Ornoy A, Sekeles E, Smith P, Simkin A, Kohn G
Am J Pathol. 1976 Jan;82(1):71-84.
Three spontaneously aborted fetuses with Type I achondrogenesis in a family with a first cousin marriage are described. Studies by light microscopy revealed abnormal cartilage, enchondral, and periosteal bone, and normal tooth development with abnormal alveolar bone. Electron microscopic studies of cultured skin fibroblasts manifested structurally normal cells. Scanning electron microscopy studies had shown deficient intercartilaginous septa in the metaphysis, with abnormally large calcifying globules. In the diaphysis, the orientation of bone trabeculae and collagen fibers within the trabeculae was disturbed. The numerous osteocytic lucunae were wide and irregular in arrangement and shape. Type 2 achondrogenesis, as studied in these fetuses, is probably a widespread mesenchymal defect, manifested by abnormal calcification and ossification of enchondral and periosteal bone.
本文描述了一个存在近亲婚姻的家庭中3例患有Ⅰ型软骨发育不全的自然流产胎儿。光镜研究显示软骨、软骨内成骨和骨膜骨异常,牙齿发育正常但牙槽骨异常。对培养的皮肤成纤维细胞进行电子显微镜研究显示细胞结构正常。扫描电子显微镜研究表明,干骺端软骨间隔缺乏,钙化球异常大。在骨干中,骨小梁的方向以及小梁内的胶原纤维排列紊乱。众多骨细胞陷窝排列和形状宽大且不规则。在这些胎儿中研究的Ⅱ型软骨发育不全可能是一种广泛的间充质缺陷,表现为软骨内成骨和骨膜骨的钙化及骨化异常。