Insights into the regulation of cellular Mn
CDG
Glycosylation
Golgi
Manganese
SPCA1
TMEM165
Journal
Biochimica et biophysica acta. Molecular basis of disease
ISSN: 1879-260X
Titre abrégé: Biochim Biophys Acta Mol Basis Dis
Pays: Netherlands
ID NLM: 101731730
Informations de publication
Date de publication:
08 2023
08 2023
Historique:
received:
08
12
2022
revised:
04
04
2023
accepted:
05
04
2023
medline:
5
6
2023
pubmed:
17
4
2023
entrez:
16
4
2023
Statut:
ppublish
Résumé
Golgi cation homeostasis is known to be crucial for many cellular processes including vesicular fusion events, protein secretion, as well as for the activity of Golgi glycosyltransferases and glycosidases. TMEM165 was identified in 2012 as the first cation transporter related to human glycosylation diseases, namely the Congenital Disorders of Glycosylation (CDG). Interestingly, divalent manganese (Mn) supplementation has been shown to suppress the observed glycosylation defects in TMEM165-deficient cell lines, thus suggesting that TMEM165 is involved in cellular Mn homeostasis. This paper demonstrates that the origin of the Golgi glycosylation defects arises from impaired Golgi Mn homeostasis in TMEM165-depleted cells. We show that Mn supplementation fully rescues the Mn content in the secretory pathway/organelles of TMEM165-depleted cells and hence the glycosylation process. Strong cytosolic and organellar Mn accumulations can also be observed in TMEM165- and SPCA1-depleted cells upon incubation with increasing Mn concentrations, thus demonstrating the crucial involvement of these two proteins in cellular Mn homeostasis. Interestingly, our results show that the cellular Mn homeostasis maintenance in control cells is correlated with the presence of TMEM165 and that the Mn-detoxifying capacities of cells, through the activity of SPCA1, rely on the Mn-induced degradation mechanism of TMEM165. Finally, this paper highlights that TMEM165 is essential in secretory pathway/organelles Mn homeostasis maintenance to ensure both Golgi glycosylation enzyme activities and cytosolic Mn detoxification.
Identifiants
pubmed: 37062452
pii: S0925-4439(23)00083-2
doi: 10.1016/j.bbadis.2023.166717
pmc: PMC10639120
mid: NIHMS1939310
pii:
doi:
Substances chimiques
Manganese
42Z2K6ZL8P
Cation Transport Proteins
0
Antiporters
0
TMEM165 protein, human
0
Types de publication
Journal Article
Research Support, Non-U.S. Gov't
Langues
eng
Sous-ensembles de citation
IM
Pagination
166717Subventions
Organisme : NIGMS NIH HHS
ID : R01 GM083144
Pays : United States
Informations de copyright
Copyright © 2023. Published by Elsevier B.V.
Déclaration de conflit d'intérêts
Declaration of competing interest The authors declare no conflict of interest and all authors were involved in the decision to publish and reviewed the article before submission.
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