Structures of the mannose-6-phosphate pathway enzyme, GlcNAc-1-phosphotransferase.
GlcNAc-1-phosphotransferase
lysosome
mannose-6-phosphate
Journal
Proceedings of the National Academy of Sciences of the United States of America
ISSN: 1091-6490
Titre abrégé: Proc Natl Acad Sci U S A
Pays: United States
ID NLM: 7505876
Informations de publication
Date de publication:
16 08 2022
16 08 2022
Historique:
entrez:
8
8
2022
pubmed:
9
8
2022
medline:
11
8
2022
Statut:
ppublish
Résumé
The mannose-6-phosphate (M6P) pathway is responsible for the transport of hydrolytic enzymes to lysosomes. N-acetylglucosamine-1-phosphotransferase (GNPT) catalyzes the first step of tagging these hydrolases with M6P, which when recognized by receptors in the Golgi diverts them to lysosomes. Genetic defects in the GNPT subunits, GNPTAB and GNPTG, cause the lysosomal storage diseases mucolipidosis types II and III. To better understand its function, we determined partial three-dimensional structures of the GNPT complex. The catalytic domain contains a deep cavity for binding of uridine diphosphate-
Identifiants
pubmed: 35939698
doi: 10.1073/pnas.2203518119
pmc: PMC9388126
doi:
Substances chimiques
Mannosephosphates
0
mannose-6-phosphate
3672-15-9
Transferases (Other Substituted Phosphate Groups)
EC 2.7.8.-
GNPTAB protein, human
EC 2.7.8.15
Types de publication
Journal Article
Research Support, U.S. Gov't, Non-P.H.S.
Research Support, N.I.H., Extramural
Research Support, Non-U.S. Gov't
Langues
eng
Sous-ensembles de citation
IM
Pagination
e2203518119Subventions
Organisme : CIHR
ID : PJT-173264
Pays : Canada
Organisme : CIHR
ID : MFE-164773
Pays : Canada
Organisme : NIGMS NIH HHS
ID : P30 GM124165
Pays : United States
Organisme : NIH HHS
ID : S10 OD021527
Pays : United States
Organisme : NIH HHS
ID : S10 OD021832
Pays : United States
Organisme : NIGMS NIH HHS
ID : P30 GM124169
Pays : United States
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