GIGYF2 gene disruption in mice results in neurodegeneration and altered insulin-like growth factor signaling
Identifieur interne : 000887 ( Main/Exploration ); précédent : 000886; suivant : 000888GIGYF2 gene disruption in mice results in neurodegeneration and altered insulin-like growth factor signaling
Auteurs : Barbara Giovannone ; William G. Tsiaras ; Suzanne De La Monte ; Jan Klysik ; Corinne Lautier ; Galina Karashchuk ; Stefano Goldwurm ; Robert J. SmithSource :
- Human Molecular Genetics [ 0964-6906 ] ; 2009-12-01.
Abstract
Grb10-Interacting GYF Protein 2 (GIGYF2) was initially identified through its interaction with Grb10, an adapter protein that binds activated IGF-I and insulin receptors. The GIGYF2 gene maps to human chromosome 2q37 within a region linked to familial Parkinson's disease (PARK11 locus), and association of GIGYF2 mutations with Parkinson's disease has been described in some but not other recent publications. This study investigated the consequences of Gigyf2 gene disruption in mice. Gigyf2 null mice undergo apparently normal embryonic development, but fail to feed and die within the first 2 post-natal days. Heterozygous Gigyf2/ mice survive to adulthood with no evident metabolic or growth defects. At 1215 months of age, the Gigyf2/ mice begin to exhibit motor dysfunction manifested as decreased balance time on a rotating horizontal rod. This is associated with histopathological evidence of neurodegeneration and rare intracytoplasmic Lewy body-like inclusions in spinal anterior horn motor neurons. There are -synuclein positive neuritic plaques in the brainstem and cerebellum, but no abnormalities in the substantia nigra. Primary cultured embryo fibroblasts from Gigyf2 null mice exhibit decreased IGF-I-stimulated IGF-I receptor tyrosine phosphorylation and augmented ERK1/2 phosphorylation. These data provide further evidence for an important role of GIGYF2 in age-related neurodegeneration and IGF pathway signaling.
Url:
DOI: 10.1093/hmg/ddp430
Affiliations:
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Smith</name><affiliation><wicri:noCountry code="syntax">???</wicri:noCountry></affiliation><affiliation><wicri:noCountry code="no comma">E-mail: rsmith4@lifespan.org</wicri:noCountry></affiliation></author></analytic><monogr></monogr><series><title level="j">Human Molecular Genetics</title><idno type="ISSN">0964-6906</idno><idno type="eISSN">1460-2083</idno><imprint><publisher>Oxford University Press</publisher><date type="published" when="2009-12-01">2009-12-01</date><biblScope unit="volume">18</biblScope><biblScope unit="issue">23</biblScope><biblScope unit="page" from="4629">4629</biblScope><biblScope unit="page" to="4639">4639</biblScope></imprint><idno type="ISSN">0964-6906</idno></series><idno type="istex">CC2507EC1926A075621F40EC5709A68C8E18DB00</idno><idno type="DOI">10.1093/hmg/ddp430</idno><idno type="ArticleID">ddp430</idno></biblStruct></sourceDesc><seriesStmt><idno type="ISSN">0964-6906</idno></seriesStmt></fileDesc><profileDesc><textClass></textClass><langUsage><language ident="en">en</language></langUsage></profileDesc></teiHeader><front><div type="abstract">Grb10-Interacting GYF Protein 2 (GIGYF2) was initially identified through its interaction with Grb10, an adapter protein that binds activated IGF-I and insulin receptors. The GIGYF2 gene maps to human chromosome 2q37 within a region linked to familial Parkinson's disease (PARK11 locus), and association of GIGYF2 mutations with Parkinson's disease has been described in some but not other recent publications. This study investigated the consequences of Gigyf2 gene disruption in mice. Gigyf2 null mice undergo apparently normal embryonic development, but fail to feed and die within the first 2 post-natal days. Heterozygous Gigyf2/ mice survive to adulthood with no evident metabolic or growth defects. At 1215 months of age, the Gigyf2/ mice begin to exhibit motor dysfunction manifested as decreased balance time on a rotating horizontal rod. This is associated with histopathological evidence of neurodegeneration and rare intracytoplasmic Lewy body-like inclusions in spinal anterior horn motor neurons. There are -synuclein positive neuritic plaques in the brainstem and cerebellum, but no abnormalities in the substantia nigra. Primary cultured embryo fibroblasts from Gigyf2 null mice exhibit decreased IGF-I-stimulated IGF-I receptor tyrosine phosphorylation and augmented ERK1/2 phosphorylation. These data provide further evidence for an important role of GIGYF2 in age-related neurodegeneration and IGF pathway signaling.</div></front></TEI><affiliations><list></list><tree><noCountry><name sortKey="De La Monte, Suzanne" sort="De La Monte, Suzanne" uniqKey="De La Monte S" first="Suzanne" last="De La Monte">Suzanne De La Monte</name><name sortKey="Giovannone, Barbara" sort="Giovannone, Barbara" uniqKey="Giovannone B" first="Barbara" last="Giovannone">Barbara Giovannone</name><name sortKey="Goldwurm, Stefano" sort="Goldwurm, Stefano" uniqKey="Goldwurm S" first="Stefano" last="Goldwurm">Stefano Goldwurm</name><name sortKey="Karashchuk, Galina" sort="Karashchuk, Galina" uniqKey="Karashchuk G" first="Galina" last="Karashchuk">Galina Karashchuk</name><name sortKey="Klysik, Jan" sort="Klysik, Jan" uniqKey="Klysik J" first="Jan" last="Klysik">Jan Klysik</name><name sortKey="Lautier, Corinne" sort="Lautier, Corinne" uniqKey="Lautier C" first="Corinne" last="Lautier">Corinne Lautier</name><name sortKey="Smith, Robert J" sort="Smith, Robert J" uniqKey="Smith R" first="Robert J." last="Smith">Robert J. Smith</name><name sortKey="Tsiaras, William G" sort="Tsiaras, William G" uniqKey="Tsiaras W" first="William G." last="Tsiaras">William G. Tsiaras</name></noCountry></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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