Althaus Karina, Greinacher Andreas
Institut für Immunologie und Transfusionsmedizin, Ernst-Moritz-Arndt-Universität Greifswald, Sauerbruchstrasse, Greifswald, Germany.
Semin Thromb Hemost. 2009 Mar;35(2):189-203. doi: 10.1055/s-0029-1220327. Epub 2009 Apr 30.
Myosin heavy chain 9 (MYH9)-related platelet disorders belong to the group of inherited thrombocytopenias. The MYH9 gene encodes the nonmuscle myosin heavy chain IIA (NMMHC-IIA), a cytoskeletal contractile protein. Several mutations in the MYH9 gene lead to premature release of platelets from the bone marrow, macrothrombocytopenia, and cytoplasmic inclusion bodies within leukocytes. Four overlapping syndromes, known as May-Hegglin anomaly, Epstein syndrome, Fechtner syndrome, and Sebastian platelet syndrome, describe different clinical manifestations of MYH9 gene mutations. Macrothrombocytopenia is present in all affected individuals, whereas only some develop additional clinical manifestations such as renal failure, hearing loss, and presenile cataracts. The bleeding tendency is usually moderate, with menorrhagia and easy bruising being most frequent. The biggest risk for the individual is inappropriate treatment due to misdiagnosis of chronic autoimmune thrombocytopenia. To date, 31 mutations of the MYH9 gene leading to macrothrombocytopenia have been identified, of which the upstream mutations up to amino acid approximately 1400 are more likely associated with syndromic manifestations than the downstream mutations. This review provides a short history of MYH9-related disorders, summarizes the clinical and laboratory characteristics, describes a diagnostic algorithm, presents recent results of animal models, and discusses aspects of therapeutic management.
肌球蛋白重链9(MYH9)相关的血小板疾病属于遗传性血小板减少症。MYH9基因编码非肌肉肌球蛋白重链IIA(NMMHC-IIA),一种细胞骨架收缩蛋白。MYH9基因的几种突变会导致血小板从骨髓中过早释放、大血小板减少以及白细胞内出现胞质包涵体。四种重叠综合征,即May-Hegglin异常、Epstein综合征、Fechtner综合征和Sebastian血小板综合征,描述了MYH9基因突变的不同临床表现。所有受影响个体均存在大血小板减少,而只有部分个体出现诸如肾衰竭、听力丧失和早老性白内障等其他临床表现。出血倾向通常为中度,月经过多和易瘀伤最为常见。个体面临的最大风险是因慢性自身免疫性血小板减少症误诊而接受不恰当治疗。迄今为止,已鉴定出31种导致大血小板减少的MYH9基因突变,其中上游直至约1400个氨基酸处的突变比下游突变更可能与综合征表现相关。本综述介绍了MYH9相关疾病的简史,总结了临床和实验室特征,描述了诊断算法,展示了动物模型的最新结果,并讨论了治疗管理方面的问题。