
95% CI)
CI: confidence interval; OR: odds ratio; RR: risk ratio
GRADE Working Group grades of evidence

High quality: We are very confident that the true effect lies close to the estimate of effect
Moderate quality: We are moderately confident in the effect estimate: The true effect is likely to be close to the estimate of effect but may be substantially different
Low quality: Our confidence in the effect estimate is limited: The true effect may be substantially different from the estimate of effect
Very low quality: We have very little confidence in the effect estimate: The true effect is likely to be substantially different from the estimate of effect
3
Informed decisions.
aImprecision (small total participant population)
bRisk of bias (incomplete blinding)

Informed decisions.
BACKGROUND while an assistant supports the head and neck. The clinician/
surgeon elevates the tongue and exposes the frenulum, which
Description of the condition is then incised with sharp, straight, blunt-ended scissors (Berry
2012; Hogan 2005). Some operators describe crushing the frenulum
The lingual frenulum is a fold of mucous membrane that extends
before incision. Direct pressure is then applied to the frenulum with
from the floor of the mouth to the midline of the underside
a piece of gauze. Bleeding is reportedly scant and is controlled by
of the tongue. It helps to stabilise the base of the tongue and
the pressure (Lalakea 2002). The incision usually is not sutured,
does not normally interfere with tongue tip movement (Marchesan
and the infant most often recovers quickly from the procedure and
2005). Tongue-tie (ankyloglossia) is a condition in which the lingual
is able to feed directly afterwards. In infants, frenotomy is usually
frenulum has an anterior attachment near the tip of the tongue and
performed without analgesia or anaesthetic (Lalakea 2002). The
may be unusually short, tight and thick (Jackson 2012). This causes
use of a laser to perform the frenotomy is becoming more frequent
virtual adhesion of the tongue tip to the floor of the mouth and
(Kotlow 2011).
can result in restricted tongue tip movement (Marchesan 2005). The
exact cause of 'tongue-tie' (ankyloglossia) is not known. Genetics Frenuloplasty, an operation that lengthens the frenulum, is the
may play a role, as the condition tends to run in some families preferred procedure for patients over one year of age (Lalakea
(Coryllos 2004). Prevalence is about 4% to 11% among newborns 2002). This intervention will not be included in this review.
(Hogan 2005; Messner 2000a; Messner 2000b; Ricke 2005).
How the intervention might work
Tongue-tie has been cited as a cause of poor breastfeeding because
the infant is unable to attach or stay latched on, and because Surgical release of the tongue-tie through frenotomy may correct
maternal nipple pain may result (Coryllos 2004 Hogan 2005). In the restriction to infant tongue movement during feeding to
older children and adults, tongue-tie has been implicated as a allow more effective breastfeeding and less maternal nipple pain
cause of speech delay, abnormal dentition, poor oral hygiene resulting from decreased friction between the infant's lower gum/
and inability to play wind instruments (Krol 2007). As an infant tongue and the nipple (Kumar 2012).
breastfeeds, the tongue moves with peristalsis over maternal
lactiferous sinuses and extracts milk. When the infant's tongue Why it is important to do this review
movement is restricted, as is the case with severe tongue- Diagnosis and management of tongue-tie remain controversial.
tie, reduced movement may affect milk extraction, and friction
may be present between the tongue or gums and the nipple, It is uncertain whether ankyloglossia is a congenital oral anomaly
causing damage to the nipple and maternal pain (Coryllos 2004 requiring treatment or a normal variant. One survey (Messner
Hogan 2005). References to tongue-tie causing speech problems 2000b) found that most lactation consultants believe tongue-tie to
date back to Aristotle in the third century BC (Obladen 2010). be a frequent cause of infant breastfeeding difficulties that could be
The association between breastfeeding difficulty and tongue-tie solved by frenotomy. In marked contrast, 90% of paediatricians and
has been recognised for at least 500 years (Obladen 2010). In 70% of otolaryngologists believe that tongue-tie never, or rarely,
recent years, with recognition and encouragement of exclusive causes a feeding problem (Messner 2000a). However, medical
breastfeeding as the optimal primary mode of infant feeding, organisations such as the American Academy of Pediatrics (Coryllos
the justification for frenotomy has shifted from improving speech 2004) and the National Institute for Health and Care Excellence
problems to improving breastfeeding (Obladen 2010), re-igniting (NICE 2005) now acknowledge that tongue-tie, or ankyloglossia, is a
the historical debate as to the role of tongue-tie in breastfeeding significant clinical entity that should be treated as early as possible
difficulties (Kumar 2012). to minimise breastfeeding problems. Given that breastfeeding
benefits both infants and mothers, it is important for the clinician
The diagnosis of tongue-tie depends on an assessment of to address any condition that may impair breastfeeding (Edmunds
the structure and function of the lingual frenulum. Diagnostic 2011).
classification systems vary from simple visual inspection and/
or palpation of the frenulum to a more complex multi-scale OBJECTIVES
classification system such as the Hazelbaker Assessment Tool for
Lingual Frenulum Function (ATLFF) (Hazelbaker 1993). The ATLFF is To determine whether frenotomy is safe and effective in improving
a highly reliable screening tool (Amir 2006) that was designed to be ability to feed orally among infants younger than three months of
used in assessment of infants younger than three months of age. It age with tongue-tie (and problems feeding).
assesses the function and appearance of the frenulum. A score of 14
indicates normal function, between 11 and 14 is acceptable and less Subgroup analysis
than 11 indicates significant tongue-tie that requires frenotomy. An
• Severity of tongue-tie before frenotomy as measured by a
appearance scale includes values to 10, and lower scores indicate
validated tool (e.g. ATLFF (scores < 11; scores ≥ 11)) (Hazelbaker
tongue-tie. A score lower than eight is suggestive of tongue-tie, but
1993).
surgery is not recommended unless a functional problem is noted.
• Gestational age at birth (< 37 weeks' gestation; 37 weeks'
Description of the intervention gestation and above).
• Method of feeding (breast or bottle).
Frenotomy and frenuloplasty are the two main surgical procedures
• Age at frenotomy (≤ 10 days of age; > 10 days to three months
used in the treatment of infants with tongue-tie (Lalakea 2002).
of age).
Frenotomy, or clipping of the frenulum, is the procedure of choice
in infants because it is relatively quick and easy to perform. The • Severity of feeding difficulty (infants with feeding difficulty
infant is swaddled and is placed supine on the examining table, affecting weight gain (as assessed by infant's not regaining birth

Informed decisions.
weight by day 14 or falling off centiles); infants with symptomatic • Excessive bleeding at the time or within 24 hours of frenotomy
feeding difficulty but thriving (greater than birth weight by day (as determined by study investigators)
14 and tracking centiles). • Infection at the site of frenotomy requiring treatment with
antibiotics within seven days of the procedure
METHODS
• Damage to the tongue or submandibular ducts noted within
Criteria for considering studies for this review seven days of the procedure (as determined by study
investigators)
Types of studies
Search methods for identification of studies
Randomised or quasi-randomised controlled trials or cluster-
randomised trials. Electronic searches
Types of participants Two review authors (JO'S, JF) independently performed electronic
database searches, including electronic searches of the Cochrane
Infants three months of age or younger with a diagnosis of tongue- Central Register of Controlled Trials (CENTRAL; 2016, Issue 1),
tie who are orally feeding. To be included, another problem must MEDLINE (1966 to January 2016), Embase (1980 to January
be present that could be related to the tongue-tie, specifically, 2016) and the Cumulative Index to Nursing and Allied Health
infant feeding problems or maternal nipple pain in a breastfeeding Literature (CINAHL; 1982 to January 2016), and searched previous
mother. We planned to exclude patients with other coexisting oral reviews including cross-references, expert informants and journal
pathology that might affect oral feeding, for example, cleft palate. handsearching. We searched MEDLINE, Embase and CINAHL
for relevant articles using the following search terms: Infant
Types of interventions AND Tongue Tie, Infant OR Newborn OR neonate (explode)
• Frenotomy versus no frenotomy. Lactation consultant [MeSH heading] AND Tongue Tie (explode) [MeSH heading] OR
interventions were accepted if provided to both groups. ankyloglossia. and Frenotomy [MeSH heading] OR Frenulotomy OR
• Frenotomy versus sham procedure. Lactation consultant Frenuloplasy [MeSH heading]. We applied no language restrictions.
interventions were accepted if provided to both groups. We also searched clinical trial registries for current and recently
completed trials (Australia and New Zealand Clinical Trials Register
Types of outcome measures (ANZCTR); clinicaltrials.gov; controlled-trials.com; who.int/ictrp;
and Oxford Database of Perinatal Trials).
Primary outcomes
• Infant feeding assessed within 48 hours, within two to seven Searching other resources
days and after seven days following the procedure with the use The search strategy included communication with expert
of a validated scale such as the LATCH score (Jensen 1994) or the informants and searches of bibliographies of reviews and trials
Infant Breastfeeding Assessment Tool (IBFAT) (Mathews 1988). for references to other trials, as well as searches of previous
The LATCH is a scoring system that assesses latch, swallowing, reviews including cross-references, abstracts and conferences and
maternal nipple, maternal comfort and assistance the mother symposia proceedings of the Perinatal Society of Australia and
needs to position the infant. Each area gets a score between New Zealand and the Pediatric Academic Societies (American
0 and 2 with a total possible score of 10. The IBFAT assesses Pediatric Society, Society for Pediatric Research and European
readiness to feed, rooting, fixing (latching on) and sucking. Society for Pediatric Research) from 1990 to 2015. We planned to
Each item is scored between 0 and 3, and a score of 10 to contact the corresponding investigator of any unpublished trials
12 represents successful breastfeeding. Bottle-feeding will be to request information. We considered unpublished studies and
assessed within 48 hours, within two to seven days and after studies reported only as abstracts as eligible for review if final trial
seven days following the procedure, subjectively, by reports of data were available. We intended to contact the corresponding
more efficient sucking and less drooling authors of identified randomised controlled trials (RCTs) to ask for
additional information about their studies if we required further
Secondary outcomes data.
• Maternal nipple pain assessed within 48 hours, within two to
seven days and after seven days following frenotomy by a Data collection and analysis
validated pain scale (e.g. Short-Form McGill Pain Questionnaire We used the standard methods of Cochrane as documented in the
(SF-MPQ)) (Melzack 1975) Cochrane Handbook for Systematic Reviews of Interventions (Higgins
• Qualitative assessment of infant feeding by parental survey 2011) and the methods of the Cochrane Neonatal Review Group
performed within 48 hours of the procedure (CNRG).
• Duration of breastfeeding (days)
Selection of studies
• Cessation of breastfeeding as assessed by maternal report
within four weeks of the procedure Review authors independently assessed for inclusion all
• Infant pain as assessed by a validated pain scale (e.g. Modified potential studies identified by the search strategy. We resolved
Behavioral Pain Scale (MBPS) (Taddio 1995), Neonatal Infant disagreements through discussion.
Pain Scale (NIPS) (Lawrence 1993), CRIES pain scale (cries,
requires oxygen, shows increased vital signs and expression and Specifically, we:
is sleepless) (Krechel 1995)) before, during and up to one hour • merged search results by using reference management software
post frenotomy and removed duplicate records of the same report;
Informed decisions.
• examined titles and abstracts to remove irrelevant reports; Measures of treatment effect
• retrieved full text of potentially relevant reports; We analysed the results of included studies using the statistical
• linked together multiple reports of the same study; package Review Manager software 5.3 (RevMan 2014).
• examined full-text reports to assess for compliance of studies
meeting eligibility criteria; We used the standard method of the CNRG, applying a fixed-
effect model for meta-analysis. In assessing treatment effects for
• corresponded with investigators, when appropriate, to clarify
dichotomous data, we reported the risk ratio (RR) and the risk
study eligibility;
difference (RD), along with 95% confidence intervals, for categorical
• at all stages noted reasons for inclusion and exclusion of articles; outcomes. If the RD was statistically significant, we calculated
and the number needed to treat for an additional beneficial outcome
• made final decisions on study inclusion and proceeded to data (NNTB) and the number needed to treat for an additional harmful
collection. outcome (NNTH) (1/RD). For outcomes measured on a continuous
scale, we used the weighted mean difference, along with 95%
Data extraction and management confidence intervals.
Review authors independently extracted data from full-text
articles using a specifically designed spreadsheet to manage the Included studies reported outcomes of change in infant feeding
information. We resolved discrepancies through discussion; if ability and in maternal pain using different validated scales. For
required, we intended to consult a review arbiter. We entered data infant feeding, researchers presented LATCH scores (a 10-point
into Review Manager software 5.3 (RevMan 2014) and checked them scale) and IBFAT scores (a 12-point scale). Included studies reported
for accuracy. maternal pain assessed by the 10-point visual analogue pain scale
and the 50-point SF-MPQ. As these different scales rely on very
Assessment of risk of bias in included studies different units of reporting and show subtle differences, we did not
combine scores for analysis, and we presented results in subgroups
We used the standardised review methods of the CNRG according to the different scales used.
(http://neonatal.cochrane.org/en/index.html) to assess the
methodological quality of included studies. Review authors Unit of analysis issues
independently assessed study quality and risk of bias using the
following criteria, as documented in the Cochrane Handbook for We combined randomised trials in a single meta-analysis using the
Systematic Reviews of Interventions (Higgins 2011). generic inverse variance method.
• Random sequence generation: Was the allocation sequence Dealing with missing data
adequately generated? We planned to contact the authors of all published studies if
• Allocation concealment: Was allocation adequately concealed? we required clarification or additional information. We planned
• Blinding of participants and personnel for each main outcome or to describe the number of participants with missing data in the
class of outcomes: Was knowledge of the allocated intervention Results section and in the Characteristics of included studies table.
adequately prevented during the study? We presented results only for available participants. We intended to
• Blinding of outcome assessors: Were the outcome assessors discuss the implications of missing data in the Discussion section
blinded? of the review.
• Incomplete outcome data for each main outcome or class of Assessment of heterogeneity
outcomes: Were incomplete data adequately addressed?
We used RevMan 5.3 (RevMan 2014) to assess the heterogeneity of
• Selective outcome reporting: Are reports of the study free of the
suggestion of selective outcome reporting? We tried to locate treatment effects between trials. We used the two formal statistical
protocols to assess outcome reporting bias. approaches described below.
• Other sources of bias: Was the study apparently free of other • Chi2 test for homogeneity: We calculated whether statistical
problems that could put it at high risk of bias? We gave particular heterogeneity is present by using the Chi2 test for homogeneity
attention to completeness of follow-up of all randomly assigned (P < 0.1). Because this test has low power when the number
infants and to the length of follow-up studies to identify whether of studies included in the meta-analysis is small, we set the
any benefits claimed were robust. probability at the 10% level of significance (Higgins 2011).
We intended to request additional information and clarification of • I2 statistic, to ensure that pooling of data was valid: We
published data from the authors of individual trials if required. We quantified the impact of statistical heterogeneity by using
assessed each trial for risk of bias based on the criteria listed above I2 statistics available in RevMan 2014, which describes the
and marked each as: percentage of total variation across studies due to heterogeneity
rather than to sampling error. We graded the degree of
• 'low' risk of bias; heterogeneity as follows: 0% to 30%: potentially trivial (not
• 'unclear' risk of bias; or important) heterogeneity; 31% to 50%: low heterogeneity;
• 'high' risk of bias. 51% to 75%: moderate heterogeneity; and 76% to 100%: high
heterogeneity. Had we found evidence of apparent or statistical
We judged each criterion as being at 'low risk' of bias, 'high risk' of heterogeneity, we planned to assess the source of heterogeneity
bias or 'unclear' risk of bias (for lack of information or uncertainty by performing sensitivity and post hoc subgroup analyses
over the potential for bias). to look for sources of bias or methodological differences

Informed decisions.
between heterogeneous trials (e.g. differences in study quality, The GRADE approach results in an assessment of the quality of a
participants, intervention regimens, outcome assessments). body of evidence according to one of four grades.
Assessment of reporting biases • High: We are very confident that the true effect lies close to that
the estimate of effect.
We tried to obtain the study protocols of all included studies to
compare outcomes reported in the protocol versus those reported • Moderate: We are moderately confident in the effect estimate:
in the findings for each of the included studies. We intended to The true effect is likely to be close to the estimate of effect but
investigate reporting and publication bias by examining the degree may be substantially different.
of asymmetry of a funnel plot if we identified 10 or more trials. • Low: Our confidence in the effect estimate is limited: The true
When we suspected reporting bias (see selective reporting bias effect may be substantially different from the estimate of effect.
above), we intended to contact study authors to ask them to • Very low: We have very little confidence in the effect estimate:
provide missing outcome data. When this was not possible and The true effect is likely to be substantially different from the
we suspected that missing data might introduce serious bias, we estimate of effect
intended to explore the impact of including such studies in the
overall assessment of results by performing a sensitivity analysis. Subgroup analysis and investigation of heterogeneity
We planned to carry out the following subgroup analyses.
Data synthesis
We performed statistical analyses according to the • Severity of tongue-tie as measured by a validated tool (e.g.
recommendations of the CNRG (http://neonatal.cochrane.org/en/ ATLFF (scores < 11; scores ≥ 11)) (Hazelbaker 1993).
index.html). We analysed all randomly assigned infants on an • Gestational age at birth (< 37 weeks' gestation; 37 weeks'
intention-to-treat (ITT) basis. We analysed treatment effects in gestation and above).
individual trials and used a fixed-effect model for meta-analysis • Method of feeding (breast or bottle).
in the first instance to combine the data. When substantial • Age at frenotomy (≤ 10 days of age; > 10 days to three months
heterogeneity existed, we examined the potential cause of of age).
heterogeneity by performing subgroup and sensitivity analyses.
• Severity of feeding difficulty (infants with feeding difficulty
When we judged the meta-analysis to be inappropriate, we
affecting weight gain (assessed by infant's not regaining birth
analysed and interpreted individual trials separately. For estimates
weight by day 14 or dropping off centiles by three months);
of typical risk ratio and risk difference, we used the Mantel-Haenszel
infants with feeding difficulty but normal weight gain).
method. For measured quantities, we used the inverse variance
method. Sensitivity analysis
Quality of evidence We intended to explore methodological heterogeneity through the
use of sensitivity analysis. We classified studies as having low risk
We used the Grading of Recommendations Assessment,
of bias if they had adequate sequence generation and allocation
Development and Evaluation (GRADE) approach, as outlined in the
concealment and reported losses less than 10% on ITT analysis.
GRADE Handbook (Schünemann 2013), to assess the quality of
evidence for infant breastfeeding when assessed by a validated
RESULTS
scale and maternal nipple pain.
Description of studies
Two review authors independently assessed the quality of the
evidence for each of the outcomes above. We considered evidence See Characteristics of included studies and Characteristics of
from RCTs as high quality but downgraded the evidence one level excluded studies.
for serious (or two levels for very serious) limitations on the basis
of the following: design (risk of bias), consistency across studies, Results of the search
directness of the evidence, precision of estimates and presence of We present a summary of our search in Figure 1. We encountered no
publication bias. We used the GRADEpro Guideline Development disagreement between assessors (JO'S, JF, DT) regarding inclusion
Tool to create Summary of findings 1 to report the quality of the or exclusion of studies, quality assessment or data extraction. We
evidence. pooled available data and analysed them as listed below.

Informed decisions.
Figure 1. Study flow diagram.

Informed decisions.
Included studies • Emond 2013 is a single-centre study performed in the United

Kingdom.
We included five small trials in this review (N = 302).
• Objective: to investigate if immediate frenotomy was
• Berry 2012 is a single-centre study performed in the United
superior to breastfeeding support.
Kingdom.
• Population: breastfeeding infants younger than two weeks
• Objective: to investigate if an immediate and sustained of age with mild to moderate tongue-tie and breastfeeding
improvement in breastfeeding occurs following frenotomy. difficulties.
• Population: infants younger than four months of age with • Intervention: infants randomised to frenotomy or standard
tongue-tie and breastfeeding difficulties. care.
• Intervention: infants randomised to frenotomy or no • Outcomes: change in breastfeeding scores and maternal pain
frenotomy. scores.
• Outcomes: subjective and objective feeding assessment and
• Hogan 2005 is a single-centre study performed in the United
maternal pain scores.
Kingdom.
• Buryk 2011 is a single-centre study performed in America.
• Objective: to investigate if frenotomy was superior to
• Objective: to investigate if frenotomy decreased maternal standard care in infants with tongue-tie and feeding
pain, improved breastfeeding scores and led to breastfeeding difficulties.
over a longer period. • Population: bottle-feeding and breastfeeding infants with
• Population: infants younger than 30 days of age with tongue-tie and feeding difficulty.
breastfeeding difficulties whose mothers experienced nipple • Intervention: infants randomised to intensive lactation
pain. support or frenotomy.
• Intervention: infants randomised to frenotomy or sham • Outcomes: subjective improvement in feeding.
procedure.
Excluded studies
• Outcomes: change in maternal pain scores and infant feeding
scores. See Characteristics of excluded studies. We excluded two studies
(Ngerncham 2013; Yousefi 2015) - the first because it was a cross-
• Dollberg 2006 is a single-centre study performed in Israel. sectional study with no control group, the second because it
compared frenotomy versus frenuloplasty in children up to 12 years
• Objective: to investigate if frenotomy in infants with of age.
ankyloglossia improves nipple pain and/or latching on
during attempts to breastfeed. Risk of bias in included studies
• Population: breastfeeding infants younger than 21 days of
age whose mothers were experiencing nipple pain. We included in the analysis randomised controlled trials that
compared frenotomy versus no frenotomy or frenotomy versus
• Intervention: infants randomised to frenotomy followed by a
sham procedure in newborn infants. Overall, the five included
sham procedure or a sham procedure followed by frenotomy.
trials had small study populations and a high incidence
• Outcomes: change in maternal pain scores and infant latch of methodological shortcomings. We discuss below specific
scores. methodological issues regarding these studies. See the risk of bias
graph in Figure 2 and summary in Figure 3.
Figure 2. 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 3. 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
Berry 2012 + + + + + - -
Buryk 2011 + ? + + + + -
Dollberg 2006 + + + + + - +
Emond 2013 + + + + + + -
Hogan 2005 + + - - + + -

Informed decisions.
Allocation of randomised participants) resulting from loss to follow-up. Buryk

2011 and Hogan 2005 had no postrandomisation exclusions.
Random sequence generation
All five studies reported the method of randomisation. Berry Selective reporting
2012, Buryk 2011, Dollberg 2006 and Hogan 2005 used computer- Two studies selectively reported outcomes. Berry 2012 reported
generated randomisation, and Emond 2013 used independent that infant latch onto the breast objectively improved but did
telephone-based randomisation. not provide data; Berry 2012 also reported on breastfeeding
success at three months but did not report results by study group.
Allocation concealment
Buryk 2011 reported no differences between groups in longer-term
Four studies described the methods used to conceal allocation. breastfeeding rates but did not present results of the two groups
Berry 2012 and Hogan 2005 used sealed envelopes created by a separately. Dollberg 2006 reported changes in pain scores before
person not involved in the trial, and Dollberg 2006 used sealed and after the intervention in the intervention arm only and on
envelopes and specified that the envelopes were opaque but did request produced scores for the control arm.
not state who created them. Emond 2013 used a randomisation
code that was kept by an independent body. Buryk 2011 did not Other potential sources of bias
state how investigators concealed allocation. All five studies offered and provided frenotomy to control infants,
suggesting lack of equipoise. Two studies - Berry 2012 and Dollberg
Blinding
2006 - performed frenotomy on all participants as part of the
Blinding of participants and research or clinical personnel study protocol. Buryk 2011, Emond 2013 and Hogan 2005 offered
frenotomy to controls and had uptakes of 77% to 97%.
All five studies reported this. Hogan 2005 did not blind participants
nor personnel. Buryk 2011 performed single-blinding (mothers Effects of interventions
only, not treating personnel), Emond 2013 also used single-blinding
(researchers blinded, mothers not blinded) and Berry 2012 and See: Summary of findings 1 Frenotomy compared with no
Dollberg 2006 blinded both participants and personnel. frenotomy or sham procedure in infants with tongue-tie and
feeding difficulties
Blinding of outcome assessment
Frenotomy versus no frenotomy or sham procedure
All five studies reported this. Berry 2012, Buryk 2011 and Dollberg
2006 blinded the assessor. Emond 2013 blinded the assessor of the Five studies compared frenotomy versus no frenotomy (Berry 2012;
primary outcome and assessors of some secondary outcomes but Emond 2013; Hogan 2005) or frenotomy versus sham procedure
did not blind the mother and did not blind the assessors of other (Buryk 2011; Dollberg 2006). We summarise the main findings in
secondary outcomes. Hogan 2005 was an unblinded study. Summary of findings 1.
Incomplete outcome data Primary outcomes
Exclusions after randomisation Infant feeding assessed with a validated tool (Analysis 1.1)
Berry 2012 had three postrandomisation exclusions (5% Four trials provided outcome data for this comparison (Berry 2012;
of randomised participants), and Dollberg 2006 had one Buryk 2011; Dollberg 2006; Emond 2013). Three studies reported
postrandomisation exclusion (4% of randomised participants). infant breastfeeding assessed with a validated scale (Buryk 2011;
Both studies reported that exclusions were due to failure of Dollberg 2006; Emond 2013) based on two different measurement
blinding. Emond 2013 had one postrandomisation exclusion (< 1% scales (Analysis 1.1; Figure 4).

Informed decisions.
Figure 4. Forest plot of comparison: 1 Frenotomy versus no frenotomy or sham procedure, outcome: 1.1 Infant
breastfeeding assessed by a validated scale.
Frenotomy Control Mean Difference Mean Difference

Study or Subgroup Mean SD Total Mean SD Total Weight IV, Fixed, 95% CI IV, Fixed, 95% CI
1.1.1 IBFAT scores following procedure

Buryk 2011 11.6 0.8 30 8.1 0.9 28 100.0% 3.50 [3.06 , 3.94]
Subtotal (95% CI) 30 28 100.0% 3.50 [3.06 , 3.94]
Heterogeneity: Not applicable
Test for overall effect: Z = 15.61 (P < 0.00001)
1.1.2 LATCH scores following procedure

Emond 2013 8.4 1.8 53 8.5 1.6 52 73.4% -0.10 [-0.75 , 0.55]
Dollberg 2006 6.8 2 25 6.8 1.9 25 26.6% 0.00 [-1.08 , 1.08]
Subtotal (95% CI) 78 77 100.0% -0.07 [-0.63 , 0.48]
Heterogeneity: Chi² = 0.02, df = 1 (P = 0.88); I² = 0%
Test for overall effect: Z = 0.26 (P = 0.80)
Test for subgroup differences: Chi² = 97.28, df = 1 (P < 0.00001), I² = 99.0% -4 -2 0 2 4

Favours control Favours frenotomy
• IBFAT score (Buryk 2011): MD 3.50 on a 12-point scale, 95% CI -0.10 on a 10-point scale, 95% CI -0.75 to 0.55; 105 participants
3.06 to 3.94; 58 participants (Analysis 1.1.1). (Analysis 1.2).
• LATCH score (Dollberg 2006; Emond 2013): MD -0.07 on a 10-
point scale, 95% CI -0.63 to 0.48; 155 participants (heterogeneity: Subgroup analysis by gestational age or by severity of feeding
Chi2 = 0.02, df = 1, P = 0.88; I2 = 0%) (Analysis 1.1.2). difficulty or method of feeding was not possible owing to lack of
data.
A fourth trial (Berry 2012) also reported that infants in the
intervention group had higher LATCH scores after the intervention Secondary outcomes
but provided no data. Effect on maternal nipple pain (Analysis 1.3)
We did not combine LATCH and IBFAT scores for meta-analysis, as Four trials reported this outcome (Berry 2012; Buryk 2011; Dollberg
studies had a high degree of heterogeneity and scales used different 2006; Emond 2013) using two different scales.
units of measure.
• Berry 2012, Dollberg 2006 and Emond 2013 assessed this
Subgroup analyses outcome after the first breastfeed using a 10-point visual
analogue pain scale. Meta-analysis showed a significant
Severity of tongue-tie
reduction in maternal nipple pain in the frenotomy group
One trial included only infants with severe tongue-tie (Buryk 2011) compared with the control group: MD -0.74, 95% CI -1.35 to -0.13
and reported improvement in infant breastfeeding (increase in (heterogeneity: Chi2 = 1.85, df = 2, P = 0.40; I2 = 0%) (Analysis
IBFAT scores) in the frenotomy group compared with the control 1.3.1).
group: MD 3.50 on a 12-point scale, 95% CI 3.06 to 3.94; 58 • Buryk 2011 used the 50-point SF-MPQ Pain Assessment Tool
participants. after five days of feeding and found a significant reduction in
the frenotomy group compared with the control group: MD -8.60,
One trial included only infants with moderate tongue-tie (Emond 95% CI -9.37 to -7.83 (Analysis 1.3.2).
2013) and found no objective improvement in feeding scores: MD
-0.10 on a 10-point scale, 95% CI -0.75 to 0.55; 105 participants. We did not combine visual analogue pain scale and SF-MPQ
scores for meta-analysis because studies had a high degree of
Timing of feeding assessment
heterogeneity and scales used different units of measure.
Two trials reported on infant breastfeeding within 48 hours of the
Qualitative assessment of infant feeding by parental survey
intervention (Buryk 2011; Dollberg 2006).
performed within 48 hours of the procedure (Analysis 1.4)
• IBFAT score (Buryk 2011): MD 3.50 on a 12-point scale, 95% CI Two trials (Berry 2012; Hogan 2005) reported high rates of
3.06 to 3.94; 58 participants. improvement in breastfeeding following frenotomy compared with
• LATCH score (Dollberg 2006): MD 0.0 on a 10-point scale, 95% CI control (typical risk ratio (RR) 3.48, 95% CI 2.18 to 5.56). The meta-
-1.08 to 1.08; 50 participants. analysis contained high levels of heterogeneity (Chi2 = 15.63, df = 1,
P < 0.0001; I2 = 94%) (Figure 5).
One trial (Emond 2013) reported no difference in LATCH scores
assessed between two and seven days after the intervention: MD

Informed decisions.
Figure 5. Forest plot of comparison: 1 Frenotomy versus no frenotomy or sham procedure, outcome: 1.6 Qualitative
assessment of infant feeding by parental survey performed within 48 hours of the procedure.
Frenotomy Control Risk Ratio Risk Ratio

Study or Subgroup Events Total Events Total Weight M-H, Fixed, 95% CI M-H, Fixed, 95% CI
Berry 2012 21 27 14 30 93.1% 1.67 [1.08 , 2.57]

Hogan 2005 27 28 1 29 6.9% 27.96 [4.07 , 192.12]
Total (95% CI) 55 59 100.0% 3.48 [2.18 , 5.56]

Total events: 48 15
Heterogeneity: Chi² = 15.63, df = 1 (P < 0.0001); I² = 94% 0.005 0.1 1 10 200
Test for overall effect: Z = 5.22 (P < 0.00001) Favours control Favours frenotomy
Test for subgroup differences: Not applicable
Hogan 2005 reported that all intervention infants (n = 8) versus no DISCUSSION

control infants (n = 9) had improved bottle-feeding.
Summary of main results
Emond 2013 obtained qualitative data from a subgroup of the study
population and found that most parents reported improvement in We included in this review five trials with a total sample size of 302
infant feeding directly after the intervention and in the days that infants. Only one small trial reported that frenotomy objectively
followed, but investigators did not state when they performed the improved infant breastfeeding (Buryk 2011). Pooled analysis of
survey. two other trials (N = 155) showed no objective change following
frenotomy (Dollberg 2006; Emond 2013) but showed consistently
Duration of breastfeeding or cessation of breastfeeding as assessed by reduced maternal nipple pain.
maternal report within four weeks of the procedure
Studies to date have not answered the clinically relevant
No trial reported the duration of breastfeeding or cessation of
question of whether frenotomy in tongue-tied infants with
breastfeeding as assessed by maternal report within four weeks.
feeding difficulties results in longer-term breastfeeding success
However, four studies reported on rates of breastfeeding at specific
and resolution of maternal pain. No studies reported duration of
time points. Berry 2012 reported that 58/59 were breastfeeding at
breastfeeding by group, and all had very high contamination rates
three months but did not report this by treatment group. Buryk
in the control group, making longer-term outcomes meaningless.
2011 reported that 36/58 were breastfeeding at two months, 23/58
at six months and 14/58 at 12 months. Study authors also did Overall completeness and applicability of evidence
not report by treatment group. Emond 2013 reported that at eight
weeks, 43/52 in the intervention group and 40/50 in the control All studies were performed at maternity hospitals in the United
group were at least partially breastfed. Hogan 2005 reported that Kingdom, America and Israel. All centres that carried out the studies
12/20 in the intervention group and 10/20 in the control group were had lactation support and frenotomy readily available. The total
breastfed for at least four months. number of infants studied to date is 302. This number is too small
to confirm the efficacy and safety of frenotomy, even though no
Infant pain assessed by a validated pain scale before, during and up to study reported any significant adverse effects. Populations studied
one hour post frenotomy to date were healthy term infants with moderate or severe tongue-
None of the included studies reported this outcome. tie. All primary outcomes were short-term, many assessed after just
one feed following intervention. Some studies followed up infants
Excessive bleeding at the time or within 24 hours of frenotomy for several months after the intervention, but as the contamination
(Analysis 1.5) rate within the control group was very high, longer-term results
No infants in any of the five trials experienced excessive bleeding are meaningless. Studies to date have not answered the clinically
following the intervention (RR not estimable; RD 0.00, 95% CI -0.03 relevant question of whether frenotomy in tongue-tied infants
to 0.03; 302 participants). results in longer-term breastfeeding success and resolution of
maternal pain.
Infection at the site of frenotomy requiring treatment with antibiotics
within seven days of the procedure (Analysis 1.6) The potential to improve infant breastfeeding while reducing
maternal nipple pain may be greatest when an infant is given the
No infants in any of the five trials experienced infection at the diagnosis of severe tongue-tie. However, only one trial has studied
intervention site requiring antibiotics (RR not estimable; RD 0.00, frenotomy in this population. Again, only one study has examined
95% CI -0.03 to 0.03; 302 participants). frenotomy in infants given the diagnosis of moderate tongue-tie
Damage to the tongue or submandibular ducts noted within seven and found that frenotomy did not have an objective effect on infant
days of the procedure (Analysis 1.7) feeding nor on maternal nipple pain but was subjectively effective.
No infants in any of the five trials experienced damage to the tongue

or submandibular ducts (RR not estimable; RD 0.00, 95% CI -0.03 to
0.03; 302 participants).

Informed decisions.
Quality of the evidence outcomes is low to insufficient and acknowledged that maternal
reported effect is greater. They identified gaps in the evidence
We assessed the quality of the evidence using the GRADE method including longer-term outcomes, applicability to infants born at
and classified evidence for major outcomes as low quality on non-tertiary centres, lack of consistency in diagnosis, effectiveness
the basis of small sample sizes (both in individual studies and in of non-surgical interventions and optimal age of intervention. Two
combined studies), inconsistent blinding and high risk of bias. The reviews acknowledged that the optimal time of intervention is
included studies had low rates of participant drop-out. Most study unknown (Bowley 2014; Power 2015). Bowley 2014 recognised that
authors responded when we requested further information. We frenotomy can improve breastfeeding problems in a tongue-tied
noted high degrees of heterogeneity between studies and observed child but advised waiting at least two weeks before performing the
that investigators used different diagnostic tools to diagnose the procedure. All reviews concluded that the procedure appears to
condition, reported differing degrees of tongue-tie severity and have a low complication rate when performed by trained operators.
used different scales to assess outcomes. The greatest weakness Many reviews reported that available studies have methodological
among studies to date is that investigators offered frenotomy to all flaws and risk of bias.
controls and provided the procedure for a large majority of them,
which strongly implies lack of equipoise about effectiveness of the AUTHORS' CONCLUSIONS
intervention during the study and reduces the quality of results.
Implications for practice
Potential biases in the review process
Frenotomy causes a short-term reduction in nipple pain among
We used the standard methods of Cochrane Neonatal in conducting breastfeeding mothers and an inconsistent positive effect on infant
this systematic review. Our inclusive search strategy would have breastfeeding. Owing to the small number of studies and the high
included all relevant studies. incidence of methodological issues, definitive benefit has not been
proven.
Agreements and disagreements with other studies or
reviews Implications for research
Several other published reviews have examined effects of Additional high-quality randomised controlled trials are needed
frenotomy for tongue-tie in young infants (Algar 2009; Bowley to confirm whether frenotomy in tongue-tied infants causes
2014; CADTH 2016; Cho 2010; Edmunds 2011; Francis 2015; Hall resolution of feeding difficulties with both short-term and longer-
2005; Hong 2013; Ito 2014; Lalakea 2003; Power 2015; Segal term follow-up. In such studies, frenotomy ideally should not be
2007; Suter 2009). All included observational studies as well as performed on control infants to allow long-term assessment of the
randomised trials. All reviews recognised a role for frenotomy effect of the intervention.
when evidence indicates feeding difficulties or maternal nipple
pain. However, review authors provided different interpretations Other major uncertainties remain unaddressed.
of the strength of available evidence supporting frenotomy. Algar
2009 Bowley 2014 Cho 2010 Edmunds 2011 Hong 2013 Ito 2014 • The effect of frenotomy on tongue-tied preterm infants has yet
Lalakea 2003 and Segal 2007 concluded that evidence is sufficient to be studied.
to recommend frenotomy in an infant with breastfeeding problems • The optimal age to perform frenotomy in infants remains
and tongue-tie. CADTH 2016 Hall 2005 Power 2015 Suter 2009 unclear.
and Francis 2015 concluded that frenotomy may be helpful • The effect of tongue-tie on early infant weight gain and on
and is safe but that definitive evidence is lacking. The most maternal difficulties in establishing a breast milk supply remains
recent review (CADTH 2016) concluded that frenotomy is a safe to be clarified.
procedure that leads to maternally perceived benefit in short-term • It has yet to be demonstrated whether frenotomy in
breastfeeding outcomes, concurring with this review that objective breastfeeding infants with tongue-tie and feeding difficulty
improvement in symptoms and long-term benefit is less certain. leads to a longer duration of breastfeeding.
CADTH 2016 concluded that it remains to be proved whether
• Whether frenotomy is a painful procedure that requires
frenotomy provides meaningful improvement in breastfeeding
analgesia or anaesthesia has yet to be established, as no study
difficulties, especially over the long term. Another recent review
to date has quantified infant pain during and after frenotomy.
(Francis 2015) included the same five randomised trials included
in this review, together with a retrospective cohort and 23 case ACKNOWLEDGEMENTS
series. Those review authors concluded that the strength of
the evidence supporting frenotomy for improved breastfeeding Thank you to the newborn research team at the Royal Women's
Hopsital for support throughout this project.

Informed decisions.
REFERENCES
References to studies included in this review Archives of Disease in Childhood 2009;94(11):911-2. [PMID:
19847004]
Berry 2012 {published data only}
Berry J, Griffiths M, Westcott C. A double-blind, randomized, Amir 2006
controlled trial of tongue-tie division and its immediate effect Amir LH, James JP, Donath SM. Reliability of the Hazelbaker
on breastfeeding. Breastfeeding Medicine 2012;7(3):189-93. assessment tool for lingual frenulum function. International
[PMID: 21999476] Breastfeeding Journal 2006;1(1):3. [PMID: 16722609]
Buryk 2011 {published data only} Bowley 2014
Buryk M, Bloom D, Shope T. Efficacy of neonatal release of Bowley DM, Arul GS. Fifteen-minute consultation: the infant
ankyloglossia. Pediatrics 2011;128(2):280-8. [PMID: 21768318] with a tongue tie. Archives of Disease in Childhood. Education
and Practice Edition 2014;99(4):127–9. [PMID: 24419208]
Dollberg 2006 {published and unpublished data}
Dollberg S, Botzer E, Grunis E, Mimouni FB. Immediate CADTH 2016
nipple pain relief after frenotomy in breast-fed infants with Canadian Agency for Drugs and Technologies in Health.
ankyloglossia: a randomized, prospective study. Journal of Frenectomy for the correction of ankyloglossia: a
Pediatric Surgery 2006;41(9):1598-1600. [PMID: 16952598] review of clinical effectiveness and guidelines. https://
www.cadth.ca/frenectomy-correction-ankyloglossia-review-
Emond 2013 {published data only}
clinical-effectiveness-and-guidelines (accessed 15 July
Emond A, Ingram J, Johnson D, Plair P, Whitelaw A, Copeland M, 2016):RC0785-000.
et al. Randomised controlled trial of early frenotomy
in breastfed infants with mild-moderate tongue-tie. Cho 2010
Archives of Diseases in Childhood Fetal and Neonatal Edition Cho A, Kelsberg G, Safranek S. Clinical inquiries. When should
2014;99(3):F189-95. [PMID: 24249695] you treat tongue-tie in a newborn? Journal of Family Practice
2010;59(12):712a-b. [PMID: 21135930]
Hogan 2005 {published data only}
Hogan M, Westcott C, Griffiths M. Randomized, controlled trial of Coryllos 2004
division of tongue-tie in infants with feeding problems. Journal Coryllos E, Genna CW, Salloum AC. Congenital tongue-
of Paediatrics and Child Health 2005;41(5-6):246-50. [PMID: tie and its impact on breastfeeding. AAP section on
15953322] breastfeeding. http://www.aap.org/breastfeeding/files/pdf/
BBM-8-27%20Newsletter.pdf 2004;Summer:(accessed 15 March
2013).
References to studies excluded from this review
Ngerncham 2013 {published data only} Edmunds 2011
Ngerncham S, Laohapensang M, Wongvisutdhi T, Ritjaroen Y, Edmunds J, Miles SC, Fulbrook P. Tongue-tie and
Painpichan N, et al. Lingual frenulum and effect on breastfeeding: a review of the literature. Breastfeeding Review
breastfeeding in Thai newborn infants. Paediatrics and 2011;19(1):19-26. [PMID: 21608523]
International Child Health 2013;33(2):86-90. [PMID: 23925281]
Francis 2015
Yousefi 2015 {published data only} Francis DO, Krishnaswami S, McPheeters M. Treatment of
Yousefi J, Namini FT, Raisolsadat SMA, Gillies R, Ashkezari A, ankyloglossia and breastfeeding outcomes: a systematic review.
Meara JG. Tongue-tie repair: z-plasty vs simple release. Iran Pediatrics 2015;135(6):e1458-66. [PMID: 25941303]
Journal of Otorhinolaryngology 2015;27(79):127–35.
GRADEpro [Computer program]
McMaster University GRADEpro [www.gradepro.org]. McMaster
References to ongoing studies University, 2014.
Ricalde 2017 {published data only} Hall 2005
Ricalde P. Prospective Evaluation of Lingual Frenotomy Hall DM, Renfrew MJ. Tongue tie. Archives of Disease in
in Newborns With Simultaneous Lip Tie for the Relief of Childhood 2005;90(12):1211-5. [PMID: 16301545]
Breastfeeding Pain. ClinicalTrials.gov.
Hazelbaker 1993
Hazelbaker AK. The Assessment Tool for Lingual Frenulum
Additional references Function (ATLFF): use in a lactation consultant private practise.
Algar 2009 Pasadena California, Pacific Oaks College, Thesis, 1993.
Algar V. Question 2. Should an infant who is breastfeeding
poorly and has a tongue tie undergo a tongue tie division?

Informed decisions.
Higgins 2011 Marchesan 2005

Higgins JPT, Green S (editors). Cochrane Handbook for Marchesan IQ. Lingual frenulum: quantitative evaluation
Systematic Reviews of Interventions Version 5.1.0. The proposal. International Journal of Orofacial Myology
Cochrane Collaboration, updated March 2011. 2005;31:39-48. [PMID: 16739711]
Hong 2013 Mathews 1988

Hong P. Ankyloglossia (tongue-tie). CMAJ 2013;185(185):E128. Mathews MK. Developing an instrument to assess infant
[DOI: 10.1503/cmaj.120785] breastfeeding behaviour in the early neonatal period. Midwifery
1988;4(4):154-65. [PMID: 3210979]
Ito 2014
Ito Y. Does frenotomy improve breast-feeding difficulties Melzack 1975
in infants with ankyloglossia? Pediatrics International Melzack R. The short-form McGill Pain Questionnaire. Pain
2014;56(4):497–505. [PMID: 24978831] 1987;30(2):191-7. [PMID: 3670870]
Jackson 2012 Messner 2000a

Jackson R. Improving breastfeeding outcomes: the impact of Messner AH, Lalakea ML. Ankyloglossia: controversies
tongue-tie. Community Practitioner 2012;85(6):42-4. [PMID: in management. International Journal of Pediatric
22779397] Otorhinolaryngology 2000;54(2-3):123-31. [PMID: 10967382]
Jensen 1994 Messner 2000b

Jensen D, Wallace S, Kelsay P. LATCH: a breastfeeding charting Messner AH, Lalakea ML, Aby J, Macmahon J, Bair E.
system and documentation tool. Journal of Obstetrics, Ankyloglossia: incidence and associated feeding difficulties.
Gynecologic, and Neonatal Nursing 1994;23(1):27-32. [PMID: Archives of Otolaryngology-Head and Neck Surgery
8176525] 2000;126(1):36-9. [PMID: 10628708]
Kotlow 2011 NICE 2005

Kotlow L. Diagnosis and treatment of ankyloglossia and National Institute of Health and Care Excellence (NICE).
tied maxillary fraenum in infants using Er:YAG and 1064 Division of ankyloglossia (tongue-tie) for breastfeeding. http://
diode lasers. European Archives of Paediatric Dentistry www.nice.org.uk/nicemedia/live/11180/31411/31411.pdf.
2011;12(2):106-12. [PMID: 21473843] (accessed 15 March 2013).
Krechel 1995 Obladen 2010

Krechel SW, Bildner J. CRIES: a new neonatal postoperative pain Obladen M. Much ado about nothing: two millennia of
measurement score—initial testing of validity and reliability. controversy on tongue-tie. Neonatology 2010;97(2):83-9. [PMID:
Paediatric Anaesthesia 1995;5(1):53-61. [PMID: 8521311] 19707023]
Krol 2007 Power 2015

Krol DM, Keels MA. Oral conditions. Pediatrics in Review Power RF, Murphy JF. Tongue-tie and frenotomy in infants
2007;28(1):15-22. [PMID: 17197455] with breastfeeding difficulties: achieving a balance. Archives of
Disease in Childhood 2015;100(5):489–94. [PMID: 25381293]
Kumar 2012
Kumar M, Kalke E. Tongue-tie, breastfeeding difficulties and the RevMan 2014
role of frenotomy. Acta Paediatrica 2012;101(7):687-9. [PMID: The Cochrane Collaboration. Review Manager (RevMan). Version
22404175] 5.3. The Cochrane Collaboration, 2014.
Lalakea 2002 Ricke 2005

Lalakea ML, Messner AH. Frenotomy and frenuloplasty: if, when Ricke LA, Baker NJ, Madlon-Kay DJ, DeFor TA. Newborn tongue-
and how. Operative Techniques in Pediatric Otolaryngology tie: prevalence and effect on breast-feeding. Journal of the
2002;13(1):93-7. [DOI: 10.1053/otot.2002.32157] American Board of Family Practice 2005;18(1):1-7. [PMID:
15709057]
Lalakea 2003
Lalakea ML, Messner AH. Ankyloglossia: does it matter? Schünemann 2013
Pediatric Clinics of North America 2003;50:381–97. Schünemann H, Brożek J, Guyatt G, Oxman A, editors, GWG.
GRADE Handbook for Grading Quality of Evidence and Strength
Lawrence 1993 of Recommendations. www.guidelinedevelopment.org/
Lawrence J, Alcock D, McGrath P, Kay J, MacMurray SB, handbook. Updated October 2013.
Dulberg C. The development of a tool to assess neonatal pain.
Neonatal Network 1993;12(6):59-66. [PMID: 8413140] Segal 2007
Segal LM, Stephenson R, Dawes M, Feldman P. Prevalence,
diagnosis, and treatment of ankyloglossia. Canadian Family
Physician 2007;53(6):1027-33. [PMID: 17872781]
Informed decisions.
Suter 2009 Taddio 1995

Suter VGA, Bornstein M. Ankyloglossia: facts and myths Taddio A, Nulman I, Koren BS, Stevens B, Koren G. A revised
in diagnosis and treatment. Journal of Periodontology measure of acute pain in infants. Journal of Pain and Symptom
2009;80(8):1204-19. [PMID: 19656020] Management 1995;10(6):456-63. [PMID: 7561228]
CHARACTERISTICS OF STUDIES
Characteristics of included studies [ordered by study ID]
Berry 2012
Study characteristics
Methods Randomised blinded controlled trial performed between October 2003 and April 2004 at an English re-
gional hospital
Participants Infants younger than 4 months of age with symptoms of breastfeeding problems and tongue-tie
Interventions Immediate frenotomy or non-division
Outcomes Primary outcomes: subjective and objective improvement in feeding - feeding score (adapted from
LATCH scoring system and Infant Breastfeeding Assessment Tool), maternal questioning, observer im-
pression
Secondary outcome: maternal pain scores
Notes Non-division infants, then divided
Risk of bias
Bias Authors' judgement Support for judgement
Random sequence genera- Low risk University of Southampton Medical Statistics and Computing Department
tion (selection bias) provided computer-generated randomisation for 60 babies; an independent
helper then placed the randomisation into sealed envelopes
Allocation concealment Low risk Sealed envelopes

(selection bias)
Blinding of participants Low risk Double-blinded

and personnel (perfor-
mance bias)
All outcomes
Blinding of outcome as- Low risk Double-blinded

sessment (detection bias)
All outcomes
Incomplete outcome data Low risk

(attrition bias)
All outcomes
Selective reporting (re- High risk Objective outcomes not reported numerically; said to be no different
porting bias)
Other bias High risk Non-divided babies offered division anyway; suggesting lack of equipoise

Informed decisions.
Buryk 2011
Methods Single-blinded randomised controlled trial performed between December 2007 and December 2008 at
an American regional military medical centre
Participants Infants (< 30 days) with breastfeeding issues found to have significant tongue-tie (Hazelbaker Assess-
ment Tool for Lingual Frenulum Function (ATLFF) scores: function scores > 11, appearance score < 8)
Exclusion criteria described
Interventions Frenotomy or sham procedure, immediately or within 1 to 2 weeks
Outcomes Primary outcome: improvement in maternal nipple pain (McGill Pain scores) and ability to breastfeed
(breastfeeding scores, Infant Breastfeeding Assessment Tool (IBFAT))
Secondary outcome: improvement in length of breastfeeding
Notes Non-division infants were offered frenotomy 2 weeks later
Risk of bias
Random sequence genera- Low risk Computerised random number generator of blocks of 4 created by a statisti-
tion (selection bias) cian and implemented by a research assistant
Allocation concealment Unclear risk Did not state how investigators concealed allocation
(selection bias)
Blinding of participants Low risk Parents blinded but told the allocation after first feed post procedure. All med-
and personnel (perfor- ical professionals involved were not blinded
mance bias)
All outcomes
Blinding of outcome as- Low risk Longer-term follow-up less significant, as all but 1 control infant had a frenoto-
sessment (detection bias) my
All outcomes

(attrition bias)
All outcomes
Selective reporting (re- Low risk

porting bias)
Other bias High risk All control infants were offered frenotomy
Dollberg 2006
Methods Randomised cross-over study performed between December 2001 and September 2004 at an Israeli
maternity hospital

Informed decisions.
Dollberg 2006 (Continued)
Participants Infants < 21 days old, all mothers had nipple pain; infants examined by neonatologist and found to
have tongue-tie
Interventions Cross-over study; randomised to sham, then feed, then frenotomy, then feed and vice versa
Outcomes Pain and latch scores after frenotomy or sham
Notes
Risk of bias
Random sequence genera- Low risk Computer-generated randomisation table

tion (selection bias)
Allocation concealment Low risk Sealed opaque envelopes

(selection bias)
Blinding of participants Low risk The mothers as well as all personnel taking care of the child after each sham or
and personnel (perfor- frenotomy procedure were masked
mance bias)
All outcomes
Blinding of outcome as- Low risk The mothers as well as all personnel taking care of the child after each sham or
sessment (detection bias) frenotomy procedure were masked
All outcomes

(attrition bias)
All outcomes
Selective reporting (re- High risk Results in control group not reported in original manuscript have been since
porting bias) provided by study authors
Other bias Low risk
Emond 2013
Methods Randomised feasibility trial of early frenotomy compared with usual care provided between December
2001 and September 2004 for infants with mild to moderate tongue-tie at a regional English maternity
hospital
Participants Mothers of babies with tongue-tie who were experiencing breastfeeding difficulties; Hazelbaker As-
sessment Tool for Lingual Frenulum Function (HTLFF) scores 6 to 12, latch score ≤ 8; infants with severe
tongue-tie excluded and sent for frenotomy
Interventions Frenotomy or not
Five days later, frenotomy offered to control group if symptoms persisted
Outcomes Primary outcome: changes in latch scores at 5 days

Informed decisions.
Emond 2013 (Continued)

Secondary outcomes: changes in Infant Breastfeeding Assessment Tool (IBFAT) score and score on
breastfeeding self-efficacy short form; changes in pain score on visual analogue scale
Notes Four of the 99 frenotomies performed as initial procedure were repeated and did not sufficiently divide
the frenulum
Risk of bias
Random sequence genera- Low risk Telephone-based block randomisation service stratified for sex and birth order
tion (selection bias)
All given routine breastfeeding support
Allocation concealment Low risk

(selection bias)
Blinding of participants Low risk Researchers were blinded, but mothers were not
mance bias)
All outcomes
Blinding of outcome as- Low risk

All outcomes

(attrition bias)
All outcomes

porting bias)
Other bias High risk First line of protocol states mothers of term infants with breastfeeding prob-
lems due to tongue-tie. Almost all infants had frenotomy at maternal request,
suggesting lack of equipoise; almost all control infants were offered frenoto-
my, suggesting lack of equipoise.
Hogan 2005
Methods Randomised controlled trial performed between March and July 2002 that recruited participants from
a regional English maternity hospital and 3 English birthing centres
Participants Infants 4 weeks of age
Interventions Frenotomy or intensive lactation support
Outcomes Subjective improvement in feeding
Notes
Risk of bias

Informed decisions.
Hogan 2005 (Continued)
Random sequence genera- Low risk Random sequence was computer generated by our department of medical
tion (selection bias) statistics and was placed in envelopes by a third party with no input from the 3
study authors
Allocation concealment Low risk Sealed envelope

(selection bias)
Blinding of participants High risk No blinding

mance bias)
All outcomes
Blinding of outcome as- High risk Study authors state that they did not have equipoise and provided no blinding
All outcomes

(attrition bias)
All outcomes

porting bias)
Other bias High risk All control infants were offered frenotomy
Characteristics of excluded studies [ordered by study ID]
Study Reason for exclusion
Ngerncham 2013 Cross-sectional study
Yousefi 2015 Compared frenotomy versus frenuloplasty. Population included children up to 12 years of age
Characteristics of ongoing studies [ordered by study ID]
Ricalde 2017
Study name Prospective Evaluation of Lingual Frenotomy in Newborns With Simultaneous Lip Tie for the Relief
of Breastfeeding Pain
Methods Randomised controlled trial of newborns in maternal infant care areas at Tampa General Hospital
Participants Term infants classified as having ankyloglossia via the HATLFF (Hazelbaker Assessment Tool for Lin-
gual Frenulum Function) and a Class III or IV maxillary labial frenum
Interventions Randomly assigned to 1 of 2 groups: Group A or Group B
Group A will receive a sham procedure for intervention #1 and a lingual frenotomy procedure for in-
tervention #2. Group B will receive a lingual frenotomy procedure for intervention #1 and a sham
procedure for intervention #2
Newborns who continue to have difficulty with breastfeeding after both interventions will undergo
intervention #3 - a labial frenotomy - and breastfeeding will be monitored afterwards

Informed decisions.
Ricalde 2017 (Continued)
Outcomes • Wong-Baker FACES Pain Rating Scale

• LATCH score
Starting date May 2015
Contact information Pat Ricalde, MD, DDS
Tampa General Hospital
Tampa, Florida, United States, 33606
813-870-6000
mailto:ricalde%40verizon.net?subject=NCT02141243, FRENOTOMY, Prospective Evaluation of Lin-

gual Frenotomy in Newborns With Simultaneous Lip Tie for the Relief of Breastfeeding Pain.
Sponsor: University of South Florida
Notes ClinicalTrials.gov identifier: NCT02141243
DATA AND ANALYSES
Comparison 1. Frenotomy versus no frenotomy or sham procedure
Outcome or subgroup title No. of studies No. of partici- Statistical method Effect size
pants
1.1 Infant breastfeeding assessed by a vali- 3 Mean Difference (IV, Subtotals only
dated scale Fixed, 95% CI)
1.1.1 IBFAT scores following procedure 1 58 Mean Difference (IV, 3.50 [3.06, 3.94]
Fixed, 95% CI)
1.1.2 LATCH scores following procedure 2 155 Mean Difference (IV, -0.07 [-0.63, 0.48]
Fixed, 95% CI)
1.2 Infant breastfeeding assessed by a vali- 1 105 Mean Difference (IV, -0.10 [-0.75, 0.55]
dated scale 2 to 7 days following procedure Fixed, 95% CI)
1.3 Maternal nipple pain assessed by a vali- 4 Mean Difference (IV, Subtotals only
dated pain scale Fixed, 95% CI)
1.3.1 Visual analogue pain scale 3 183 Mean Difference (IV, -0.74 [-1.35,
Fixed, 95% CI) -0.13]
1.3.2 SF-MPQ pain scale 1 58 Mean Difference (IV, -8.60 [-9.37,

Fixed, 95% CI) -7.83]
1.4 Qualitative assessment of infant feed- 2 114 Risk Ratio (M-H, Fixed, 3.48 [2.18, 5.56]
ing by parental survey performed within 48 95% CI)
hours of procedure

Informed decisions.
Outcome or subgroup title No. of studies No. of partici- Statistical method Effect size
pants
1.5 Excessive bleeding at the time or within 5 302 Risk Difference (M-H, 0.00 [-0.03, 0.03]
24 hours of frenotomy (as determined by Fixed, 95% CI)
study investigators)
1.6 Infection at the site of frenotomy re- 5 302 Risk Ratio (M-H, Fixed, Not estimable
quiring treatment with antibiotics within 7 95% CI)
days of procedure
1.7 Damage to the tongue and/or sub- 5 302 Risk Ratio (M-H, Fixed, Not estimable
mandibular ducts noted within 7 days of 95% CI)
procedure (as determined by study investi-
gators)
Analysis 1.1. Comparison 1: Frenotomy versus no frenotomy or sham

procedure, Outcome 1: Infant breastfeeding assessed by a validated scale

1.1.1 IBFAT scores following procedure

Buryk 2011 11.6 0.8 30 8.1 0.9 28 100.0% 3.50 [3.06 , 3.94]
Subtotal (95% CI) 30 28 100.0% 3.50 [3.06 , 3.94]
1.1.2 LATCH scores following procedure

Emond 2013 8.4 1.8 53 8.5 1.6 52 73.4% -0.10 [-0.75 , 0.55]
Dollberg 2006 6.8 2 25 6.8 1.9 25 26.6% 0.00 [-1.08 , 1.08]
Subtotal (95% CI) 78 77 100.0% -0.07 [-0.63 , 0.48]

Favours control Favours frenotomy
Analysis 1.2. Comparison 1: Frenotomy versus no frenotomy or sham procedure, Outcome

2: Infant breastfeeding assessed by a validated scale 2 to 7 days following procedure

Emond 2013 8.4 1.8 53 8.5 1.6 52 100.0% -0.10 [-0.75 , 0.55]
Total (95% CI) 53 52 100.0% -0.10 [-0.75 , 0.55]

Test for overall effect: Z = 0.30 (P = 0.76) -1 -0.5 0 0.5 1
Test for subgroup differences: Not applicable Favours control Favours frenotomy

Informed decisions.
Analysis 1.3. Comparison 1: Frenotomy versus no frenotomy or sham

procedure, Outcome 3: Maternal nipple pain assessed by a validated pain scale
Favours frenotomy Control Mean Difference Mean Difference

1.3.1 Visual analogue pain scale

Berry 2012 1.6 1.5 14 2.9 1.5 14 30.3% -1.30 [-2.41 , -0.19]
Dollberg 2006 5.3 2.2 25 5.5 1.9 25 28.8% -0.20 [-1.34 , 0.94]
Emond 2013 3 2.4 53 3.7 2.6 52 40.8% -0.70 [-1.66 , 0.26]
Subtotal (95% CI) 92 91 100.0% -0.74 [-1.35 , -0.13]
1.3.2 SF-MPQ pain scale

Buryk 2011 4.9 1.5 30 13.5 1.5 28 100.0% -8.60 [-9.37 , -7.83]
Subtotal (95% CI) 30 28 100.0% -8.60 [-9.37 , -7.83]

Favours frenotomy Favours control
Analysis 1.4. Comparison 1: Frenotomy versus no frenotomy or sham procedure, Outcome 4:

Qualitative assessment of infant feeding by parental survey performed within 48 hours of procedure

Berry 2012 21 27 14 30 93.1% 1.67 [1.08 , 2.57]

Hogan 2005 27 28 1 29 6.9% 27.96 [4.07 , 192.12]
Total (95% CI) 55 59 100.0% 3.48 [2.18 , 5.56]

Total events: 48 15
Heterogeneity: Chi² = 15.63, df = 1 (P < 0.0001); I² = 94% 0.005 0.1 1 10 200
Test for overall effect: Z = 5.22 (P < 0.00001) Favours control Favours frenotomy

Excessive bleeding at the time or within 24 hours of frenotomy (as determined by study investigators)
Experimental Control Risk Difference Risk Difference

Berry 2012 0 27 0 30 18.9% 0.00 [-0.07 , 0.07]

Hogan 2005 0 28 0 29 18.9% 0.00 [-0.07 , 0.07]
Emond 2013 0 53 0 52 34.8% 0.00 [-0.04 , 0.04]
Dollberg 2006 0 14 0 11 8.2% 0.00 [-0.15 , 0.15]
Buryk 2011 0 30 0 28 19.2% 0.00 [-0.06 , 0.06]
Total (95% CI) 152 150 100.0% 0.00 [-0.03 , 0.03]

Total events: 0 0
Heterogeneity: Chi² = 0.00, df = 4 (P = 1.00); I² = 0% -1 -0.5 0 0.5 1
Test for overall effect: Z = 0.00 (P = 1.00) Favours frenotomy Favours control

Informed decisions.

Infection at the site of frenotomy requiring treatment with antibiotics within 7 days of procedure

Berry 2012 0 28 0 29 Not estimable

Buryk 2011 0 53 0 52 Not estimable
Dollberg 2006 0 14 0 11 Not estimable
Emond 2013 0 30 0 28 Not estimable
Hogan 2005 0 27 0 30 Not estimable
Total (95% CI) 152 150 Not estimable

Total events: 0 0
Heterogeneity: Not applicable 0.01 0.1 1 10 100
Test for overall effect: Not applicable Favours frenotomy Favours control
Analysis 1.7. Comparison 1: Frenotomy versus no frenotomy or sham procedure, Outcome 7: Damage to the
tongue and/or submandibular ducts noted within 7 days of procedure (as determined by study investigators)
Experimental Control Risk Ratio Risk Ratio

Berry 2012 0 28 0 29 Not estimable

Buryk 2011 0 53 0 52 Not estimable
Dollberg 2006 0 14 0 11 Not estimable
Emond 2013 0 30 0 28 Not estimable
Hogan 2005 0 27 0 30 Not estimable
Total (95% CI) 152 150 Not estimable

Total events: 0 0
Heterogeneity: Not applicable 0.01 0.1 1 10 100
Test for overall effect: Not applicable Favours frenotomy Favours control
WHAT'S NEW
Date Event Description
8 June 2021 Amended Minor changes to Dollberg 2006.
• Risk of bias table:

◦ Blinding of participants and personnel (performance bias):
previously " not blinded". Now states: "The mothers as well
as all personnel taking care of the child after each sham or
frenotomy procedure were masked".
◦ Blinding of outcome assessment (detection bias): previous-
ly " not blinded". Now states: "The mothers as well as all per-
sonnel taking care of the child after each sham or frenotomy
procedure were masked".
• Reference: previously stated: 'published data only'. Now
states: "published and unpublished data".

Informed decisions.
HISTORY
Protocol first published: Issue 4, 2014
Review first published: Issue 3, 2017
Date Event Description
7 May 2021 Amended Minor change to wording for clarity in effects of intervention sec-
tion regarding outcome 1.1.
16 June 2017 Amended number of figures reduced to 8
10 July 2008 Amended Converted to new review format
CONTRIBUTIONS OF AUTHORS
CO'D and DB wrote the first draft of the protocol.
JO'S and JF wrote subsequent drafts of the protocol.
PD, SJ, DT and CO'D commented on and reviewed the protocol.
JO'S and JF wrote the review. PD, SJ, DT and CO'D commented on and reviewed the review.
DECLARATIONS OF INTEREST
None declared.
SOURCES OF SUPPORT
Internal sources
• The Royal Women's Hospital Foundation, Parkville, Melbourne, Australia, Other
External sources
• NMHRC Program Grant, Australia
Part of Dr O'Shea's salary

• Eunice Kennedy Shriver National Institute of Child Health and Human Development, National Institutes of Health, Department of Health
and Human Services, USA
Editorial support for the Cochrane Neonatal Review Group has been funded with Federal funds from the Eunice Kennedy Shriver
National Institute of Child Health and Human Development, National Institutes of Health, Department of Health and Human Services,
USA, under Contract No. HHSN275201600005C
• National Institute for Health Research, UK
Editorial support for the Cochrane Neonatal Review Group has been funded with funds from a UK National Institute of Health Research
Grant (NIHR) Cochrane Programme Grant (13/89/12). The views expressed in this publication are those of the review authors and are
not necessarily those of the NHS, the NIHR or the UK Department of Health
DIFFERENCES BETWEEN PROTOCOL AND REVIEW

We added to this review the methods and plan for 'Summary of findings' tables and GRADE recommendations, which were not included
in the original protocol.
INDEX TERMS
Medical Subject Headings (MeSH)

Ankyloglossia [*surgery]; *Breast Feeding [adverse effects]; Gestational Age; Lingual Frenum [*surgery]; Mastodynia [etiology];
Nipples; Pain Measurement; Randomized Controlled Trials as Topic

Informed decisions.
MeSH check words

Female; Humans; Infant, Newborn


Frenotomy For Tongue Tie in Newborn Infants

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Cochrane

Frenotomy for tongue-tie in newborn infants (Review)

Frenotomy for tongue-tie in newborn infants (Review)

Frenotomy for tongue-tie in newborn infants (Review) i

Frenotomy for tongue-tie in newborn infants

Contact: Joyce E O'Shea, joyce.o'shea@nhs.net, jem.oshea@gmail.com.

Editorial group: Cochrane Neonatal Group.

Also, to perform subgroup analysis to determine the following.

• Method of feeding (breast or bottle).

• Age at frenotomy (≤ 10 days of age; > 10 days to three months of age).

Frenotomy for tongue-tie in newborn infants (Review) 1

Data collection and analysis

PLAIN LANGUAGE SUMMARY

Surgical release of tongue-tie for the treatment of tongue-tie in young babies

Frenotomy for tongue-tie in newborn infants (Review) 2

Patient or population: tongue-tie in newborn infants

Cochrane Database of Systematic Reviews

GRADE Working Group grades of evidence

Frenotomy for tongue-tie in newborn infants (Review) 4

Frenotomy for tongue-tie in newborn infants (Review) 5

Frenotomy for tongue-tie in newborn infants (Review) 7

Frenotomy for tongue-tie in newborn infants (Review) 8

Figure 1. Study flow diagram.

Frenotomy for tongue-tie in newborn infants (Review) 9

Included studies • Emond 2013 is a single-centre study performed in the United

Random sequence generation (selection bias)

0% 25% 50% 75% 100%

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

Frenotomy for tongue-tie in newborn infants (Review) 10

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

Selective reporting (reporting bias)

Frenotomy for tongue-tie in newborn infants (Review) 11

Allocation of randomised participants) resulting from loss to follow-up. Buryk

Incomplete outcome data Primary outcomes

Frenotomy for tongue-tie in newborn infants (Review) 12

Frenotomy Control Mean Difference Mean Difference

1.1.1 IBFAT scores following procedure

1.1.2 LATCH scores following procedure

Test for subgroup differences: Chi² = 97.28, df = 1 (P < 0.00001), I² = 99.0% -4 -2 0 2 4

Frenotomy for tongue-tie in newborn infants (Review) 13

Frenotomy Control Risk Ratio Risk Ratio

Berry 2012 21 27 14 30 93.1% 1.67 [1.08 , 2.57]

Total (95% CI) 55 59 100.0% 3.48 [2.18 , 5.56]

Hogan 2005 reported that all intervention infants (n = 8) versus no DISCUSSION

No infants in any of the five trials experienced damage to the tongue

Frenotomy for tongue-tie in newborn infants (Review) 14

Frenotomy for tongue-tie in newborn infants (Review) 15

Frenotomy for tongue-tie in newborn infants (Review) 16

Higgins 2011 Marchesan 2005

Hong 2013 Mathews 1988

Jackson 2012 Messner 2000a

Jensen 1994 Messner 2000b

Kotlow 2011 NICE 2005

Krechel 1995 Obladen 2010

Krol 2007 Power 2015

Lalakea 2002 Ricke 2005

Suter 2009 Taddio 1995

Characteristics of included studies [ordered by study ID]

Interventions Immediate frenotomy or non-division