Received: 18 January 2021 | Revised: 23 June 2021 | Accepted: 28 June 2021

DOI: 10.1111/joor.13229

ORIGINAL ARTICLE

Implementation of biopsychosocial supported

self-­management for chronic primary oro-­facial pain
including temporomandibular disorders: A theory, person and
evidence-­based approach

Vishal R. Aggarwal1 | Jianhua Wu1 | Frank Fox1 | Daniel Howdon2 |

Elspeth Guthrie2 | Alan Mighell1

1
School of Dentistry, Faculty of Medicine &
Health, University of Leeds, Leeds, UK Abstract
2
Faculty of Medicine & Health, Leeds Background: Aims of the study were to:
Institute of Health Sciences, University of
• Implement supported self-­management for chronic primary oro-­facial pain in a
Leeds, Leeds, UK
clinical setting.
Correspondence
• Evaluate its impact on consultation rates, pain severity, interference with life and
Vishal R. Aggarwal, Clinical Associate
Professor in Acute Dental Care and Chronic patient experience.
Pain, School of Dentistry, Faculty of
Methods: Sixty-­six patients with chronic primary oro-­facial pain received the inter-
Medicine & Health, University of Leeds,
Worsley Building, Clarendon Way, Leeds vention at a facial pain clinic at Leeds Dental Institute, UK. Brief Pain Inventory (BPI)
LS2 9LU.
scores measured pain severity and interference with life before and after the inter-
Email: v.r.k.aggarwal@leeds.ac.uk
vention. Process mining outlined patient care pathways. Monthly consultation rates
Funding information
measured 12 months before and after the intervention were used to evaluate bur-
None.
den on healthcare services and economic impact. Patient feedback was assessed via
Patient and Public involvement discussion groups.
Results: Mean BPI scores significantly improved after intervention—­from 5.70 (SD
1.89) to 3.78 (SD 2.34) (p < .001); mean pain interference score reduced from 19.95
(SD 9.41) to 12.05 (SD 9.64) (p < .001). Average monthly consultations significantly
(p = .001) reduced from 0.42/month before the intervention to 0.16/month after
the intervention. Economic assessment showed cost savings of £293 per patient per
year. Process mining showed high rates of service usage with 31 patients also at-
tending 51 other specialist services between them. Patient and Public Involvement
discussion groups with 5 patients identified that the intervention was a ‘constant
companion’ and should be implemented at the outset in the care pathway.
Conclusion: Supported self-­management for chronic primary oro-­facial pain has a
positive impact on health outcomes (physical functioning, pain intensity and patient
experience), as well as service usage and healthcare costs when implemented in a
secondary care clinical setting. Reconfiguring current care pathways to upscale early
implementation of such interventions should be a priority for future testing.

J Oral Rehabil. 2021;00:1–11. wileyonlinelibrary.com/journal/joor© 2021 John Wiley & Sons Ltd | 1
2 | AGGARWAL et al.

KEYWORDS

biopsychosocial, burning mouth syndrome, chronic oro-­facial pain, Self-­management,

Temporomandibular disorders

1 | I NTRO D U C TI O N considerable out-­of-­pocket costs (£333 per person per 6-­month

period) and indirect (£1242 per person per 6-­month period) costs
Persistent pain in the face or mouth (oro-­facial pain) is a frequent related to lost time at work.16
cause for consultation in both primary dental and medical care, and There is growing evidence from systematic reviews that self-­
a substantial proportion of people (7% of the population) develop management interventions incorporating a biopsychosocial ap-
both chronic and disabling pain.1,2 People with chronic oro-­facial proach are effective, in comparison with usual care, in improving
pain also report an increase in pain levels and interference with life outcomes (long-­term pain intensity and depression) in patients
and are thus more likely to seek treatment and take medication.3 with oro-­f acial conditions like TMD.17 Using synthesis of evidence
Clinically, chronic oro-­facial pain is the characteristic feature of from this review and further qualitative and quantitative7,17 work
a number of conditions such as temporomandibular joint disorder our team developed an evidence-­based biopsychosocial interven-
(TMD), burning mouth syndrome and persistent idiopathic facial pain tion for chronic oro-­f acial pain18 in accordance with the medical
that are difficult to diagnose and manage successfully.4-­7 The most research council guidance on development of complex interven-
recent IASP (international association for studies on pain) classifica- tions.19 In addition to synthesis of evidence from both quantita-
8
tion for ICD-­11 and the International Classification of Orofacial Pain tive17 and qualitative studies,7 we adapted an existing manual,
9
(ICOP) classified these chronic oro-­facial pain conditions as chronic ‘Managing chronic widespread pain’, which was shown to be both
primary pains defined as ‘chronic pain in one or more anatomical re- clinically and cost-­effective in a large randomised controlled trial
gions that persists or recurs for longer than 3 months and is associ- for managing chronic widespread pain in primary care in both the
ated with significant emotional distress and/or significant functional short (6 months) and longer term (24 months). 20 We adopted a co-­
8
disability’. This has been confirmed by epidemiological evidence production and co-­design approach with patient and clinician stake-
showing that chronic oro-­facial pain co-­occurs with other chronic holders to develop our intervention manual. 21 This was evaluated
pain syndromes2 and is associated with high levels of psychological in a proof-­of-­concept trial,18 which showed that the intervention
distress and aspects of illness behaviour that are indicative of the was acceptable to patients22 and could be delivered by telephone
1,2
process of somatisation and that these psychological factors pre- or face-­to-­f ace by facilitators from clinical and non-­clinical back-
dict the onset and persistence of chronic oro-­facial pain.10 grounds.18 Although the proof-­of-­concept trial was not powered
Despite this, management of patients with chronic oro-­facial to measure effectiveness, there was a significant improvement
pain, in particular TMD, tends to focus on correction of local me- in some outcome measures indicating potential effectiveness.18
chanical factors like teeth grinding and malocclusion rather than Additional components (posture control and habit reversal) were
addressing underlying psychosocial factors which are aetiological. identified which led to further refinement of the intervention. 23
Cochrane systematic reviews have shown little or no beneficial Overall, the intervention incorporated a biopsychosocial approach
effects of physical therapies such as irreversible occlusal adjust- with principles of cognitive behaviour therapy for the psychoso-
ments11 and oral splints12,13 that are targeted towards the correction cial components and posture control for physical self-­regulation. 23
of these factors. This intervention aligns with recent recommendations by the US
Indeed, an audit of 101 consecutive referrals of patients with National Academies high profile international expert committee
chronic oro-­facial pain to secondary care14 showed that they had that advocated therapist-­guided self-­management approaches that
been treated in nine different hospitals; referred to fifteen distinct combine a psychoeducational rationale with techniques drawn
specialties, with a mean of seven consultations per specialty. Of 341 from cognitive-­b ehavioural therapy (CBT) and biofeedback for
treatment attempts, only 24% yielded a successful outcome and temporomandibular disorders. 24
14
these did not account for natural remission or placebo response.
This imposes a huge burden on already stretched healthcare re-
sources as current care pathways associated with multiple consulta- 1.1 | Aims
4,5,7
tions in secondary care do nothing to improve pain.
TMD alone is thought to cost the US 4 billion dollars annually.15 The aim of the current investigation was to therefore to implement
Whilst data are unavailable for overall economic cost in the UK, a and evaluate our self-­management intervention in a healthcare set-
study examining the costs to the UK National Health Service6 showed ting for patients with chronic oro-­facial pain.
that consultation costs comprised 75% of cumulative healthcare util- Specific objectives were to:
isation costs, or an estimated £1,318 on average, for patients with
persistent oro-­facial pain. Economic analysis for indirect and out-­of-­ 1. Assess the impact of the intervention on pain intensity and
pocket costs show that patients with persistent oro-­facial pain have interference with life.
AGGARWAL et al. | 3

2. Assess the impact of the intervention on consultation rates within • Step 2: Understanding how the pain is affecting me—­Impact as-
medical and dental specialties. sessment and goal setting allows tailoring to the patient's needs.
3. Evaluate the economic impact of our intervention. • Step 3: My programme—­focuses on a biopsychosocial approach
4. Evaluate patient feedback on their experiences with the using three evidence-­based interventions—­posture control, be-
intervention. havioural activation and cognitive restructuring.
• Step 4: Continuing to manage my pain, and recovery techniques—­
discusses episodic nature of pain and relapse prevention.
2 | M ATE R I A L S A N D M E TH O DS

2.1 | Study design 2.4 | Outcome measures and data analysis

The study was a pragmatic prospective evaluation of our biopsy- 1. Pain severity and interference with life: This was measured using
chosocial supported self-­management intervention. 20 Patients were the Brief Pain Inventory (BPI), 25 which includes patient-­rated
referred to a facial pain clinic which solely implemented the inter- visual analogue scales and measures pain location, intensity (least,
vention and was located at Leeds Dental Institute, UK. Patients at- worse and average over the past week) and interference with
tended the clinic from October 2016 and were followed up from their activities of daily-­living. It is recommended by IMMPACT26 core
first appointment up to and including December 2018. Anonymous outcomes measures for chronic pain trials. BPI was completed
data were extracted for these patients and their Dental Institute re- blind and independent of the treatment provider by patients
cords were linked to their medical outpatient clinic records at Leeds before their first appointment and similarly after their last ap-
General Infirmary. Both the Dental Institute and General Infirmary pointment. All scores were calculated and entered into the data
are part of Leeds Teaching Hospital Trust. set also blind and independent of the treatment provider. For
the purpose of analysis, means and standard deviations of BPI
scores were compared before and after treatment. The paired
2.2 | Participants sample t-­test was used in all comparisons as we tested the
pre-­ and post-­treatment BPI scores for each patient. Statistical
Sixty-­six patients attending the clinic were referred from the oral sur- significance was set at p<0.05.
gery, oral medicine and restorative dentistry departments at Leeds 2. Consultation rates: Average monthly consultation rates were
Dental Institute. Oral surgery and restorative dentistry included measured before and after the intervention to assess impact
patients with chronic temporomandibular disorders (principally in- on consultations both within dental and medical specialities
ternal derangements and muscle disorders (myalgia and myofascial at the Dental Institute and Leeds General Infirmary respec-
pain N = 46) whereas those from oral medicine had chronic burning tively. Average monthly consultations were used to account
mouth (BMS) or persistent idiopathic facial pain (N = 20). for the differing pre-­clinic and post-­clinic attendance times.
Consultation rates before the intervention were measured in
the 12 months prior to the first facial pain clinic appointment
2.3 | Intervention and appointments after were measured for the 12 months
after the final facial pain clinic appointment (or the data cut-­
The intervention has been described previously.18 Briefly, up to off date of December 2018 if sooner). The paired sample t-­test
8 sessions of face-­to-­face or telephone supported self-­management was used in all comparisons as we tested the pre-­ and post-­
were offered over a period of up to 12 weeks and were delivered treatment consultation rates for each patient. Statistical sig-
by a clinician from a dental background (VA) with training and prior nificance was set at p < .05.
experience in delivering the intervention. New patient appointments 3. Economic analysis: We estimated hospital costs for each ap-
were allocated a 60min appointment while follow-­up appointments pointment in our data, before and after the intervention, based
were allocated a 40min appointment. All patients received the treat- on the NHS Specialty Code specified for the appointment. 28 We
23
ment guide divided into 4 steps, and which is easy to read (age estimated all planned appointments irrespective of whether the
equivalent 12–­13 years (Flesch-­Kincaid level 6.4)), engaging and al- patient attended or ultimately failed to attend.
lows patients to personalise it to their needs: 4. Care Pathways: Process mining, an emerging data analysis tech-
nique, was used to map care pathway to illustrate the most com-
• Step 1: What is ‘managing my chronic oro-­facial pain’ all about?—­ mon sequences of treatment experienced by the patient cohort
explanation of the origins of physical symptoms of subtypes of in the hospitals’ dental and medical specialities. Its purpose is to
chronic oro-­facial pain and psychological processes involved in rapidly deliver comprehensive representations of care pathways
pain pathways using, for example the gate control theory of pain. and, for this case, revealing new insights to the utilisation of ser-
Introduces self-­management, emphasising the role of the patient vices. The process mining tool selected was Disco27 and the data
as the agent of change/ taking control. used is detailed in the study design above.
4 | AGGARWAL et al.

5. Subgroup analysis: we investigated differences in consultation 1.89) to 3.78 (SD 2.34) post-­treatment. Mean pain interference score
rates for those with TMD (N = 46) and BMS (N = 20) separately also significantly reduced (p<0.001) from pre-­treatment scores of
to determine whether the intervention had different effects on 19.95 (SD 9.41) to 12.05 (SD 9.64) post-­treatment.
consultation rates, pain intensity and pain severity for subtypes
of chronic oro-­facial pain. Paired sample t-­test were used in these
comparisons as we tested the pre-­ and post-­treatment consul- 3.2 | Consultation rates and cost savings
tation rates and pain severity and interference for each patient.
Statistical significance was set at p < .05. Average monthly specialist clinic visit reduced from 0.23/month be-
6. Patient feedback and experience was elucidated through Patient fore the intervention to 0.15/month after the intervention (p-­value
and Public Involvement (PPI) meetings where five patients shared =0.008) measured over a 12-­month period before and after the in-
their lived experience of chronic oro-­facial pain including their ex- tervention. Mean follow-­up time was 384.82 days (SD 197.16).
periences of using our supported self-­management intervention. An initial comparison of average monthly costs observed
All patients consented to participation at the meetings (N=3) and 12 months before and 12 months after the patient's consultations
transcription and publication of audio recordings. yielded a large fall in those observed afterwards (£69.85 before vs
£21.50 after, p < .001, n = 66). Over a full 12-­month period, this
represents a cost saving of £580 per patient.
2.5 | Ethical approval The average number of sessions per patient of the facial pain
clinic was 2.73 (SD 1.66 range 1–­7). These are presented in Figure 1,
Ethical approval for a larger project ‘Linking dental and medical pa- which shows number of patients attending that range of sessions.
tient records for research’ was used to conduct the current study. The average tariff per patient for a consultant-­led face-­to-­face new
Approval for that project was granted by HRA and HCRW (IRAS ref: patient consultation in an oral medicine setting is £123.07 and for
277767; Research Ethics committee ref: 20/WM/0127). a consultant-­led face-­to-­face review is £95.13 (based on local tariff
rates at Leeds Dental Institute). These were the costs applied to fa-
cilitator time. For 2.73 sessions, this gives a total consultation cost
3 | R E S U LT S of £287. Based on cost saved from consultation rates above (£580),
this represents a cost saving of (£580 -­ £287) or £293 per patient
3.1 | Pain outcomes per year.

Of 66 patients who attended the clinic, 22 completed the Brief Pain

Inventory (BPI) before and after clinical consultation. For these pa- 3.2.1 | Service Usage and comorbidities
tients, mean pain severity scores and mean pain interference scores
were calculated before and after treatment using the standard BPI Patients with chronic primary oro-­facial pain had high rates of ser-
scoring. 25 This showed that mean pain severity scores significantly vice usage attending 51 different clinic services within both dental
reduced (p<0.001) from 5.70 pre-­treatment (standard deviation (SD) and medical specialties. Figure 2 shows services that had more than

F I G U R E 1 Bar chart showing number

of sessions attended on the facial pain
(OMPAIN) clinic (x-­axis) plotted against
the number of patients attending those
sessions (y-­axis)
AGGARWAL et al. | 5

F I G U R E 2 Consultations of the 66
patients (who received the intervention)
across medical and dental services.
The graph shows those with more than
10 consultations per service. AUDI,
Audiology; CARD, Cardiology; CHMO,
Medical oncology; COLO, Colorectal;
DERM, Dermatology; DIAB, Diabetic
medicine; DMS, Oral medicine; ENDO,
Endocrinology; ENT, Ear, Nose and
Throat; GAST, Gastroenterology; GYNA,
Gynaecology; NEUR, Neurology; OBST,
Obstetrics; ODON, Orthodontics; OPH,
Ophthalmoloy; ORAL, oral surgery; PAIN,
Pain management; PHYS, Physiotherapy;
RDEN, Restorative dentistry; RHEU,
Rheumatology; THOR, Respiratory
medicine; TRAU, Trauma; UROL, Urology;
VA-­M, facial pain clinic.

F I G U R E 3 Process mining care pathways of 66 patients attending the facial pain clinic (VA-­M). Thirty-­seven patients were discharged
following attendance to the clinic represented by the stop symbol on the bottom left-­hand corner. Key pathways to VA-­M highlighted
with red arrows. AUDI, Audiology; DERM, Dermatology; DMS, Oral medicine; ENT, Ear, Nose and Throat; GAST, Gastroenterology;
GYNA, Gynaecology; GYNA, Gynaecology; NEUR, Neurology; OPH, Ophthalmoloy; ORAL, oral surgery; PAIN, Pain management; PHYS,
Physiotherapy; RDEN, Restorative dentistry; RHEU, Rheumatology; TRAU, Trauma; UROL, Urology; VA-­M, facial pain clinic

10 consultations. Not surprisingly, the most common services were Botox injections with 81 consultations. Medical services included
dental and included oral surgery (ORAL), oral medicine (DMS), our those to which comorbid pain conditions present. A total of 48 (73%)
pain clinic (VA-­M) and restorative dentistry (RDEN) in descending of patients had comorbidities with the most frequent comorbidity
order. The most frequent procedure in the oral surgery clinic was clinical services attended out with dentistry being: Gynaecology
6 | AGGARWAL et al.

(GYNA for chronic pelvic pain), Rheumatology (RHEU for fibromy- the intervention (Figure 5). However, whilst there was a drop in pain
algia) and Gastroenterology (GAST for irritable bowel syndrome)—­ intensity for BMS patients with mean 5.8 (2.1) before to 5.4 (2.7)
conditions that frequently co-­occur with chronic oro-­facial pain. 2 after the intervention (Figure 5), this was not statistically significant
Figure 3 shows the pathway and number of patients who attended (p = .39).
each of these services. The process model clearly shows the 66 pa- Pain interference for TMD improved after the intervention from
tients attending the pain clinic (VA-­M) and the total number of at- a mean BPI of 4.7 (2.7) before to 2.7 (3.2) after the intervention al-
tendances to be 180. It also shows that 25 of these patients came though not statistically significant (p = .069) (Figure 6). There was
from ‘ORAL’ and 32 came from ‘DMS’. These pathways are indicated also improvement in pain interference for BMS patients with mean
by red arrows in Figure 3. Attendance at services such as PAIN (pain 5.4 (2.7) before to 3.2 (2.1) after the intervention (Figure 6) but this
management) and PHYS (physiotherapy) included those patients was not statistically significant (p = 0.42).
that were managed by multiple services and others such as ENT,
Audiology and Ophthalmology where patients are referred for mul-
tiple investigations to exclude an organic cause. 3.5 | Patient feedback

Patient and Public Involvement (PPI) meetings (N=3) were attended

3.3 | Attrition by 5 patients and one lay chair who had previous expertise in patient
involvement in research. Patient feedback related to three key do-
There were 16 (24%) patients who attended one session on the pain mains: personal experiences, experiences of the current healthcare
clinic (Figure 1). Of these, 7 needed further appointments but did not system and experiences of the supported self-­management inter-
attend. The attrition rate was therefore 7/66 (11%). vention. Table 1 shows the key domains along with corresponding
feedback. Chronic oro-­facial pain affected every aspect of patients’
lives and suffering was both physical and psychosocial. Experiences
3.4 | Subgroup Analysis of the current healthcare system involved multiple referrals and
invasive and irreversible treatments, which did nothing to improve
Differences in consultation rates for TMD (n=46) and BMS (n=20) symptoms, imposed financial burden on the patients and produced
are presented in Figure 4. This shows consultation rates dropped sig- harmful side effects and addiction.
nificantly (p<0.01) for TMD patients from a mean of 0.48 (SD 0.30) Patients reported that the intervention improved their under-
before the intervention to 0.18 (SD 0.36) after the intervention. standing of the pain and could readily be shared with their work
Similarly for BMS consultation rates dropped significantly (p<0.01) and social networks to allow them to understand the pain which re-
from a mean of 0.29 (SD 0.16) before the intervention to 0.11 (SD sulted in more empathy. Patients were able tailor the intervention as
0.27) after the intervention. needed according to their symptom severity and use what worked
Pain severity for TMD also significantly (p < .01) improved after for them. All five patients found it to be a constant ‘support’ and
the intervention from a mean BPI of 6.4 (2.0) before to 3.4 (2.3) after recommended its use at the onset of symptoms.

F I G U R E 4 Box plots showing monthly

consultation rates before and after the
intervention for subtypes of chronic
oro-­facial pain. BMS, burning mouth
syndrome; TMD, Temporomandibular
Disorders
AGGARWAL et al. | 7

F I G U R E 5 Box plots showing pain

severity scores measured using the
Brief Pain Inventory before and after
the intervention for subtypes of chronic
oro-­facial pain. BMS, burning mouth
syndrome; TMD, Temporomandibular
Disorders

F I G U R E 6 Box plots showing pain

interference scores measured using the
Brief Pain Inventory before and after
the intervention for subtypes of chronic
oro-­facial pain. BMS, burning mouth
syndrome; TMD, Temporomandibular
Disorders

4 | D I S CU S S I O N the patient manual was a ‘constant companion’ to self-­manage ex-

acerbations in symptoms particularly during the ongoing COVID-­19
To our knowledge, this is the first successful implementation of a pandemic. The intervention could also be delivered flexibly either
biopsychosocial intervention that targets chronic primary oro-­facial face-­to-­face or remotely by telephone—­the latter being particularly
pains and allows the patient to take control and self-­manage their useful during the current pandemic.
pain. The intervention had positive impact on pain intensity and The intervention has several strengths. First, we used an
quality of life measured using the Brief Pain Inventory subscales. evidence-­based approach.17 Second, the self-­management inter-
It also reduced the burden on healthcare services by reducing con- vention model (biopsychosocial) aligns with recommendations both
sultation rates within both medical and dental specialties and sub- within the United Kingdom (NICE guidance29) and internationally24
sequent costs to the healthcare service. Expert patient feedback for management of chronic primary oro-­facial pain including TMD
recommended use of the intervention at the outset and as a first-­line which advocate therapist-­guided self-­management approaches that
treatment. Patients in the Patient and Public Involvement (PPI) group combine a psychoeducational rationale with techniques drawn from
reported that the intervention could be tailored to their needs and cognitive-­behavioural therapy (CBT) and biofeedback for chronic
8 | AGGARWAL et al.

TA B L E 1 Patient Feedback from 3

Domains Corresponding feedback
PPI user group meetings identifying key
Personal Experiences • We have been grinding and clenching our teeth both during the day themes related to personal experiences
(AF, BF, CF, DF, EF,) and night since we were at least 15yrs of age (over 30 years ago). of current care pathways and benefits of
We have suffered from quite intense pain and at times have been using supported self-­management
left unable to eat solid foods at all. Hard or crunchy foods have
been a challenge for many years as the repetitive chewing can be
extremely painful in the jaw so we have tended to avoid these.
• We have had quite severe sleeping difficulties, waking up multiple
times throughout the night and feeling fatigued most days, even
after an apparent 8–­10 hours sleep!
• The inside of our mouth is often sore and bleeding where we
have bitten it with our grinding and this leads us to bite it again
aggravating the unusual jaw movement.
• No one can ‘see’ our pain so no one understands it and we have to
put up with it.
Experiences of the • Being ‘passed from pillar to post’ within primary and secondary
current healthcare care in search for a diagnosis and not receiving the appropriate care
system for their personal condition quickly enough, when the appropriate
resources are actually available.
• Often patients are not referred by their primary care practitioner
(GP or dentist) for pain management at time of diagnosis
• (BF) Receiving the wrong medication, that is given medication for
epilepsy which can have side effects.
• (CF) In her experience, some drugs prescribed can lead to reliance,
for example antidepressants, strong painkillers.
• Irreversible, invasive treatment still ongoing, that is teeth extraction
in some cases, where there is nothing wrong with the patient's
teeth but are being extracted due to the pain. This may also cause
long term damage (AF, CF, BF)
• BF stated that after 30 years of great discomfort at dental
appointments, an appointment was finally made for the hospital.
The hospital health professional, advised not to use her splint as it
was not working.
• Cost implications—­These should not be underestimated for
patients, they are paying out for years of treatment (both NHS and
private), that does not necessary help (ALL).
Experiences with • Members use it any stage of pain. CF when she has flare ups
the supported considers it a ‘constant companion’ more so during the COVID
self-­management lockdown.
intervention • BF has distributed it to friends and employers who, having read it
have understood her pain issues and shown empathy.
• Four members of the group, who have used the guidance would like
to see it available to patients who are in pain now. They recognise
that the guide is not a one-­off solution and patients will use the
techniques that work for them.
• CF had seen doctors and dentists interchangeably on a regular
basis, but found the guide was the most effective information for
her condition. She also shared how she went for a routine dental
check-­up recently, and the dentist said she has many patients with
the same problem; so CF shared the guide. The dentist said she had
problems with time allocation with patients but CF suggested she
give it to them and call them back at a later date.

primary oro-­facial pain29 and temporomandibular disorders. 24 Third, was acceptable to patients and could be feasibly delivered face-­to-­
we incorporated a theoretical (biopsychosocial) and methodological face or by telephone by healthcare workers from dental and psy-
framework19 in developing the intervention, which involved syn- chology backgrounds. The intervention theory, design, content and
thesis of evidence from both quantitative17 and qualitative7 studies evaluation are presented in the logic model in Figure 7. Through
20
and adaptation of an existing intervention, which had been shown PPI meetings, we captured actual patient experience as they trav-
to be cost-­effective for chronic widespread pain. We optimised the elled through their care pathway and this showed how patients are
design using a co-­production approach with patient and clinicians passed from pillar to post and undergo multiple invasive and irre-
and the findings of our pilot trial18 showed that the intervention versible treatments that are harmful—­f indings shared by previous
AGGARWAL et al. | 9

F I G U R E 7 Logic model showing the process of intervention development including the following: theoretical underpinnings,
methodological frameworks, process evaluation using co-­production and co-­design approaches; feasibility and pilot work; implementation
and future upscaling based on patient input and current care pathways

research. 5,7,13 Our intervention was seen as a first step by patients Our study was limited as there was no control group, which
to improving their care as they were able to tailor it to better meets would be only possible in a randomised controlled trial that would
their needs. They suggested prioritisation of early self-­management require substantial resources. In our pragmatic study, the best we
throughout chronic oro-­facial pain care pathways to prevent pain could achieve was using patients’ attendance and BPI scores be-
negatively impacting their lives. From a commissioning perspective, fore being referred to the clinic as a proxy for a control group and
our intervention not only has a sound evidence base but is safe and compare this to attendance and BPI scores after receiving the in-
efficient to deliver and positively impacts on the emotional experi- tervention. Whilst consultation data were available for all patients,
ence of delivering and receiving care. In addition, the intervention not everyone completed BPIs which was another limitation. BPIs
saves consultation costs although the cost savings of the inter- were incomplete as those who were discharged from the clinic at
vention (£293 per year per patient) are likely an underestimate as the last appointment were not required to attend again, and there-
we only included costs of face-­to-­face consultations (whereas the fore, if they did not return their BPIs after discharge, we could not
intervention can be delivered by telephone) and additional proce- get hold of the patients. Finally, the PPI groups by their very nature
dures such as botox, splints and surgery that were undertaken for were made up of those who wished to contribute to inform further
patients in other clinics prior to attending our pain clinic were not development of the intervention by sharing of their experiences
included. A telephone only service which has been the alternative of using it. This group did not include non-­responders for whom
during COVID would result in a consultation cost saving of £511 per the intervention may not have been effective. Previous research
patient per year (consultation savings £580 minus telephone costs has shown that non-­responders to psychosocial management of
25.24*2.73 = £68.90) based on a £25.24 tariff for a telephone con- TMD using cognitive behaviour therapy accounted for 16% of the
sultation. In addition, we did not include primary care costs (from sample and although they did not differ from treatment respond-
where patients were initially referred) as these were unavailable. ers on demographics or temporomandibular joint pathology, they
Whilst this affects the generalizability of the findings, the cost sav- reported more psychiatric symptoms, poorer coping, and higher
ings are likely to have been an underestimate as prior primary care levels of catastrophizing. 30 These patients may be vulnerable to
consultation and treatment costs would have increased consulta- seeking alternative treatments and unnecessary investigations that
tion costs pre-­intervention. not only burden healthcare services but more importantly expose
10 | AGGARWAL et al.

patients to potentially harmful treatment. Future research there- to acknowledge the support received from staff at Leeds Dental
fore needs to understand the long term needs of non-­responders Institute both in setting up and maintaining the facial pain clinical
so as to avoid referrals or exposure to ineffective treatments, un- service and also for helping with anoymising and extracting the data.
necessary investigations and/or potentially harmful medication (eg
opiates). Strategies to increase patient engagement in focus groups C O N FL I C T O F I N T E R E S T
particularly from non-­responders have been previously described There are no conflicts of interest to declare.
and need to be implemented in future research evaluating inter-
ventions such as ours. 31 Our subgroup analysis showed that the AU T H O R C O N T R I B U T I O N S
intervention was potentially more effective for TMD patients com- V.R. Aggarwal contributed to conception and design, intervention
pared with BMS patients. Apart from pain severity for BMS (which delivery, data acquisition and interpretation, drafting of the article
only reduced by 0.4 points on the BPI), all other values for pain and final approval of the version to be published. J. Wu contributed
severity and interference for TMD and pain interference for BMS to acquisition, statistical analysis, interpretation of data, drafting of
improved by greater than 1 point on the BPI which is deemed clin- the article and final approval of the version to be published. F. Fox
26
ically significant by IMMPACT. The lack of statistical significance contributed to acquisition, analysis (process mining), interpretation
is likely to be down to the small number of BMS patients (n=20). of data, drafting of the article and final approval of the version to
Previous studies have successfully implemented behavioural in- be published. D. Howdon contributed to economic analysis, inter-
terventions using trained dental nurses.32 Our intervention may be pretation of data, drafting of the article and final approval of the
suitable for such pathways as we have shown that it can be delivered version to be published. E. Guthrie contributed to conception study
by trained healthcare workers from a dental background18 and could design, drafting of the article and final approval of the version to be
fit the remit for extended roles (nurse practitioners/dental thera- published. A. Mighell contributed to conception, study design, clinic
pists) akin to those commonly used in medical care to manage long-­ supervision, drafting of the article and final approval of the version
33,34
term conditions with nurse-­led enablement of self-­management. to be published. All authors have read the article, discussed the re-
Future work therefore needs to investigate the cost-­effectiveness sults and commented on the manuscript.
(in a pragmatic randomised controlled trial) of our upscaling our inter-
vention to re-­configure current care pathways so that interventions PEER REVIEW
such as ours are accessible across healthcare services to improve The peer review history for this article is available at https://publo​
outcomes for patients with chronic primary oro-­facial pain.35 Future ns.com/publo​n/10.1111/joor.13229.
work also needs to explore mechanisms by which these interven-
tions brings about change and understand characteristics of patients DATA AVA I L A B I L I T Y S TAT E M E N T
who respond to such treatments as well as those who do not as non-­ The data that support the findings of this study are available on
responders may be particularly vulnerable to seeking further unnec- request from the corresponding author, [VRA]. The data are not
essary investigations and alternative (often harmful) treatments. publicly available due to NHS trust information governance and re-
strictions and their containing information that could compromise
the privacy of research participants.
5 | CO N C LU S I O N S
ORCID
We have implemented, for the first time, a biopsychosocial interven- Vishal R. Aggarwal https://orcid.org/0000-0003-0838-9682
tion that supports patients to self-­manage chronic primary oro-­facial
pain. The intervention had a positive impact on both health out- REFERENCES
comes (physical functioning, pain intensity and patient experience) 1. Aggarwal VR, McBeth J, Zakrzewska JM, Lunt M, Macfarlane GJ.
and non-­health outcomes (consultation rates and costs). Future work Are reports of mechanical dysfunction in chronic oro-­facial pain
related to somatisation? A population based study. Eur J Pain.
needs to upscale such interventions to explore whether early sup-
2008;12(4):501-­507.
ported self-­management can improve outcomes for these patients 2. Aggarwal VR, McBeth J, Lunt M, Zakrzewska J, Macfarlane GJ.
by re-­configuring current clinical pathways for optimal referral, tri- Epidemiology of chronic symptoms that are frequently unex-
age and management of chronic primary oro-­facial pain within pri- plained: do they share common associated factors? Int J Epidemiol.
2006;35(2):468-­476.
mary and secondary care services.
3. Macfarlane TV, Blinkhorn AS, Davies RM, Kincey J, Worthington
HV. Oro-­facial pain in the community: prevalence and associated
AC K N OW L E D G E M E N T S impact. Community Dent Oral Epidemiol. 2002b;30(1):52-­60.
We would like to thank Dr Elizabeth Bradley for her help with data 4. Breckons M, Bissett S, Exley C, Araujo-­Soares V, Durham J. Care
pathways in persistent orofacial pain: qualitative evidence from the
entry of BPI scores and transcription of PPI meetings. Thanks also
DEEP study. J Dent Res Clin Trans Res. 2017;2(1):48-­57.
goes to our patient user group who provided valuable feedback 5. Durham J, Exley C, Wassell R, Steele JG. ‘Management is a black
on their difficulties of accessing care and their experiences of art'–­professional ideologies with respect to temporomandibular
self-­management of chronic oro-­facial pain. Finally, we would like disorders. Br Dent J. 2007;202:E29-­E33.
AGGARWAL et al. | 11

6. Durham J, Shen J, Breckons M, et al. Healthcare cost and impact based intervention to manage Chronic Orofacial Pain. Br Dent J.
of persistent orofacial pain the deep study cohort. J Dent Res. 2016;220:459-­463.
2016;95(10):1147-­1154. 23. Lovell K, Richards D, Keeley P, Goldthorpe J, Aggarwal V. Self-­
7. Peters S, Goldthorpe J, McElroy C, et al. Managing chronic orofacial Management of Chronic Oro-­facial Pain including TMD. University
pain: A qualitative study of patients’, doctors’, and dentists’ experi- of Leeds 2019. https://licen​sing.leeds.ac.uk/produ​c t/self-­manag​
ences. Br J Health Psychol. 2015;20(4):777-­791. ement​-­of-­chron​ic-­orofa​cial-­pain-­inclu​ding-­tmd
8. Nicholas M, Vlaeyen J, Rief W, et al. The IASP classification of 24. National Academies of Sciences, Engineering, and Medicine.
chronic pain for ICD-­11. Pain. 2019;160(1):28-­37. Temporomandibular Disorders: Priorities for Research and Care.
9. International Classification of Orofacial Pain. Cephalalgia. Washington, DC: The National Academies Press. 2020. https://doi.
2020;40(2):129-­221. https://doi.org/10.1177/03331​02419​893823 org/10.17226/​25652
10. Aggarwal VR, Macfarlane GJ, Farragher T, McBeth J. Risk fac- 25. Tan G, Jensen MP, Thornby JI, Shanti BF. Validation of the brief pain
tors for the onset of chronic oro-­facial pain –­ results from the inventory for chronic nonmalignant pain. J Pain. 2004;5(2):133-­137.
Cheshire Oro-­facial Pain prospective population study. Pain. 26. Dworkin RH, Turk DC, Wyrwich KW, et al. Interpreting the clinical
2010;149(2):354-­359. importance of treatment outcomes in chronic pain clinical trials:
11. Koh H, Robinson PG. Occlusal adjustment for treating and pre- IMMPACT recommendations. J Pain. 2008;9:105-­121.
venting temporomandibular joint disorders. J Oral Rehabil. 27. Process mining for Professionals. https://fluxi​con.com/disco/
2004;31(4):287-­292. 28. NHS Data model and Dictionary. https://www.datad​ictio​nary.nhs.
12. Al-­Ani Z, Gray RJ, Davies SJ, Sloan P, Glenny AM. Stabilization splint uk/web_site_conte​n t/suppo​r ting_infor​m atio​n /main_speci​a lty_
therapy for the treatment of temporomandibular myofascial pain: a and_treat​ment_funct​ion_codes_table.asp?shown​av=1
systematic review. J Dent Educ. 2005;69(11):1242-­1250. 29. Chronic pain (primary and secondary) in over 16s: assessment of
13. Riley P, Glenny AM, Worthington H, Jacobsen E, Robertson C, all chronic pain and management of chronic primary pain. https://
Durham J, Davies S, Petersen H, Boyers D. Oral splints for patients www.nice.org.uk/guida​n ce/ng193/​c hapt​e r/Recom​m enda​t ions​
with temporomandibular disorders or bruxism: a systematic review #manag​ing-­chron​ic-­prima​r y-­pain accessed 04/5/2021
and economic evaluation. Final report submitted to NIHR Health 30. Litt MD, Porto FB. Determinants of pain treatment response and
Technology Assessment Programme (Project number: 16/146). nonresponse: identification of TMD patient subgroups. J Pain.
2018. 2013;14(11):1502-­1513.
14. Beecroft E, Durham J, Thomson P. Retrospective examination of 31. Vat LE, Finlay T, Jan Schuitmaker-­Warnaar T, et al. Evaluating the
the healthcare ‘journey’ of chronic orofacial pain patients referred “return on patient engagement initiatives” in medicines research
to oral and maxillofacial surgery. Br Dent J. 2013;214:E12. and development: A literature review. Health Expect. 2020;23:5-­18.
15. Gatchel RJ, Stowell AW, Wildenstein L, Riggs R, Ellis E III. Efficacy 32. Pine CM, Adair PM, Burnside G, et al. Dental RECUR Randomized
of an early intervention for patients with acute temporomandibular Trial to Prevent Caries Recurrence in Children. J Dent Res.
disorder-­related pain: a one-­year outcome study. J Am Dent Assoc. 2020;99(2):168-­174. https://doi.org/10.1177/00220​3 4519​886808
2006;137(3):339-­3 47. 33. Coventry P, Lovell K, Dickens C, et al. Integrated primary care
16. Breckons M, Shen J, Bunga J, Vale L, Durham J. DEEP Study: Indirect for patients with mental and physical multimorbidity: cluster ran-
and Out-­of-­pocket Costs of Persistent Orofacial Pain. J Dent Res. domised controlled trial of collaborative care for patients with de-
2018;97(11):1200-­1206. pression comorbid with diabetes or cardiovascular disease. BMJ.
17. Aggarwal VR, Fu Y, Main CJ, Wu J. The effectiveness of self-­ 2015;350:h638.
management interventions in adults with chronic orofacial pain: A 34. Kendall S, Wilson P, Procter S, et al. The nursing contribution to
systematic review, Meta-­analysis and Meta-­regression. Eur J Pain. chronic disease management: A whole systems approach. Hatfield,
2019;23:849-­865. UK: Queen's Printer and Controller of HMSO; 2010.
18. Goldthorpe J, Lovell K, Peters S, McGowan L, Aggarwal VR. A 35. Hurst L, Mahtani K, Pluddemann A, Lewis S, Harvey K, Briggs A,
randomised controlled pilot trial of a guided self-­help interven- Boylan A-­M, Bajwa R, Haire K, Entwistle A, Handa A, Heneghan
tion to manage Chronic Orofacial Pain. J Oral Fac Pain Headache. C. Defining Value-­based Healthcare in the NHS: CEBM report May
2017;31(1):61-­71. 2019. https://www.cebm.net/2019/04/defin​ing-­value​-­based​-­healt​
19. Craig P, Dieppe P, Macintyre S, Michie S, Nazareth I, Petticrew M. hcare​-­in-­the-­nhs/
Developing and evaluating complex interventions: the new Medical
Research Council guidance. BMJ. 2008;337:a1655.
20. McBeth J, Prescott G, Scotland G, et al. Cognitive behavior therapy,
How to cite this article: Aggarwal VR, Wu J, Fox F, Howden
exercise, or both for treating chronic widespread pain. Arch Intern
D, Guthrie E, Mighell A. Implementation of biopsychosocial
Med. 2012;172(1):48-­57.
21. Lovell K, Richards D, Keeley P, Goldthorpe J, Aggarwal V. Self-­ supported self-­management for chronic primary oro-­facial
Management of Chronic Oro-­facial Pain. University of Manchester pain including temporomandibular disorders: A theory,
2018. https://www.click​2go.umip.com/i/coa/chron​ic_orofa​cial_ person and evidence-­based approach. J Oral Rehabil.
pain_manual.html
2021;00:1–­11. https://doi.org/10.1111/joor.13229
22. Goldthorpe J, Peters S, Lovell K, McGowan L, Aggarwal V. I just
wanted someone to tell me it wasn't all in my mind and do some-
thing for me": Qualitative exploration of acceptability of a CBT