FAM111A regulates replication origin activation and cell fitness.
Journal
Life science alliance
ISSN: 2575-1077
Titre abrégé: Life Sci Alliance
Pays: United States
ID NLM: 101728869
Informations de publication
Date de publication:
12 2023
12 2023
Historique:
received:
25
04
2023
revised:
19
09
2023
accepted:
19
09
2023
medline:
6
10
2023
pubmed:
5
10
2023
entrez:
4
10
2023
Statut:
epublish
Résumé
FAM111A is a replisome-associated protein and dominant mutations within its trypsin-like peptidase domain are linked to severe human developmental syndrome, the Kenny-Caffey syndrome. However, FAM111A functions remain unclear. Here, we show that FAM111A facilitates efficient activation of DNA replication origins. Upon hydroxyurea treatment, FAM111A-depleted cells exhibit reduced single-stranded DNA formation and a better survival rate. Unrestrained expression of FAM111A WT and patient mutants causes accumulation of DNA damage and cell death, only when the peptidase domain remains intact. Unrestrained expression of FAM111A WT also causes increased single-stranded DNA formation that relies on S phase entry, FAM111A peptidase activity but not its binding to proliferating cell nuclear antigen. Altogether, these data unveil how FAM111A promotes DNA replication under normal conditions and becomes harmful in a disease context.
Identifiants
pubmed: 37793778
pii: 6/12/e202302111
doi: 10.26508/lsa.202302111
pmc: PMC10551639
pii:
doi:
Substances chimiques
DNA, Single-Stranded
0
Peptide Hydrolases
EC 3.4.-
FAM111A protein, human
0
Receptors, Virus
0
Types de publication
Journal Article
Research Support, Non-U.S. Gov't
Langues
eng
Sous-ensembles de citation
IM
Subventions
Organisme : Cancer Research UK
ID : C57404/A21782
Pays : United Kingdom
Organisme : Wellcome Trust
ID : 206293/Z/17/Z
Pays : United Kingdom
Organisme : Medical Research Council
ID : MC_UU_00007/15
Pays : United Kingdom
Informations de copyright
© 2023 Rios-Szwed et al.
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