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                <title level="j">Movement Disorders</title>
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                <term xml:lang="en">Treatment Outcome</term>
                <term xml:lang="en">Middle Aged</term>
                <term xml:lang="en">Male</term>
                <term xml:lang="en">Huntington disease</term>
                <term xml:lang="en">Humans</term>
                <term xml:lang="en">Follow-Up Studies</term>
                <term xml:lang="en">Female</term>
                <term xml:lang="en">Double-Blind Method</term>
                <term xml:lang="en">Delayed-Action Preparations</term>
                <term xml:lang="en">Cystine Depleting Agents</term>
                <term xml:lang="en">Cysteamine</term>
                <term xml:lang="en">Aged</term>
                <term xml:lang="en">Adult</term>
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              <classCode scheme="halDomain" n="sdv.mhep">Life Sciences [q-bio]/Human health and pathology</classCode>
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              <p>BACKGROUND: Cysteamine has been demonstrated as potentially effective in numerous animal models of Huntington's disease.METHODS: Ninety-six patients with early-stage Huntington's disease were randomized to 1200 mg delayed-release cysteamine bitartrate or placebo daily for 18 months. The primary end point was the change from baseline in the UHDRS Total Motor Score. A linear mixed-effects model for repeated measures was used to assess treatment effect, expressed as the least-squares mean difference of cysteamine minus placebo, with negative values indicating less deterioration relative to placebo.RESULTS: At 18 months, the treatment effect was not statistically significant - least-squares mean difference, -1.5 ± 1.71 (P = 0.385) - although this did represent less mean deterioration from baseline for the treated group relative to placebo. Treatment with cysteamine was safe and well tolerated.CONCLUSIONS: Efficacy of cysteamine was not demonstrated in this study population of patients with Huntington's disease. Post hoc analyses indicate the need for definitive future studies. © 2017 International Parkinson and Movement Disorder Society.</p>
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