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

Children Pub Date : 2024-07-26 DOI:10.3390/children11080900
A. Broutin, Jean-Pierre Salles, V. Porquet-Bordes, Thomas Edouard, Frédéric Vaysse, E. Noirrit-Esclassan
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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.
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成骨不全症患者的口腔特征、与口腔健康相关的生活质量以及遭受欺凌的情况:一项横断面研究
背景/目的:成骨不全症(OI)是一种罕见的遗传疾病,不仅会导致骨质脆弱,还会导致牙齿错位和牙本质发育不全(DI)。本研究旨在评估与对照组相比,牙齿畸形的严重程度是否会影响口腔健康相关的生活质量(OHRQoL)以及儿童 OI 群体遭受欺凌的情况。研究方法在口腔和放射检查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。使用 P-CPQ、COHIP(34) 和 BCS-A 问卷分别评估外部和自我感觉的 OHRQoL 和受欺凌情况。研究结果我们在 OI 组中纳入了 39 名患者,平均年龄为 11.3 岁(±4.8 SD);在对照组中纳入了 45 名患者,平均年龄为 12.3 岁(±3.2 SD)。两组患者在咬合垂直和横向尺寸方面无明显差异。与中度或轻度 OI 患者相比,出现骨折、骨骼畸形和身材矮小的重度 OI 患者的前方(p < 0.05)和后方开牙合(p < 0.05)以及 DI(p < 0.05)明显增多。口腔疾病(p = 0.01),尤其是口腔健康(p < 0.05)和自我形象(p < 0.001)方面,对自我感觉的健康和生活质量(OHRQoL)产生了负面影响,但与口腔疾病的严重程度无关。虽然有更多的口腔疾病患者表示曾被人取笑(21.4%的患者面对面取笑,7.1%的患者通过网络取笑,而对照组分别为14.6%和2.4%),但两组患者遭受欺凌的程度并无显著差异。结论对口腔畸形患者的牙齿咬合不正和口腔健康进行干预将有助于改善他们的生活质量和自我形象。
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