| Levodopa is not a useful treatment for Lesch-Nyhan disease. | |
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MedLine Citation:
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PMID: 21506156 Owner: NLM Status: MEDLINE |
Abstract/OtherAbstract:
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Lesch-Nyhan disease (LND) is characterized by dystonia, cognitive abnormalities, and self-injurious behavior. No effective therapies are available. LND is associated with a presynaptic dopaminergic deficit, but the reported effects of dopamine replacement therapy are conflicting. The current prospective open-label study assesses the effects of levodopa on both neurological and behavioral features of LND. All 6 study participants discontinued levodopa early, due to lack of effect and sometimes worsening of motor function. The results provide important clues for pathophysiological mechanisms and suggestions for future treatment options. |
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Authors:
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Jasper E Visser; David J Schretlen; Bastiaan R Bloem; Hyder A Jinnah |
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Publication Detail:
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Type: Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't Date: 2011-01-31 |
Journal Detail:
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Title: Movement disorders : official journal of the Movement Disorder Society Volume: 26 ISSN: 1531-8257 ISO Abbreviation: Mov. Disord. Publication Date: 2011 Mar |
Date Detail:
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Created Date: 2011-04-20 Completed Date: 2011-08-15 Revised Date: 2013-05-24 |
Medline Journal Info:
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Nlm Unique ID: 8610688 Medline TA: Mov Disord Country: United States |
Other Details:
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Languages: eng Pagination: 746-9 Citation Subset: IM |
Copyright Information:
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Copyright © 2011 Movement Disorder Society. |
Affiliation:
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Department of Neurology, Donders Institute for Brain, Cognition and Behavior, Radboud University Nijmegen Medical Centre, Nijmegen, The Netherlands. j.visser@neuro.umcn.nl |
Export Citation:
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APA/MLA Format Download EndNote Download BibTex |
| MeSH Terms | |
Descriptor/Qualifier:
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Adult Antiparkinson Agents / contraindications* Child Child, Preschool Female Humans Lesch-Nyhan Syndrome / drug therapy* Levodopa / contraindications* Male Treatment Outcome |
| Grant Support | |
ID/Acronym/Agency:
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HD053312/HD/NICHD NIH HHS; R01 HD053312/HD/NICHD NIH HHS |
| Chemical | |
Reg. No./Substance:
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0/Antiparkinson Agents; 0/Levodopa |
| Comments/Corrections | |
From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine
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