Hassoun H, Palek J
Department of Biomedical Research, St. Elizabeth's Medical Center of Boston, Tufts University Medical School, MA 02135, USA.
Blood Rev. 1996 Sep;10(3):129-47. doi: 10.1016/s0268-960x(96)90021-1.
Hereditary spherocytosis is a common and very heterogeneous hemolytic anemia caused by defects of the red cell membrane proteins. In recent years, major advances in our understanding of the red cell membrane skeleton and a better characterization of its individual components have allowed a brighter insight into the pathogenesis of the disease. In this article, we present an overview of the erythrocyte skeleton and its individual constituents. We also review the clinical aspects of the disease and describe the currently known molecular defects involving the membrane proteins which have been shown to play an essential role in the underlying mechanism of hereditary spherocytosis. Finally we examine several models that have been proposed in an attempt to clarify the mechanism leading from the initial molecular insult to the clinical phenotype.
遗传性球形红细胞增多症是一种常见且异质性很强的溶血性贫血,由红细胞膜蛋白缺陷引起。近年来,我们对红细胞膜骨架的认识取得了重大进展,对其各个组成部分也有了更清晰的表征,这使我们对该疾病的发病机制有了更深入的了解。在本文中,我们概述了红细胞骨架及其各个组成部分。我们还回顾了该疾病的临床方面,并描述了目前已知的涉及膜蛋白的分子缺陷,这些缺陷已被证明在遗传性球形红细胞增多症的潜在机制中起着至关重要的作用。最后,我们研究了几种为阐明从最初的分子损伤到临床表型的机制而提出的模型。