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IV型埃勒斯-当洛综合征的临床和超微结构异质性。

Clinical and ultrastructural heterogeneity of type IV Ehlers-Danlos syndrome.

作者信息

Byers P H, Holbrook K A, McGillivray B, MacLeod P M, Lowry R B

出版信息

Hum Genet. 1979 Mar 12;47(2):141-50. doi: 10.1007/BF00273196.

Abstract

Ehlers-Danlos syndrome (EDS) type IV is a clinically and genetically heterogeneous disorder characterized by thin skin, prominent venous vascular markings, markedly increased bruising, and an increased likelihood of large bowel and large artery rupture. We studied two type IV EDS patients. Both have decreased amounts of type IIII collagen in skin, but ultrastructural examination of dermis showed massive dilation of rough endoplasmic reticulum in dermal fibroblasts in one, but not the other. Both had a major population of collagen fibrils of small diameter. Although previous studies suggested absent synthesis of type III collagen as the hallmark of one type of EDS IV, several abnormalities in metabolism of that type of collagen may be responsible for the phenotype in these disorders. Such disorders are likely to provide better understanding of the function of specific collagens in tissues.

摘要

IV型埃勒斯-当洛综合征(EDS)是一种临床和遗传异质性疾病,其特征为皮肤薄、静脉血管纹路明显、瘀伤显著增加以及大肠和大动脉破裂的可能性增加。我们研究了两名IV型EDS患者。两人皮肤中III型胶原蛋白的含量均减少,但对真皮的超微结构检查显示,其中一人的真皮成纤维细胞中粗面内质网大量扩张,另一人则没有。两人都有大量小直径的胶原纤维。尽管先前的研究表明III型胶原蛋白合成缺失是一种IV型EDS的标志,但该类型胶原蛋白代谢中的几种异常情况可能是这些疾病表型的原因。此类疾病可能有助于更好地理解特定胶原蛋白在组织中的功能。

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