{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,11]],"date-time":"2025-10-11T01:16:38Z","timestamp":1760145398528,"version":"build-2065373602"},"reference-count":44,"publisher":"MDPI AG","issue":"8","license":[{"start":{"date-parts":[[2024,7,26]],"date-time":"2024-07-26T00:00:00Z","timestamp":1721952000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"Association de l\u2019Ost\u00e9ogen\u00e8se Imparfaite"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Children"],"abstract":"<jats:p>Background\/Objectives: Osteogenesis imperfecta (OI) is a rare genetic disease that is responsible for bone fragility, but also for dental malocclusions and dentinogenesis imperfecta (DI). The aim of this study was to assess whether the severity of dental malocclusion influenced the oral health-related quality of life (OHRQoL) and exposure to bullying in a paediatric OI population compared with a control group. Methods: Dental and occlusal characteristics were noted during oral and radiographic examination. The severity of malocclusion was assessed using the PAR index. P-CPQ, COHIP(34), and BCS-A questionnaires were used to evaluate, respectively, externally and self-perceived OHRQoL and bullying. Results: We included 39 patients with a mean age of 11.3 (\u00b14.8 SD) in the OI group, and 45 patients with a mean age of 12.3 (\u00b13.2 SD) in the control group. There were no significant differences between the two groups in terms of occlusal vertical and transverse dimensions. Patients with severe OI, presenting with bone fractures, bones deformities, and short stature, had significantly more anterior (p &lt; 0.05) and posterior openbites (p &lt; 0.05) and more DI (p &lt; 0.05) compared to patients who had moderate or mild OI. Self-perceived OHRQoL was negatively impacted by the disease (p = 0.01), particularly in the domains of oral health (p &lt; 0.05) and self-image (p &lt; 0.001), but not by its severity. Exposure to bullying did not differ significantly between the two groups, although more patients with OI reported being teased (21.4% face to face and 7.1% online vs. 14.6% and 2.4% in the control group). Conclusion: Interventions for dental malocclusion and oral health in OI patients would help to improve their quality of life and self-image.<\/jats:p>","DOI":"10.3390\/children11080900","type":"journal-article","created":{"date-parts":[[2024,7,26]],"date-time":"2024-07-26T14:39:08Z","timestamp":1722004748000},"page":"900","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Orofacial Features, Oral Health-Related Quality of Life, and Exposure to Bullying in Osteogenesis Imperfecta: A Cross-Sectional Study"],"prefix":"10.3390","volume":"11","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-5295-6184","authenticated-orcid":false,"given":"Alice","family":"Broutin","sequence":"first","affiliation":[{"name":"Paediatric Dentistry, University Toulouse III, CHU Toulouse, Reference Centre for Rare Oral Diseases, Centre for Anthropobiology & Genomics of Toulouse (CAGT) CNRS UMR 5288, 31400 Toulouse, France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jean-Pierre","family":"Salles","sequence":"additional","affiliation":[{"name":"Endocrine, Bone Diseases and Genetics Unit, Reference Centre for Rare Diseases of Calcium and Phosphate Metabolism, OSCAR Network, ERN BOND, Children\u2019s Hospital, Toulouse University Hospital, 31300 Toulouse, France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0507-0230","authenticated-orcid":false,"given":"Val\u00e9rie","family":"Porquet-Bordes","sequence":"additional","affiliation":[{"name":"Endocrine, Bone Diseases and Genetics Unit, Reference Centre for Rare Diseases of Calcium and Phosphate Metabolism, OSCAR Network, ERN BOND, Children\u2019s Hospital, Toulouse University Hospital, 31300 Toulouse, France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9222-5009","authenticated-orcid":false,"given":"Thomas","family":"Edouard","sequence":"additional","affiliation":[{"name":"Endocrine, Bone Diseases and Genetics Unit, Reference Centre for Rare Diseases of Calcium and Phosphate Metabolism, OSCAR Network, ERN BOND, Children\u2019s Hospital, Toulouse University Hospital, 31300 Toulouse, France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Fr\u00e9d\u00e9ric","family":"Vaysse","sequence":"additional","affiliation":[{"name":"Paediatric Dentistry, University Toulouse III, CHU Toulouse, Reference Centre for Rare Oral Diseases, Centre for Anthropobiology & Genomics of Toulouse (CAGT) CNRS UMR 5288, 31400 Toulouse, France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7039-6405","authenticated-orcid":false,"given":"Emmanuelle","family":"Noirrit-Esclassan","sequence":"additional","affiliation":[{"name":"Paediatric Dentistry, University Toulouse III, CHU Toulouse, Reference Centre for Rare Oral Diseases, ADES UMR 7268 CNRS EFS, 13005 Marseille, France"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2024,7,26]]},"reference":[{"key":"ref_1","unstructured":"Feingold, K.R., Anawalt, B., Blackman, M.R., Boyce, A., Chrousos, G., Corpas, E., de Herder, W.W., Dhatariya, K., Dungan, K., and Hofland, J. (2000). Osteogenesis Imperfecta. Endotext, MDText.com, Inc."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"36","DOI":"10.1186\/s13023-023-02643-3","article-title":"Systematic Review of Health Related-Quality of Life in Adults with Osteogenesis Imperfecta","volume":"18","author":"Crowley","year":"2023","journal-title":"Orphanet J. Rare Dis."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"123","DOI":"10.1186\/s13023-023-02728-z","article-title":"Quality of Life of Pediatric and Adult Individuals with Osteogenesis Imperfecta: A Meta-Analysis","volume":"18","author":"Wehrli","year":"2023","journal-title":"Orphanet J. Rare Dis."},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"102","DOI":"10.1016\/j.pmn.2022.08.014","article-title":"Multimodal Pain Management of Children Diagnosed with Osteogenesis Imperfecta: An Integrative Literature Review","volume":"24","author":"Dlesk","year":"2023","journal-title":"Pain. Manag. Nurs."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"314","DOI":"10.1111\/odi.13715","article-title":"Malocclusion in Individuals with Osteogenesis Imperfecta: A Systematic Review and Meta-Analysis","volume":"28","author":"Prado","year":"2022","journal-title":"Oral. Dis."},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"232","DOI":"10.1093\/ejo\/cjl035","article-title":"The Craniofacial Characteristics of Osteogenesis Imperfecta Patients","volume":"29","author":"Chang","year":"2007","journal-title":"Eur. J. Orthod."},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"1264","DOI":"10.1177\/0022034511399918","article-title":"Oral Health-Related Quality of Life","volume":"90","author":"Sischo","year":"2011","journal-title":"J. Dent. Res."},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"238","DOI":"10.1093\/ejo\/cju046","article-title":"The Impact of Malocclusion on the Quality of Life among Children and Adolescents: A Systematic Review of Quantitative Studies","volume":"37","author":"Dimberg","year":"2015","journal-title":"Eur. J. Orthod."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"1881","DOI":"10.1007\/s00784-015-1681-3","article-title":"The Impact of Malocclusions on Oral Health-Related Quality of Life in Children\u2014A Systematic Review and Meta-Analysis","volume":"20","author":"Kragt","year":"2016","journal-title":"Clin. Oral. Investig."},{"key":"ref_10","doi-asserted-by":"crossref","unstructured":"Broutin, A., Blanchet, I., Canceill, T., and Noirrit-Esclassan, E. (2023). Association between Dentofacial Features and Bullying from Childhood to Adulthood: A Systematic Review. Children, 10.","DOI":"10.3390\/children10060934"},{"key":"ref_11","unstructured":"(2024, March 28). Centre de R\u00e9f\u00e9rence Maladies Osseuses Constitutionnelles, Fili\u00e8re OSCAR. Protocole National de Diagnostic et de Soins (PNDS) Osteogenese Imparfaite. HAS. 2017. Available online: https:\/\/www.has-sante.fr\/upload\/docs\/application\/pdf\/2016-12\/pnds_-_osteogenese_imparfaite.pdf."},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"1470","DOI":"10.1002\/ajmg.a.36545","article-title":"Osteogenesis Imperfecta: Clinical Diagnosis, Nomenclature and Severity Assessment","volume":"164","author":"Sillence","year":"2014","journal-title":"Am. J. Med. Genet. Part A"},{"key":"ref_13","doi-asserted-by":"crossref","unstructured":"Nguyen, H.T.T., Vu, D.C., Nguyen, D.M., Dang, Q.D., Tran, V.K., Le, H., and Tong, S.M. (2021). Dentinogenesis Imperfecta and Caries in Osteogenesis Imperfecta among Vietnamese Children. Dent. J., 9.","DOI":"10.3390\/dj9050049"},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"477","DOI":"10.1093\/ejo\/cjp031","article-title":"Malocclusion and Occlusal Traits in an Urban Iranian Population. An Epidemiological Study of 11- to 14-Year-Old Children","volume":"31","author":"Eslamipour","year":"2009","journal-title":"Eur. J. Orthod."},{"key":"ref_15","doi-asserted-by":"crossref","first-page":"125","DOI":"10.1093\/ejo\/14.2.125","article-title":"The Development of the PAR Index (Peer Assessment Rating): Reliability and Validity","volume":"14","author":"Richmond","year":"1992","journal-title":"Eur. J. Orthod."},{"key":"ref_16","doi-asserted-by":"crossref","unstructured":"Razanamihaja, N., Boy-Lef\u00e8vre, M.-L., Jordan, L., Tapiro, L., Berdal, A., de la Dure-Molla, M., and Azogui-Levy, S. (2018). Parental\u2013Caregivers Perceptions Questionnaire (P-CPQ): Translation and Evaluation of Psychometric Properties of the French Version of the Questionnaire. BMC Oral. Health, 18.","DOI":"10.1186\/s12903-018-0670-8"},{"key":"ref_17","doi-asserted-by":"crossref","first-page":"176","DOI":"10.1186\/s12955-015-0371-9","article-title":"Validation of the Child Oral Health Impact Profile (COHIP) French Questionnaire among 12 Years-Old Children in New Caledonia","volume":"13","author":"Pichot","year":"2015","journal-title":"Health Qual. Life Outcomes"},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"75","DOI":"10.1111\/bjep.12223","article-title":"Development and Validation of the Bullying and Cyberbullying Scale for Adolescents: A Multi-Dimensional Measurement Model","volume":"89","author":"Thomas","year":"2019","journal-title":"Br. J. Educ. Psychol."},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"336","DOI":"10.1016\/j.ajodo.2012.10.016","article-title":"Evaluation of the Severity of Malocclusions in Children Affected by Osteogenesis Imperfecta with the Peer Assessment Rating and Discrepancy Indexes","volume":"143","author":"Rizkallah","year":"2013","journal-title":"Am. J. Orthod. Dentofac. Orthop."},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"19","DOI":"10.1002\/cre2.53","article-title":"Occlusal Features and Need for Orthodontic Treatment in Persons with Osteogenesis Imperfecta","volume":"3","author":"Nguyen","year":"2017","journal-title":"Clin. Exp. Dent. Res."},{"key":"ref_21","doi-asserted-by":"crossref","unstructured":"De Nova-Garc\u00eda, M.J., Bernal-Barroso, F., Mourelle-Mart\u00ednez, M.R., Gallardo-L\u00f3pez, N.E., Di\u00e9guez-P\u00e9rez, M., Feijoo-Garc\u00eda, G., and Burgue\u00f1o-Torres, L. (2022). Evaluation of the Severity of Malocclusion in Children with Osteogenesis Imperfecta. J. Clin. Med., 11.","DOI":"10.3390\/jcm11164862"},{"key":"ref_22","doi-asserted-by":"crossref","first-page":"225","DOI":"10.1038\/sj.bdj.4801530","article-title":"A Comparison of the Index of Complexity Outcome and Need (ICON) with the Peer Assessment Rating (PAR) and the Index of Orthodontic Treatment Need (IOTN)","volume":"193","author":"Fox","year":"2002","journal-title":"Br. Dent. J."},{"key":"ref_23","doi-asserted-by":"crossref","unstructured":"Blanck-Lubarsch, M., Hohoff, A., Wiechmann, D., and Stamm, T. (2014). Orthodontic Treatment of Children\/Adolescents with Special Health Care Needs: An Analysis of Treatment Length and Clinical Outcome. BMC Oral. Health, 14.","DOI":"10.1186\/1472-6831-14-67"},{"key":"ref_24","doi-asserted-by":"crossref","first-page":"142","DOI":"10.1002\/ajmg.a.30523","article-title":"Craniofacial Features in Osteogenesis Imperfecta: A Cephalometric Study","volume":"133A","author":"Kolkka","year":"2005","journal-title":"Am. J. Med. Genet. Part A"},{"key":"ref_25","first-page":"121","article-title":"Osteogenesis Imperfecta: Clinical, Cephalometric, and Biochemical Investigations of OI Types I, III, and IV","volume":"17","author":"Jensen","year":"1997","journal-title":"J. Craniofac Genet. Dev. Biol."},{"key":"ref_26","first-page":"Doc06","article-title":"The Effect of Bisphosphonate Medication on Orthodontics and Orthognathic Surgery in Patients with Osteogenesis Imperfecta","volume":"8","author":"Friedrich","year":"2019","journal-title":"GMS Interdiscip. Plast. Reconstr. Surg. DGPW"},{"key":"ref_27","first-page":"30","article-title":"Dental Manifestations of Osteogenesis Imperfecta and Abnormalities of Collagen I Metabolism","volume":"18","author":"Lund","year":"1998","journal-title":"J. Craniofac Genet. Dev. Biol."},{"key":"ref_28","doi-asserted-by":"crossref","first-page":"65","DOI":"10.1080\/000163502753509446","article-title":"Dental Aberrations in Children and Adolescents with Osteogenesis Imperfecta","volume":"60","author":"Malmgren","year":"2002","journal-title":"Acta Odontol. Scand."},{"key":"ref_29","first-page":"34","article-title":"Oral cavity features in patients suffering from osteogenesis imperfecta","volume":"193","author":"Alania","year":"2011","journal-title":"Georgian Med. News"},{"key":"ref_30","doi-asserted-by":"crossref","unstructured":"Thuesen, K.J., Gj\u00f8rup, H., Hald, J.D., Schmidt, M., Harsl\u00f8f, T., Langdahl, B., and Haubek, D. (2018). The Dental Perspective on Osteogenesis Imperfecta in a Danish Adult Population. BMC Oral. Health, 18.","DOI":"10.1186\/s12903-018-0639-7"},{"key":"ref_31","doi-asserted-by":"crossref","first-page":"621","DOI":"10.1016\/j.oooo.2019.07.001","article-title":"Dental Alterations on Panoramic Radiographs of Patients with Osteogenesis Imperfecta in Relation to Clinical Diagnosis, Severity, and Bisphosphonate Regimen Aspects: A STROBE-Compliant Case-Control Study","volume":"128","author":"Ribeiro","year":"2019","journal-title":"Oral Surg. Oral Med. Oral Pathol. Oral Radiol."},{"key":"ref_32","doi-asserted-by":"crossref","first-page":"72","DOI":"10.1080\/00016350310001398","article-title":"Assessment of Dysplastic Dentin in Osteogenesis Imperfecta and Dentinogenesis Imperfecta","volume":"61","author":"Malmgren","year":"2003","journal-title":"Acta Odontol. Scand."},{"key":"ref_33","doi-asserted-by":"crossref","unstructured":"Mart\u00edn-Vacas, A., de Nova, M.J., Sagastizabal, B., Garc\u00eda-Barbero, \u00c1.E., and Vera-Gonz\u00e1lez, V. (2022). Morphological Study of Dental Structure in Dentinogenesis Imperfecta Type I with Scanning Electron Microscopy. Healthcare, 10.","DOI":"10.3390\/healthcare10081453"},{"key":"ref_34","doi-asserted-by":"crossref","first-page":"285","DOI":"10.1097\/DBP.0000000000000155","article-title":"Bullying in Medically Fragile Youth: A Review of Risks, Protective Factors, and Recommendations for Medical Providers","volume":"36","author":"Faith","year":"2015","journal-title":"J. Dev. Behav. Pediatr."},{"key":"ref_35","first-page":"13","article-title":"Activities and Participation in Young Adults with Osteogenesis Imperfecta","volume":"4","author":"Montpetit","year":"2011","journal-title":"J. Pediatr. Rehabil. Med."},{"key":"ref_36","doi-asserted-by":"crossref","first-page":"201","DOI":"10.1002\/ajmg.a.20257","article-title":"Personality and Stereotype in Osteogenesis Imperfecta: Behavioral Phenotype or Response to Life\u2019s Hard Challenges?","volume":"122A","author":"Ablon","year":"2003","journal-title":"Am. J. Med. Genet. Part. A"},{"key":"ref_37","doi-asserted-by":"crossref","first-page":"27","DOI":"10.21307\/aoj-2020-030","article-title":"Considering Malocclusion as a Disability","volume":"35","author":"Blanch","year":"2019","journal-title":"Australas. Orthod. J."},{"key":"ref_38","doi-asserted-by":"crossref","first-page":"187","DOI":"10.1186\/s13023-018-0935-y","article-title":"Oral Health-Related Quality of Life in Children and Adolescents with Osteogenesis Imperfecta: Cross-Sectional Study","volume":"13","author":"Najirad","year":"2018","journal-title":"Orphanet J. Rare Dis."},{"key":"ref_39","doi-asserted-by":"crossref","first-page":"261","DOI":"10.1007\/s40368-021-00664-9","article-title":"Oral Health-Related Quality of Life in Children with Osteogenesis Imperfecta","volume":"23","author":"Riddington","year":"2022","journal-title":"Eur. Arch. Paediatr. Dent."},{"key":"ref_40","doi-asserted-by":"crossref","first-page":"160","DOI":"10.1111\/joor.13114","article-title":"Oral Health-related Quality of Life in X-linked Hypophosphataemia and Osteogenesis Imperfecta","volume":"48","author":"Hald","year":"2021","journal-title":"J. Oral. Rehabil."},{"key":"ref_41","doi-asserted-by":"crossref","first-page":"480","DOI":"10.1016\/j.adaj.2020.03.040","article-title":"Malocclusion Traits and Oral Health-Related Quality of Life in Children with Osteogenesis Imperfecta (Cross-Sectional Study)","volume":"151","author":"Najirad","year":"2020","journal-title":"J. Am. Dent. Assoc."},{"key":"ref_42","doi-asserted-by":"crossref","first-page":"169","DOI":"10.1007\/s00784-024-05568-8","article-title":"The Impact of Craniofacial and Dental Osteogenesis Imperfecta Manifestations on Oral Health-Related Quality of Life of Children and Adolescents","volume":"28","author":"Pantoja","year":"2024","journal-title":"Clin. Oral. Investig."},{"key":"ref_43","doi-asserted-by":"crossref","first-page":"20","DOI":"10.1111\/j.1600-0528.2007.0002.x","article-title":"Reliability and Convergent and Discriminant Validity of the Child Oral Health Impact Profile (COHIP Child\u2019s Version)","volume":"35","author":"Broder","year":"2007","journal-title":"Community Dent. Oral Epidemiol."},{"key":"ref_44","doi-asserted-by":"crossref","first-page":"189","DOI":"10.1016\/S1079-2104(99)70272-6","article-title":"Evaluation of Oral Problems in an Osteogenesis Imperfecta Population","volume":"87","author":"Marini","year":"1999","journal-title":"Oral Surg. Oral Med. Oral Pathol. Oral Radiol. Endodontology"}],"container-title":["Children"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2227-9067\/11\/8\/900\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,10]],"date-time":"2025-10-10T15:24:24Z","timestamp":1760109864000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2227-9067\/11\/8\/900"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,7,26]]},"references-count":44,"journal-issue":{"issue":"8","published-online":{"date-parts":[[2024,8]]}},"alternative-id":["children11080900"],"URL":"https:\/\/doi.org\/10.3390\/children11080900","relation":{},"ISSN":["2227-9067"],"issn-type":[{"type":"electronic","value":"2227-9067"}],"subject":[],"published":{"date-parts":[[2024,7,26]]}}}