Atypical parkinsonism–associated retromer mutant alters endosomal sorting of specific cargo proteins

Mutations in the retromer complex, which is involved in sorting integral membrane proteins from endosomes to cellular compartments, are associated with atypical parkinsonism, but how these mutations affect retromer function remains unclear. Through a quantitative proteomic analysis of the retromer interactome, McMillan et al. reveal a new mechanism for perturbed endosomal sorting in parkinsonism.

• Publication year

  2016

• Venue

  Journal of Cell Biology

• Publication date

  2016-08-15

• Fields of study

  Biology, Medicine

• Identifiers
  DOI 10.1083/jcb.201604057PMID 27528657PMCID 4987296
• External record

  Open on Semantic Scholar

• Source metadata

  Semantic Scholar, PubMed

• No claims are published for this paper.

• No concepts are published for this paper.

• Sorting nexin 27 couples PTHR trafficking to retromer for signal regulation in osteoblasts during bone growth

  2016cited by this paper
• COMMD1 is linked to the WASH complex and regulates endosomal trafficking of the copper transporter ATP7A

  2015cited by this paper
• Rab5-family guanine nucleotide exchange factors bind retromer and promote its recruitment to endosomes

  2015cited by this paper
• A defect in the retromer accessory protein, SNX27, manifests by infantile myoclonic epilepsy and neurodegeneration

  2015cited by this paper
• De novo deleterious genetic variations target a biological network centered on Aβ peptide in early-onset Alzheimer disease

  2015cited by this paper
• Genetic variability of the retromer cargo recognition complex in parkinsonism

  2015cited by this paper
• Genetic variation of the retromer subunits VPS26A/B-VPS29 in Parkinson's disease.

  2014cited by this paper
• Identification of molecular heterogeneity in SNX27–retromer-mediated endosome-to-plasma-membrane recycling

  2014cited by this paper
• RME-8 coordinates the activity of the WASH complex with the function of the retromer SNX dimer to control endosomal tubulation

  2014influential reference
• VARP Is Recruited on to Endosomes by Direct Interaction with Retromer, Where Together They Function in Export to the Cell Surface

  2014cited by this paper
• esyN: Network Building, Sharing and Publishing

  2014cited by this paper
• Retromer: a master conductor of endosome sorting.

  2014cited by this paper
• DNAJC13 mutations in Parkinson disease.

  2014cited by this paper
• Retromer Binding to FAM21 and the WASH Complex Is Perturbed by the Parkinson Disease-Linked VPS35(D620N) Mutation

  2014cited by this paper
• A unique PDZ domain and arrestin-like fold interaction reveals mechanistic details of endocytic recycling by SNX27-retromer

  2014cited by this paper
• Mutation in VPS35 associated with Parkinson’s disease impairs WASH complex association and inhibits autophagy

  2014cited by this paper
• Loss of sorting nexin 27 contributes to excitatory synaptic dysfunction via modulation of glutamate receptor recycling in Down syndrome

  2013influential reference
• RAB7L1 interacts with LRRK2 to modify intraneuronal protein sorting and Parkinson's disease risk.

  2013cited by this paper
• A global analysis of SNX27–retromer assembly and cargo specificity reveals a function in glucose and metal ion transport

  2013cited by this paper
• Multivariate proteomic profiling identifies novel accessory proteins of coated vesicles

  2012cited by this paper
• SNX17 protects integrins from degradation by sorting between lysosomal and recycling pathways

  2012cited by this paper
• Recruitment of the endosomal WASH complex is mediated by the extended 'tail' of Fam21 binding to the retromer protein Vps35.

  2012cited by this paper
• The retromer complex – endosomal protein recycling and beyond

  2012cited by this paper
• Identification of Alzheimer disease-associated variants in genes that regulate retromer function.

  2012cited by this paper
• SNX27 mediates retromer tubule entry and endosome-to-plasma membrane trafficking of signaling receptors

  2011cited by this paper
• Polycystin-2 takes different routes to the somatic and ciliary plasma membrane

  2011cited by this paper
• Mechanism underlying selective regulation of G protein-gated inwardly rectifying potassium channels by the psychostimulant-sensitive sorting nexin 27

  2011cited by this paper
• Sorting nexins provide diversity for retromer-dependent trafficking events

  2011cited by this paper
• VPS35 mutations in Parkinson disease.

  2011cited by this paper
• A mutation in VPS35, encoding a subunit of the retromer complex, causes late-onset Parkinson disease.

  2011cited by this paper
• Deficiency of Sorting Nexin 27 (SNX27) Leads to Growth Retardation and Elevated Levels of N-Methyl-d-Aspartate Receptor 2C (NR2C)

  2011cited by this paper
• The cell biology of polycystic kidney disease

  2010cited by this paper
• Family-wide characterization of the DENN domain Rab GDP-GTP exchange factors

  2010cited by this paper
• The cargo-selective retromer complex is a recruiting hub for protein complexes that regulate endosomal tubule dynamics

  2010cited by this paper
• SNX27 mediates PDZ-directed sorting from endosomes to the plasma membrane

  2010cited by this paper
• A FAM21-containing WASH complex regulates retromer-dependent sorting.

  2009cited by this paper
• Regulation of endosomal clathrin and retromer‐mediated endosome to Golgi retrograde transport by the J‐domain protein RME‐8

  2009cited by this paper
• Membrane recruitment of the cargo-selective retromer subcomplex is catalysed by the small GTPase Rab7 and inhibited by the Rab-GAP TBC1D5

  2009cited by this paper
• Retromer deficiency observed in Alzheimer's disease causes hippocampal dysfunction, neurodegeneration, and Aβ accumulation

  2008cited by this paper
• The retromer complex and clathrin define an early endosomal retrograde exit site

  2007cited by this paper
• Model‐guided microarray implicates the retromer complex in Alzheimer's disease

  2005cited by this paper
• A Novel Mammalian Retromer Component, Vps26B

  2005cited by this paper
• Role of the mammalian retromer in sorting of the cation-independent mannose 6-phosphate receptor

  2004cited by this paper
• New sorting nexin (SNX27) and NHERF specifically interact with the 5-HT4(a) receptor splice variant: roles in receptor targeting

  2004cited by this paper
• Cargo-selective endosomal sorting for retrieval to the Golgi requires retromer

  2004cited by this paper
• A Membrane Coat Complex Essential for Endosome-to-Golgi Retrograde Transport in Yeast

  1998cited by this paper

• Identification of a VPS29 isoform with restricted association to Retriever and Retromer accessory proteins through autoinhibition

  2025influential citation
• Endosomal protein DENND10 promotes developmental competence of neurite extension

  2025cites this paper
• MicroRNAs and rs1803274 SNP-based BuChe downregulation are associated with metabolic syndrome through ghrelin hydrolysis and expression quantitative trait loci regulation in PD patients

  2025cites this paper
• VPS29C: Adding complexity to the Retromer complex

  2025cites this paper
• Mapping of endosomal proximity proteomes reveals Retromer as a hub for RAB GTPase regulation

  2025cites this paper
• Hemochromatosis neural archetype reveals iron disruption in motor circuits

  2024cites this paper
• The Entamoeba histolytica Vps26 (EhVps26) retromeric protein is involved in phagocytosis: Bioinformatic and experimental approaches

  2024cites this paper
• Sorting of cargo in the tubular endosomal network

  2022cites this paper
• The Retromer Complex: From Genesis to Revelations.

  2021cites this paper
• Toward Understanding the Molecular Role of SNX27/Retromer in Human Health and Disease

  2021cites this paper
• SNX27-driven membrane localisation of OTULIN antagonises linear ubiquitination and NF-κB signalling activation

  2021cites this paper
• NEK1-mediated retromer trafficking promotes blood–brain barrier integrity by regulating glucose metabolism and RIPK1 activation

  2021cites this paper
• FERARI and cargo adaptors coordinate cargo flow through sorting endosomes

  2021cites this paper
• Ca2+ administration prevents α-synuclein proteotoxicity by stimulating calcineurin-dependent lysosomal proteolysis

  2021cites this paper
• The Dopamine Transporter Recycles via a Retromer-Dependent The Dopamine Transporter Recycles via a Retromer-Dependent Postendocytic Mechanism: Tracking Studies Using a Novel Postendocytic Mechanism: Tracking Studies Using a Novel Fluorophore-Coupling Approach Fluorophore-Coupling Approach

  2021cites this paper
• FERARI and cargo adaptors coordinate cargo �ow through sorting endosomes

  2021cites this paper
• Explosion in the complexity of membrane protein recycling.

  2020cites this paper
• Unveiling the cryo-EM structure of retromer.

  2020cites this paper
• De novo macrocyclic peptides for inhibiting, stabilizing, and probing the function of the retromer endosomal trafficking complex

  2020cites this paper
• Acute inactivation of retromer and ESCPE-1 leads to time-resolved defects in endosomal cargo sorting

  2020cites this paper
• Sorting nexin-27 regulates AMPA receptor trafficking through the synaptic adhesion protein LRFN2

  2020cites this paper
• Neuronal functions of clathrin-associated endocytic sorting adaptors – from molecules to disease

  2020cites this paper
• Acute perturbation of retromer and ESCPE-1 leads to functionally distinct and temporally resolved defects in endosomal cargo sorting

  2019cites this paper
• Unraveling Novel Mechanisms of Neurodegeneration Through a Large-Scale Forward Genetic Screen in Drosophila

  2019cites this paper
• Endocytic iron trafficking and mitochondria in Parkinson's disease.

  2019cites this paper
• Coatopathies: Genetic Disorders of Protein Coats.

  2019cites this paper
• Sorting nexin 27 rescues neuroligin 2 from lysosomal degradation to control inhibitory synapse number.

  2019cites this paper
• SNX3-retromer requires an evolutionary conserved MON2:DOPEY2:ATP9A complex to mediate Wntless sorting and Wnt secretion

  2018cites this paper
• Sorting nexin-21 is a scaffold for the endosomal recruitment of huntingtin

  2018cites this paper
• Epithelial Na+ Channel: Reciprocal Control by COMMD10 and Nedd4-2

  2018cites this paper
• Alteration of endosomal trafficking is associated with early-onset parkinsonism caused by SYNJ1 mutations

  2018cites this paper
• Retromer associates with the cytoplasmic amino-terminus of polycystin-2

  2018influential citation
• Sequence-dependent cargo recognition by SNX-BARs mediates retromer-independent transport of CI-MPR

  2017cites this paper
• Disorders in Hepatic Copper Secretion: Wilson's Disease and Pleomorphic Syndromes

  2017cites this paper
• Membrane protein trafficking in Drosophila photoreceptor cells.

  2017cites this paper
• The Dopamine Transporter Recycles via a Retromer-Dependent Postendocytic Mechanism: Tracking Studies Using a Novel Fluorophore-Coupling Approach

  2017cites this paper
• The emerging role of retromer in neuroprotection

  2017influential citation
• Retriever is a multiprotein complex for retromer-independent endosomal cargo recycling

  2017cites this paper
• Biglycan expression in the melanoma microenvironment promotes invasiveness via increased tissue stiffness inducing integrin-β1 expression

  2017cites this paper
• SUMOylation of FOXP1 regulates transcriptional repression via CtBP1 to drive dendritic morphogenesis

  2017cites this paper
• Sequence-dependent cargo recognition by SNX-BARs mediates retromer-independent transport of CI-MPR

  2017cites this paper
• Post-translational modifications: How to modulate Rab7 functions

  2017cites this paper
• GPCR Signaling and Trafficking: The Long and Short of It.

  2017cites this paper
• SNX27 REGULATES GPR17 RECYCLING AND THE KINETICS OF OLIGODENDROCYTE DIFFERENTIATION: IMPLICATION IN DOWN SYNDROME

  2017cites this paper
• Novel Mechanisms Regulating Dopamine Transporter Endocytic Trafficking: Ack1-Controlled Endocytosis And Retromer-Mediated Recycling

  2017cites this paper
• Parkinson’s Disease-associated proteins: studying their role in mitophagy with cellular and Drosophila models

  2017cites this paper
• Retriever , a multiprotein complex for retromer-independent 1 endosomal cargo recycling 2

  2017cites this paper
• Updated Insight into the Physiological and Pathological Roles of the Retromer Complex

  2017cites this paper
• (2017). Sequence-dependent cargo recognition by SNX-BARs mediates retromer-independent transport of CI-MPR. Cell Biology

  2017cites this paper
• Sequence-dependent cargo recognition by SNX-BARs mediates retromer-independent transport of CI-MPR

  2017cites this paper