Orofacial Features, Oral Health-Related Quality of Life, and Exposure to Bullying in Osteogenesis Imperfecta: A Cross-Sectional Study

Author:

Broutin Alice1ORCID,Salles Jean-Pierre2,Porquet-Bordes Valérie2ORCID,Edouard Thomas2ORCID,Vaysse Frédéric1,Noirrit-Esclassan Emmanuelle3ORCID

Affiliation:

1. 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

2. Endocrine, Bone Diseases and Genetics Unit, Reference Centre for Rare Diseases of Calcium and Phosphate Metabolism, OSCAR Network, ERN BOND, Children’s Hospital, Toulouse University Hospital, 31300 Toulouse, France

3. Paediatric Dentistry, University Toulouse III, CHU Toulouse, Reference Centre for Rare Oral Diseases, ADES UMR 7268 CNRS EFS, 13005 Marseille, France

Abstract

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 (±4.8 SD) in the OI group, and 45 patients with a mean age of 12.3 (±3.2 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 < 0.05) and posterior openbites (p < 0.05) and more DI (p < 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 < 0.05) and self-image (p < 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.

Funder

Association de l’Ostéogenèse Imparfaite

Publisher

MDPI AG

Reference44 articles.

1. 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.

2. Systematic Review of Health Related-Quality of Life in Adults with Osteogenesis Imperfecta;Crowley;Orphanet J. Rare Dis.,2023

3. Quality of Life of Pediatric and Adult Individuals with Osteogenesis Imperfecta: A Meta-Analysis;Wehrli;Orphanet J. Rare Dis.,2023

4. Multimodal Pain Management of Children Diagnosed with Osteogenesis Imperfecta: An Integrative Literature Review;Dlesk;Pain. Manag. Nurs.,2023

5. Malocclusion in Individuals with Osteogenesis Imperfecta: A Systematic Review and Meta-Analysis;Prado;Oral. Dis.,2022

同舟云学术

1.学者识别学者识别

2.学术分析学术分析

3.人才评估人才评估

"同舟云学术"是以全球学者为主线,采集、加工和组织学术论文而形成的新型学术文献查询和分析系统,可以对全球学者进行文献检索和人才价值评估。用户可以通过关注某些学科领域的顶尖人物而持续追踪该领域的学科进展和研究前沿。经过近期的数据扩容,当前同舟云学术共收录了国内外主流学术期刊6万余种,收集的期刊论文及会议论文总量共计约1.5亿篇,并以每天添加12000余篇中外论文的速度递增。我们也可以为用户提供个性化、定制化的学者数据。欢迎来电咨询!咨询电话:010-8811{复制后删除}0370

www.globalauthorid.com

TOP

Copyright © 2019-2024 北京同舟云网络信息技术有限公司
京公网安备11010802033243号  京ICP备18003416号-3