Leśniewicz R, Galicka A, Anchim T, Skotnicki M, Wołczyński S, Urban J
Zakładu Genetyki Klinicznej AM w Białymstoku.
Ginekol Pol. 2001 Jun;72(6):456-65.
Osteogenesis imperfecta (OI types I, II, III, IV) is a heterogeneous group of genetically disorders of connective tissue. Quantitative or qualitative abnormalities of type I collagen form pathogenetical basis of the disease. They are caused by mutations in genes encoding collagen proteins or enzymes involved in collagen biosynthesis. The clinical features of each type usually correspond to the type of mutation. Typical manifestations are fragile bones with multiple bone fractures and bone deformities. Currently applied diagnosis in utero of OI II and sometimes OI III may be performed. Diagnosis of other OI phenotypes cannot be made until after birth. We present three cases of OI II (four children) diagnosed, in utero, by ultrasound examination. The analysis in work include: 1. the prenatal sonographic features of OI type II 2. the biochemical properties of collagen in the above cases 3. genetic counselling of the families affected by OI.
成骨不全症(I型、II型、III型、IV型)是一组结缔组织遗传性疾病的异质性群体。I型胶原蛋白的定量或定性异常构成了该疾病的发病基础。它们是由编码胶原蛋白或参与胶原蛋白生物合成的酶的基因突变引起的。每种类型的临床特征通常与突变类型相对应。典型表现为骨骼脆弱,伴有多处骨折和骨骼畸形。目前可以对II型成骨不全症进行宫内诊断,有时也可对III型进行宫内诊断。其他成骨不全症表型在出生后才能诊断。我们介绍了3例通过超声检查在宫内诊断为II型成骨不全症的病例(4名儿童)。本研究分析内容包括:1. II型成骨不全症的产前超声特征;2. 上述病例中胶原蛋白的生化特性;3. 受成骨不全症影响家庭的遗传咨询。