ArticleNature neuroscience2015
Human mutant huntingtin disrupts vocal learning in transgenic songbirds.
Article in Nature neuroscience, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 19 papers.
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Who cites it
19 citing papers in PubMed, 36 citations in OpenAlex.
- Genome Editing in the Chicken: From PGC-Mediated Germline Transmission to Advanced Applications.International journal of molecular sciences · 2025Review
- Review
- Noncanonical DNA structures are drivers of genome evolution.Trends in genetics : TIG · 2023Review
- Reversible inhibition of the basal ganglia prolongs repetitive vocalization but only weakly affects sequencing at branch points in songbirds.Cerebral cortex communications · 2023Article
- Generation and characterization of genome-modified chondrocyte-like cells from the zebra finch cell line immortalized by c-MYC expression.Frontiers in zoology · 2022Article
- A Model of Discovery: The Role of Imaging Established and Emerging Non-mammalian Models in Neuroscience.Frontiers in molecular neuroscience · 2022Review
- Article
- Efficient gene transfer into zebra finch germline-competent stem cells using an adenoviral vector system.Scientific reports · 2021Article
- Article
- Highly Efficient Genome Modification of Cultured Primordial Germ Cells with Lentiviral Vectors to Generate Transgenic Songbirds.Stem cell reports · 2021Article
- Gene manipulation to test links between genome, brain and behavior in developing songbirds: a test case.The Journal of experimental biology · 2020Review
- Avian genomics lends insights into endocrine function in birds.General and comparative endocrinology · 2018Review
- Primordial germ cell-mediated transgenesis and genome editing in birds.Journal of animal science and biotechnology · 2018Review
- To transduce a zebra finch: interrogating behavioral mechanisms in a model system for speech.Journal of comparative physiology. A, Neuroethology, sensory, neural, and behavioral physiology · 2017Review
- Gene editing in birds takes flight.Mammalian genome : official journal of the International Mammalian Genome Society · 2017Review
- Advantages of comparative studies in songbirds to understand the neural basis of sensorimotor integration.Journal of neurophysiology · 2017Review
- A reliable and flexible gene manipulation strategy in posthatch zebra finch brain.Scientific reports · 2017Article
- Focal expression of mutant huntingtin in the songbird basal ganglia disrupts cortico-basal ganglia networks and vocal sequences.Proceedings of the National Academy of Sciences of the United States of America · 2016Article
- Review
Corrections and comments
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Authors and funding
10 authors at 3 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
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.
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.