Tsipouras P, Børresen A L, Dickson L A, Berg K, Prockop D J, Ramirez F
Am J Hum Genet. 1984 Nov;36(6):1172-9.
Mild osteogenesis imperfecta (OI type I and OI type IV) is characterized by postnatal onset of fractures, absence of skeletal deformity, presenile hearing loss with or without blue sclerae, and dentinogenesis imperfecta. Using one common DNA polymorphism associated with the pro alpha 2(I) human collagen gene, we found genetic heterogeneity in this disorder. In three families, the OI phenotype segregated independently of the DNA polymorphism, whereas in one family, the OI phenotype cosegregated with a DNA polymorphism in a manner suggesting linkage. Use of DNA polymorphisms associated with both type I procollagen genes should provide a tool to unravel the molecular heterogeneity of various heritable disorders of the connective tissue.
轻度成骨不全(Ⅰ型和Ⅳ型成骨不全)的特征为出生后出现骨折、无骨骼畸形、伴有或不伴有蓝色巩膜的老年前听力丧失以及牙本质生成不全。利用一种与人类前α2(Ⅰ)型胶原基因相关的常见DNA多态性,我们发现该疾病存在遗传异质性。在三个家系中,成骨不全表型与DNA多态性独立分离,而在一个家系中,成骨不全表型与一种DNA多态性共分离,其方式提示存在连锁关系。使用与Ⅰ型前胶原基因相关的DNA多态性应能为揭示各种遗传性结缔组织疾病的分子异质性提供一种工具。