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成骨不全症——临床与分子多样性

Osteogenesis imperfecta--clinical and molecular diversity.

作者信息

Roughley P J, Rauch F, Glorieux F H

机构信息

Genetics Unit, Shriners Hospital for Children, Montreal, Canada.

出版信息

Eur Cell Mater. 2003 Jun 30;5:41-7; discussion 47. doi: 10.22203/ecm.v005a04.

Abstract

Osteogenesis imperfecta is a heritable disorder of bone formation resulting in low bone mass and a propensity to fracture. It exhibits a broad range of clinical severity, ranging from multiple fracturing in utero and perinatal death to normal adult stature and a low fracture incidence. The disorder is currently classified into seven types based on differences in clinical presentation and bone architecture. Mutation in one of the type I collagen genes is commonly associated with osteogenesis imperfecta, but is not a prerequisite for the diagnosis. Indeed, the newer forms of osteogenesis imperfecta (types V, VI and VII) are not associated with type I collagen gene defects. Amongst the type I collagen gene mutations that can occur, missense base substitutions involving glycine codons in the exons encoding the central triple-helix forming domain predominate. Such mutations can occur in all the classical forms of osteogenesis imperfecta (types I-IV), but genotype/phenotype correlations are complex and often unpredictable. Treatment of osteogenesis imperfecta by bisphosphonate therapy can improve bone mass in all types of the disorder, and while not being a cure for the disorder does improve the quality of life of the patient.

摘要

成骨不全症是一种遗传性骨形成障碍疾病,会导致骨量降低和易于骨折。它表现出广泛的临床严重程度,从子宫内多发骨折和围产期死亡到正常成人身高及低骨折发生率不等。目前,该疾病根据临床表现和骨骼结构的差异分为七种类型。I型胶原蛋白基因中的突变通常与成骨不全症相关,但并非诊断的必要条件。实际上,成骨不全症的新类型(V型、VI型和VII型)与I型胶原蛋白基因缺陷无关。在可能发生的I型胶原蛋白基因突变中,涉及编码中央三螺旋形成结构域的外显子中甘氨酸密码子的错义碱基替换占主导。此类突变可发生在成骨不全症的所有经典类型(I-IV型)中,但基因型/表型相关性复杂且往往不可预测。双膦酸盐疗法治疗成骨不全症可改善所有类型该疾病的骨量,虽然无法治愈该疾病,但确实能提高患者的生活质量。

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