{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T21:23:44Z","timestamp":1791321824107,"version":"4.3.1"},"reference-count":84,"publisher":"The Company of Biologists","issue":"5-6","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2009,4,30]]},"abstract":"<jats:title>SUMMARY<\/jats:title><jats:p>A polyglutamine expansion in the huntingtin (HTT) gene causes neurodegeneration in Huntington\u2019s disease (HD), but the in vivo function of the native protein (Htt) is largely unknown. Numerous biochemical and in vitro studies have suggested a role for Htt in neuronal development, synaptic function and axonal trafficking. To test these models, we generated a null mutant in the putative Drosophila HTT homolog (htt, hereafter referred to asdhtt) and, surprisingly, found that dhtt mutant animals are viable with no obvious developmental defects. Instead, dhtt is required for maintaining the mobility and long-term survival of adult animals, and for modulating axonal terminal complexity in the adult brain. Furthermore, removing endogenous dhtt significantly accelerates the neurodegenerative phenotype associated with a Drosophila model of polyglutamine Htt toxicity (HD-Q93), providing in vivo evidence that disrupting the normal function of Htt might contribute to HD pathogenesis.<\/jats:p>","DOI":"10.1242\/dmm.000653","type":"journal-article","created":{"date-parts":[[2009,4,7]],"date-time":"2009-04-07T01:09:02Z","timestamp":1239066542000},"page":"247-266","source":"Crossref","is-referenced-by-count":91,"title":["Inactivation of Drosophila Huntingtin affects long-term adult functioning and the pathogenesis of a Huntington\u2019s disease model"],"prefix":"10.1242","volume":"2","author":[{"given":"Sheng","family":"Zhang","sequence":"first","affiliation":[{"name":"Department of Genetics"},{"name":"Present address: Research Center for Neurodegenerative Diseases, The Brown Foundation Institute of Molecular Medicine, University of Texas Health Science Center at Houston, 1825 Pressler Street, Houston, TX 77030, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mel B.","family":"Feany","sequence":"additional","affiliation":[{"name":"Department of Pathology and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sudipta","family":"Saraswati","sequence":"additional","affiliation":[{"name":"The Picower Institute for Learning and Memory, Department of Biology and Department of Brain and Cognitive Sciences, Massachusetts Institute of Technology, Cambridge, MA 02139, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J. Troy","family":"Littleton","sequence":"additional","affiliation":[{"name":"The Picower Institute for Learning and Memory, Department of Biology and Department of Brain and Cognitive Sciences, Massachusetts Institute of Technology, Cambridge, MA 02139, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Norbert","family":"Perrimon","sequence":"additional","affiliation":[{"name":"Department of Genetics"},{"name":"Howard Hughes Medical Institute, Brigham and Women\u2019s Hospital, 77 Avenue Louis Pasteur, Harvard Medical School, Boston, MA 02115, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","reference":[{"key":"2021042500045938200_b1-0020247","doi-asserted-by":"crossref","first-page":"115","DOI":"10.1038\/ng1095-115","article-title":"HEAT repeats in the Huntington\u2019s disease protein","volume":"11","author":"Andrade","year":"1995","journal-title":"Nat 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