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