Keyword search (3,448 papers available)


TRAPPing a neurological disorder: from yeast to humans.

Author(s): Lipatova Z, Van Bergen N, Stanga D, Sacher M, Christodoulou J, Segev N

Autophagy. 2020 Mar 02;: Authors: Lipatova Z, Van Bergen N, Stanga D, Sacher M, Christodoulou J, Segev N

Article GUID: 32116085
Deficiencies in vesicular transport mediated by TRAPPC4 are associated with severe syndromic intellectual disability.

Author(s): Van Bergen NJ, Guo Y, Al-Deri N, Lipatova Z, Stanga D, Zhao S, Murtazina R, Gyurkovska V, Pehlivan D, Mitani T, Gezdirici A, Antony J, Colli...

The conserved transport protein particle (TRAPP) complexes regulate key trafficking events and are required for autophagy. TRAPPC4, like its yeast Trs23 orthologue, is a core component of the TRAPP...

Article GUID: 31794024
Characterization of three TRAPPC11 variants suggests a critical role for the extreme carboxy terminus of the protein.

Author(s): Milev MP, Stanga D, Schänzer A, Nascimento A, Saint-Dic D, Ortez C, Benito DN, Barrios DG, Colomer J, Badosa C, Jou C, Gallano P, Gonzalez-Q...

Sci Rep. 2019 Oct 01;9(1):14036 Authors: Milev MP, Stanga D, Schänzer A, Nascimento A, Saint-Dic D, Ortez C, Benito DN, Barrios DG, Colomer J, Badosa C, Jou C, Gallano P, Gonzalez-Quereda L, ...

Article GUID: 31575891
Bi-allelic mutations in TRAPPC2L result in a neurodevelopmental disorder and have an impact on RAB11 in fibroblasts.

Author(s): Milev MP, Graziano C, Karall D, Kuper WFE, Al-Deri N, Cordelli DM, Haack TB, Danhauser K, Iuso A, Palombo F, Pippucci T, Prokisch H, Saint-D...

J Med Genet. 2018 Nov;55(11):753-764 Authors: Milev MP, Graziano C, Karall D, Kuper WFE, Al-Deri N, Cordelli DM, Haack TB, Danhauser K, Iuso A, Palombo F, Pippucci T, Prokisch H, Saint-Dic D, Seri...

Article GUID: 30120216
TRAPPC11 functions in autophagy by recruiting ATG2B-WIPI4/WDR45 to preautophagosomal membranes.

Author(s): Stanga D, Zhao Q, Milev MP, Saint-Dic D, Jimenez-Mallebrera C, Sacher M

Traffic. 2019 May;20(5):325-345 Authors: Stanga D, Zhao Q, Milev MP, Saint-Dic D, Jimenez-Mallebrera C, Sacher M

Article GUID: 30843302
Title:TRAPPC11 functions in autophagy by recruiting ATG2B-WIPI4/WDR45 to preautophagosomal membranes.
Authors:Stanga D,  Zhao Q,  Milev MP,  Saint-Dic D,  Jimenez-Mallebrera C,  Sacher M
Link:https://www.ncbi.nlm.nih.gov/pubmed/30843302?dopt=Abstract
Category:Traffic
PMID:30843302
Dept Affiliation: CONCORDIA
1 Concordia University, Department of Biology, Montreal, Quebec, Canada.
2 University of Montreal, Department of Medicine and Institute for Research in Immunology and Cancer, Montreal, Quebec, Canada.
3 Neuromuscular Unit, Neuropaediatrics Department, Institut de Recerca Sant Joan de Déu, Hospital Sant Joan de Déu and CIBERER, Barcelona, Spain.
4 McGill University, Department of Anatomy and Cell Biology, Quebec, Canada.

Description:

TRAPPC11 functions in autophagy by recruiting ATG2B-WIPI4/WDR45 to preautophagosomal membranes.

Traffic. 2019 May;20(5):325-345

Authors: Stanga D, Zhao Q, Milev MP, Saint-Dic D, Jimenez-Mallebrera C, Sacher M

Abstract

TRAPPC11 has been implicated in membrane traffic and lipid-linked oligosaccharide synthesis, and mutations in TRAPPC11 result in neuromuscular and developmental phenotypes. Here, we show that TRAPPC11 has a role upstream of autophagosome formation during macroautophagy. Upon TRAPPC11 depletion, LC3-positive membranes accumulate prior to, and fail to be cleared during, starvation. A proximity biotinylation assay identified ATG2B and its binding partner WIPI4/WDR45 as TRAPPC11 interactors. TRAPPC11 depletion phenocopies that of ATG2 and WIPI4 and recruitment of both proteins to membranes is defective upon reduction of TRAPPC11. We find that a portion of TRAPPC11 and other TRAPP III proteins localize to isolation membranes. Fibroblasts from a patient with TRAPPC11 mutations failed to recruit ATG2B-WIPI4, suggesting that this interaction is physiologically relevant. Since ATG2B-WIPI4 is required for isolation membrane expansion, our study suggests that TRAPPC11 plays a role in this process. We propose a model whereby the TRAPP III complex participates in the formation and expansion of the isolation membrane at several steps.

PMID: 30843302 [PubMed - in process]