Antero-ventral internal pallidum stimulation improves behavioral disorders in Lesch-Nyhan disease.
Identifieur interne : 002528 ( PubMed/Curation ); précédent : 002527; suivant : 002529Antero-ventral internal pallidum stimulation improves behavioral disorders in Lesch-Nyhan disease.
Auteurs : Laura Cif [France] ; Brigitte Biolsi ; Sophie Gavarini ; Aude Saux ; Santiago Gil Robles ; Cornel Tancu ; Xavier Vasques ; Philippe CoubesSource :
- Movement disorders : official journal of the Movement Disorder Society [ 0885-3185 ] ; 2007.
English descriptors
- KwdEn :
- MESH :
- complications : Lesch-Nyhan Syndrome.
- etiology : Behavioral Symptoms.
- methods : Electric Stimulation Therapy.
- physiopathology : Globus Pallidus.
- radiation effects : Globus Pallidus.
- therapy : Behavioral Symptoms.
- Adolescent, Humans, Male.
Abstract
The Lesch-Nyhan syndrome is an X-linked recessive disorder caused by a deficiency in hypoxanthine-guanine phosphoribosyl transferase, a purine salvage enzyme. Affected individuals exhibit a characteristic neurobehavioral disorder with delayed acquisition of motor skills, dystonia, severe self-mutilations, and aggressive behavior. Deep brain stimulation has been previously proposed for controlling isolated involuntary movements and psychiatric disorders. We applied a double bilateral simultaneous stimulation to limbic and motor internal pallidum in one patient for controlling both behavioral and movement disorders, respectively. The injurious compulsions disappeared; dystonia and dyskinesia were decreased at 28 months follow-up.
DOI: 10.1002/mds.21723
PubMed: 17853483
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pubmed:17853483Le document en format XML
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<author><name sortKey="Cif, Laura" sort="Cif, Laura" uniqKey="Cif L" first="Laura" last="Cif">Laura Cif</name>
<affiliation wicri:level="1"><nlm:affiliation>CHRU Montpellier, Service de Neurochirurgie, Montpellier, France.</nlm:affiliation>
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<author><name sortKey="Biolsi, Brigitte" sort="Biolsi, Brigitte" uniqKey="Biolsi B" first="Brigitte" last="Biolsi">Brigitte Biolsi</name>
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<author><name sortKey="Gavarini, Sophie" sort="Gavarini, Sophie" uniqKey="Gavarini S" first="Sophie" last="Gavarini">Sophie Gavarini</name>
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<author><name sortKey="Saux, Aude" sort="Saux, Aude" uniqKey="Saux A" first="Aude" last="Saux">Aude Saux</name>
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<author><name sortKey="Robles, Santiago Gil" sort="Robles, Santiago Gil" uniqKey="Robles S" first="Santiago Gil" last="Robles">Santiago Gil Robles</name>
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<author><name sortKey="Tancu, Cornel" sort="Tancu, Cornel" uniqKey="Tancu C" first="Cornel" last="Tancu">Cornel Tancu</name>
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<author><name sortKey="Vasques, Xavier" sort="Vasques, Xavier" uniqKey="Vasques X" first="Xavier" last="Vasques">Xavier Vasques</name>
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<author><name sortKey="Coubes, Philippe" sort="Coubes, Philippe" uniqKey="Coubes P" first="Philippe" last="Coubes">Philippe Coubes</name>
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<term>Electric Stimulation Therapy (methods)</term>
<term>Globus Pallidus (physiopathology)</term>
<term>Globus Pallidus (radiation effects)</term>
<term>Humans</term>
<term>Lesch-Nyhan Syndrome (complications)</term>
<term>Male</term>
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<keywords scheme="MESH" qualifier="therapy" xml:lang="en"><term>Behavioral Symptoms</term>
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<front><div type="abstract" xml:lang="en">The Lesch-Nyhan syndrome is an X-linked recessive disorder caused by a deficiency in hypoxanthine-guanine phosphoribosyl transferase, a purine salvage enzyme. Affected individuals exhibit a characteristic neurobehavioral disorder with delayed acquisition of motor skills, dystonia, severe self-mutilations, and aggressive behavior. Deep brain stimulation has been previously proposed for controlling isolated involuntary movements and psychiatric disorders. We applied a double bilateral simultaneous stimulation to limbic and motor internal pallidum in one patient for controlling both behavioral and movement disorders, respectively. The injurious compulsions disappeared; dystonia and dyskinesia were decreased at 28 months follow-up.</div>
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<Abstract><AbstractText>The Lesch-Nyhan syndrome is an X-linked recessive disorder caused by a deficiency in hypoxanthine-guanine phosphoribosyl transferase, a purine salvage enzyme. Affected individuals exhibit a characteristic neurobehavioral disorder with delayed acquisition of motor skills, dystonia, severe self-mutilations, and aggressive behavior. Deep brain stimulation has been previously proposed for controlling isolated involuntary movements and psychiatric disorders. We applied a double bilateral simultaneous stimulation to limbic and motor internal pallidum in one patient for controlling both behavioral and movement disorders, respectively. The injurious compulsions disappeared; dystonia and dyskinesia were decreased at 28 months follow-up.</AbstractText>
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