Denecke Jonas, Marquardt Thorsten
University Hospital of Rostock, Department of Pediatrics, Rembrandtstrabetae 16/17, 18057 Rostock, Germany.
Biochim Biophys Acta. 2009 Sep;1792(9):915-20. doi: 10.1016/j.bbadis.2008.12.005. Epub 2008 Dec 25.
Congenital diserythropoietic anemias (CDA) were classified according to bone marrow changes and biochemical features 40 years ago. A consistent finding in CDA type II, the most frequent subgroup of CDAs is a relevant hypoglycosylation of erythrocyte membrane proteins. It is a matter of debate if the hypoglycosylation is the primary cause of the disorder or a phenomenon secondary to other pathomechanisms. The molecular cause of the disorder is still unknown although some enzyme deficiencies have been proposed to cause CDA II in the last 2 decades and a linkage analysis locating the CDA II gene in a 5 cM region on chromosome 20 was done in 1997. In this review biochemical and genetic data are discussed and diagnostic methods based on biochemical observations of the recent years are reviewed.
先天性红细胞生成异常性贫血(CDA)在40年前是根据骨髓变化和生化特征进行分类的。CDAⅡ型是CDA最常见的亚组,其一个一致的发现是红细胞膜蛋白存在明显的低糖基化。低糖基化是该疾病的主要原因还是其他病理机制继发的现象仍存在争议。尽管在过去20年中有人提出一些酶缺乏会导致CDAⅡ型,并且在1997年进行了连锁分析将CDAⅡ型基因定位在20号染色体上5厘摩的区域,但该疾病的分子病因仍然未知。在这篇综述中,我们讨论了生化和遗传数据,并回顾了基于近年来生化观察的诊断方法。