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Titolo:
Behavioral abnormalities of Zic1 and Zic2 mutant mice: Implications as models for human neurological disorders
Autore:
Ogura, H; Aruga, J; Mikoshiba, K;
Indirizzi:
RIKEN, Brain Sci Inst, Lab Dev Neurobiol, Wako, Saitama 3510198, Japan RIKEN Wako Saitama Japan 3510198 Neurobiol, Wako, Saitama 3510198, Japan Eisai & Co Ltd, Tsukuba Res Labs, Tsukuba, Ibaraki 3002635, Japan Eisai & Co Ltd Tsukuba Ibaraki Japan 3002635 kuba, Ibaraki 3002635, Japan Univ Tokyo, Inst Med Sci, Dept Basic Med Sci, Div Mol Neurobiol, Tokyo 1088639, Japan Univ Tokyo Tokyo Japan 1088639 , Div Mol Neurobiol, Tokyo 1088639, Japan
Titolo Testata:
BEHAVIOR GENETICS
fascicolo: 3, volume: 31, anno: 2001,
pagine: 317 - 324
SICI:
0001-8244(200105)31:3<317:BAOZAZ>2.0.ZU;2-M
Fonte:
ISI
Lingua:
ENG
Soggetto:
ACOUSTIC STARTLE RESPONSE; NEURAL CREST DEVELOPMENT; PREPULSE INHIBITION; MOUSE ZIC1; RATS; LESIONS; PERFORMANCE; RECEPTOR; PROTEIN; ANXIETY;
Keywords:
cerebellar function; hypotonia; startle response; sensorimotor gating abnormality; Joubert syndrome; gene targeting;
Tipo documento:
Article
Natura:
Periodico
Settore Disciplinare:
Social & Behavioral Sciences
Life Sciences
Citazioni:
35
Recensione:
Indirizzi per estratti:
Indirizzo: Aruga, J RIKEN, Brain Sci Inst, Lab Dev Neurobiol, 2-1 Hirosawa, Wako, Saitama 3510198, Japan RIKEN 2-1 Hirosawa Wako Saitama Japan 3510198 tama 3510198, Japan
Citazione:
H. Ogura et al., "Behavioral abnormalities of Zic1 and Zic2 mutant mice: Implications as models for human neurological disorders", BEHAV GENET, 31(3), 2001, pp. 317-324

Abstract

Zic1 and Zic2 encode closely related zinc finger proteins expressed in dorsal neural tube and its derivatives. In previous studies, we showed that the homozygous Zic1 null mutation (Zic1(-/-)) results in cerebellar malformation with severe ataxia and that holoprosencephaly and spina bifida occur inhomozygotes for Zic2 knockdown mutation (Zic2(kd/kd)). Since human ZIC2 haploin-sufficiency is a cause of holoprosencephaly, the Zic2(kd/kd) mice areregarded as an animal model for holoprosencephaly in humans. In this study, the behavioral characteristics of the Zic1 and Zic2 mutant mice were investigated in heterozygotes (Zic1(-/+) or Zic2(kd/+)), and significant abnormalities were found in the hanging, spontaneous locomotor activity, stationary rod (Zic1(-/+)), acoustic startle response, and prepulse inhibition tests (Zic2(kd/+)). The abnormalities in the Zic1(-/+) mice may be explained inpart by the hypotonia caused by hypoplasia of the cerebellar anterior vermis, and these mice are regarded as a model of Joubert syndrome. In contrast, the sensorimotor gating abnormality in the Zic2(kd/+) mice may be attributable to the presumed abnormality in the dorsomedial forebrain, which was strongly affected in the Zic2(kd/kd) Mice. Zic2(kd/+) mice can serve as a model for diseases involving sensorimotor gating abnormalities, such as schizophrenia.

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Documento generato il 19/01/20 alle ore 11:49:58