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PMID: 16314489 Published · ppublish English Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, Non-P.H.S.

Inactivation of NF1 in CNS causes increased glial progenitor proliferation and optic glioma formation.

Development (Cambridge, England) ·Vol. 132 ·No. 24 ·2005-12-00 ·Pages 5577-88

Zhu Y, Harada T, Liu L, Lush ME, Guignard F, Harada C, Burns DK, Bajenaru ML, Gutmann DH, Parada LF

Abstract

The gene responsible for neurofibromatosis type 1 (NF1) encodes a tumor suppressor that functions as a negative regulator of the Ras proto-oncogene. Individuals with germline mutations in NF1 are predisposed to the development of benign and malignant tumors of the peripheral and central nervous system (CNS). Children with this disease suffer a high incidence of optic gliomas, a benign but potentially debilitating tumor of the optic nerve; and an increased incidence of malignant astrocytoma, reactive astrogliosis and intellectual deficits. In the present study, we have sought insight into the molecular and cellular basis of NF1-associated CNS pathologies. We show that mice genetically engineered to lack NF1 in CNS exhibit a variety of defects in glial cells. Primary among these is a developmental defect resulting in global reactive astrogliosis in the adult brain and increased proliferation of glial progenitor cells leading to enlarged optic nerves. As a consequence, all of the mutant optic nerves develop hyperplastic lesions, some of which progress to optic pathway gliomas. These data point to hyperproliferative glial progenitors as the source of the optic tumors and provide a genetic model for NF1-associated astrogliosis and optic glioma.

MeSH Terms
Animals Astrocytes/metabolism,pathology Brain/growth & development,metabolism,pathology Cell Differentiation Cell Lineage Cell Proliferation Hyperplasia Mice Mice, Knockout Mutation Neurofibromin 1/genetics,physiology Neuroglia/metabolism,pathology Optic Nerve/pathology Optic Nerve Glioma/genetics,metabolism,pathology Stem Cells/physiology
Chemicals
Neurofibromin 1
Authors & Affiliations
10 authors, click to expand affiliations / ORCID
Zhu Yuan
Center for Developmental Biology and Kent Waldrep Foundation Center for Basic Research on Nerve Growth and Regeneration, University of Texas Southwestern Medical Center, Dallas, TX 75390-9133, USA. yuanzhu@umich.edu
Harada Takayuki
Liu Li
Lush Mark E
Guignard Frantz
Harada Chikako
Burns Dennis K
Bajenaru M Livia
Gutmann David H
Parada Luis F
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Article Info
Journal
Development (Cambridge, England)
Abbr.
Development
ISSN
0950-1991
Published
2005-12-00
Pages
5577-88
Language
English
Region
England
NLM ID
8701744
PMCID
PMC2760350
Subset
IM
Grants
NINDS NIH HHS · P50 NS052606 · United States
NINDS NIH HHS · P50 NS052606-01 · United States
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