Levodopa is not a useful treatment for Lesch‐Nyhan disease
Identifieur interne : 001705 ( Istex/Curation ); précédent : 001704; suivant : 001706Levodopa is not a useful treatment for Lesch‐Nyhan disease
Auteurs : Jasper E. Visser [Pays-Bas] ; David J. Schretlen [États-Unis] ; Bastiaan R. Bloem [Pays-Bas] ; Hyder A. Jinnah [États-Unis]Source :
- Movement Disorders [ 0885-3185 ] ; 2011-03.
English descriptors
Abstract
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. © 2011 Movement Disorder Society
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DOI: 10.1002/mds.23478
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Disord.</title><idno type="ISSN">0885-3185</idno><idno type="eISSN">1531-8257</idno><imprint><publisher>Wiley Subscription Services, Inc., A Wiley Company</publisher><pubPlace>Hoboken</pubPlace><date type="published" when="2011-03">2011-03</date><biblScope unit="vol">26</biblScope><biblScope unit="issue">4</biblScope><biblScope unit="page" from="746">746</biblScope><biblScope unit="page" to="749">749</biblScope></imprint><idno type="ISSN">0885-3185</idno></series><idno type="istex">466B646823D61BEDDFA107E2CBFE202706039751</idno><idno type="DOI">10.1002/mds.23478</idno><idno type="ArticleID">MDS23478</idno></biblStruct></sourceDesc><seriesStmt><idno type="ISSN">0885-3185</idno></seriesStmt></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Lesch‐Nyhan disease</term><term>dyskinesias</term><term>dystonia</term><term>levodopa</term><term>self‐injurious behavior</term><term>treatment</term></keywords></textClass><langUsage><language ident="en">en</language></langUsage></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">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. © 2011 Movement Disorder Society</div></front></TEI></record>Pour manipuler ce document sous Unix (Dilib)
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