Haploinsufficiency of Dyrk1A in mice leads to specific alterations in the development and regulation of motor activity

被引:57
作者
Fotaki, V
de Lagrán, MM
Estivill, X
Arbonés, M
Diersson, M
机构
[1] Genom Regulat Ctr, Genes & Dis Program, Barcelona 08003, Spain
[2] Pompeu Fabra Univ, Life Sci & Hlth Dept, Barcelona, Spain
关键词
D O I
10.1037/0735-7044.118.4.815
中图分类号
B84 [心理学]; C [社会科学总论]; Q98 [人类学];
学科分类号
03 ; 0303 ; 030303 ; 04 ; 0402 ;
摘要
DYRK1A is a protein kinase proposed to be involved in neurogenesis. Gene-targeting disruption of Dyrk1A in mice leads to decreased body and brain size, with no severe disturbance of behavior. In this study, the authors focused on the motor profile of Dyrk1A(+/-) mice. These mice presented impairment of neuromotor development with decreased activity, suggesting a physiological role of Dyrk1A in the maturation of the neuromotor system. In the adult, a marked hypoactivity and alteration of specific motor parameters were detected. These results are in agreement with the significant expression of Dyrk1A in structures related to motor function and support a role of Dyrk1A in the control of motor function.
引用
收藏
页码:815 / 821
页数:7
相关论文
共 24 条
[1]   Neurodevelopmental delay, motor abnormalities and cognitive deficits in transgenic mice overexpressing Dyrk1A (minibrain), a murine model of Down's syndrome [J].
Altafaj, X ;
Dierssen, M ;
Baamonde, C ;
Martí, E ;
Visa, J ;
Guimerà, J ;
Oset, M ;
González, JR ;
Flórez, J ;
Fillat, C ;
Estivill, X .
HUMAN MOLECULAR GENETICS, 2001, 10 (18) :1915-1923
[2]   DYRK1A accumulates in splicing speckles through a novel targeting signal and induces speckle disassembly [J].
Alvarez, M ;
Estivill, X ;
de la Luna, S .
JOURNAL OF CELL SCIENCE, 2003, 116 (15) :3099-3107
[3]   Atm-deficient mice: A paradigm of ataxia telangiectasia [J].
Barlow, C ;
Hirotsune, S ;
Paylor, R ;
Liyanage, M ;
Eckhaus, M ;
Collins, F ;
Shiloh, Y ;
Crawley, JN ;
Ried, T ;
Tagle, D ;
WynshawBoris, A .
CELL, 1996, 86 (01) :159-171
[4]  
Becker W, 1999, PROG NUCLEIC ACID RE, V62, P1
[5]   Differing substrate specificities of members of the DYRK family of arginine-directed protein kinases [J].
Campbell, LE ;
Proud, CG .
FEBS LETTERS, 2002, 510 (1-2) :31-36
[6]   Motor dysfunction in a mouse model for Down syndrome [J].
Costa, ACS ;
Walsh, K ;
Davisson, MT .
PHYSIOLOGY & BEHAVIOR, 1999, 68 (1-2) :211-220
[7]   Motor phenotypic alterations in TgDyrk1a transgenic mice implicate DYRK1A in Down syndrome motor dysfunction [J].
de Lagrán, MM ;
Altafaj, X ;
Gallego, X ;
Martí, E ;
Estivill, X ;
Sahún, I ;
Fillat, C ;
Dierssen, M .
NEUROBIOLOGY OF DISEASE, 2004, 15 (01) :132-142
[8]   Neurobehavioral development of two mouse lines commonly used in transgenic studies [J].
Dierssen, M ;
Fotaki, V ;
de Lagrán, MM ;
Gratacós, M ;
Arbonés, M ;
Fillat, C ;
Estivill, X .
PHARMACOLOGY BIOCHEMISTRY AND BEHAVIOR, 2002, 73 (01) :19-25
[9]   Dyrk1A haploinsufficiency affects viability and causes developmental delay and abnormal brain morphology in mice [J].
Fotaki, V ;
Dierssen, M ;
Alcántara, S ;
Martínez, S ;
Martí, E ;
Casas, C ;
Visa, J ;
Soriano, E ;
Estivill, X ;
Arbonés, ML .
MOLECULAR AND CELLULAR BIOLOGY, 2002, 22 (18) :6636-6647
[10]   Human minibrain homologue (MNBH/DYRK1):: Characterization, alternative splicing, differential tissue expression, and overexpression in Down syndrome [J].
Guimera, J ;
Casas, C ;
Estivill, X ;
Pritchard, M .
GENOMICS, 1999, 57 (03) :407-418