Myotubularin phosphatases: policing 3-phosphoinositides

Fred Robinson, Jack E. Dixon

Research output: Contribution to journalArticle

164 Citations (Scopus)

Abstract

In eukaryotic cells, phosphatidylinositol is subject to differential phosphorylation, resulting in the production of seven distinct phosphatidylinositol phosphates, often referred to as phosphoinositides (PIs). PIs have numerous distinct roles in cellular regulation and membrane trafficking. Recently, myotubularin family PI 3-phosphatases have emerged as key regulators of phosphatidylinositol 3-phosphate and phosphatidylinositol 3,5-bisphosphate, two PIs that regulate traffic within the endosomal-lysosomal pathway. Mutations in several myotubularin genes lead to myotubular myopathy and Charcot-Marie-Tooth peripheral neuropathy. Strikingly, nearly half of the members of the human myotubularin family appear to be catalytically inactive. Several inactive myotubularins have essential functions in mammals. Recent work in mammalian cells and model organisms is shedding light on the roles of myotubularins in membrane traffic.

Original languageEnglish (US)
Pages (from-to)403-412
Number of pages10
JournalTrends in Cell Biology
Volume16
Issue number8
DOIs
StatePublished - Aug 2006
Externally publishedYes

Fingerprint

Phosphatidylinositols
Phosphoric Monoester Hydrolases
Congenital Structural Myopathies
Phosphatidylinositol Phosphates
Membranes
Eukaryotic Cells
Peripheral Nervous System Diseases
Mammals
Tooth
Phosphorylation
myotubularin
Mutation
Genes

ASJC Scopus subject areas

  • Cell Biology

Cite this

Myotubularin phosphatases : policing 3-phosphoinositides. / Robinson, Fred; Dixon, Jack E.

In: Trends in Cell Biology, Vol. 16, No. 8, 08.2006, p. 403-412.

Research output: Contribution to journalArticle

Robinson, Fred ; Dixon, Jack E. / Myotubularin phosphatases : policing 3-phosphoinositides. In: Trends in Cell Biology. 2006 ; Vol. 16, No. 8. pp. 403-412.
@article{8ff5196cdf1744668ec7449cb8d3e6d5,
title = "Myotubularin phosphatases: policing 3-phosphoinositides",
abstract = "In eukaryotic cells, phosphatidylinositol is subject to differential phosphorylation, resulting in the production of seven distinct phosphatidylinositol phosphates, often referred to as phosphoinositides (PIs). PIs have numerous distinct roles in cellular regulation and membrane trafficking. Recently, myotubularin family PI 3-phosphatases have emerged as key regulators of phosphatidylinositol 3-phosphate and phosphatidylinositol 3,5-bisphosphate, two PIs that regulate traffic within the endosomal-lysosomal pathway. Mutations in several myotubularin genes lead to myotubular myopathy and Charcot-Marie-Tooth peripheral neuropathy. Strikingly, nearly half of the members of the human myotubularin family appear to be catalytically inactive. Several inactive myotubularins have essential functions in mammals. Recent work in mammalian cells and model organisms is shedding light on the roles of myotubularins in membrane traffic.",
author = "Fred Robinson and Dixon, {Jack E.}",
year = "2006",
month = "8",
doi = "10.1016/j.tcb.2006.06.001",
language = "English (US)",
volume = "16",
pages = "403--412",
journal = "Trends in Cell Biology",
issn = "0962-8924",
publisher = "Elsevier Limited",
number = "8",

}

TY - JOUR

T1 - Myotubularin phosphatases

T2 - policing 3-phosphoinositides

AU - Robinson, Fred

AU - Dixon, Jack E.

PY - 2006/8

Y1 - 2006/8

N2 - In eukaryotic cells, phosphatidylinositol is subject to differential phosphorylation, resulting in the production of seven distinct phosphatidylinositol phosphates, often referred to as phosphoinositides (PIs). PIs have numerous distinct roles in cellular regulation and membrane trafficking. Recently, myotubularin family PI 3-phosphatases have emerged as key regulators of phosphatidylinositol 3-phosphate and phosphatidylinositol 3,5-bisphosphate, two PIs that regulate traffic within the endosomal-lysosomal pathway. Mutations in several myotubularin genes lead to myotubular myopathy and Charcot-Marie-Tooth peripheral neuropathy. Strikingly, nearly half of the members of the human myotubularin family appear to be catalytically inactive. Several inactive myotubularins have essential functions in mammals. Recent work in mammalian cells and model organisms is shedding light on the roles of myotubularins in membrane traffic.

AB - In eukaryotic cells, phosphatidylinositol is subject to differential phosphorylation, resulting in the production of seven distinct phosphatidylinositol phosphates, often referred to as phosphoinositides (PIs). PIs have numerous distinct roles in cellular regulation and membrane trafficking. Recently, myotubularin family PI 3-phosphatases have emerged as key regulators of phosphatidylinositol 3-phosphate and phosphatidylinositol 3,5-bisphosphate, two PIs that regulate traffic within the endosomal-lysosomal pathway. Mutations in several myotubularin genes lead to myotubular myopathy and Charcot-Marie-Tooth peripheral neuropathy. Strikingly, nearly half of the members of the human myotubularin family appear to be catalytically inactive. Several inactive myotubularins have essential functions in mammals. Recent work in mammalian cells and model organisms is shedding light on the roles of myotubularins in membrane traffic.

UR - http://www.scopus.com/inward/record.url?scp=33746729798&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=33746729798&partnerID=8YFLogxK

U2 - 10.1016/j.tcb.2006.06.001

DO - 10.1016/j.tcb.2006.06.001

M3 - Article

C2 - 16828287

AN - SCOPUS:33746729798

VL - 16

SP - 403

EP - 412

JO - Trends in Cell Biology

JF - Trends in Cell Biology

SN - 0962-8924

IS - 8

ER -