| Literature DB >> 26436900 |
Wan-Chun Liu1, Jessica Kohn1, Sarah K Szwed1, Eben Pariser1, Sharon Sepe1, Bhagwattie Haripal1, Naoki Oshimori2, Martin Marsala3, Atsushi Miyanohara4, Ramee Lee5.
Abstract
Speech and vocal impairments characterize many neurological disorders. However, the neurogenetic mechanisms of these disorders are not well understood, and current animal models do not have the necessary circuitry to recapitulate vocal learning deficits. We developed germline transgenic songbirds, zebra finches (Taneiopygia guttata) expressing human mutant huntingtin (mHTT), a protein responsible for the progressive deterioration of motor and cognitive function in Huntington's disease (HD). Although generally healthy, the mutant songbirds had severe vocal disorders, including poor vocal imitation, stuttering, and progressive syntax and syllable degradation. Their song abnormalities were associated with HD-related neuropathology and dysfunction of the cortical-basal ganglia (CBG) song circuit. These transgenics are, to the best of our knowledge, the first experimentally created, functional mutant songbirds. Their progressive and quantifiable vocal disorder, combined with circuit dysfunction in the CBG song system, offers a model for genetic manipulation and the development of therapeutic strategies for CBG-related vocal and motor disorders.Entities:
Mesh:
Substances:
Year: 2015 PMID: 26436900 DOI: 10.1038/nn.4133
Source DB: PubMed Journal: Nat Neurosci ISSN: 1097-6256 Impact factor: 24.884