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Cited by 35Neurogenesis and neuroplasticity mechanismsCellular transport and secretionSphingolipid Metabolism and Signaling

PI(3,5)P2 biosynthesis regulates oligodendrocyte differentiation by intrinsic and extrinsic mechanisms

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Mironova, Yevgeniya A, Lenk, Guy M, Lin, Jing-Ping, Lee, Seung Joon, Twiss, Jeffery L, Vaccari, Ilaria, Bolino, Alessandra, Havton, Leif A +5 more·2016·eLife
DOI 10.7554/elife.13023PMC PMC4889328
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Abstract

Proper development of the CNS axon-glia unit requires bi-directional communication between axons and oligodendrocytes (OLs). We show that the signaling lipid phosphatidylinositol-3,5-bisphosphate [PI(3,5)P2] is required in neurons and in OLs for normal CNS myelination. In mice, mutations of Fig4, Pikfyve or Vac14, encoding key components of the PI(3,5)P2 biosynthetic complex, each lead to impaired OL maturation, severe CNS hypomyelination and delayed propagation of compound action potentials. Primary OLs deficient in Fig4 accumulate large LAMP1+ and Rab7+ vesicular structures and exhibit reduced membrane sheet expansion. PI(3,5)P2 deficiency leads to accumulation of myelin-associated glycoprotein (MAG) in LAMP1+perinuclear vesicles that fail to migrate to the nascent myelin sheet. Live-cell imaging of OLs after genetic or pharmacological inhibition of PI(3,5)P2 synthesis revealed impaired trafficking of plasma membrane-derived MAG through the endolysosomal system in primary cells and brain tissue. Collectively, our studies identify PI(3,5)P2 as a key regulator of myelin membrane trafficking and myelinogenesis.

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  • Phosphoinositides including PIP2 are abundant in CNS myelin and have been implicated in myelin protein trafficking, cytoskeletal coupling, and paranodal organization.

    “Beyond microglia, phosphoinositides including PIP2 are abundant in CNS myelin and have been implicated in myelin protein trafficking, cytoskeletal coupling, and paranodal organization [38–41]”

    From PLCγ2 deficiency compromises systemic immune tolerance and erodes myelin homeostasis while enhancing oxidative metabolism in the mouse brain (2026) · cites this paper as [40]

    The Discussion (page unavailable) identifies PI(3,5)P2 as important for trafficking myelin proteins through the endolysosomal system. Our copy does not establish that phosphoinositides, including PIP2, are abundant in CNS myelin or implicate them in cytoskeletal coupling or paranodal organization. (LLM, conf=0.86)

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Authors
Mironova, Y. A., Lenk, G. M., Lin, J.-P., Lee, S. J., Twiss, J. L., Vaccari, I., Bolino, A., Havton, L. A., Min, S. H., Abrams, C. S., Shrager, P., Meisler, M. H., & Giger, R. J.
Affiliations
  • University of Michigan (US)
  • University of South Carolina (US)
  • IRCCS Ospedale San Raffaele (IT)
  • University of California, Los Angeles (US)
  • University of Pennsylvania (US)
  • University of Rochester Medicine (US)
Corresponding author
Giger, Roman J
ORCID
Venue
eLife
Publisher
eLife Sciences Publications, Ltd
Published
2016-03-23
Volume
5
ISSN
2050-084X (ISSN-L)
DOI
10.7554/elife.13023
PMC
PMC4889328
OpenAlex
W2317771024
Cited by
35
References
81
Citations by year
2026: 4 · 2025: 2 · 2024: 1 · 2023: 4 · 2022: 5 · 2021: 2 · 2020: 4 · 2019: 6 · 2018: 5 · 2017: 2
FWCI
2.21
Type
journal article
Language
en
Open access
Licence
Locations
  • eLife (publishedVersion, cc-by)
  • Europe PMC (PubMed Central) (publishedVersion, cc-by)
  • PubMed Central (publishedVersion, cc-by)
  • PubMed (publishedVersion)
  • Archivio Istituzionale della Ricerca - Università Vita-Salute San Raffaele (submittedVersion, cc-by)
  • DOAJ (DOAJ: Directory of Open Access Journals) (submittedVersion, cc-by-sa)
Funders
  • National Institute of Child Health and Human Development (T32HD007505)
  • National Institute of General Medical Sciences (T32GM007315, R01GM24872)
  • Dr. Miriam and Sheldon G. Adelson Medical Research Foundation (APNRR)
  • National Heart, Lung, and Blood Institute (HL040387, HL120846)
  • National Institute of Neurological Disorders and Stroke (R01NS081281)
  • Schmitt Program on Integrative Brain Research
Topics
  • Life Sciences › Neuroscience › Developmental Neuroscience › Neurogenesis and neuroplasticity mechanisms
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Cell Biology › Cellular transport and secretion
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Sphingolipid Metabolism and Signaling
Keywords
phosphatidylinositol 3,5-bisphosphate, oligodendrocyte differentiation, myelination, myelin-associated glycoprotein, FIG4, PIKfyve, VAC14, LAMP1, Rab7, endolysosomal trafficking, hypomyelination
MeSH terms
Animals, Cell Differentiation, Myelin Sheath, Neurons, Oligodendroglia, Gene Deletion, Phosphatidylinositol Phosphates, Mice

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