Brodie S G, Lachman R S, Crandall B F, Fox M A, Rimoin D L, Cohn D H, Wilcox W R
Ahmanson Department of Pediatrics, Steven Spielberg Pediatric Research Center, UCLA School of Medicine, Los Angeles, California, USA.
Am J Med Genet. 1998 Nov 16;80(3):247-51.
The mesomelic chondrodysplasias are a heterogeneous group of dwarfing disorders characterized by shortness of the middle segments of limbs. We report on a 25-week fetus with disproportionate shortness of limbs with an apparently distinct form of mesomelic dysplasia. Radiographic findings at necropsy included ulnar deviation of hands, talipes equinovarus, distal tapering of the humeri, and hypoplastic fibulae, radii, and ulnae. Chondro-osseous morphology showed mild shortness of the physeal columns, overgrowth of perichondral bone, peripheral ingrowth of mesenchymal cells into the physis, and numerous areas of fibrillar degeneration with rings of collagen surrounding the chondrocytes. Ultrastructural findings included a degenerated territorial matrix, pericellular halos of collagen, and dilated loops of rough endoplasmic reticulum in chondrocytes. The radiographic appearance of the long bones is distinct from that of previously described mesomelic dysplasias. The chondro-osseous morphologic findings and the distal tapering of the humerus are somewhat reminiscent of atelosteogenesis type II, but the pattern of matrix degeneration and the presence of inclusion bodies in the chondrocytes distinguish it from disorders of sulfate transport.
中肢短小性软骨发育异常是一组异质性的侏儒症疾病,其特征为肢体中段短小。我们报告了一名25周大的胎儿,其肢体不成比例地短小,呈现出一种明显独特的中肢发育异常形式。尸检时的影像学表现包括手部尺骨偏斜、马蹄内翻足、肱骨远端变细以及腓骨、桡骨和尺骨发育不全。软骨-骨形态学显示生长板柱轻度短小、软骨周骨过度生长、间充质细胞向生长板周围内生,以及许多纤维变性区域,软骨细胞周围有胶原环。超微结构表现包括退化的区域基质、软骨细胞周围的胶原晕以及粗面内质网扩张的环。长骨的影像学表现与先前描述的中肢发育异常不同。软骨-骨形态学发现以及肱骨远端变细在一定程度上让人联想到II型骨发育不全,但基质变性模式和软骨细胞中包涵体的存在使其与硫酸盐转运障碍性疾病相区别。