Borochowitz Z, Sabo E, Misselevitch I, Boss J H
Simon Winter Institute for Human Genetics, Bnai-Zion Medical Center, Technion-Israel Institute of Technology, Haifa.
Am J Med Genet. 1998 Mar 19;76(3):238-44. doi: 10.1002/(sici)1096-8628(19980319)76:3<238::aid-ajmg7>3.0.co;2-m.
Second-semester ultrasonography of a female fetus documented short femora and humeri and dislocation of the radii. Based on the clinical and postmortem radiological findings, autosomal-recessive omodysplasia was diagnosed. The physeal plates of the long tubular bones were assessed by computer-assisted image analysis. The dimensions and orientation of the chondrocytic lacunae in the physeal plates of the omodysplastic fetus were compared with those in the physeal plates of fetuses without gross limb abnormalities (oligohydramnios, n = 2; hydrocephalus, n = 2; Down syndrome, n = 1). The pathological characteristics of the omodysplastic physeal plates were an expanded zone of proliferating cartilage and an increased number of closely packed, small chondrocytes. We propose that a genetic, functional deficiency of the physeal cells, underlying the short-limbed dwarfism of autosomal-recessive omodysplasia, is partially compensated, albeit ineffectively, by an increased number of small chondrocytes in the proliferating zone of the physeal plate.
对一名女性胎儿进行的第二学期超声检查显示股骨和肱骨短小以及桡骨脱位。基于临床和尸检放射学结果,诊断为常染色体隐性遗传性骨发育不全。通过计算机辅助图像分析评估长管状骨的生长板。将骨发育不全胎儿生长板中软骨细胞腔的尺寸和方向与无肢体明显异常胎儿(羊水过少,n = 2;脑积水,n = 2;唐氏综合征,n = 1)生长板中的进行比较。骨发育不全生长板的病理特征是增殖软骨区扩大以及紧密排列的小软骨细胞数量增加。我们提出,常染色体隐性遗传性骨发育不全导致的短肢侏儒症背后的生长板细胞遗传性功能缺陷,虽效果不佳,但部分地通过生长板增殖区中小软骨细胞数量的增加得到了补偿。