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[一种新的范可尼贫血样疾病,醛降解缺陷综合征:两种共同作用于基因组和造血作用的防御机制]

[A new Fanconi anemia-like disorder, aldehyde degradation deficiency syndrome: two defense mechanisms working together for the genome and hematopoiesis].

作者信息

Takata Minoru

机构信息

Laboratory of DNA Damage Signaling, Department of Late Effects Studies, Radiation Biology Center, Graduate School of Biostudies, Kyoto University.

出版信息

Rinsho Ketsueki. 2023;64(7):639-645. doi: 10.11406/rinketsu.64.639.

Abstract

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.

摘要

范可尼贫血(FA)是一种遗传性骨髓衰竭综合征,有人认为它是由DNA修复缺陷引起的,这种缺陷会消除因醛类导致的内源性DNA损伤。在7名临床症状类似FA的日本再生障碍性贫血儿童中,我们鉴定出了编码甲醛降解酶的ADH5基因的双等位基因变异以及一个杂合的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,现称为醛降解缺陷综合征(ADDS)。我们认为,这种疾病是由于造血细胞分化过程中组蛋白去甲基化产生的甲醛清除缺陷所致。针对甲醛的治疗靶点可能会减轻FA以及ADDS的造血功能缺陷。

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