Potassium channel blocker 4‐aminopyridine is effective in interictal cerebellar symptoms in episodic ataxia type 2 — A video case report
Identifieur interne : 002683 ( Main/Curation ); précédent : 002682; suivant : 002684Potassium 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 [Allemagne] ; Martin Wolz [Allemagne] ; Heinz Reichmann [Allemagne] ; Alexander Storch [Allemagne]Source :
- Movement Disorders [ 0885-3185 ] ; 2008-07-15.
Descripteurs français
- Pascal (Inist)
English descriptors
- KwdEn :
- 4-Aminopyridine (therapeutic use), 4‐aminopyridine, Case study, Cerebellar ataxia, Female, Gait (drug effects), Gait Ataxia (drug therapy), Humans, Middle Aged, Nervous system diseases, Potassium Channel Blockers (therapeutic use), Potassium channel antagonist, SARA, Treatment Outcome, episodic ataxia type 2, interictal ataxia, video case report.
- 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. © 2008 Movement Disorder Society
Url:
DOI: 10.1002/mds.22071
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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="2008-07-15">2008-07-15</date><biblScope unit="vol">23</biblScope><biblScope unit="issue">9</biblScope><biblScope unit="page" from="1314">1314</biblScope><biblScope unit="page" to="1316">1316</biblScope></imprint><idno type="ISSN">0885-3185</idno></series><idno type="istex">5A29F579CCD897BEF9D98809889B96228D2F0AFA</idno><idno type="DOI">10.1002/mds.22071</idno><idno type="ArticleID">MDS22071</idno></biblStruct></sourceDesc><seriesStmt><idno type="ISSN">0885-3185</idno></seriesStmt></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>4-Aminopyridine (therapeutic use)</term><term>4‐aminopyridine</term><term>Case study</term><term>Cerebellar ataxia</term><term>Female</term><term>Gait (drug effects)</term><term>Gait Ataxia (drug therapy)</term><term>Humans</term><term>Middle Aged</term><term>Nervous system diseases</term><term>Potassium Channel Blockers (therapeutic use)</term><term>Potassium channel antagonist</term><term>SARA</term><term>Treatment Outcome</term><term>episodic ataxia type 2</term><term>interictal ataxia</term><term>video case report</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></keywords><keywords scheme="MESH" xml:lang="en"><term>Female</term><term>Humans</term><term>Middle Aged</term><term>Treatment Outcome</term></keywords><keywords scheme="Pascal" xml:lang="fr"><term>Antagoniste canal potassium</term><term>Ataxie cérébelleuse</term><term>Etude cas</term><term>Pathologie du système nerveux</term></keywords></textClass><langUsage><language ident="en">en</language></langUsage></profileDesc></teiHeader><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. © 2008 Movement Disorder Society</div></front></TEI><double idat="0885-3185:2008:Lohle M:potassium:channel:blocker"><INIST><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en" level="a">Potassium Channel Blocker 4-Aminopyridine is Effective in Interictal Cerebellar Symptoms in Episodic Ataxia Type 2 : A Video Case Report</title><author><name sortKey="Lohle, Matthias" sort="Lohle, Matthias" uniqKey="Lohle M" first="Matthias" last="Löhle">Matthias Löhle</name><affiliation wicri:level="3"><inist:fA14 i1="01"><s1>Department of Neurology, Technical University of Dresden</s1><s2>Dresden</s2><s3>DEU</s3><sZ>1 aut.</sZ><sZ>2 aut.</sZ><sZ>3 aut.</sZ><sZ>4 aut.</sZ><sZ>5 aut.</sZ></inist:fA14><country>Allemagne</country><placeName><region type="land" nuts="1">Saxe (Land)</region><region type="district" nuts="2">District de Dresde</region><settlement type="city">Dresde</settlement></placeName></affiliation></author><author><name sortKey="Schrempf, Wiebke" sort="Schrempf, Wiebke" uniqKey="Schrempf W" first="Wiebke" last="Schrempf">Wiebke Schrempf</name><affiliation wicri:level="3"><inist:fA14 i1="01"><s1>Department of Neurology, Technical University of Dresden</s1><s2>Dresden</s2><s3>DEU</s3><sZ>1 aut.</sZ><sZ>2 aut.</sZ><sZ>3 aut.</sZ><sZ>4 aut.</sZ><sZ>5 aut.</sZ></inist:fA14><country>Allemagne</country><placeName><region type="land" nuts="1">Saxe (Land)</region><region type="district" nuts="2">District de Dresde</region><settlement type="city">Dresde</settlement></placeName></affiliation></author><author><name sortKey="Wolz, Martin" sort="Wolz, Martin" uniqKey="Wolz M" first="Martin" last="Wolz">Martin Wolz</name><affiliation wicri:level="3"><inist:fA14 i1="01"><s1>Department of Neurology, Technical University of Dresden</s1><s2>Dresden</s2><s3>DEU</s3><sZ>1 aut.</sZ><sZ>2 aut.</sZ><sZ>3 aut.</sZ><sZ>4 aut.</sZ><sZ>5 aut.</sZ></inist:fA14><country>Allemagne</country><placeName><region type="land" nuts="1">Saxe (Land)</region><region type="district" nuts="2">District de Dresde</region><settlement type="city">Dresde</settlement></placeName></affiliation></author><author><name sortKey="Reichmann, Heinz" sort="Reichmann, Heinz" uniqKey="Reichmann H" first="Heinz" last="Reichmann">Heinz Reichmann</name><affiliation wicri:level="3"><inist:fA14 i1="01"><s1>Department of Neurology, Technical University of Dresden</s1><s2>Dresden</s2><s3>DEU</s3><sZ>1 aut.</sZ><sZ>2 aut.</sZ><sZ>3 aut.</sZ><sZ>4 aut.</sZ><sZ>5 aut.</sZ></inist:fA14><country>Allemagne</country><placeName><region type="land" nuts="1">Saxe (Land)</region><region type="district" nuts="2">District de Dresde</region><settlement type="city">Dresde</settlement></placeName></affiliation></author><author><name sortKey="Storch, Alexander" sort="Storch, Alexander" uniqKey="Storch A" first="Alexander" last="Storch">Alexander Storch</name><affiliation wicri:level="3"><inist:fA14 i1="01"><s1>Department of Neurology, Technical University of Dresden</s1><s2>Dresden</s2><s3>DEU</s3><sZ>1 aut.</sZ><sZ>2 aut.</sZ><sZ>3 aut.</sZ><sZ>4 aut.</sZ><sZ>5 aut.</sZ></inist:fA14><country>Allemagne</country><placeName><region type="land" nuts="1">Saxe (Land)</region><region type="district" nuts="2">District de Dresde</region><settlement type="city">Dresde</settlement></placeName></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">INIST</idno><idno type="inist">08-0396197</idno><date when="2008">2008</date><idno type="stanalyst">PASCAL 08-0396197 INIST</idno><idno type="RBID">Pascal:08-0396197</idno><idno type="wicri:Area/PascalFrancis/Corpus">001189</idno><idno type="wicri:Area/PascalFrancis/Curation">001B30</idno><idno type="wicri:Area/PascalFrancis/Checkpoint">001132</idno><idno type="wicri:doubleKey">0885-3185:2008:Lohle M:potassium:channel:blocker</idno><idno type="wicri:Area/Main/Merge">003740</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a">Potassium Channel Blocker 4-Aminopyridine is Effective in Interictal Cerebellar Symptoms in Episodic Ataxia Type 2 : A Video Case Report</title><author><name sortKey="Lohle, Matthias" sort="Lohle, Matthias" uniqKey="Lohle M" first="Matthias" last="Löhle">Matthias Löhle</name><affiliation wicri:level="3"><inist:fA14 i1="01"><s1>Department of Neurology, Technical University of Dresden</s1><s2>Dresden</s2><s3>DEU</s3><sZ>1 aut.</sZ><sZ>2 aut.</sZ><sZ>3 aut.</sZ><sZ>4 aut.</sZ><sZ>5 aut.</sZ></inist:fA14><country>Allemagne</country><placeName><region type="land" nuts="1">Saxe (Land)</region><region type="district" nuts="2">District de Dresde</region><settlement type="city">Dresde</settlement></placeName></affiliation></author><author><name sortKey="Schrempf, Wiebke" sort="Schrempf, Wiebke" uniqKey="Schrempf W" first="Wiebke" last="Schrempf">Wiebke Schrempf</name><affiliation wicri:level="3"><inist:fA14 i1="01"><s1>Department of Neurology, Technical University of Dresden</s1><s2>Dresden</s2><s3>DEU</s3><sZ>1 aut.</sZ><sZ>2 aut.</sZ><sZ>3 aut.</sZ><sZ>4 aut.</sZ><sZ>5 aut.</sZ></inist:fA14><country>Allemagne</country><placeName><region type="land" nuts="1">Saxe (Land)</region><region type="district" nuts="2">District de Dresde</region><settlement type="city">Dresde</settlement></placeName></affiliation></author><author><name sortKey="Wolz, Martin" sort="Wolz, Martin" uniqKey="Wolz M" first="Martin" last="Wolz">Martin Wolz</name><affiliation wicri:level="3"><inist:fA14 i1="01"><s1>Department of Neurology, Technical University of Dresden</s1><s2>Dresden</s2><s3>DEU</s3><sZ>1 aut.</sZ><sZ>2 aut.</sZ><sZ>3 aut.</sZ><sZ>4 aut.</sZ><sZ>5 aut.</sZ></inist:fA14><country>Allemagne</country><placeName><region type="land" nuts="1">Saxe (Land)</region><region type="district" nuts="2">District de Dresde</region><settlement type="city">Dresde</settlement></placeName></affiliation></author><author><name sortKey="Reichmann, Heinz" sort="Reichmann, Heinz" uniqKey="Reichmann H" first="Heinz" last="Reichmann">Heinz Reichmann</name><affiliation wicri:level="3"><inist:fA14 i1="01"><s1>Department of Neurology, Technical University of Dresden</s1><s2>Dresden</s2><s3>DEU</s3><sZ>1 aut.</sZ><sZ>2 aut.</sZ><sZ>3 aut.</sZ><sZ>4 aut.</sZ><sZ>5 aut.</sZ></inist:fA14><country>Allemagne</country><placeName><region type="land" nuts="1">Saxe (Land)</region><region type="district" nuts="2">District de Dresde</region><settlement type="city">Dresde</settlement></placeName></affiliation></author><author><name sortKey="Storch, Alexander" sort="Storch, Alexander" uniqKey="Storch A" first="Alexander" last="Storch">Alexander Storch</name><affiliation wicri:level="3"><inist:fA14 i1="01"><s1>Department of Neurology, Technical University of Dresden</s1><s2>Dresden</s2><s3>DEU</s3><sZ>1 aut.</sZ><sZ>2 aut.</sZ><sZ>3 aut.</sZ><sZ>4 aut.</sZ><sZ>5 aut.</sZ></inist:fA14><country>Allemagne</country><placeName><region type="land" nuts="1">Saxe (Land)</region><region type="district" nuts="2">District de Dresde</region><settlement type="city">Dresde</settlement></placeName></affiliation></author></analytic><series><title level="j" type="main">Movement disorders</title><title level="j" type="abbreviated">Mov. disord.</title><idno type="ISSN">0885-3185</idno><imprint><date when="2008">2008</date></imprint></series></biblStruct></sourceDesc><seriesStmt><title level="j" type="main">Movement disorders</title><title level="j" type="abbreviated">Mov. disord.</title><idno type="ISSN">0885-3185</idno></seriesStmt></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Case study</term><term>Cerebellar ataxia</term><term>Nervous system diseases</term><term>Potassium channel antagonist</term></keywords><keywords scheme="Pascal" xml:lang="fr"><term>Pathologie du système nerveux</term><term>Antagoniste canal potassium</term><term>Ataxie cérébelleuse</term><term>Etude cas</term></keywords></textClass></profileDesc></teiHeader><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></front></TEI></INIST><ISTEX><TEI wicri:istexFullTextTei="biblStruct"><teiHeader><fileDesc><titleStmt><title xml:lang="en">Potassium channel blocker 4‐aminopyridine is effective in interictal cerebellar symptoms in episodic ataxia type 2 — A video case report</title><author><name sortKey="Lohle, Matthias" sort="Lohle, Matthias" uniqKey="Lohle M" first="Matthias" last="Löhle">Matthias Löhle</name></author><author><name sortKey="Schrempf, Wiebke" sort="Schrempf, Wiebke" uniqKey="Schrempf W" first="Wiebke" last="Schrempf">Wiebke Schrempf</name></author><author><name sortKey="Wolz, Martin" sort="Wolz, Martin" uniqKey="Wolz M" first="Martin" last="Wolz">Martin Wolz</name></author><author><name sortKey="Reichmann, Heinz" sort="Reichmann, Heinz" uniqKey="Reichmann H" first="Heinz" last="Reichmann">Heinz Reichmann</name></author><author><name sortKey="Storch, Alexander" sort="Storch, Alexander" uniqKey="Storch A" first="Alexander" last="Storch">Alexander Storch</name></author></titleStmt><publicationStmt><idno type="wicri:source">ISTEX</idno><idno type="RBID">ISTEX:5A29F579CCD897BEF9D98809889B96228D2F0AFA</idno><date when="2008" year="2008">2008</date><idno type="doi">10.1002/mds.22071</idno><idno type="url">https://api.istex.fr/document/5A29F579CCD897BEF9D98809889B96228D2F0AFA/fulltext/pdf</idno><idno type="wicri:Area/Istex/Corpus">000D56</idno><idno type="wicri:Area/Istex/Curation">000D56</idno><idno type="wicri:Area/Istex/Checkpoint">001239</idno><idno type="wicri:doubleKey">0885-3185:2008:Lohle M:potassium:channel:blocker</idno><idno type="wicri:source">PubMed</idno><idno type="RBID">pubmed:18442126</idno><idno type="wicri:Area/PubMed/Corpus">002244</idno><idno type="wicri:Area/PubMed/Curation">002244</idno><idno type="wicri:Area/PubMed/Checkpoint">002134</idno><idno type="wicri:Area/Ncbi/Merge">002117</idno><idno type="wicri:Area/Ncbi/Curation">002117</idno><idno type="wicri:Area/Ncbi/Checkpoint">002117</idno><idno type="wicri:Area/Main/Merge">003261</idno></publicationStmt><sourceDesc><biblStruct><analytic><title level="a" type="main" xml:lang="en">Potassium channel blocker 4‐aminopyridine is effective in interictal cerebellar symptoms in episodic ataxia type 2 — A video case report</title><author><name sortKey="Lohle, Matthias" sort="Lohle, Matthias" uniqKey="Lohle M" first="Matthias" last="Löhle">Matthias Löhle</name><affiliation wicri:level="3"><country xml:lang="fr">Allemagne</country><wicri:regionArea>Department of Neurology, Technical University of Dresden, Dresden</wicri:regionArea><placeName><region type="land" nuts="1">Saxe (Land)</region><region type="district" nuts="2">District de Dresde</region><settlement type="city">Dresde</settlement></placeName></affiliation></author><author><name sortKey="Schrempf, Wiebke" sort="Schrempf, Wiebke" uniqKey="Schrempf W" first="Wiebke" last="Schrempf">Wiebke Schrempf</name><affiliation wicri:level="3"><country xml:lang="fr">Allemagne</country><wicri:regionArea>Department of Neurology, Technical University of Dresden, Dresden</wicri:regionArea><placeName><region type="land" nuts="1">Saxe (Land)</region><region type="district" nuts="2">District de Dresde</region><settlement type="city">Dresde</settlement></placeName></affiliation></author><author><name sortKey="Wolz, Martin" sort="Wolz, Martin" uniqKey="Wolz M" first="Martin" last="Wolz">Martin Wolz</name><affiliation wicri:level="3"><country xml:lang="fr">Allemagne</country><wicri:regionArea>Department of Neurology, Technical University of Dresden, Dresden</wicri:regionArea><placeName><region type="land" nuts="1">Saxe (Land)</region><region type="district" nuts="2">District de Dresde</region><settlement type="city">Dresde</settlement></placeName></affiliation></author><author><name sortKey="Reichmann, Heinz" sort="Reichmann, Heinz" uniqKey="Reichmann H" first="Heinz" last="Reichmann">Heinz Reichmann</name><affiliation wicri:level="3"><country xml:lang="fr">Allemagne</country><wicri:regionArea>Department of Neurology, Technical University of Dresden, Dresden</wicri:regionArea><placeName><region type="land" nuts="1">Saxe (Land)</region><region type="district" nuts="2">District de Dresde</region><settlement type="city">Dresde</settlement></placeName></affiliation></author><author><name sortKey="Storch, Alexander" sort="Storch, Alexander" uniqKey="Storch A" first="Alexander" last="Storch">Alexander Storch</name><affiliation wicri:level="3"><country xml:lang="fr">Allemagne</country><wicri:regionArea>Department of Neurology, Technical University of Dresden, Dresden</wicri:regionArea><placeName><region type="land" nuts="1">Saxe (Land)</region><region type="district" nuts="2">District de Dresde</region><settlement type="city">Dresde</settlement></placeName></affiliation></author></analytic><monogr></monogr><series><title level="j">Movement Disorders</title><title level="j" type="abbrev">Mov. 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="2008-07-15">2008-07-15</date><biblScope unit="vol">23</biblScope><biblScope unit="issue">9</biblScope><biblScope unit="page" from="1314">1314</biblScope><biblScope unit="page" to="1316">1316</biblScope></imprint><idno type="ISSN">0885-3185</idno></series><idno type="istex">5A29F579CCD897BEF9D98809889B96228D2F0AFA</idno><idno type="DOI">10.1002/mds.22071</idno><idno type="ArticleID">MDS22071</idno></biblStruct></sourceDesc><seriesStmt><idno type="ISSN">0885-3185</idno></seriesStmt></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>4-Aminopyridine (therapeutic use)</term><term>4‐aminopyridine</term><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>SARA</term><term>Treatment Outcome</term><term>episodic ataxia type 2</term><term>interictal ataxia</term><term>video case report</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></keywords><keywords scheme="MESH" xml:lang="en"><term>Female</term><term>Humans</term><term>Middle Aged</term><term>Treatment Outcome</term></keywords></textClass><langUsage><language ident="en">en</language></langUsage></profileDesc></teiHeader><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. © 2008 Movement Disorder Society</div></front></TEI></ISTEX></double></record>Pour manipuler ce document sous Unix (Dilib)
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