MovDisordV3, Ncbi, Curation, bibRecord, 002117

Potassium channel blocker 4-aminopyridine is effective in interictal cerebellar symptoms in episodic ataxia type 2--a video case report.

Identifieur interne : 002117 ( Ncbi/Curation ); précédent : 002116; suivant : 002118

Potassium channel blocker 4-aminopyridine is effective in interictal cerebellar symptoms in episodic ataxia type 2--a video case report.

Auteurs : Matthias Löhle [Allemagne] ; Wiebke Schrempf ; Martin Wolz ; Heinz Reichmann ; Alexander Storch

Source :

Movement disorders : official journal of the Movement Disorder Society [ 1531-8257 ] ; 2008.

RBID : pubmed:18442126

English descriptors

KwdEn :
- 4-Aminopyridine (therapeutic use), Female, Gait (drug effects), Gait Ataxia (drug therapy), Humans, Middle Aged, Potassium Channel Blockers (therapeutic use), Treatment Outcome.
MESH :
- chemical , therapeutic use : 4-Aminopyridine, Potassium Channel Blockers.
- drug effects : Gait.
- drug therapy : Gait Ataxia.
- Female, Humans, Middle Aged, Treatment Outcome.

Abstract

Episodic ataxia type 2 (EA2) is an autosomal-dominant hereditary disorder clinically characterized by recurrent attacks of vertigo, imbalance and ataxia. Studies have shown that 4-aminopyridine (4-AP) is capable to prevent these attacks. However, there are no reports whether 4-AP is able to attenuate interictal cerebellar ataxia. Using the scale for assessment and rating of ataxia (SARA), we examined the efficacy of 4-AP on interictal ataxia in a 63-year-old female patient who suffered from EA2 since the age of 57. EA2 was diagnosed based on clinical criteria and not genetically proven. When treatment with 4-AP was paused the patient was suffering from marked gait and stance ataxia. After re-initiation of treatment with 5 mg 4-AP t.i.d., there was pronounced improvement in gait and stance ataxia. Within 24 hours SARA score lowered from 8.5 to 4.5 points. We conclude that 4-AP may be beneficial for interictal cerebellar ataxia in late onset EA2.

DOI: 10.1002/mds.22071
PubMed: 18442126

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Le document en format XML

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<author><name sortKey="Lohle, Matthias" sort="Lohle, Matthias" uniqKey="Lohle M" first="Matthias" last="Löhle">Matthias Löhle</name>
<affiliation wicri:level="3"><nlm:affiliation>Department of Neurology, Technical University of Dresden, Dresden, Germany. matthias.loehle@neuro.med.tu-dresden.de</nlm:affiliation>
<country xml:lang="fr">Allemagne</country>
<wicri:regionArea>Department of Neurology, Technical University of Dresden, Dresden</wicri:regionArea>
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<author><name sortKey="Schrempf, Wiebke" sort="Schrempf, Wiebke" uniqKey="Schrempf W" first="Wiebke" last="Schrempf">Wiebke Schrempf</name>
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<author><name sortKey="Wolz, Martin" sort="Wolz, Martin" uniqKey="Wolz M" first="Martin" last="Wolz">Martin Wolz</name>
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<author><name sortKey="Reichmann, Heinz" sort="Reichmann, Heinz" uniqKey="Reichmann H" first="Heinz" last="Reichmann">Heinz Reichmann</name>
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<author><name sortKey="Storch, Alexander" sort="Storch, Alexander" uniqKey="Storch A" first="Alexander" last="Storch">Alexander Storch</name>
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<wicri:regionArea>Department of Neurology, Technical University of Dresden, Dresden</wicri:regionArea>
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<author><name sortKey="Schrempf, Wiebke" sort="Schrempf, Wiebke" uniqKey="Schrempf W" first="Wiebke" last="Schrempf">Wiebke Schrempf</name>
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<author><name sortKey="Wolz, Martin" sort="Wolz, Martin" uniqKey="Wolz M" first="Martin" last="Wolz">Martin Wolz</name>
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<author><name sortKey="Reichmann, Heinz" sort="Reichmann, Heinz" uniqKey="Reichmann H" first="Heinz" last="Reichmann">Heinz Reichmann</name>
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<series><title level="j">Movement disorders : official journal of the Movement Disorder Society</title>
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<term>Female</term>
<term>Gait (drug effects)</term>
<term>Gait Ataxia (drug therapy)</term>
<term>Humans</term>
<term>Middle Aged</term>
<term>Potassium Channel Blockers (therapeutic use)</term>
<term>Treatment Outcome</term>
</keywords>
<keywords scheme="MESH" type="chemical" qualifier="therapeutic use" xml:lang="en"><term>4-Aminopyridine</term>
<term>Potassium Channel Blockers</term>
</keywords>
<keywords scheme="MESH" qualifier="drug effects" xml:lang="en"><term>Gait</term>
</keywords>
<keywords scheme="MESH" qualifier="drug therapy" xml:lang="en"><term>Gait Ataxia</term>
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<keywords scheme="MESH" xml:lang="en"><term>Female</term>
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<front><div type="abstract" xml:lang="en">Episodic ataxia type 2 (EA2) is an autosomal-dominant hereditary disorder clinically characterized by recurrent attacks of vertigo, imbalance and ataxia. Studies have shown that 4-aminopyridine (4-AP) is capable to prevent these attacks. However, there are no reports whether 4-AP is able to attenuate interictal cerebellar ataxia. Using the scale for assessment and rating of ataxia (SARA), we examined the efficacy of 4-AP on interictal ataxia in a 63-year-old female patient who suffered from EA2 since the age of 57. EA2 was diagnosed based on clinical criteria and not genetically proven. When treatment with 4-AP was paused the patient was suffering from marked gait and stance ataxia. After re-initiation of treatment with 5 mg 4-AP t.i.d., there was pronounced improvement in gait and stance ataxia. Within 24 hours SARA score lowered from 8.5 to 4.5 points. We conclude that 4-AP may be beneficial for interictal cerebellar ataxia in late onset EA2.</div>
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	Movement Disorders (revue)
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Movement Disorders (revue)

Potassium channel blocker 4-aminopyridine is effective in interictal cerebellar symptoms in episodic ataxia type 2--a video case report.

Potassium channel blocker 4-aminopyridine is effective in interictal cerebellar symptoms in episodic ataxia type 2--a video case report.

Source :

English descriptors

Abstract

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Links to Exploration step

Le document en format XML

Pour manipuler ce document sous Unix (Dilib)

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Pour générer des pages wiki