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成骨不全症:诊断与治疗

Osteogenesis imperfecta: diagnosis and treatment.

作者信息

Biggin A, Munns C F

机构信息

Institute of Endocrinology and Diabetes, Children's Hospital Westmead, Locked Bag 4001, Westmead, NSW, 2145, Australia,

出版信息

Curr Osteoporos Rep. 2014 Sep;12(3):279-88. doi: 10.1007/s11914-014-0225-0.

Abstract

Osteogenesis imperfecta (OI) is a genetic bone fragility disorder characterized by low bone mass, skeletal deformity, and variable short stature. OI is predominantly caused by dominant mutations affecting type 1 collagen synthesis, with a number of other genes implicated in OI over recent years. The clinical severity of OI can vary greatly, even within families who share a common mutation. Optimal management of OI requires a multidisciplinary approach involving pediatrician, endocrinologist (bone and mineral physician), rehabilitation specialist, orthopedic surgeon, dentist, geneticist, social worker/psychologist, physiotherapist, and occupational therapist. Bisphosphonate therapy remains the mainstay of medical treatment in OI and has been shown to decrease bone pain, enhance well-being, improve muscle strength and mobility and decrease fracture incidence. Novel therapies are beginning to emerge as more is understood about the signaling pathways involved in bone formation. The following summarizes the diagnosis, genetic heterogeneity and management of OI in pediatric practice.

摘要

成骨不全症(OI)是一种遗传性骨脆性疾病,其特征为骨量低、骨骼畸形以及身材矮小程度不一。OI主要由影响I型胶原蛋白合成的显性突变引起,近年来还有许多其他基因与OI有关。即使在具有共同突变的家族中,OI的临床严重程度也可能有很大差异。OI的最佳管理需要多学科方法,涉及儿科医生、内分泌学家(骨与矿物质医生)、康复专家、整形外科医生、牙医、遗传学家、社会工作者/心理学家、物理治疗师和职业治疗师。双膦酸盐治疗仍然是OI药物治疗的主要手段,已被证明可减轻骨痛、提高幸福感、增强肌肉力量和活动能力并降低骨折发生率。随着对骨形成所涉及的信号通路有了更多了解,新的治疗方法开始出现。以下总结了儿科实践中OI的诊断、遗传异质性和管理。

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