FIG4 regulates lysosome membrane homeostasis independent of phosphatase function

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FIG4 regulates lysosome membrane homeostasis independent of phosphatase function is …
instance of (P31):
scholarly articleQ13442814

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P356DOI10.1093/HMG/DDV505
P932PMC publication ID4743688
P698PubMed publication ID26662798

P50authorThomas E. LloydQ55712562
Kathleen M CunninghamQ85994577
P2093author name stringKe Zhang
Rajnish Bharadwaj
P2860cites workMucolipidosis type IV is caused by mutations in a gene encoding a novel transient receptor potential channelQ24290404
The Human Phosphatidylinositol Phosphatase SAC1 Interacts with the Coatomer I ComplexQ24296999
Core protein machinery for mammalian phosphatidylinositol 3,5-bisphosphate synthesis and turnover that regulates the progression of endosomal transport. Novel Sac phosphatase joins the ArPIKfyve-PIKfyve complexQ24307982
VAC14 nucleates a protein complex essential for the acute interconversion of PI3P and PI(3,5)P(2) in yeast and mouseQ24318746
The phosphoinositide kinase PIKfyve/Fab1p regulates terminal lysosome maturation in Caenorhabditis elegansQ24548664
ArPIKfyve regulates Sac3 protein abundance and turnover: disruption of the mechanism by Sac3I41T mutation causing Charcot-Marie-Tooth 4J disorderQ24621406
A mutation in VPS35, encoding a subunit of the retromer complex, causes late-onset Parkinson diseaseQ24633417
Assembly of a Fab1 phosphoinositide kinase signaling complex requires the Fig4 phosphoinositide phosphataseQ24643186
Fab1 phosphatidylinositol 3-phosphate 5-kinase controls trafficking but not silencing of endocytosed receptorsQ24671705
The Vac14p-Fig4p complex acts independently of Vac7p and couples PI3,5P2 synthesis and turnoverQ24683845
Phosphoinositides in cell regulation and membrane dynamicsQ27861051
Vacuole Size Control: Regulation of PtdIns(3,5)P2Levels by the Vacuole-associated Vac14-Fig4 Complex, a PtdIns(3,5)P2-specific PhosphataseQ27933521
Mammalian cell morphology and endocytic membrane homeostasis require enzymatically active phosphoinositide 5-kinase PIKfyveQ28210351
Recent advances in retromer biologyQ28308202
Pathogenic mechanism of the FIG4 mutation responsible for Charcot-Marie-Tooth disease CMT4JQ28478398
Mutation of FIG4 causes neurodegeneration in the pale tremor mouse and patients with CMT4JQ28511673
PI(3,5)P2 controls membrane trafficking by direct activation of mucolipin Ca2+ release channels in the endolysosomeQ29543488
Identification of functional elements and regulatory circuits by Drosophila modENCODEQ29617551
Cbl-associated protein regulates assembly and function of two tension-sensing structures in DrosophilaQ30451385
Mutant huntingtin impairs post-Golgi trafficking to lysosomes by delocalizing optineurin/Rab8 complex from the Golgi apparatusQ30486228
Neuronal expression of Fig4 is both necessary and sufficient to prevent spongiform neurodegeneration.Q30523191
Rescue of neurodegeneration in the Fig4 null mouse by a catalytically inactive FIG4 transgeneQ30696655
Mutations in the small GTP-ase late endosomal protein RAB7 cause Charcot-Marie-Tooth type 2B neuropathyQ33904567
Distinct pathogenic processes between Fig4-deficient motor and sensory neurons.Q34171101
Distinctive genetic and clinical features of CMT4J: a severe neuropathy caused by mutations in the PI(3,5)P₂ phosphatase FIG4.Q34195421
Molecular reactions of protein phosphatases--insights from structure and chemistryQ34465999
Minos as a genetic and genomic tool in Drosophila melanogasterQ34589640
A synthetic maternal-effect selfish genetic element drives population replacement in DrosophilaQ34613883
Loss of Fig4 in both Schwann cells and motor neurons contributes to CMT4J neuropathyQ34764035
Deleterious variants of FIG4, a phosphoinositide phosphatase, in patients with ALS.Q34913669
Defective autophagy in neurons and astrocytes from mice deficient in PI(3,5)P2.Q35005794
VPS35 mutations in Parkinson diseaseQ35103751
Reactivation of Lysosomal Ca2+ Efflux Rescues Abnormal Lysosomal Storage in FIG4-Deficient CellsQ35547893
Endogenously tagged rab proteins: a resource to study membrane trafficking in DrosophilaQ35606386
The Fab1 phosphatidylinositol kinase pathway in the regulation of vacuole morphology.Q36173537
Fig4 deficiency: a newly emerged lysosomal storage disorder?Q36592491
Yunis-Varón syndrome is caused by mutations in FIG4, encoding a phosphoinositide phosphataseQ36817367
Murine Fig4 is dispensable for muscle development but required for muscle functionQ37351972
Role of the phosphoinositide phosphatase FIG4 gene in familial epilepsy with polymicrogyriaQ37657008
Membrane dynamics and fusion at late endosomes and vacuoles--Rab regulation, multisubunit tethering complexes and SNAREsQ37891288
Phosphatidylinositol-3,5-bisphosphate: no longer the poor PIP2.Q37898748
PIKfyve regulation of endosome-linked pathwaysQ41852844
Motor deficit in a Drosophila model of mucolipidosis type IV due to defective clearance of apoptotic cells.Q42254064
Drosophila larval NMJ dissectionQ42584951
Fig4 expression in the rodent nervous system and its potential role in preventing abnormal lysosomal accumulation.Q51837174
Genetic manipulation and monitoring of autophagy in Drosophila.Q51941406
Electron Microscopy of Drosophila Larval Neuromuscular JunctionsQ52707871
Novel FIG4 mutations in Yunis–Varon syndromeQ59697627
Yunis-varon syndrome: further delineation of cardiovascular and endocrine outcomeQ87416926
P433issue4
P304page(s)681-692
P577publication date2015-12-11
P1433published inHuman Molecular GeneticsQ2720965
P1476titleFIG4 regulates lysosome membrane homeostasis independent of phosphatase function
P478volume25

Reverse relations

cites work (P2860)
Q52329270Autophagy as a common pathway in amyotrophic lateral sclerosis.
Q52315057Control of vacuole membrane homeostasis by a resident PI-3,5-kinase inhibitor.
Q38885442Defective phosphoinositide metabolism in autism
Q91235415Draft genome assembly of Tenualosa ilisha, Hilsa shad, provides resource for osmoregulation studies
Q91194191Identification of CR43467 encoding a long non-coding RNA as a novel genetic interactant with dFIG4, a CMT-causing gene
Q54111267Loss-of-function mutation in Hippo suppressed enlargement of lysosomes and neurodegeneration caused by dFIG4 knockdown.
Q90095321Neuron-specific knockdown of solute carrier protein SLC25A46a induces locomotive defects, an abnormal neuron terminal morphology, learning disability, and shortened lifespan
Q36957125PI(3,5)P2 biosynthesis regulates oligodendrocyte differentiation by intrinsic and extrinsic mechanisms
Q38600541PIKfyve activity regulates reformation of terminal storage lysosomes from endolysosomes
Q97531610The role of lipids in autophagy and its implication in neurodegeneration
Q89543124Whole exome sequencing establishes diagnosis of Charcot-Marie-Tooth 4J, 1C, and X1 subtypes
Q47723480Yunis-Varón syndrome caused by biallelic VAC14 mutations

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