Suppr超能文献

红细胞膜疾病发病机制研究进展。

Advances in understanding the pathogenesis of red cell membrane disorders.

机构信息

Department of Molecular Medicine and Medical Biotechnologies, Federico II" University of Naples, Naples, Italy.

CEINGE - Biotecnologie Avanzate, Naples, Italy.

出版信息

Br J Haematol. 2019 Oct;187(1):13-24. doi: 10.1111/bjh.16126. Epub 2019 Jul 31.

Abstract

Hereditary erythrocyte membrane disorders are caused by mutations in genes encoding various transmembrane or cytoskeletal proteins of red blood cells. The main consequences of these genetic alterations are decreased cell deformability and shortened erythrocyte survival. Red blood cell membrane defects encompass a heterogeneous group of haemolytic anaemias caused by either (i) altered membrane structural organisation (hereditary spherocytosis, hereditary elliptocytosis, hereditary pyropoikilocytosis and Southeast Asian ovalocytosis) or (ii) altered membrane transport function (overhydrated hereditary stomatocytosis, dehydrated hereditary stomatocytosis or xerocytosis, familial pseudohyperkalaemia and cryohydrocytosis). Herein we provide a comprehensive review of the recent literature on the molecular genetics of erythrocyte membrane defects and their reported clinical consequences. We also describe the effect of low-expression genetic variants on the high inter- and intra-familial phenotype variability of erythrocyte structural defects.

摘要

遗传性红细胞膜缺陷是由编码红细胞各种跨膜或细胞骨架蛋白的基因突变引起的。这些遗传改变的主要后果是细胞变形能力下降和红细胞寿命缩短。红细胞膜缺陷包括一组异质性溶血性贫血,由以下原因引起:(i) 膜结构组织改变(遗传性球形红细胞增多症、遗传性椭圆形红细胞增多症、遗传性热不稳定血影蛋白溶血性贫血和东南亚椭圆形红细胞增多症)或 (ii) 膜转运功能改变(过度水化遗传性口形红细胞增多症、脱水遗传性口形红细胞增多症或棘形红细胞增多症、家族性假性高钾血症和冷沉淀红细胞增多症)。本文综述了红细胞膜缺陷的分子遗传学及其临床后果的最新文献。我们还描述了低表达遗传变异对红细胞结构缺陷的高家族内和家族间表型变异性的影响。

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验