TY - JOUR
T1 - Reversal of neurological defects in a mouse model of Rett syndrome
AU - Guy, J.
AU - Gan, J.
AU - Selfridge, J.
AU - Cobb, Stuart
AU - Bird, A.
PY - 2007/2/23
Y1 - 2007/2/23
N2 - Rett syndrome is an autism spectrum disorder caused by mosaic expression of mutant copies of the X-linked MECP2 gene in neurons. However, neurons do not die, which suggests that this is not a neurodegenerative disorder. An important question for future therapeutic approaches to this and related disorders concerns phenotypic reversibility. Can viable but defective neurons be repaired, or is the damage done during development without normal MeCP2 irrevocable? Using a mouse model, we demonstrate robust phenotypic reversal, as activation of MeCP2 expression leads to striking loss of advanced neurological symptoms in both immature and mature adult animals.
AB - Rett syndrome is an autism spectrum disorder caused by mosaic expression of mutant copies of the X-linked MECP2 gene in neurons. However, neurons do not die, which suggests that this is not a neurodegenerative disorder. An important question for future therapeutic approaches to this and related disorders concerns phenotypic reversibility. Can viable but defective neurons be repaired, or is the damage done during development without normal MeCP2 irrevocable? Using a mouse model, we demonstrate robust phenotypic reversal, as activation of MeCP2 expression leads to striking loss of advanced neurological symptoms in both immature and mature adult animals.
UR - http://www.scopus.com/inward/record.url?eid=2-s2.0-33847266846&partnerID=8YFLogxK
U2 - 10.1126/science.1138389
DO - 10.1126/science.1138389
M3 - Article
AN - SCOPUS:33847266846
SN - 0036-8075
VL - 315
SP - 1143
EP - 1147
JO - Science
JF - Science
IS - 5815
ER -