[一种新的范可尼贫血样疾病,醛降解缺乏综合征:两种防御机制共同作用于基因组和造血]。

Minoru Takata
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引用次数: 0

摘要

范可尼贫血(FA)是一种遗传性骨髓衰竭综合征,已被认为是由DNA修复缺陷引起的,该缺陷可消除由醛类引起的内源性DNA损伤。在7名临床表现与FA相似的日本再生障碍性贫血儿童中,我们发现了编码甲醛降解酶的ADH5基因的双等位变异和杂合ALDH2变异(rs671)。我们得出结论,ADH5/ALDH2的联合缺陷导致了一种新的疾病,现在称为醛降解缺陷综合征(add)。我们认为这种疾病是由造血细胞分化过程中组蛋白去甲基化产生的甲醛去除缺陷引起的。靶向治疗甲醛可以减少FA和add的造血功能缺陷。
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[A new Fanconi anemia-like disorder, aldehyde degradation deficiency syndrome: two defense mechanisms working together for the genome and hematopoiesis].

Fanconi anemia (FA), a hereditary bone marrow failure syndrome, has been suggested to be caused by a defect in DNA repair that removes endogenous DNA damage due to aldehydes. In seven Japanese children with aplastic anemia who clinically resembled FA, we identified biallelic variants of the ADH5 gene, encoding formaldehyde degrading enzyme, and a heterozygous ALDH2 variant (rs671). We conclude that the combined defects in ADH5/ALDH2 caused a new disorder now termed Aldehyde Degradation Deficiency Syndrome (ADDS). We suggest that this disease is caused by defective removal of formaldehyde produced by histone demethylation during hematopoietic cell differentiation. Therapeutic targeting of formaldehyde may reduce the hematopoietic deficits of FA as well as ADDS.

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