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


Journal

[Rinsho ketsueki] The Japanese journal of clinical hematology
ISSN: 0485-1439
Titre abrégé: Rinsho Ketsueki
Pays: Japan
ID NLM: 2984782R

Informations de publication

Date de publication:
2023
Historique:
medline: 8 8 2023
pubmed: 7 8 2023
entrez: 6 8 2023
Statut: ppublish

Résumé

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.

Identifiants

pubmed: 37544724
doi: 10.11406/rinketsu.64.639
doi:

Substances chimiques

Aldehyde Dehydrogenase, Mitochondrial EC 1.2.1.3
Aldehydes 0
Formaldehyde 1HG84L3525
ALDH2 protein, human EC 1.2.1.3

Types de publication

English Abstract Journal Article

Langues

jpn

Sous-ensembles de citation

IM

Pagination

639-645

Auteurs

Minoru Takata (M)

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

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Classifications MeSH