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Article

FIG4 regulates lysosome membrane homeostasis independent of phosphatase function.

Human molecular genetics - 15 Feb 2016

Bharadwaj Rajnish, Cunningham Kathleen M, Zhang Ke, Lloyd Thomas E

Abstract excerpt

FIG4 is a phosphoinositide phosphatase that is mutated in several diseases including Charcot-Marie-Tooth Disease 4J (CMT4J) and Yunis-Varon syndrome (YVS). To investigate the mechanism of disease pathogenesis, we generated Drosophila models of FIG4-related diseases. Fig4 null mutant animals are viable but exhibit marked enlargement of the lysosomal compartment in muscle cells and neurons, accompanied by an...

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Identifiers

PMID: 26662798
DOI: 10.1093/hmg/ddv505
PMCID: PMC4743688

Genes

FIG4

Provenance

PubMed metadata and Literature Corpus relationships. Corpus snapshot updated 7 Sept 2026. Manifest publication-graph-cohort-2026-09-07.1.

Source receipt: pubmed, retrieved 7 Sept 2026.

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FIG4 regulates lysosome membrane homeostasis independent of phosphatase function.PMID 26662798Read ↗

Yunis-Varón syndrome is caused by mutations in FIG4, encoding a phosphoinositide phosphatase.Semantic proximity Read ↗

Protective role of the lipid phosphatase Fig4 in the adult nervous system.Semantic proximity Read ↗

Rescue of neurodegeneration in the Fig4 null mouse by a catalytically inactive FIG4 transgene.Semantic proximity Read ↗

Mutation of FIG4 causes a rapidly progressive, asymmetric neuronal degeneration.Semantic proximity Read ↗

The Lysosomal Storage Disorder Due to fig4a Mutation Causes Robust Liver Vacuolation in Zebrafish.Semantic proximity Read ↗

Mouse models of PI(3,5)P2 deficiency with impaired lysosome function.Semantic proximity Read ↗

Mutation of FIG4 causes neurodegeneration in the pale tremor mouse and patients with CMT4J.Semantic proximity Read ↗

Novel FIG4 mutations in Yunis-Varon syndrome.Semantic proximity Read ↗

Altered phenotypes due to genetic interaction between the mouse phosphoinositide biosynthesis genes Fig4 and Pip4k2c.Semantic proximity Read ↗

Deleterious variants of FIG4, a phosphoinositide phosphatase, in patients with ALS.Semantic proximity Read ↗

Pathogenic mechanism of the FIG4 mutation responsible for Charcot-Marie-Tooth disease CMT4J.Semantic proximity Read ↗

Loss-of-function mutation in Hippo suppressed enlargement of lysosomes and neurodegeneration caused by dFIG4 knockdown.Semantic proximity Read ↗

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