Trihexyphenidyl for acute life-threatening episodes due to a dystonic movement disorder in Rett syndrome.
Identifieur interne : 001A31 ( PubMed/Curation ); précédent : 001A30; suivant : 001A32Trihexyphenidyl for acute life-threatening episodes due to a dystonic movement disorder in Rett syndrome.
Auteurs : Artemis D. Gika [Royaume-Uni] ; Elaine Hughes ; Sushma Goyal ; Matthew Sparkes ; Jean-Pierre LinSource :
- Movement disorders : official journal of the Movement Disorder Society [ 1531-8257 ] ; 2010.
English descriptors
- KwdEn :
- Adolescent, Antiparkinson Agents (therapeutic use), Child, Dystonic Disorders (drug therapy), Dystonic Disorders (etiology), Electrocardiography (methods), Electroencephalography (methods), Electromyography (methods), Female, Humans, Rett Syndrome (complications), Trihexyphenidyl (therapeutic use), Video Recording (methods), Young Adult.
- MESH :
- chemical , therapeutic use : Antiparkinson Agents, Trihexyphenidyl.
- complications : Rett Syndrome.
- drug therapy : Dystonic Disorders.
- etiology : Dystonic Disorders.
- methods : Electrocardiography, Electroencephalography, Electromyography, Video Recording.
- Adolescent, Child, Female, Humans, Young Adult.
Abstract
In Rett syndrome (RS), acute life-threatening episodes (ALTEs) are usually attributed to epilepsy or autonomic dysfunction but they can represent a movement disorder (MD). We describe three girls with RS who experienced ALTEs from an early age. These were long considered epileptic until video-EEG in Patients 1 and 3 revealed their non-epileptic nature. A primary dystonic mechanism was suspected and Patients 1 and 2 were treated with Trihexyphenidyl with significantly reduced frequency of the ALTEs. Patient 3 died before Trihexyphenidyl was tried. Trihexyphenidyl in RS patients with similar presentations can modify the dystonia and prevent ALTEs.
DOI: 10.1002/mds.22926
PubMed: 20063434
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<author><name sortKey="Gika, Artemis D" sort="Gika, Artemis D" uniqKey="Gika A" first="Artemis D" last="Gika">Artemis D. Gika</name>
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<wicri:regionArea>Department of Paediatric Neurology, Evelina Children's Hospital, Guy's and St Thomas' NHS Foundation Trust, London</wicri:regionArea>
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<author><name sortKey="Sparkes, Matthew" sort="Sparkes, Matthew" uniqKey="Sparkes M" first="Matthew" last="Sparkes">Matthew Sparkes</name>
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<author><name sortKey="Lin, Jean Pierre" sort="Lin, Jean Pierre" uniqKey="Lin J" first="Jean-Pierre" last="Lin">Jean-Pierre Lin</name>
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<front><div type="abstract" xml:lang="en">In Rett syndrome (RS), acute life-threatening episodes (ALTEs) are usually attributed to epilepsy or autonomic dysfunction but they can represent a movement disorder (MD). We describe three girls with RS who experienced ALTEs from an early age. These were long considered epileptic until video-EEG in Patients 1 and 3 revealed their non-epileptic nature. A primary dystonic mechanism was suspected and Patients 1 and 2 were treated with Trihexyphenidyl with significantly reduced frequency of the ALTEs. Patient 3 died before Trihexyphenidyl was tried. Trihexyphenidyl in RS patients with similar presentations can modify the dystonia and prevent ALTEs.</div>
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<Abstract><AbstractText>In Rett syndrome (RS), acute life-threatening episodes (ALTEs) are usually attributed to epilepsy or autonomic dysfunction but they can represent a movement disorder (MD). We describe three girls with RS who experienced ALTEs from an early age. These were long considered epileptic until video-EEG in Patients 1 and 3 revealed their non-epileptic nature. A primary dystonic mechanism was suspected and Patients 1 and 2 were treated with Trihexyphenidyl with significantly reduced frequency of the ALTEs. Patient 3 died before Trihexyphenidyl was tried. Trihexyphenidyl in RS patients with similar presentations can modify the dystonia and prevent ALTEs.</AbstractText>
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