Congenital CNS hypomyelination in the Fig4 null mouse is rescued by neuronal expression of the PI(3,5)P(2) phosphatase Fig4.

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Congenital CNS hypomyelination in the Fig4 null mouse is rescued by neuronal expression of the PI(3,5)P(2) phosphatase Fig4. is …
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scholarly articleQ13442814

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P356DOI10.1523/JNEUROSCI.1482-11.2011
P932PMC publication ID3711465
P698PubMed publication ID22131434

P50authorGuy M. LenkQ37604576
P2093author name stringPeter Shrager
Miriam H Meisler
Cole J Ferguson
Roman J Giger
Jesse J Winters
Vessela I Giger-Mateeva
P2860cites workDeath receptor 6 negatively regulates oligodendrocyte survival, maturation and myelinationQ24310284
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VAC14 nucleates a protein complex essential for the acute interconversion of PI3P and PI(3,5)P(2) in yeast and mouseQ24318746
Astrocytes promote myelination in response to electrical impulsesQ24547572
ArPIKfyve regulates Sac3 protein abundance and turnover: disruption of the mechanism by Sac3I41T mutation causing Charcot-Marie-Tooth 4J disorderQ24621406
The lysosomal sialic acid transporter sialin is required for normal CNS myelinationQ24648839
Myelin gene regulatory factor is a critical transcriptional regulator required for CNS myelinationQ24657511
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
Pathogenic mechanism of the FIG4 mutation responsible for Charcot-Marie-Tooth disease CMT4JQ28478398
Mice lacking sodium channel beta1 subunits display defects in neuronal excitability, sodium channel expression, and nodal architectureQ28506321
Mutation of FIG4 causes neurodegeneration in the pale tremor mouse and patients with CMT4JQ28511673
Molecular basis of the interactions of the Nogo-66 receptor and its homolog NgR2 with myelin-associated glycoprotein: development of NgROMNI-Fc, a novel antagonist of CNS myelin inhibitionQ28567486
Paranodin, a glycoprotein of neuronal paranodal membranesQ28572513
LINGO-1 negatively regulates myelination by oligodendrocytesQ28588853
The Nogo-66 receptor homolog NgR2 is a sialic acid-dependent receptor selective for myelin-associated glycoproteinQ28771372
A gain-of-function mutation in the sodium channel gene Scn2a results in seizures and behavioral abnormalitiesQ49049294
Neuropilin-2 is required in vivo for selective axon guidance responses to secreted semaphorins.Q52170285
Dependence of nodal sodium channel clustering on paranodal axoglial contact in the developing CNS.Q52174918
The relationship between internodal length and fibre diameter in the spinal cord of the cat.Q52436528
A program for simulation of nerve equations with branching geometries.Q52538070
Astrocytes, but not olfactory ensheathing cells or Schwann cells, promote myelination of CNS axons in vitro.Q53496575
Changing phosphoinositides "on the fly": how trafficking vesicles avoid an identity crisis.Q54724417
A computational test of the requirements for conduction in demyelinated axonsQ84063207
PI(3,5)P2 controls membrane trafficking by direct activation of mucolipin Ca2+ release channels in the endolysosomeQ29543488
Oligodendrocytes: biology and pathologyQ30051584
Induction of myelination in the central nervous system by electrical activity.Q30453890
Negative regulation of central nervous system myelination by polysialylated-neural cell adhesion moleculeQ30454391
Mutation of FIG4 causes a rapidly progressive, asymmetric neuronal degenerationQ30489465
Update on genetic disorders affecting white matterQ30628463
Animal models for autoimmune demyelinating disorders of the nervous systemQ33826625
Distinctive genetic and clinical features of CMT4J: a severe neuropathy caused by mutations in the PI(3,5)P₂ phosphatase FIG4.Q34195421
Cell death and control of cell survival in the oligodendrocyte lineageQ34460930
The molecular basis of mucolipidosis type IV.Q34748134
Progeny of Olig2-expressing progenitors in the gray and white matter of the adult mouse cerebral cortex.Q34848711
Deleterious variants of FIG4, a phosphoinositide phosphatase, in patients with ALS.Q34913669
Phosphatidylinositol 3,5-bisphosphate and Fab1p/PIKfyve underPPIn endo-lysosome functionQ34959368
Astrocytic tissue inhibitor of metalloproteinase-1 (TIMP-1) promotes oligodendrocyte differentiation and enhances CNS myelinationQ34960361
Defective autophagy in neurons and astrocytes from mice deficient in PI(3,5)P2.Q35005794
Differentiation and death of premyelinating oligodendrocytes in developing rodent brainQ36273807
Control of local protein synthesis and initial events in myelination by action potentialsQ36350920
Optical measurement of conduction in single demyelinated axonsQ36410502
Autosomal-recessive forms of demyelinating Charcot-Marie-Tooth diseaseQ36507147
Distinct stages of myelination regulated by gamma-secretase and astrocytes in a rapidly myelinating CNS coculture systemQ37116136
Allopregnanolone treatment delays cholesterol accumulation and reduces autophagic/lysosomal dysfunction and inflammation in Npc1-/- mouse brainQ37179554
Integrin-mediated axoglial interactions initiate myelination in the central nervous systemQ37263985
Disruption of Nectin-like 1 cell adhesion molecule leads to delayed axonal myelination in the CNS.Q37309625
Endothelin-1 regulates oligodendrocyte developmentQ37368275
Phosphatidylinositol metabolism and membrane fusionQ37386315
Mechanisms and consequences of impaired lipid trafficking in Niemann-Pick type C1-deficient mammalian cellsQ37470965
Neurodegeneration in Niemann-Pick Type C disease and Huntington's disease: impact of defects in membrane traffickingQ37550648
The complex world of oligodendroglial differentiation inhibitorsQ37869047
Phosphatidylinositol-3,5-bisphosphate: no longer the poor PIP2.Q37898748
Control of oligodendrocyte number in the developing rat optic nerveQ40753787
Myelin-associated glycoprotein is a myelin signal that modulates the caliber of myelinated axons.Q41712768
Anatomy of rat semaphorin III/collapsin-1 mRNA expression and relationship to developing nerve tracts during neuroembryogenesisQ42526905
Neuron-specific expression of a hamster prion protein minigene in transgenic mice induces susceptibility to hamster scrapie agentQ42677331
Transgenic mice expressing beta-galactosidase in mature neurons under neuron-specific enolase promoter controlQ46168479
Notch receptor activation inhibits oligodendrocyte differentiationQ47713081
Axonal coding of action potentials in demyelinated nerve fibersQ48233190
P4510describes a project that usesImageJQ1659584
P433issue48
P407language of work or nameEnglishQ1860
P921main subjectcongenital disorderQ727096
P304page(s)17736-17751
P577publication date2011-11-01
P1433published inJournal of NeuroscienceQ1709864
P1476titleCongenital CNS hypomyelination in the Fig4 null mouse is rescued by neuronal expression of the PI(3,5)P(2) phosphatase Fig4
P478volume31

Reverse relations

cites work (P2860)
Q44216960ALS-associated protein FIG4 is localized in Pick and Lewy bodies, and also neuronal nuclear inclusions, in polyglutamine and intranuclear inclusion body diseases.
Q91444964Cerebral hypomyelination associated with biallelic variants of FIG4
Q36592491Fig4 deficiency: a newly emerged lysosomal storage disorder?
Q89452029Gene therapies for axonal neuropathies: Available strategies, successes to date, and what to target next
Q91194191Identification of CR43467 encoding a long non-coding RNA as a novel genetic interactant with dFIG4, a CMT-causing gene
Q28571064LDL receptor-related protein-1 is a sialic-acid-independent receptor for myelin-associated glycoprotein that functions in neurite outgrowth inhibition by MAG and CNS myelin.
Q47130977LRP1 regulates peroxisome biogenesis and cholesterol homeostasis in oligodendrocytes and is required for proper CNS myelin development and repair.
Q42133744Loss of PIKfyve in platelets causes a lysosomal disease leading to inflammation and thrombosis in mice
Q33764669Mouse models of PI(3,5)P2 deficiency with impaired lysosome function.
Q37351972Murine Fig4 is dispensable for muscle development but required for muscle function
Q30523191Neuronal expression of Fig4 is both necessary and sufficient to prevent spongiform neurodegeneration.
Q34672075Npc1 acting in neurons and glia is essential for the formation and maintenance of CNS myelin
Q36957125PI(3,5)P2 biosynthesis regulates oligodendrocyte differentiation by intrinsic and extrinsic mechanisms
Q26823539Phosphatidylinositol 3,5-bisphosphate: low abundance, high significance
Q27012953Phosphoinositides: tiny lipids with giant impact on cell regulation
Q50488733Preferential conduction block of myelinated axons by nitric oxide.
Q52311182Protective Role of the Lipid Phosphatase Fig4 in the Adult Nervous System.
Q30696655Rescue of neurodegeneration in the Fig4 null mouse by a catalytically inactive FIG4 transgene
Q28508873Ten-m3 is required for the development of topography in the ipsilateral retinocollicular pathway
Q36031613Th Cell Diversity in Experimental Autoimmune Encephalomyelitis and Multiple Sclerosis
Q50432878The DYT6 Dystonia Protein THAP1 Regulates Myelination within the Oligodendrocyte Lineage.
Q90957586The K2P -channel TASK1 affects Oligodendroglial differentiation but not myelin restoration
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Q38634867The extended human PTPome: a growing tyrosine phosphatase family
Q30362381Transient auditory nerve demyelination as a new mechanism for hidden hearing loss.
Q36817367Yunis-Varón syndrome is caused by mutations in FIG4, encoding a phosphoinositide phosphatase