成骨不全症患者的口面部特征、口腔健康相关生活质量及受欺凌情况：一项横断面研究

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

作者信息

Broutin Alice, Salles Jean-Pierre, Porquet-Bordes Valérie, Edouard Thomas, Vaysse Frédéric, Noirrit-Esclassan Emmanuelle

机构信息

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.

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.

出版信息

Children (Basel). 2024 Jul 26;11(8):900. doi: 10.3390/children11080900.

DOI:10.3390/children11080900

PMID:39201835

原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11352506/

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 ( < 0.05) and posterior openbites ( < 0.05) and more DI ( < 0.05) compared to patients who had moderate or mild OI. Self-perceived OHRQoL was negatively impacted by the disease ( = 0.01), particularly in the domains of oral health ( < 0.05) and self-image ( < 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.

摘要

背景/目的：成骨不全症（OI）是一种罕见的遗传性疾病，不仅会导致骨骼脆弱，还会引发牙齿咬合不正和牙本质发育不全（DI）。本研究的目的是评估与对照组相比，小儿OI患者的牙齿咬合不正严重程度是否会影响其口腔健康相关生活质量（OHRQoL）以及遭受欺凌的情况。

方法

在口腔和影像学检查过程中记录牙齿和咬合特征。使用PAR指数评估咬合不正的严重程度。分别使用P-CPQ、COHIP(34)和BCS-A问卷来评估外部和自我感知的OHRQoL以及欺凌情况。

结果

我们纳入了OI组39例平均年龄为11.3（±4.8标准差）岁的患者，以及对照组45例平均年龄为12.3（±3.2标准差）岁的患者。两组在咬合垂直和横向维度方面无显著差异。与患有中度或轻度OI的患者相比，患有严重OI且出现骨折、骨骼畸形和身材矮小的患者，前牙开合（<0.05）和后牙开合（<0.05）以及DI更多（<0.05）。自我感知的OHRQoL受到该疾病的负面影响（=0.01），特别是在口腔健康（<0.05）和自我形象（<0.001）方面，但不受其严重程度影响。两组之间遭受欺凌的情况没有显著差异，尽管更多的OI患者报告受到取笑（面对面21.4%，在线7.1%，而对照组为14.6%和2.4%）。