Human mutant huntingtin disrupts vocal learning in transgenic songbirds.

TitleHuman mutant huntingtin disrupts vocal learning in transgenic songbirds.
Publication TypeJournal Article
Year of Publication2015
AuthorsLiu W-C, Kohn J, Szwed SK, Pariser E, Sepe S, Haripal B, Oshimori N, Marsala M, Miyanohara A, Lee R
JournalNat Neurosci
Volume18
Issue11
Pagination1617-22
Date Published2015 Nov
ISSN1546-1726
KeywordsAnimals, Animals, Genetically Modified, Basal Ganglia, Finches, Humans, Huntingtin Protein, Learning, Nerve Tissue Proteins, Neurons, Songbirds, Vocalization, Animal
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.

DOI10.1038/nn.4133
Alternate JournalNat. Neurosci.
PubMed ID26436900
Grant ListK99 CA178197 / CA / NCI NIH HHS / United States