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Received: 25 January 2021 | Accepted: 25 February 2021 DOI: 10.1111/joor.13175 ORIGINAL ARTICLE Diagnostic criteria for temporomandibular disorders (DC/TMD) for children and adolescents: An international Delphi study—­ Part 1-­Development of Axis I Roberto Rongo1 | EwaCarin Ekber...

Received: 25 January 2021 | Accepted: 25 February 2021 DOI: 10.1111/joor.13175 ORIGINAL ARTICLE Diagnostic criteria for temporomandibular disorders (DC/TMD) for children and adolescents: An international Delphi study—­ Part 1-­Development of Axis I Roberto Rongo1 | EwaCarin Ekberg2 | Ing-­Marie Nilsson2,3 | Amal Al-­Khotani4,5 | Per Alstergren2,4,6 | Paulo Cesar Rodrigues Conti7,8 | Justin Durham9,10 | Jean-­Paul Goulet11 | Christian Hirsch12 | Stanimira I. Kalaykova13 | Flavia P. Kapos14 | Osamu Komiyama15 | Michail Koutris16 | Thomas List2,4 | Frank Lobbezoo16 | 17 18 19 Richard Ohrbach | Christopher C. Peck | Claudia Restrepo | 20 2,17 2,4,6,21 Maria Joao Rodrigues | Sonia Sharma | Peter Svensson | 16 22 1 Corine M. Visscher | Kerstin Wahlund | Ambra Michelotti 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, 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 Newcastle Upon Tyne Hospitals NHS Foundation Trust, Newcastle Upon Tyne, UK 11 Faculty of Dental Medicine, Laval University, Quebec, QC, Canada 12 Clinic of Pediatric Dentistry, University of Leipzig, Leipzig, Germany 13 Department of Oral Function and Prosthetic Dentistry, College of Dental Sciences, Radboud University Medical Center, Nijmegen, The Netherlands 14 Department of Epidemiology, University of Washington, Seattle, WA, USA 15 Division of Oral Function and Rehabilitation, Nihon University School of Dentistry at Matsudo, Matsudo, Japan 16 Department of Orofacial pain and Dysfunction, Academic Centre for Dentistry Amsterdam (ACTA, University of Amsterdam and Vrije Universiteit Amsterdam, Amsterdam, The Netherlands 17 Department of Oral Diagnostic Sciences, University at Buffalo, Buffalo, NY, USA 18 Faculty of Medicine and Health, The University of Sydney, Sydney, NSW, Australia 19 CES-­LPH Research Group, Universidad CES, Medellin, Colombia 20 Institute for Occlusion and Orofacial Pain Faculty of Medicine, University of Coimbra, Coimbra, Portugal 21 Section of Orofacial Pain and Jaw Function, School of Dentistry and Oral Health, Aarhus, Denmark 22 Department of Stomatognathic Physiology, Kalmar County Hospital, Kalmar, Sweden This is an open access article under the terms of the Creative Commons Attribution License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited. © 2021 2021 The Authors. Journal of Oral Rehabilitation published by John Wiley & Sons Ltd 836 |  wileyonlinelibrary.com/journal/joor J Oral Rehabil. 2021;48:836–845. RONGO et al. | 837 Correspondence Roberto Rongo, Department of Abstract Neuroscience, Reproductive Sciences Background: Since in children and adolescence prevalence is assessed mainly on self-­ and Oral Sciences –­University of Naples ‘Federico II’, Via Pansini 5, 80131 Naples, reported or proxy-­reported signs and symptoms; there is a need to develop a more Italy. comprehensive standardised process for the collection of clinical information and the Emails: [email protected] and [email protected] diagnosis of TMD in these populations. Objective: To develop new instruments and to adapt the diagnostic criteria for tem- poromandibular disorders (DC/TMD) for the evaluation of TMD in children and adolescents. Method: A modified Delphi method was used to seek international consensus among TMD experts. Fourteen clinicians and researchers in the field of oro-­facial pain and TMD worldwide were invited to participate in a workshop initiated by the International Network for Orofacial Pain and Related Disorders Methodology (INfORM scientific network) at the General Session of the International Association for Dental Research (IADR, London 2018), as the first step in the Delphi process. Participants discussed the protocols required to make physical diagnoses included in the Axis I of the DC/ TMD. Thereafter, nine experts in the field were added, and the first Delphi round was created. This survey included 60 statements for Axis I, and the experts were asked to respond to each statement on a five-­item Likert scale ranging from ‘Strongly disagree’ to ‘Strongly agree’. Consensus level was set at 80% agreement for the first round, and at 70% for the next. Results: After three rounds of the Delphi process, a consensus among TMD experts was achieved and two adapted DC/TMD protocols for Axis I physical diagnoses for children and adolescents were developed. Conclusion: Through international consensus among TMD experts, this study adapted the Axis I of the DC/TMD for use in evaluating TMD in children and adolescents. KEYWORDS adolescents, children, Delphi study, diagnostic criteria, temporomandibular disorders 1 | I NTRO D U C TI O N for females.7,8 However, in children under the age of 10 prevalence is assessed mainly on self-­reported or proxy-­reported signs and symp- Temporomandibular disorders (TMDs) are a group of musculoskel- toms; hence, there is a need to develop a more comprehensive stan- etal disorders that involve the temporomandibular joints (TMJs), dardised process for the collection of clinical information and the masticatory muscles and associated tissues or structures.1 TMDs diagnosis of TMD in children and adolescents, so that reliability and are a significant public health problem with a prevalence in adults validity can be assessed and improved for this population.16 of 5%–­30% according to different pathologies, different age range Since 2014, the international standard for the assessment of and different assessment forms. 2–­4 Reported TMD prevalence in TMDs is the Diagnostic Criteria for TMD (DC/TMD).1 The DC/TMD children and adolescents varies widely in the literature from 4.2% consists of two axes and their respective instruments: Axis I for to 68%, depending on population and method of assessment.5–­9 physical diagnoses and Axis II for assessment of psychosocial sta- Painful and dysfunctional TMD might be associated with emotional tus and pain-­related disability. The DC/TMD is validated for several stress, depression, sleep and hormonal disturbances, and functional diagnoses as based on a standardised assessment protocol including complications.10–­12 In turn, the patient's daily life will be adversely history and clinical examination. A diagnostic algorithm utilising both affected by the presence of a TMD; in addition, TMDs result in in- history and clinical data permits to have very high sensitivity and creased pursuit of medical care which consumes both time and specificity for some TMD subgroups and consequently excellent di- money.13 TMDs are a considerable health problem in children and agnostic accuracy for TMD in adults.1 adolescents; several studies consistently show that TMD prevalence The DC/TMD is validated for individuals who are 18 or more years increases with age from childhood to adolescence,7–­9,14,15 especially of age; consequently, using the DC/TMD in children and adolescents 838 | 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 1. Al-­Khotani Amal TMD/Oro-­facial Pain in children and adolescents; Paediatric Ministry of Health (Saudi Arabia) Dentistry; Paediatric Psychology, Epidemiology 2. Alstergren Pera TMD/Oro-­facial Pain; Rheumatological disease; TMJ Malmö University (Sweden) physiology 3. Durham Justina TMD/Oro-­facial Pain; TMD pathophysiology; TMD treatment Newcastle University (United Kingdom) 4. Ekberg EwaCarina TMD/Oro-­facial Pain; TMD pathophysiology; TMD treatment Malmö University (Sweden) 5. Goulet Jean-­Paul TMD/Oro-­facial Pain; TMD treatment; Oral disease Laval University (Canada) 6. Hirsch Christian Epidemiology; TMD/Oro-­facial Pain in children and University of Leipzig (Germany) adolescents; TMD treatment 7. Kalaykova Stanimira I.a TMD/Oro-­facial pain; Dental Sleep Disorders; Oral physiology Radboud University Medical Centre (The Netherlands) 8. Kapos Flavia P.a TMD/Oro-­facial Pain; Epidemiology; TMD diagnosis University of Washington (United States of America) 9. Komiyama Osamu TMD/Oro-­facial Pain; TMD pathophysiology; TMD treatment Nihon University (Japan) 10. Koutris Michaila TMD/Oro-­facial pain; Dental Sleep Disorders; TMD ACTA (The Netherlands) pathophysiology 11. List Thomasa TMD/Oro-­facial Pain; Oral physiology; TMD treatment Malmö University (Sweden) 12. Lobbezoo Frank TMD/Oro-­facial Pain; Oral Movement Disorders; Dental Sleep ACTA (The Netherlands) Disorders 13. Michelotti Ambraa TMD/Oro-­facial Pain; TMD treatment; Orthodontics University of Naples Federico II (Italy) 14. Nilsson Ing-­Mariea Epidemiology; TMD/Oro-­facial Pain in children and Malmö University (Sweden) adolescents; TMD treatment 15. Ohrbach Richarda TMD/Oro-­facial Pain; Psychology; Epidemiology University of Buffalo (United States of America) 16. Peck Christopher C.a TMD/Oro-­facial Pain; TMD treatment; Neuroscience University of Sydney (Australia) a 17. Restrepo Claudia TMD/Oro-­facial Pain in children and adolescents; Paediatric Universidad CES (Colombia) Dentistry; Dental Sleep Disorders 18. Rodrigues Conti Paulo Cesar TMD/Oro-­facial Pain; TMD diagnosis; TMD treatment Universidade de São Paulo (Brazil) 19. Rodrigues Maria Joaoa TMD/Oro-­facial Pain; Dental Sleep Disorders; TMD treatment University of Coimbra (Portugal) 20. Sharma Soniaa TMD/Oro-­facial Pain; Epidemiology; TMD diagnosis University of Buffalo (United States of America) 21. Svensson Peter TMD/Oro-­facial pain; Neuroscience; Oral physiology Aarhus University (Denmark) 22. Visscher Corine M. TMD/Oro-­facial pain; Physiotherapy; Dental Sleep Disorders ACTA (The Netherlands) 23. Wahlund Kerstin Epidemiology; TMD/Oro-­facial Pain in children and Malmö University (Sweden) adolescents; TMD treatment a Experts that participated in the workshop in London 2018. requires a form of adaptation for each age group.1 Adaptation of the an effective method moving towards testable hypotheses.19 One of DC/TMD includes (1) a separate language review for both question- the methods used for decision-­making among experts is the Delphi naires and clinical examination, due to the difference in understand- method. This method includes a series of questions and statements ing and speaking skills between adults, adolescents and children,17 that are regrouped in different ‘rounds’ and thereby presents several and (2) modified protocols for clinical assessment. Adaptation and advantages over other consensus techniques: it is anonymous and assessment of content and construct validity of the Spanish transla- there is less possibility that some experts may influence the opinions of tion of the DC/TMD Axis I were performed in 7-­ to 11-­year-­old chil- other experts as they might in a face-­to-­face setting.20 Moreover, as it dren in Colombia, finding high internal consistency (.72 ≤ Cronbach's is usually performed online, experts from different geographic regions 18 alpha ≤.94). can easily be included. The Delphi method is well recognised as legiti- In the development or adaptation of diagnostic systems for which mate and suitable for addressing highly complex problems, such as the high-­quality evidence is not yet available, relying on experience of development of a new diagnostic instrument, and as being flexible and international experts in the field to achieve consensus on a topic is adaptable to different research contexts and data collection.21 RONGO et al. | 839 FIGURE 1 Flow chart of Delphi rounds The aim of this study was to develop new instruments to diag- demographics, screening, health, Symptom Questionnaire, clinical ex- nose TMDs in children and in adolescents by the adaptation of DC/ amination, imaging, and diagnosis as presented in the DC/TMD. TMD Axis I through an international Delphi study with a consensus Twenty-­three experts worldwide (Table 1) were invited by e-mail among TMD experts. This paper is focused on the Delphi process re- to participate in the Delphi process; this included 14 experts who lated to the DC/TMD Axis I, while the Delphi process related to the had previously participated in the workshop in London (exclud- DC/TMD Axis II and the full examination protocols of the DC/TMD ing the facilitator RR) and a further 9 experts who were identified for children and adolescents will be described in future publications. among different competences, such as surgeons, orthodontists, oro-­facial pain specialists, paediatric dentists, physiotherapists, psy- chologists and epidemiologists; 100% of the invited experts agreed 2 | M ATE R I A L S A N D M E TH O DS to participate. An expert was defined as a person with at least five years of experience in the clinical management of TMD patients, The modified Delphi method was used to seek international consen- experience in using the DC/TMD and research interest in TMDs sus for Axis I assessment among TMD experts.22 Development of based on publications in international peer-­reviewed journals. The the adaptations of the DC/TMD started at a workshop promoted by experts were asked to answer each statement on a five-­item Likert the International Network for Orofacial Pain and Related Disorders scale ranging from ‘Strongly disagree’ to ‘Strongly agree’. In addition, Methodology (INfORM) at the General Session of the International comments could be provided for each statement. Agreement on Association of Dental Research in London in 2018. Fourteen TMD ex- each statement was reached if the sum of experts replying ‘Agree or perts (RO, SS, FK, CR, MJR, JD, MK, SK, AM, TL, PA, ECE, IMN, CP) and Strongly agree’ or the sum of experts replying ‘Disagree or Strongly the Delphi facilitator (RR) participated in the meeting and created a list disagree’ was equal to or higher than the selected threshold for each of key issues, related to the applicability of DC/TMD for children and round. Threshold level for consensus was set at 80% agreement (18 adolescents. After this workshop, the facilitator (RR), who did not par- out of 23) for the first round and at 70% (16 out of 23) for the next ticipate in the online Delphi survey, constructed a survey of 60 state- rounds. 23,24 The Survey Monkey® cloud-­based software (SVMK) ments based on the key issues pertaining to physical diagnoses (Axis I) was used to develop the online survey. Together with the invitation as outlined by the experts. Each of these statements was subsequently to participate in the survey, each expert received a letter of instruc- assessed during the Delphi online survey. The statements addressed tions and a list of references with full-­text versions of all the papers. 840 | RONGO et al. The Delphi process is shown in Figure 1; after Delphi round-­1, TA B L E 2 Round of agreement achievement the facilitator and the organising committee (ECE, IMN, AM) eval- Round Round Round No uated the results. Based on comments from the experts, existing 1 2 3 agreement statements were either rephrased or removed or new statements Structure were added, resulting in a total of 26 statements for Delphi round-­2. Age x A similar process of evaluating the experts’ replies and comments Short and x was used at the second round which led to 15 statements for Delphi Complete Forms round-­3. Evaluation of the replies and data analysis was performed History blinded; that is, the organising committee (ECE, IMN, AM) did not Screening x know the experts’ panels identities. At the end of each round, the ex- Demographics, x perts received a document with the instructions for the next round health and a summary of the previous round's evaluation. Only the facilita- questionnaire, tor (RR) kept the code list to match responses to the experts’ identi- Symptom ties. Final consensus was achieved in November 2019. The present Questionnaire manuscript was sent to all the TMD experts who were invited to be Clinical examination co-­authors, and the manuscript was finalised in September 2020. Mandatory x commands Jaw opening x 3 | R E S U LT S Jaw lateral and xa protrusive excursions The results of the three Delphi rounds are shown in Table 2. The Sounds during jaw x response rate was 100%; that is, all experts responded to all state- opening ments in each of the three rounds. Sounds during xa Delphi round-­1 resulted in 45% (27 out of the 60 statements) jaw lateral and agreement among the experts. Of the remaining 33 statements that protrusive did not achieve consensus at Delphi round-­1, 12 statements were excursions excluded, 4 were retained as-­is, 17 were rephrased based on ex- Muscular and joint x perts’ answers and comments, and 5 new statements were added, palpation for a total of 26 statements presented at round-­2. In the Delphi Referred pain xa round-­2, 11 out of 26 statements (42%) reached the 70% agreement. Familiar pain x Out of the 15 statements not reaching consensus, 8 were rephrased, Imaging 6 were excluded, 1 was retained as-­is, and 6 new statements were Imaging x added according to the experts’ comments to create a survey of 15 a Agreement achieved only in adolescents. statements presented at round-­3. Finally, in the Delphi round-­3, 7 out of 15 statements (47%) reached the 70% consensus. Out of the 8 statements not reaching consensus, 1 statement with clear disagree- adolescents. There was agreement to use the 3Q/TMD question- ment was excluded, and 7 statements, albeit with a trend towards naire25 as an instrument for TMD screening in both age groups. agreement, failed to achieve consensus (Figure 1). Finally, the clinical diagnostic classification already present in the DC/TMD was retained. 3.1 | Demographics, screening, health and Symptom Questionnaire 3.2 | Clinical examination During the Delphi survey, experts agreed to define adolescents from The experts agreed not to use the mandatory commands for the 10 years of age or older. Participants agreed to create two different clinical examination such as used in the adult version of the instru- Axis I protocols: one for children and one for adolescents. For each ment but sought to provide instructions for the clinician to explain of the child and adolescent DC/TMD protocols, consensus indicated the concepts included in the DC/TMD. that both a short version for screening and a comprehensive version needed to be created. There was agreement among the experts to include three general health questionnaires: one for children, one 3.2.1 | Adolescents for adolescents and one for their parents, two demographic ques- tionnaires one for children and one for adolescents, and a rephrased Experts agreed to maintain the examination of jaw movements form of the Symptom Questionnaire, each adapted for children and (opening, closing, protrusion and laterotrusion) including the report RONGO et al. | 841 of pain on movement, as it is used in the protocol in the adult version, TMD diagnostic protocol in order to be adopted for use in children using cut-­off measurements for limited opening already present in and in adolescents. the literature (≤36 mm, 3rd percentile at 10 years of age). 26 Muscle and joint palpation pain and the evaluation of joint noises were not modified with respect to the adult version. Indeed, regarding the 4.1 | Demographics, screening, health and Symptom muscle and TMJ pain assessment, the experts agreed to maintain Questionnaire both the 30-­day time frame as the default period for symptoms rel- evant to the diagnosis and the amount of pressure as recommended Adolescents were defined from 10 years of age or older, according by the DC/TMD for adult, and they agreed to ask for familiar and to the World Health Organization (WHO) definition. 27 However, the referred pain. Agreement was also achieved among experts to main- ability of the individual to understand and respond to the questions is tain the examination of joint noises during all mandibular move- not related only to age. Hence, for individuals transitioning between ments, as it is in the DC/TMD for adults. childhood and adolescence, the child or adolescent assessment pro- tocol should be selected depending on the patients’ cognitive devel- opment. Children's cognitive development shows four main stages: 3.2.2 | Children sensorimotor period (birth-­2 years of age), preoperational period (2–­7 years old), concrete operational period (7 years old-­puberty), Experts agreed only to maintain the examination of jaw opening and formal operations (puberty to adulthood), and although the stages closing movements, including pain on movement, as it is in the DC/ are sequential, their time frame is flexible. 28,29 The identification TMD for adults, using cut-­off measurements for limited opening of children's cognitive development may be possible through spe- (≤32 mm, 3rd percentile at 6 years of age). 26 No agreement, how- cific tests such as the Differential Ability Scales-­II (DAS-­II)30 or the ever, was obtained in retaining or not the assessment of lateral and Kaufman Assessment Battery for Children (KABC),31 but these tools protrusive movements. would increase the time burden of the consultation (around 60 min- Regarding muscle and joint palpation, the recommendations utes to complete the test). Therefore, in research setting the DC/ for children were to assess muscle pain by palpation of the mas- TMD for children should be used in subjects

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