Fanconi anemia and Aldehyde Degradation Deficiency Syndrome: Metabolism and DNA repair protect the genome and hematopoiesis from endogenous DNA damage

Mu, Anfeng; Hira, Asuka; Mori, Minako; Okamoto, Yusuke; Takata, Minoru

doi:10.1016/j.dnarep.2023.103546

DNA Repair

2023

DOI: 10.1016/j.dnarep.2023.103546

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Fanconi anemia and Aldehyde Degradation Deficiency Syndrome: Metabolism and DNA repair protect the genome and hematopoiesis from endogenous DNA damage

Anfeng Mu,

Asuka Hira,

Minako Mori

et al.

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2024

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Cited by 3 publications

References 58 publications

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The role of SLFN11 in DNA replication stress response and its implications for the Fanconi anemia pathway

Mu,

Okamoto,

Katsuki

et al. 2024

DNA Repair

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The role of SLFN11 in DNA replication stress response and its implications for the Fanconi anemia pathway

Mu,

Okamoto,

Katsuki

et al. 2024

DNA Repair

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Transcription reprogramming and endogenous DNA damage

2024

DNA Repair

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Distinct DNA repair mechanisms prevent formaldehyde toxicity during development, reproduction and aging

Rieckher,

Gallrein,

Alquezar-Artieda

et al. 2024

Nucleic Acids Research

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Formaldehyde (FA) is a recognized environmental and metabolic toxin implicated in cancer development and aging. Inherited mutations in the FA-detoxifying enzymes ADH5 and ALDH2 genes lead to FA overload in the severe multisystem AMeD syndrome. FA accumulation causes genome damage including DNA–protein-, inter- and intra-strand crosslinks and oxidative lesions. However, the influence of distinct DNA repair systems on organismal FA resistance remains elusive. We have here investigated the consequence of a range of DNA repair mutants in a model of endogenous FA overload generated by downregulating the orthologs of human ADH5 and ALDH2 in C. elegans. We have focused on the distinct components of nucleotide excision repair (NER) during developmental growth, reproduction and aging. Our results reveal three distinct modes of repair of FA-induced DNA damage: Transcription-coupled repair (TCR) operating NER-independently during developmental growth or through NER during adulthood, and, in concert with global-genome (GG-) NER, in the germline and early embryonic development. Additionally, we show that the Cockayne syndrome B (CSB) factor is involved in the resolution of FA-induced DNA–protein crosslinks, and that the antioxidant and FA quencher N-acetyl-l-cysteine (NAC) reverses the sensitivity of detoxification and DNA repair defects during development, suggesting a therapeutic intervention to revert FA-pathogenic consequences.

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Fanconi anemia and Aldehyde Degradation Deficiency Syndrome: Metabolism and DNA repair protect the genome and hematopoiesis from endogenous DNA damage

Cited by 3 publications

References 58 publications

The role of SLFN11 in DNA replication stress response and its implications for the Fanconi anemia pathway

The role of SLFN11 in DNA replication stress response and its implications for the Fanconi anemia pathway

Transcription reprogramming and endogenous DNA damage

Distinct DNA repair mechanisms prevent formaldehyde toxicity during development, reproduction and aging

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