Schwann cell hyperplasia and tumors in transgenic mice expressing a naturally occurring mutant NF2 protein

被引:101
作者
Giovannini, M
Robanus-Maandag, E
Niwa-Kawakita, M
van der Valk, M
Woodruff, JM
Goutebroze, L
Mérel, P
Berns, A
Thomas, G [1 ]
机构
[1] Inst Curie, INSERM, U434, F-75005 Paris, France
[2] Fdn Jean Dausset, CEPH, F-75010 Paris, France
[3] Netherlands Canc Inst, Ctr Biomed Genet, NL-1066 CX Amsterdam, Netherlands
[4] Mem Sloan Kettering Canc Ctr, Dept Pathol, New York, NY 10021 USA
[5] Netherlands Canc Inst, Div Mol Genet, NL-1066 CX Amsterdam, Netherlands
关键词
neurofibromatosis type 2; schwannomin; Schwann cell tumors; tumor suppressor gene; dominant mutant; transgenic mice;
D O I
10.1101/gad.13.8.978
中图分类号
Q2 [细胞生物学];
学科分类号
071009 ; 090102 ;
摘要
Specific mutations in some tumor suppressor genes such as p53 can act in a dominant fashion. We tested whether this mechanism may also apply for the neurofibromatosis type-2 gene (NF2) which, when mutated, leads to schwannoma development. Transgenic mice were generated that express, in Schwann cells, mutant NF2 proteins prototypic of natural mutants observed in humans. Mice expressing a NF2 protein with an interstitial deletion in the amino-terminal domain showed high prevalence of Schwann cell-derived tumors and Schwann cell hyperplasia, whereas those expressing a carboxy-terminally truncated protein were normal. Our results indicate that a subset of mutant NF2 alleles observed in patients may encode products with dominant properties when overexpressed in specific cell lineages.
引用
收藏
页码:978 / 986
页数:9
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