Diagnostic Criteria for Temporomandibular Disorders in Children and Adolescents: An International Delphi Study-Part 2-Development of Axis II PDF

Received: 11 April 2021 | Revised: 27 October 2021 | Accepted: 16 December 2021 DOI: 10.1111/joor.13301 ORIGINAL ARTICLE Diagnostic criteria for temporomandibular disorders in children and adolescents: An international Delphi study-­Part 2-­Development of Axis II Roberto Rongo1 |...

Received: 11 April 2021 | Revised: 27 October 2021 | Accepted: 16 December 2021 DOI: 10.1111/joor.13301 ORIGINAL ARTICLE Diagnostic criteria for temporomandibular disorders in children and adolescents: An international Delphi study-­Part 2-­Development of Axis II Roberto Rongo1 | EwaCarin Ekberg2 | Ing-­Marie Nilsson2,3 | Amal Al-­Khotani4,5 | 2,4,6 7,8 9 Per Alstergren | Paulo Cesar Rodrigues Conti | Justin Durham | Jean-­Paul Goulet10 | Christian Hirsch11 | Stanimira I. Kalaykova12 | Flavia P. Kapos13 | 14,15,16 17 18 2,4 Christopher D. King | Osamu Komiyama | Michail Koutris | Thomas List | Frank Lobbezoo18 | Richard Ohrbach19 | Tonya M. Palermo20,21 | Christopher C. Peck22 | Chris Penlington9 | Claudia Restrepo23 | Maria Joao Rodrigues24 | Sonia Sharma2,19 | Peter Svensson2,4,6,25 | Corine M. Visscher18 | Kerstin Wahlund26 | Ambrosina Michelotti1 1 Department of Neurosciences, Reproductive Sciences and Oral Sciences, School of Orthodontics, University of Naples Federico II, Naples, Italy 2 Department of Orofacial Pain and Jaw Function, Faculty of Odontology, Malmö University, Malmö, Sweden 3 Center for Oral Rehabilitation, FTV Östergötland, Norrköping, Sweden 4 Scandinavian Center for Orofacial Neurosciences, Jeddah, Sweden 5 East Jeddah Hospital, Ministry of Health, Jeddah, Saudi Arabia 6 Department of Dental Medicine, Karolinska Institute, Huddinge, Sweden 7 Department of Prosthodontics and Periodontology, Bauru School of Dentistry, University of São Paulo, Bauru, Brazil 8 Bauru Orofacial Pain Group, University of São Paulo, Bauru, Brazil 9 School of Dental Sciences, Newcastle University, Newcastle-­Upon-­Tyne, UK 10 Faculty of Dental Medicine, Laval University, Quebec City, Quebec, Canada 11 Clinic of Pediatric Dentistry, University of Leipzig, Leipzig, Germany 12 Department of Oral Function and Prosthetic Dentistry, College of Dental Sciences, Radboud University Medical Center, Nijmegen, The Netherlands 13 Department of Epidemiology, University of Washington, Seattle, Washington, USA 14 Division of Behavioral Medicine and Clinical Psychology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, USA 15 Center for Understanding Pediatric Pain (CUPP), Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, USA 16 Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio, USA 17 Division of Oral Function and Rehabilitation, Nihon University School of Dentistry at Matsudo, Matsudo, Japan 18 Department of Orofacial pain and Dysfunction, Academic Centre for Dentistry Amsterdam (ACTA), University of Amsterdam and Vrije Universiteit Amsterdam, Amsterdam, The Netherlands 19 Department of Oral Diagnostic Sciences, University at Buffalo, Buffalo, New York, USA 20 Department of Anesthesiology and Pain Medicine, University of Washington School of Medicine, Seattle, Washington, USA 21 Seattle Children's Research Institute, Seattle, Washington, USA 22 Faculty of Medicine and Health, University of Sydney, Westmead, New South Wales, Australia 23 CES-­LPH Research Group, Universidad CES, Medellin, Colombia 24 Institute for Occlusion and Orofacial Pain Faculty of Medicine, University of Coimbra, Coimbra, Portugal 25 Section for Orofacial Pain and Jaw Function, School of Dentistry and Oral Health, Aarhus, Denmark 26 Department of Stomatognathic Physiology, Kalmar County Hospital, Kalmar, Sweden J Oral Rehabil. 2022;49:541–552. wileyonlinelibrary.com/journal/joor© 2021 John Wiley & Sons Ltd | 541  | 13652842, 2022, 5, Downloaded from https://onlinelibrary.wiley.com/doi/10.1111/joor.13301 by Uni Federico Ii Di Napoli, Wiley Online Library on [11/07/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License 542 RONGO et al. Correspondence Roberto Rongo, Department of Abstract Neurosciences, Reproductive Sciences Background: Unlike the psychosocial assessment established for adults in the and Oral Sciences -­University of Naples “Federico II” Via Pansini, 5 -­80131 Naples, Diagnostic Criteria for Temporomandibular Disorders (DC/TMD), a standardised psy- Italy. chosocial assessment for children and adolescents with TMD complaints has not yet Emails: [email protected]; [email protected] been established. Objectives: To develop a new standardised instrument set to assess the psychosocial Funding information International Network for Orofacial pain functioning in children and adolescents by adapting the psychosocial status and pain-­ and Related disorders Methodology related disability (Axis II) of the adult DC/TMD and by including new instruments. (INfORM) Group; International Association for Dental Research Methods: A modified Delphi method was used to survey 23 international TMD experts and four international experts in pain-­related psychological factors for consensus re- garding assessment tools for psychosocial functioning and pain-­related disability in children and adolescents. The TMD experts reviewed 29 Axis II statements at round 1, 13 at round 2 and 2 at round 3. Agreement was set at 80% for first-­round consen- sus level and 70% for each of the second and third rounds. The psychological experts completed a complementary Delphi survey to reach a consensus on tools to use to assess more complex psychological domains in children and adolescents. For the psy- chological experts, the first round included 10 open-­ended questions on preferred screening tools for depression, anxiety, catastrophising, sleep problems and stress in children (ages 6–­9 years old) and adolescents (ages 10–­19 years old) as well as on other domains suggested for investigation. In the second round, the psychological experts received a 9-­item questionnaire to prioritise the suggested instruments from most to least recommended. Results: The TMD experts, after three Delphi rounds, reached consensus on the changes of DC/TMD to create a form to evaluate Axis II in children and adolescents with TMD complaints. The psychological experts added tools to assess depression and anxiety, sleep disorders, catastrophising, stress and resilience. Conclusion: Through international expert consensus, this study adapted Axis II of the adult DC/TMD to assess psychosocial functioning and pain-­related disability in chil- dren and adolescents. The adapted Axis II protocols will be validated in the target populations. 1 | BAC KG RO U N D to developing psychological symptoms.8 Many studies have found a higher prevalence of psychological and social disorders in TMD Temporomandibular disorders (TMDs) include pain and/or impaired patients, when compared to general populations.9,10 In addition, 1 function and are often associated with psychological factors. The psychological factors can influence patient's response to treatment, biopsychosocial model has been used to describe the complex na- including an impact on the disorder's prognosis.11,12 Hence, psycho- 2 ture of TMD along with its etiology. This model describes a dy- social assessment of patients with a TMD is a mandatory step for namic relationship between physiological, psychological and social the clinicians to fully appraise the global impact of the disorder and factors and thus underlines the importance of the patient's func- contribute to better clinical decision making.3 tional status.3 Hence, the aetiology of TMD is considered as mul- The Research Diagnostic Criteria for Temporomandibular tifactorial, and psychosocial factors can play a significant role in Disorders (RDC/TMD), published in 1992, introduced a standardised the onset and in persistence of TMD.4,5 For example, psychological evaluation protocol for TMD patients with a dual-­axis diagnostic symptoms, such as stress and anxiety, can induce parafunctional system based on the biopsychosocial model and included both a behaviours, which, in turn, can contribute to the development of physical diagnoses (Axis I) and a biobehavioural assessment of pain-­ TMD symptoms.6,7In addition, long-­term TMD symptoms contribute related disabilities (Axis II).13  | 13652842, 2022, 5, Downloaded from https://onlinelibrary.wiley.com/doi/10.1111/joor.13301 by Uni Federico Ii Di Napoli, Wiley Online Library on [11/07/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License RONGO et al. 543 Thereafter, the Diagnostic Criteria for TMD (DC/TMD) intro- at the General Session of the International Association of Dental duced new tools to assess psychosocial factors, providing short Research (IADR) in London in 2018. The organising committee was screening and comprehensive versions.14 designated in an INfORM workshop during the IADR in Seoul in The incidence of self-­reported TMD pain increases in ado- 2016. The members of the committee initiated the development of lescents with age.15,16 The prevalence of signs and symptoms in new set of instruments to evaluate TMD in children and adolescents cross-­s ectional studies also is positively associated with age in given their clinical and research expertise in this age group. Before children and adolescents.17–­19 Similarly, psychological and psycho- London 2018, the organising committee performed a literature re- social factors affect children's and adolescents’ well-­b eing, 20,21 view aimed to find references to be sent to all participants to the and a strong association between pain and psychological comor- workshop and then to all participants to the Delphi. The literature 20,22 bidities has been found in these populations. At present, in- review aimed to search instruments that evaluate psychological sufficient standardisation of tools and the limited data available constructs in children and adolescents. Fourteen TMD experts, (PA, have made it difficult to estimate to what extent children and ad- JD, ECE, SK, FK, MK, TL, AM, IMN, CCP, RO, CR, MJR, SS) and the olescents are affected by TMD, what impact these disorders have Delphi facilitator (RR) attending the workshop, created a list of key on the emotional and psychosocial domains, and what impact the issues about the applicability of DC/TMD to children and adoles- psychosocial factors have on TMDs. Indeed, most of the studies cents. Thereafter, the facilitator (RR), who did not participate in the have used either -­unvalidated assessment instruments or instru- online Delphi survey, developed a questionnaire with 29 statements ments validated only in adults. 23 based on the key issues relating to the Axis II assessment raised by To overcome this lack of a standardised set of instruments for the experts during the London workshop. The statements were better research, a Delphi study was planned to identify the most rel- about pain intensity/physical function, pain location, jaw function evant psychosocial domains in TMD and to find the best instruments limitation, depression/anxiety/physical symptoms, parafunction and to screen these domains in children and adolescents. Requirements other domains not included in the DC/TMD for adults. Twenty-­three for the ideal instruments included reliable and valid for the age range experts worldwide (Table 1) were invited by email to participate in as well as brief and easy to use. Preferably, the instruments would be the process. It includes the 14 experts who had participated in the already available in multiple languages. The Delphi method is often London workshop (excluding the facilitator RR), and 9 other experts used to achieve consensus among experts, and it is especially rec- (AA-­K , PCRC, JPG, CH, FL, OK, PS, CV, KW) who were identified ognised as valid and suitable for addressing highly complex prob- as having different competencies, such as oral surgery, orthodon- lems, such as the development of a new diagnostic protocol, and as tics, orofacial pain, paediatric dentistry, physiotherapy and epidemi- being flexible and adaptable to different research contexts and data ology. All 23 invited experts agreed to participate. A TMD expert collection. 24 was defined as a person with at least 5 years of experience in the In a previous Delphi study that adapted the DC/TMD Axis I for clinical management of TMD patients, experience in using the DC/ physical diagnosis, two different protocols were developed: one for TMD and having research interest in TMD as demonstrated by their children up to the age of 10 (from here forward, age 6–­9); and one publications in international peer-­reviewed journals. The experts for adolescents, from age 10–­19 years, according to the definition were asked to respond to each statement on a five-­point Likert scale of adolescent defined according to the World Health Organization ranging from ‘Strongly disagree’ to ‘Strongly agree’; further, they as age 10 years and older. 25,26 Similarly, the aim of this international could comment on each statement. Consensus to retain or reject a Delphi study was to reach consensus regarding the adaptation of the statement was reached when the percentage of experts answering, adult DC/TMD Axis II and the inclusion of new instruments to cre- ‘Agree or Strongly agree’ or the percentage of experts answering ate two new standardised set of instruments, one for children and ‘Disagree or Strongly disagree’ was equal to or higher than the se- one for adolescents, for the assessment of psychosocial functioning lected threshold for each round. The threshold level for consensus and pain-­related disability related to TMDs. The final aim of this new was set at 80% (18 out of 23 experts) for the first round and at 70% set of instruments is to help clinicians in diagnosis and treatment of (16 out of 23 experts) for the subsequent rounds. The survey was children and adolescents with TMDs and to help researchers to use created on Survey Monkey® (SVMK, San Mateo, CA, USA), an online standardised instruments during a research protocol. This set of in- survey development cloud-­based software. With the invitation to struments should be validated in the future research studies. participate in the survey, each expert received a letter of instruc- tions, a list of references and corresponding full-­text papers. The Delphi Technique is shown in Figure 1. After Round 1, the 2 | M ATE R I A L S A N D M E TH O DS facilitator and the organising committee (ECE, IMN, AM) analysed the results. Based on the experts' comments, the statements were The modified Delphi method was used to reach consensus among either rephrased or removed and/or new statements were added TMD experts on how to adapt to children and adolescents, the Axis when necessary, resulting in a total of 13 statements for Round 2. A II assessment for adults. Development of the adaptation of the DC/ similar process of analysing the experts' replies and comments was TMD started at a workshop promoted by the International Network conducted at the end of the second round, resulting in 2 statements for Orofacial Pain and Related Disorders Methodology (INfORM) for Round 3.  | 13652842, 2022, 5, Downloaded from https://onlinelibrary.wiley.com/doi/10.1111/joor.13301 by Uni Federico Ii Di Napoli, Wiley Online Library on [11/07/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License 544 RONGO et al. TA B L E 1 List of experts included in the Delphi study with area of expertise and affiliations Name Surname Area of expertise Affiliations TMD experts 1. Al-­Khotani Amal TMD/Orofacial Pain in children and adolescents; Ministry of Health (Saudi Arabia) Paediatric Dentistry; Paediatric Psychology, Epidemiology 2. Alstergren Per* TMD/Orofacial Pain; Rheumatological disease; Malmö University (Sweden) TMJ physiology 3. Durham Justin* TMD/Orofacial Pain; TMD pathophysiology; TMD Newcastle University (United Kingdom) treatment 4. Ekberg EwaCarin* TMD/Orofacial Pain; TMD pathophysiology; TMD Malmö University (Sweden) treatment 5. Goulet Jean-­Paul TMD/Orofacial Pain; TMD treatment; Oral disease Laval University (Canada) 6. Hirsch Christian Epidemiology; TMD/Orofacial Pain in children and University of Leipzig (Germany) adolescents; TMD treatment 7. Kalaykova Stanimira I* TMD/Orofacial pain; Dental Sleep Disorders; Radboud University Medical Centre (The Oral physiology Netherlands) 8. Kapos Flavia P* TMD/Orofacial Pain; University of Washington (United States of Epidemiology; America) TMD diagnosis 9. Komiyama Osamu TMD/Orofacial Pain; TMD pathophysiology; TMD Nihon University (Japan) treatment 10. Koutris Michail* TMD/Orofacial pain; Dental Sleep Disorders; ACTA (The Netherlands) TMD pathophysiology 11. List Thomas* TMD/Orofacial Pain; Oral physiology; TMD Malmö University (Sweden) treatment 12. Lobbezoo Frank TMD/Orofacial Pain; Oral Movement Disorders; ACTA (The Netherlands) Dental Sleep Disorders 13. Michelotti Ambrosina* TMD/Orofacial Pain; TMD treatment; University of Naples Federico II (Italy) Orthodontics 14. Nilsson Ing-­Marie* Epidemiology; TMD/Orofacial Pain in children and Malmö University (Sweden) adolescents; TMD treatment 15. Ohrbach Richard* TMD/Orofacial Pain; University of Buffalo (United States of Psychology; Epidemiology America) 16. Peck Christopher C.* TMD/Orofacial Pain; TMD treatment; University of Sydney (Australia) Neuroscience 17. Restrepo Claudia* TMD/Orofacial Pain in children and adolescents; Universidad CES (Colombia) Paediatric Dentistry; Dental Sleep Disorders 18. Rodrigues Conti Paulo TMD/Orofacial Pain; TMD diagnosis; TMD Universidade de São Paulo (Brazil) Cesar treatment 19. Rodrigues Maria Joao* TMD/Orofacial Pain; Dental Sleep Disorders; TMD University of Coimbra (Portugal) treatment 20. Sharma Sonia* TMD/Orofacial Pain; Epidemiology; Malmö University (Sweden) TMD diagnosis University of Buffalo (United States of America) 21. Svensson Peter TMD/Orofacial pain; Neuroscience; Oral Aarhus University (Denmark) physiology 22. Visscher Corine M. TMD/Orofacial pain; ACTA (The Netherlands) Physiotherapy; Dental Sleep Disorders 23. Wahlund Kerstin Epidemiology; TMD/Orofacial Pain in children and Malmö University (Sweden) adolescents; TMD treatment Psychological experts 24. Bryant Caroline Psychology in patients with chronic disease North Tyneside GH (United Kingdom)  | 13652842, 2022, 5, Downloaded from https://onlinelibrary.wiley.com/doi/10.1111/joor.13301 by Uni Federico Ii Di Napoli, Wiley Online Library on [11/07/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License RONGO et al. 545 TA B L E 1 (Continued) Name Surname Area of expertise Affiliations 25. King Christopher D. Pain in children and adolescents; NIH/NIDCR K99/ Cincinnati Children's Hospital (United States R00 in TMD of America) 26. Penlington Chris Pain Management Newcastle University (United Kingdom) Psychological treatment for Long Term Conditions 27. Palermo Tonya M. Paediatric chronic and recurrent pain, sleep University of Washington (United States of disorders in children America) Psychological treatment of paediatric chronic pain *Experts that participated in the workshop in London 2018. Literature review conducted by organizing committee Workshop London 2018 Working group 15 experts outline of areas of discussion. Item generation by the facilitator First round 23 invited experts 29 statements 10 agreements 7 excluded 1 added Second round 23 invited experts 13 statements First round 4 psychological experts 10 open-ended questions 6 agreements 3 excluded Third round 23 invited experts 2 2tatements Second round 3 psychological experts 9 questions 2 agreements F I G U R E 1 Flowchart of Delphi rounds At the end of each round, the TMD experts received a document Temporomandibular disorders experts were invited to sug- with the instructions for the subsequent round and a summary of gest experts in the field of child and adolescent psychology. They the previous round. provided to the organising committee a list of 4 international ex- In addition to the TMD experts survey, in October 2019 the fa- perts who were not members of the INfORM group (CB, CK, CP, cilitator (RR) conducted a complementary survey involving experts in TP). These experts in psychology were invited to participate in the child and adolescent psychology (Delphi Psych). The psychological ex- Delphi Psyc survey by email; they also received instructions asking perts were involved to compliment the TMD experts in important psy- them to indicate instruments to measure psychological domains in chological domains with no agreement or areas identified with limited children and adolescents. knowledge regarding instruments in the Delphi survey. The expert in Delphi Psych Round 1 included 10 open-­ended questions re- psychological domains was defined as internationally well-­recognised garding the tools they preferred to measure depression, anxiety, researcher and/or clinician, having research interest in psychological catastrophising, sleep problems and stress in children (ages 6–­9) and aspects related to pain in children and adolescents and/or experience in adolescents (ages 10–­19). These psychological experts were also in clinical setting in treating children or adolescents with pain. asked to suggest any other constructs that should be considered in a  | 13652842, 2022, 5, Downloaded from https://onlinelibrary.wiley.com/doi/10.1111/joor.13301 by Uni Federico Ii Di Napoli, Wiley Online Library on [11/07/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License 546 RONGO et al. standard assessment protocol for the psychosocial domains in young rephrased, and one was added. Thus, 13 statements were presented patients with TMD pain. in Delphi Round 2; of these, >70% agreement was reached for six In Delphi Psyc Round 2, the experts received a survey with out of 13 statements, five were dropped, and two were rephrased nine questions regarding the suggested instruments, a list of and proposed for Delphi Round 3. Finally, both statements proposed references and the corresponding full-­text papers, and a table in Round 3 reached >70% agreement. indicating the most important psychometric properties of the in- During the survey, consensus was reached for all domains but struments, which consisted of Cronbach alpha, instrument struc- one. For pain intensity/physical function, experts agreed to adapt ture, validity and sensitivity and specificity if available. In the first the Graded Chronic Pain Scale (GCPS) 2.0, rephrasing the ques- six statements of the survey, the psychological experts had to rank tions and using a Faces Pain Scale–­Revised (FPS-­R) rather than a the suggested screening tools for depression and anxiety, sleep numeric rating scale (NRS) for children only. For the pain location, problems and stress from most to least recommended, based on experts agreed that the figures in the pain drawing must be a child their opinion of the questionnaires (eg relevance, utility, psycho- in the child version and an adolescent in the adolescent version. metric properties). Ranking depends by scores that were calcu- Furthermore, the drawings should include pre-­selected areas for lated as the mean of the scores assigned by the experts (sum of both children and adolescents. In the DC/TMD for adults, the rec- scores divided by three). The maximum score was influenced by ommended instrument to assess jaw limitation is the Jaw Function the number of items, that is, if 5 items were included in the list, the Limitation Scale with 20 items for a comprehensive evaluation. In maximum score was 5. this Delphi the experts agreed to assess jaw limitation by means of The other three statements were on the assessment of resilience JFLS-­20 for adolescents and JFLS-­8 for children. To assess para- in children and adolescents. The analyses of the replies and of the function in children and adolescents, it was decided that a shorter data were performed blinded, that is, the organising committee (ECE, form of the Oral Behaviour Checklist (OBC) should be used for both IMN, AM) did not know the identities of the expert panel members. children and adolescents. Finally, regarding domains not initially in- Only the facilitator (RR) kept the code list to match responses to the cluded in the DC/TMD for adults, it was decided that catastrophis- experts' identities. At the end of each round, the external experts ing, sleep disorders and stress would be assessed. The TMD experts received a document with the instructions for the subsequent round agreed to assess catastrophising in children and adolescents using and a summary of the previous round. Final consensus was achieved the Pain Catastrophising Scale for Children (PCS-­C)27 and for parent in April 2020. The present manuscript was sent to all participating reporting on their own worry/catastrophising about their child's pain experts, who were invited to be co-­authors, and the manuscript was using the PCS-­Parents (PCS-­P). 28 However, the TMD experts did not finalised in March 2021. have recommendation for tools to screen for sleep disorders or for stress. Furthermore, TMD experts did not agree on the screening tools for depression and anxiety in either children or adolescents. 3 | R E S U LT S These and additional domains were also addressed by the psycho- logical experts. 3.1 | Delphi among TMD experts The results of the three rounds of the Delphi for TMD experts are 3.2 | Delphi among psychological experts shown in Table 2. The response rate was 100% with all the experts responding to all the statements in each of the three rounds regard- The results of the two rounds of the Delphi Psyc are shown in ing Axis II for children and adolescents. Table 3. Of the four experts invited, all replied to the statements In Delphi Round 1, there was >80% agreement among the ex- in the first round and three in the second round. In Delphi Psyc perts for 10 of the 29 statements. Based on the TMD experts' an- Round 1, these experts suggested five questionnaires to assess swers, of the remaining 19 statements, seven were dropped, 12 were depression and/or anxiety in children and eight questionnaires for TA B L E 2 Round of agreement Round 1 Round 2 Round 3 No agreement achievement Domain Agreement Agreement Agreement Agreement Pain intensity/Physical x function Pain location x Jaw limitation x Parafunction x Depression and anxiety x Other domains x  TA B L E 3 Suggested instruments and best instruments (in bold text) ranking in the Delphi for psychological experts Round 1–­2 RONGO et al. Children Adolescents Domain Instrument Score Instrument Score Depression and 1. Revised Child Anxiety and Depression Scale 4.33 (Maximum score 5) 1. Revised Child Anxiety and Depression Scale (RCADS) 6.00 (Maximum score 8) anxiety (RCADS-­SV) 2. Center for Epidemiology Studies-­Depression Child 4.00 2. Generalised anxiety Disorder (GAD−7) 5.67 (CES-­DC) 3. Patient-­Reported Outcomes Measurement 3.67 3. Patient-­Reported Outcomes Measurement Information 5.33 Information System (PROMIS) System (PROMIS) 4. Mood and Feelings Questionnaire (MFQ) 2.00 4. Screen for Child Anxiety Related Disorders (SCARED) 5.33 5. Clinical Outcomes in Routine Evaluation (CORE) 1.00 5. Center for Epidemiology Studies-­Depression Child 5.33 (CES-­DC) 6. Patient Health Questionnaire-­ADOLESCENTS (PHQ-­A) 4.67 7. Mood and Feelings Questionnaire (MFQ) 2.67 8. Clinical Outcomes in Routine Evaluation (CORE) 1.00 Catastrophising Pain Catastrophising Scale for Children (PCS-­C) Pain Catastrophising Scale for Children (PCS-­C) Pain Catastrophising Scale for Parents (PCS-­P) Pain Catastrophising Scale for Parents (PCS-­P)3 Sleep disorders 1. Children's Sleep Habits Questionnaire (CSHQ) 4.67 (Maximum score 5) 1. Adolescent Sleep-­Wake Scale (ASWS) 6.00 (Maximum score 7) 2. Children's Report of Sleep Patterns (CRSP) 3.00 2. Insomnia Severity Index (ISI) 5.67 3. Patient-­Reported Outcomes Measurement 2.67 3. Patient-­Reported Outcomes Measurement Information 3.67 Information System (PROMIS) System (PROMIS) Sleep-­Related Disturbance 4. Sleep-­Related Disturbance Patient-­Reported 2.33 4. Children's Report of Sleep Patterns (CRSP) 3.33 Outcomes Measurement Information System (PROMIS) Sleep-­Related Impairment 5. BEARS 2.33 5. Patient-­Reported Outcomes Measurement Information 3.33 System (PROMIS) Sleep-­Related Impairment 6. BEARS 3.00 7. Adolescent Sleep Hygiene Scale (ASHS) 2.67 Stress 1. Perceived Stress Scale for Children (PSS-­C) 2.67 (Maximum score 3) 1. Perceived Stress Scale for Children (PSS-­C) 3.67 (Maximum score 5) 2. Strengths and Difficulties Questionnaire (SDQ) 2.33 2. Adolescent Stress Questionnaire -­Shortened (ASQ) 3.33 3. Paediatric Pain Screening Tool (PPST) 1.00 3. Responses to Stress Questionnaire (RSQ) 3.33 4. Strengths and Difficulties Questionnaire (SDQ) 2.67 5. Paediatric Pain Screening Tool (PPST) 2 Resilience Adolescent Resilience Questionnaire (ARQ) | Scores were calculated as the mean of the scores assigned by the experts (sum of scores divided by three). The maximum score was influenced by the number of items, that is, if 5 items were included in the list, the maximum score was 5. 547 13652842, 2022, 5, Downloaded from https://onlinelibrary.wiley.com/doi/10.1111/joor.13301 by Uni Federico Ii Di Napoli, Wiley Online Library on [11/07/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License  | 13652842, 2022, 5, Downloaded from https://onlinelibrary.wiley.com/doi/10.1111/joor.13301 by Uni Federico Ii Di Napoli, Wiley Online Library on [11/07/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License 548 RONGO et al. adolescents. In Delphi Psyc Round 2, the experts rated the Revised 4.1 | Delphi among TMD experts 29 Child Anxiety and Depression Scale-­S hort Version (RCADS-­S V) as the best tool to assess depression and anxiety in both chil- The TMD experts agreed to use the GCPS 2.0 to assess pain inten- dren and adolescents. For sleep disorders, the experts suggested sity and related disability domain in both children and adolescents, five tools for children and seven tools for adolescents. In Delphi rephrasing the items. The GCPS is a valid, reliable tool for adults, Psyc Round 2, the experts rated the Children's Sleep Habits which was found to be strongly associated to the management of Questionnaire (CSHQ) 30 as the best tool to evaluate sleep disor- TMD pain.3,35 It presents eight questions, six of which include a 31 ders in children and the Adolescent Sleep-­Wake Scale (ASWS) as numeric rating scale (NRS) from 0 to 10. Considering that children the best for adolescents. Regarding stress, the psychological ex- under the age of 10 may have limited ordinal numerical competence, perts suggested three tools for children and five for adolescents. the experts agreed to substitute the NRS with the Faces Pain Scale-­ In Delphi Psyc Round2, the Perceived Stress Scale for Children Revised.36,37 Moreover, the experts agreed to rephrase GCPS 2.0 32 (PSS-­C) was deemed the best to screen for stress in both chil- items for both children and adolescents to take into consideration dren and adolescents. As the experts suggested one questionnaire activities typically performed by these two distinct age groups. to assess catastrophising in children and adolescents (PCS-­C) and The pain drawing is an important visual aid for patients and one for parents reporting catastrophising about their child's pain clinicians alike: the patient uses it to indicate the location and the (PCS-­P) in Delphi Psyc Round 1, consistent with the recommenda- spread of his/her pain on the face and neck, inside the mouth and tions of the TMD experts, this domain was not included in Delphi on other sites on the body. In the adult version, the image used is Psyc Round 2. Finally, in Delphi Psyc Round 1, experts suggested of a stylised adult man without hair14 with whom a child or young investigating resilience only in adolescents, suggesting in Round adolescent may not easily identify. Hence, the TMD experts agreed 2 the Adolescent Resilience Questionnaire (ARQ). 33 TMD experts to substitute the images on the pain drawing with images of a child accepted the instruments suggested by the experts in psychology. for the child version and of an adolescent for the adolescent version. All the authors participated to in the correction and approved the Still, it could be argued that a ‘typical’ adolescent face is not easy manuscript. to portray, given the ongoing changes occurring during this growth period. The experts agreed to include pre-­selected areas on the pain drawing to facilitate locating and reporting the painful areas. The 4 | DISCUSSION advantage of using pre-­selected areas is that it could make the tool more reliable and easier to use on electronic devices; increasing the This Delphi study established a new set of instruments by adapting ‘standardisation’ of this tool, that could be useful in research studies. the existing DC/TMD for adults and including new constructs and The disadvantages could be the limited freedom in selecting areas questionnaires to assess psychosocial status in children and adoles- that are painful. cents with TMD complaints. Temporomandibular disorders can provoke alterations and lim- This Delphi study was designed to create expert consensus in itations in normal jaw function. The DC/TMD for adults includes standardising evaluation measures for Axis II in the assessment of two instruments to assess patients' self-­reported jaw function TMD in children and adolescents. The final aim of this new set of limitation: the JFLS-­20 and the JFLS-­8.38 The JFLS-­20, that is, the instruments is to support clinicians and to provide researchers with complete form of the questionnaire, assesses three different con- standardised instruments to be used in research protocols. structs related to jaw function—­mastication (items 1–­6), vertical jaw The Delphi group consisted of 23 international experts from sev- mobility (items 7–­10) and verbal and non-­verbal expression (items eral countries who routinely work with the DC/TMD. This was pos- 13–­20)—­and global functional limitation. The JFLS-­8, instead, is sible thanks to the dissemination of the DC/TMD for adults, which a short version that assesses global functional limitation using a has already been translated into almost 20 languages.34 limited number of items from the three constructs. The TMD ex- Since psychosocial functioning is evaluated in Axis II, an addi- perts agreed to assess jaw function limitation using the JFLS-­20 in tional Delphi survey was organised for experts in psychosocial disci- adolescents and the JFLS-­8 in children. Both questionnaires will be plines whose task was to reach an agreement on the recommended adapted and assessed for content validity in both age groups. The questionnaires to measure some domains of Axis II. The psycholog- experts agreed to use the JFLS-­8 in children because of its brevity ical experts contributed to broadening and updating knowledge on and because the concepts used in the questionnaire, such as kissing, the questionnaires used to screen and to investigate the psychoso- yawning, chewing and talking are easy to understand.38 In children, cial functioning of children and adolescents with pain. Further, the the FPS-­R will be tested to replace the NRS of the JFLS-­8. psychological experts introduced new tools and new domains in Axis The role of parafunction in TMD onset, duration and manage- II for this population. ment is still unclear. The OPPERA study suggested a strong asso- At the end of these two Delphi processes, the experts identi- ciation between self-­reports of jaw parafunctions and TMD onset fied a set of instruments enabling the creation of a DC/TMD Axis in adults.39 Perrotta et al.,18 de Oliveira Reis et al.,40 and Fernandes II for children and adolescents, that will be validated in the future et al.41 found an association between awake and sleep bruxism and studies. TMD pain in children and adolescents as well. The TMD experts  | 13652842, 2022, 5, Downloaded from https://onlinelibrary.wiley.com/doi/10.1111/joor.13301 by Uni Federico Ii Di Napoli, Wiley Online Library on [11/07/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License RONGO et al. 549 agreed to assess the frequency of oral behaviours in children and psychological experts agreed on using the Revised Child Anxiety and 42 adolescents by means of the Oral Behaviours Checklist (OBC), a Depression Scale-­Short Version (RCADS-­SV) to assess depression 21-­item self-­report that uses a 5-­point ordinal scale. However, as the and anxiety in both children and adolescents. The RCADS-­SV, the TMD experts considered the OBC too long to be used in children and short version of the RCADS, consists of 25 items―10 for major de- adolescents, they suggested creating a short version, based on stud- pressive disorders and 15 for anxiety. This widely used instrument 43 44 ies conducted by Michelotti et al., Cioffi et al. and Donnarumma has good psychometric properties and is validated in the population et al.45 on a 6-­item OBC that still must be validated in children and ages 7–­18 years. 29,55 adolescents. To assess sleep disorders, the psychological experts suggested The TMD experts agreed to add other constructs to Axis II a questionnaire for children and another one for adolescents. The screening, such as catastrophising, sleep disorders and stress. Children's Sleep Habits Questionnaire (CSHQ) is a 45-­item parent Since the publication of the DC/TMD for adults in 2014, several questionnaire that screens for both behaviour-­based and medically studies indicate that other factors that influence onset, chronifi- based sleep problems. This questionnaire has an acceptable inter- cation and therapy outcomes of TMD. Pain catastrophising is ‘an nal consistency and acceptable reliability and is usually used in the exaggerated negative “mental set” brought to bear during actual population ages 4–­10 years.30 This widespread tool has been used in 46 or anticipated painful experience’ ; it is associated with poor more than 600 published studies. prognosis, possible pain persistence and poor patient treatment For adolescents, instead, the psychological experts suggested compliance in adults with several pain syndromes,47 including the Adolescent Sleep-­Wake Scale (ASWS) to assess sleep quality TMD.48,49 Pain catastrophising has also been investigated in chil- in youth ages 12–­18 years. The ASWS is a 28-­item questionnaire dren with chronic pain, with a positive association found between with a 6-­point scale (‘always’, ‘frequently-­if not always’, ‘quite often’, higher catastrophising and increased pain intensity, increased ‘sometimes’, ‘once in a while’, and ‘never’) for which overall internal disability, increased anxiety and depression. 50 Sleep quality has consistency has been found to be good for the total scale.31 Essner been associated with TMD incidence in adults. 39 Similarly, several et al.56 developed a short ASWS-­10 scale. studies on children and adolescents have reported that sleep has To screen for stress, the psychological experts suggested as in- a causal role in the chronification of pain and in the worsening strument useful for both children and adolescents, the Perceived of psychological symptoms. 51,52 Patients have indicated stress as Stress Scale -­ Children (PSS-­C),32 a 14-­item instrument that can be a factor that initiates, exacerbates and perpetuates their pain. 6 used in children ages 5–­18 years. Likewise, perceived stress is associated with increased pain in- Regarding catastrophising, the psychological experts suggested tensity in adolescents with musculoskeletal pain. 53 Hence, the the same questionnaires in the first round as those suggested by the experts in our study agreed on the need to search for new tools TMD experts for parents (PCS-­P) and for children and adolescents to screen for these three constructs in children and adolescents. (PCS-­C). The TMD experts reached an agreement on using the PCS-­C and Finally, the psychological experts suggested a further domain to the PCS-­P to assess catastrophising. PCS-­C is a 13-­item question- be assessed only in adolescents: resilience. Resilience has been de- naire for subjects between the ages of 8 and 16 years, while the fined as positive developmental outcomes in the face of adversity PCS-­P is a parent-­reported measure to describe the parent's own or stress,33 it is as an emerging area of study in paediatric chronic catastrophic thinking about their child's pain; it is indicated for in- pain and no studies have evaluated this domain in youth with TMD. dividuals between the ages of 9 and 16. Understanding parents' The Adolescent Resilience Questionnaire (ARQ) is an 88-­item ques- catastrophic thinking and behaviour has been important in other tionnaire with five scales in the self-­domain that assess confidence, paediatric chronic pain conditions where parent and family fac- emotional insight, negative cognition, social skills and empathy/tol- tors have been found to relate to child's pain, disability and school erance, and two scales in the family and in the peer domains that attendance. 54 To identify assessment tools for sleep disorders, assess connectedness and availability. The ARQ also includes two stress, depression, anxiety and physical symptoms, the experts scales in the school domain—­supportive environment and connect- decided to create a parallel Delphi for psychological experts. edness―and one scale in the community domain—­connectedness. This instrument can identify those adolescents who are positively engaged with their families, peers, school and environment, who 4.2 | Delphi among psychological experts show more resilient behaviour in the face of adversity, as well as those adolescents with more negative or poor engagement who may A separate Delphi was created to involve experts in the field of psy- be more vulnerable in situations of adversity. This instrument is rec- chology, with better knowledge of measures useful to investigate ommended initially in the research setting to determine how it might the psychological aspects related to pain in children and adoles- be associated with onset, duration and management of TMD. There cents. In Delphi Psyc Round 1, the experts suggested several instru- has to date been little if any research into positive attributes that ments, based on their experience and their knowledge. In Delphi may act as protective factors in this population. In order to be of Psyc Round 2, all the suggested instruments were ranked, and the use clinically, it would be helpful if a shorter questionnaire for resil- questionnaire that had the highest score was included in Axis II. The ience could be developed and validated. The psychological experts  | 13652842, 2022, 5, Downloaded from https://onlinelibrary.wiley.com/doi/10.1111/joor.13301 by Uni Federico Ii Di Napoli, Wiley Online Library on [11/07/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License 550 RONGO et al. included in this survey were all from North America and Europe, and C O N FL I C T O F I N T E R E S T S this may represent a limitation. The authors declare that they have no conflict of interest. Developing a unique standardised set of tools to assess the psy- chosocial functioning and pain-­related disability of young patients AU T H O R C O N T R I B U T I O N S with TMD in research and clinical settings will be a breakthrough RR, ECE, IMN, AM Conception and design of study;RR, ECE, IMN, in the orofacial pain field. The association between musculoskele- AM Acquisition of data; RR, ECE, IMN, AM Data analysis and/orin- tal pain and psychological factors is well known. Sleep disorders, terpretation; AA-­K , PA, PCRC, JD, ECE, JPG, CH, SK, FK, CK, OK, catastrophising and stress are associated with pain intensity, pain MK, TL, FL, AM,IMN, RO, TP, CCP, CP, CR, MJR, RR, SS, PS, CMV, disability and pain persistence; depression and quality of life have KW Drafting of manuscriptand/or critical revision AA-­K , PA, PCRC, been found to be associated with pain developing from acute to JD, ECE, JPG, CH, SK, FK, CK, OK, MK,TL, FL, AM, IMN, RO, TP, persistent.57 Some studies have evaluated the association between CCP, CP, CR, MJR, RR, SS, PS, CMV, KW Approval offinal version of painful TMD and depression or anxiety using the RDC/TMD20,22,23,58 manuscript for the clinical examination but have used different questionnaires for psychological assessment, thereby limiting the possibility of PEER REVIEW comparisons. Having a standardised protocol to assess Axis I and The peer review history for this article is available at https://publo​ Axis II would improve the quality of the research as well as clinicians' ns.com/publo​n/10.1111/joor.13301. ability to detect children and adolescents at higher risk of developing long-­lasting pain. The need for tools to assess TMD in children has DATA AVA I L A B I L I T Y S TAT E M E N T been clear since the publication of at least two previous studies.59,60 The data that support the findings of this study are available from However, both analysed only DC/TMD Axis I, without including any the corresponding author [RR], upon reasonable request. Axis II assessments. In our Delphi studies, TMD experts developed and adapted in- ORCID struments to assess Axis I26 of the DC/TMD in children and ado- Roberto Rongo https://orcid.org/0000-0002-9741-794X lescents and, with the support of psychological experts, selected Ing-­Marie Nilsson https://orcid.org/0000-0002-0550-8925 instruments for Axis II evaluation. The proposed changes of the DC/ Amal Al-­Khotani https://orcid.org/0000-0001-7168-9835 TMD will be assessed for validity and reliability. The TMD experts Paulo Cesar Rodrigues Conti https://orcid. were recruited from different countries including Europe, North org/0000-0003-0413-4658 and South America, Asia and Oceania while the psychological ex- Flavia P. Kapos https://orcid.org/0000-0002-6224-273X perts were recruited from North America and Europe. This might Frank Lobbezoo https://orcid.org/0000-0001-9877-7640 represent a limitation, because of sociocultural background. Future Richard Ohrbach https://orcid.org/0000-0002-9266-9734 research with broader global representation may help to validate, Claudia Restrepo https://orcid.org/0000-0002-0695-7562 improve and adapt assessment tools to reflect different sociocul- Sonia Sharma https://orcid.org/0000-0002-1887-7420 tural contexts’ needs and experiences. Corine M. Visscher https://orcid.org/0000-0002-4448-6781 REFERENCES 5 | CO N C LU S I O N S 1. Okeson J. Management of Temporomandibular Disorders and Occlusion, 7th ed. Mosby; 2013. 2. Suvinen TI, Reade PC, Kemppainen P, Könönen M, Dworkin SF. 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