Status Dystonicus: Predictors of Outcome and Progression Patterns of Underlying Disease
Identifieur interne : 000052 ( PascalFrancis/Checkpoint ); précédent : 000051; suivant : 000053Status Dystonicus: Predictors of Outcome and Progression Patterns of Underlying Disease
Auteurs : Alfonso Fasano [Italie] ; Lucia Ricciardi [Italie] ; Anna Rita Bentivoglio [Italie] ; Carlotta Canavese [Italie] ; Giovanna Zorzi [Italie] ; Igor Petrovic [Serbie] ; Nikola Kresojevic [Serbie] ; Vladimir S. Kostic [Serbie] ; Marina Svetel [Serbie] ; Norbert Kovacs [Hongrie] ; Istvan Balas [Hongrie] ; Agathe Roubertie [France] ; Devendra Mishra [Inde] ; Paolo Mariotti [Italie] ; Teresa Temudo [Portugal] ; Nardo Nardocci [Italie]Source :
- Movement disorders [ 0885-3185 ] ; 2012.
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- Pascal (Inist)
English descriptors
Abstract
Background: Status dystonicus (SD) is a rare, lifethreatening disorder characterized by acute worsening of generalized dystonia. Methods: This study was conducted to characterize the pathogenesis, clinical course, and prognosis of SD. We reviewed the records of six centers and analyzed them together with all the cases previously reported in the literature. Results: Eighty-nine episodes occurring in 68 patients were studied. The majority of patients were males (64.7%), were <15 years of age (58.8%), and had secondary dystonia as the underlying condition (37.8%). The episodes were mainly characterized by tonic muscle spasms (68.5%), with phasic forms more common in secondary forms and among females. Almost all cases needed a multistaged approach, with surgery being the most successful strategy. Neurological conditions preceding the episode worsened in 16.2% of cases (ending in death in 10.3%). Conclusions: The course and outcome of SD is highly variable; male gender and prevalent tonic phenotype predict a poor outcome.
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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en" level="a">Status Dystonicus: Predictors of Outcome and Progression Patterns of Underlying Disease</title><author><name sortKey="Fasano, Alfonso" sort="Fasano, Alfonso" uniqKey="Fasano A" first="Alfonso" last="Fasano">Alfonso Fasano</name><affiliation wicri:level="3"><inist:fA14 i1="01"><s1>Istituto di Neurologia, Università Cattolica del Sacro Cuore</s1><s2>Rome</s2><s3>ITA</s3><sZ>1 aut.</sZ><sZ>2 aut.</sZ><sZ>3 aut.</sZ></inist:fA14><country>Italie</country><placeName><settlement type="city">Rome</settlement><region nuts="2">Latium</region></placeName></affiliation><affiliation wicri:level="3"><inist:fA14 i1="02"><s1>AFaR-Fatebenefratelli Association for Biomedical Research, Neuromed Institute</s1><s2>Rome</s2><s3>ITA</s3><sZ>1 aut.</sZ></inist:fA14><country>Italie</country><placeName><settlement type="city">Rome</settlement><region nuts="2">Latium</region></placeName></affiliation></author><author><name sortKey="Ricciardi, Lucia" sort="Ricciardi, Lucia" uniqKey="Ricciardi L" first="Lucia" last="Ricciardi">Lucia Ricciardi</name><affiliation wicri:level="3"><inist:fA14 i1="01"><s1>Istituto di Neurologia, Università Cattolica del Sacro Cuore</s1><s2>Rome</s2><s3>ITA</s3><sZ>1 aut.</sZ><sZ>2 aut.</sZ><sZ>3 aut.</sZ></inist:fA14><country>Italie</country><placeName><settlement type="city">Rome</settlement><region nuts="2">Latium</region></placeName></affiliation><affiliation wicri:level="1"><inist:fA14 i1="03"><s1>Department of Neurosciences, Psychiatry and Anaesthesiological Science, University of Messina</s1><s2>Messina</s2><s3>ITA</s3><sZ>2 aut.</sZ></inist:fA14><country>Italie</country><wicri:noRegion>Messina</wicri:noRegion></affiliation></author><author><name sortKey="Bentivoglio, Anna Rita" sort="Bentivoglio, Anna Rita" uniqKey="Bentivoglio A" first="Anna Rita" last="Bentivoglio">Anna Rita Bentivoglio</name><affiliation wicri:level="3"><inist:fA14 i1="01"><s1>Istituto di Neurologia, Università Cattolica del Sacro Cuore</s1><s2>Rome</s2><s3>ITA</s3><sZ>1 aut.</sZ><sZ>2 aut.</sZ><sZ>3 aut.</sZ></inist:fA14><country>Italie</country><placeName><settlement type="city">Rome</settlement><region nuts="2">Latium</region></placeName></affiliation></author><author><name sortKey="Canavese, Carlotta" sort="Canavese, Carlotta" uniqKey="Canavese C" first="Carlotta" last="Canavese">Carlotta Canavese</name><affiliation wicri:level="3"><inist:fA14 i1="04"><s1>Neurological Institute "Carlo Besta</s1><s2>Milan</s2><s3>ITA</s3><sZ>4 aut.</sZ><sZ>5 aut.</sZ><sZ>16 aut.</sZ></inist:fA14><country>Italie</country><placeName><settlement type="city">Milan</settlement><region nuts="2">Lombardie</region></placeName></affiliation></author><author><name sortKey="Zorzi, Giovanna" sort="Zorzi, Giovanna" uniqKey="Zorzi G" first="Giovanna" last="Zorzi">Giovanna Zorzi</name><affiliation wicri:level="3"><inist:fA14 i1="04"><s1>Neurological Institute "Carlo Besta</s1><s2>Milan</s2><s3>ITA</s3><sZ>4 aut.</sZ><sZ>5 aut.</sZ><sZ>16 aut.</sZ></inist:fA14><country>Italie</country><placeName><settlement type="city">Milan</settlement><region nuts="2">Lombardie</region></placeName></affiliation></author><author><name sortKey="Petrovic, Igor" sort="Petrovic, Igor" uniqKey="Petrovic I" first="Igor" last="Petrovic">Igor Petrovic</name><affiliation wicri:level="1"><inist:fA14 i1="05"><s1>Institute of Neurology, Clinical Center of Serbia, School of Medicine</s1><s2>Belgrade</s2><s3>SRB</s3><sZ>6 aut.</sZ><sZ>7 aut.</sZ><sZ>8 aut.</sZ><sZ>9 aut.</sZ></inist:fA14><country>Serbie</country><wicri:noRegion>Belgrade</wicri:noRegion></affiliation></author><author><name sortKey="Kresojevic, Nikola" sort="Kresojevic, Nikola" uniqKey="Kresojevic N" first="Nikola" last="Kresojevic">Nikola Kresojevic</name><affiliation wicri:level="1"><inist:fA14 i1="05"><s1>Institute of Neurology, Clinical Center of Serbia, School of Medicine</s1><s2>Belgrade</s2><s3>SRB</s3><sZ>6 aut.</sZ><sZ>7 aut.</sZ><sZ>8 aut.</sZ><sZ>9 aut.</sZ></inist:fA14><country>Serbie</country><wicri:noRegion>Belgrade</wicri:noRegion></affiliation></author><author><name sortKey="Kostic, Vladimir S" sort="Kostic, Vladimir S" uniqKey="Kostic V" first="Vladimir S." last="Kostic">Vladimir S. Kostic</name><affiliation wicri:level="1"><inist:fA14 i1="05"><s1>Institute of Neurology, Clinical Center of Serbia, School of Medicine</s1><s2>Belgrade</s2><s3>SRB</s3><sZ>6 aut.</sZ><sZ>7 aut.</sZ><sZ>8 aut.</sZ><sZ>9 aut.</sZ></inist:fA14><country>Serbie</country><wicri:noRegion>Belgrade</wicri:noRegion></affiliation></author><author><name sortKey="Svetel, Marina" sort="Svetel, Marina" uniqKey="Svetel M" first="Marina" last="Svetel">Marina Svetel</name><affiliation wicri:level="1"><inist:fA14 i1="05"><s1>Institute of Neurology, Clinical Center of Serbia, School of Medicine</s1><s2>Belgrade</s2><s3>SRB</s3><sZ>6 aut.</sZ><sZ>7 aut.</sZ><sZ>8 aut.</sZ><sZ>9 aut.</sZ></inist:fA14><country>Serbie</country><wicri:noRegion>Belgrade</wicri:noRegion></affiliation></author><author><name sortKey="Kovacs, Norbert" sort="Kovacs, Norbert" uniqKey="Kovacs N" first="Norbert" last="Kovacs">Norbert Kovacs</name><affiliation wicri:level="1"><inist:fA14 i1="06"><s1>Department of Neurology, University of Pecs</s1><s2>Pecs</s2><s3>HUN</s3><sZ>10 aut.</sZ><sZ>11 aut.</sZ></inist:fA14><country>Hongrie</country><wicri:noRegion>Pecs</wicri:noRegion></affiliation></author><author><name sortKey="Balas, Istvan" sort="Balas, Istvan" uniqKey="Balas I" first="Istvan" last="Balas">Istvan Balas</name><affiliation wicri:level="1"><inist:fA14 i1="06"><s1>Department of Neurology, University of Pecs</s1><s2>Pecs</s2><s3>HUN</s3><sZ>10 aut.</sZ><sZ>11 aut.</sZ></inist:fA14><country>Hongrie</country><wicri:noRegion>Pecs</wicri:noRegion></affiliation></author><author><name sortKey="Roubertie, Agathe" sort="Roubertie, Agathe" uniqKey="Roubertie A" first="Agathe" last="Roubertie">Agathe Roubertie</name><affiliation wicri:level="1"><inist:fA14 i1="07"><s1>CHU Montpellier, Hôpital Gui de Chauliac, Service de Neuropédiatrie</s1><s2>Montpellier</s2><s3>FRA</s3><sZ>12 aut.</sZ></inist:fA14><country>France</country><placeName><settlement type="city">Montpellier</settlement></placeName></affiliation><affiliation wicri:level="1"><inist:fA14 i1="08"><s1>INSERM U827, Institut Universitaire de Recherche Clinique</s1><s2>Montpellier</s2><s3>FRA</s3><sZ>12 aut.</sZ></inist:fA14><country>France</country><placeName><settlement type="city">Montpellier</settlement></placeName></affiliation></author><author><name sortKey="Mishra, Devendra" sort="Mishra, Devendra" uniqKey="Mishra D" first="Devendra" last="Mishra">Devendra Mishra</name><affiliation wicri:level="1"><inist:fA14 i1="09"><s1>Department of Pediatrics, Maulana Azad Medical College and LN Hospital, University of Delhi</s1><s2>Delhi</s2><s3>IND</s3><sZ>13 aut.</sZ></inist:fA14><country>Inde</country><wicri:noRegion>Delhi</wicri:noRegion></affiliation></author><author><name sortKey="Mariotti, Paolo" sort="Mariotti, Paolo" uniqKey="Mariotti P" first="Paolo" last="Mariotti">Paolo Mariotti</name><affiliation wicri:level="3"><inist:fA14 i1="10"><s1>Istituto di Neuropsichiatria Infantile, Università Cattolica del Sacro Cuore</s1><s2>Rome</s2><s3>ITA</s3><sZ>14 aut.</sZ></inist:fA14><country>Italie</country><placeName><settlement type="city">Rome</settlement><region nuts="2">Latium</region></placeName></affiliation></author><author><name sortKey="Temudo, Teresa" sort="Temudo, Teresa" uniqKey="Temudo T" first="Teresa" last="Temudo">Teresa Temudo</name><affiliation wicri:level="1"><inist:fA14 i1="11"><s1>Unidade de Neuropediatria, Serviço de Pediatria, Hospital Geral de Santo Antonio</s1><s2>Porto</s2><s3>PRT</s3><sZ>15 aut.</sZ></inist:fA14><country>Portugal</country><wicri:noRegion>Porto</wicri:noRegion></affiliation></author><author><name sortKey="Nardocci, Nardo" sort="Nardocci, Nardo" uniqKey="Nardocci N" first="Nardo" last="Nardocci">Nardo Nardocci</name><affiliation wicri:level="3"><inist:fA14 i1="04"><s1>Neurological Institute "Carlo Besta</s1><s2>Milan</s2><s3>ITA</s3><sZ>4 aut.</sZ><sZ>5 aut.</sZ><sZ>16 aut.</sZ></inist:fA14><country>Italie</country><placeName><settlement type="city">Milan</settlement><region nuts="2">Lombardie</region></placeName></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">INIST</idno><idno type="inist">12-0248755</idno><date when="2012">2012</date><idno type="stanalyst">PASCAL 12-0248755 INIST</idno><idno type="RBID">Pascal:12-0248755</idno><idno type="wicri:Area/PascalFrancis/Corpus">000185</idno><idno type="wicri:Area/PascalFrancis/Curation">002B29</idno><idno type="wicri:Area/PascalFrancis/Checkpoint">000052</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a">Status Dystonicus: Predictors of Outcome and Progression Patterns of Underlying Disease</title><author><name sortKey="Fasano, Alfonso" sort="Fasano, Alfonso" uniqKey="Fasano A" first="Alfonso" last="Fasano">Alfonso Fasano</name><affiliation wicri:level="3"><inist:fA14 i1="01"><s1>Istituto di Neurologia, Università Cattolica del Sacro Cuore</s1><s2>Rome</s2><s3>ITA</s3><sZ>1 aut.</sZ><sZ>2 aut.</sZ><sZ>3 aut.</sZ></inist:fA14><country>Italie</country><placeName><settlement type="city">Rome</settlement><region nuts="2">Latium</region></placeName></affiliation><affiliation wicri:level="3"><inist:fA14 i1="02"><s1>AFaR-Fatebenefratelli Association for Biomedical Research, Neuromed Institute</s1><s2>Rome</s2><s3>ITA</s3><sZ>1 aut.</sZ></inist:fA14><country>Italie</country><placeName><settlement type="city">Rome</settlement><region nuts="2">Latium</region></placeName></affiliation></author><author><name sortKey="Ricciardi, Lucia" sort="Ricciardi, Lucia" uniqKey="Ricciardi L" first="Lucia" last="Ricciardi">Lucia Ricciardi</name><affiliation wicri:level="3"><inist:fA14 i1="01"><s1>Istituto di Neurologia, Università Cattolica del Sacro Cuore</s1><s2>Rome</s2><s3>ITA</s3><sZ>1 aut.</sZ><sZ>2 aut.</sZ><sZ>3 aut.</sZ></inist:fA14><country>Italie</country><placeName><settlement type="city">Rome</settlement><region nuts="2">Latium</region></placeName></affiliation><affiliation wicri:level="1"><inist:fA14 i1="03"><s1>Department of Neurosciences, Psychiatry and Anaesthesiological Science, University of Messina</s1><s2>Messina</s2><s3>ITA</s3><sZ>2 aut.</sZ></inist:fA14><country>Italie</country><wicri:noRegion>Messina</wicri:noRegion></affiliation></author><author><name sortKey="Bentivoglio, Anna Rita" sort="Bentivoglio, Anna Rita" uniqKey="Bentivoglio A" first="Anna Rita" last="Bentivoglio">Anna Rita Bentivoglio</name><affiliation wicri:level="3"><inist:fA14 i1="01"><s1>Istituto di Neurologia, Università Cattolica del Sacro Cuore</s1><s2>Rome</s2><s3>ITA</s3><sZ>1 aut.</sZ><sZ>2 aut.</sZ><sZ>3 aut.</sZ></inist:fA14><country>Italie</country><placeName><settlement type="city">Rome</settlement><region nuts="2">Latium</region></placeName></affiliation></author><author><name sortKey="Canavese, Carlotta" sort="Canavese, Carlotta" uniqKey="Canavese C" first="Carlotta" last="Canavese">Carlotta Canavese</name><affiliation wicri:level="3"><inist:fA14 i1="04"><s1>Neurological Institute "Carlo Besta</s1><s2>Milan</s2><s3>ITA</s3><sZ>4 aut.</sZ><sZ>5 aut.</sZ><sZ>16 aut.</sZ></inist:fA14><country>Italie</country><placeName><settlement type="city">Milan</settlement><region nuts="2">Lombardie</region></placeName></affiliation></author><author><name sortKey="Zorzi, Giovanna" sort="Zorzi, Giovanna" uniqKey="Zorzi G" first="Giovanna" last="Zorzi">Giovanna Zorzi</name><affiliation wicri:level="3"><inist:fA14 i1="04"><s1>Neurological Institute "Carlo Besta</s1><s2>Milan</s2><s3>ITA</s3><sZ>4 aut.</sZ><sZ>5 aut.</sZ><sZ>16 aut.</sZ></inist:fA14><country>Italie</country><placeName><settlement type="city">Milan</settlement><region nuts="2">Lombardie</region></placeName></affiliation></author><author><name sortKey="Petrovic, Igor" sort="Petrovic, Igor" uniqKey="Petrovic I" first="Igor" last="Petrovic">Igor Petrovic</name><affiliation wicri:level="1"><inist:fA14 i1="05"><s1>Institute of Neurology, Clinical Center of Serbia, School of Medicine</s1><s2>Belgrade</s2><s3>SRB</s3><sZ>6 aut.</sZ><sZ>7 aut.</sZ><sZ>8 aut.</sZ><sZ>9 aut.</sZ></inist:fA14><country>Serbie</country><wicri:noRegion>Belgrade</wicri:noRegion></affiliation></author><author><name sortKey="Kresojevic, Nikola" sort="Kresojevic, Nikola" uniqKey="Kresojevic N" first="Nikola" last="Kresojevic">Nikola Kresojevic</name><affiliation wicri:level="1"><inist:fA14 i1="05"><s1>Institute of Neurology, Clinical Center of Serbia, School of Medicine</s1><s2>Belgrade</s2><s3>SRB</s3><sZ>6 aut.</sZ><sZ>7 aut.</sZ><sZ>8 aut.</sZ><sZ>9 aut.</sZ></inist:fA14><country>Serbie</country><wicri:noRegion>Belgrade</wicri:noRegion></affiliation></author><author><name sortKey="Kostic, Vladimir S" sort="Kostic, Vladimir S" uniqKey="Kostic V" first="Vladimir S." last="Kostic">Vladimir S. Kostic</name><affiliation wicri:level="1"><inist:fA14 i1="05"><s1>Institute of Neurology, Clinical Center of Serbia, School of Medicine</s1><s2>Belgrade</s2><s3>SRB</s3><sZ>6 aut.</sZ><sZ>7 aut.</sZ><sZ>8 aut.</sZ><sZ>9 aut.</sZ></inist:fA14><country>Serbie</country><wicri:noRegion>Belgrade</wicri:noRegion></affiliation></author><author><name sortKey="Svetel, Marina" sort="Svetel, Marina" uniqKey="Svetel M" first="Marina" last="Svetel">Marina Svetel</name><affiliation wicri:level="1"><inist:fA14 i1="05"><s1>Institute of Neurology, Clinical Center of Serbia, School of Medicine</s1><s2>Belgrade</s2><s3>SRB</s3><sZ>6 aut.</sZ><sZ>7 aut.</sZ><sZ>8 aut.</sZ><sZ>9 aut.</sZ></inist:fA14><country>Serbie</country><wicri:noRegion>Belgrade</wicri:noRegion></affiliation></author><author><name sortKey="Kovacs, Norbert" sort="Kovacs, Norbert" uniqKey="Kovacs N" first="Norbert" last="Kovacs">Norbert Kovacs</name><affiliation wicri:level="1"><inist:fA14 i1="06"><s1>Department of Neurology, University of Pecs</s1><s2>Pecs</s2><s3>HUN</s3><sZ>10 aut.</sZ><sZ>11 aut.</sZ></inist:fA14><country>Hongrie</country><wicri:noRegion>Pecs</wicri:noRegion></affiliation></author><author><name sortKey="Balas, Istvan" sort="Balas, Istvan" uniqKey="Balas I" first="Istvan" last="Balas">Istvan Balas</name><affiliation wicri:level="1"><inist:fA14 i1="06"><s1>Department of Neurology, University of Pecs</s1><s2>Pecs</s2><s3>HUN</s3><sZ>10 aut.</sZ><sZ>11 aut.</sZ></inist:fA14><country>Hongrie</country><wicri:noRegion>Pecs</wicri:noRegion></affiliation></author><author><name sortKey="Roubertie, Agathe" sort="Roubertie, Agathe" uniqKey="Roubertie A" first="Agathe" last="Roubertie">Agathe Roubertie</name><affiliation wicri:level="1"><inist:fA14 i1="07"><s1>CHU Montpellier, Hôpital Gui de Chauliac, Service de Neuropédiatrie</s1><s2>Montpellier</s2><s3>FRA</s3><sZ>12 aut.</sZ></inist:fA14><country>France</country><placeName><settlement type="city">Montpellier</settlement></placeName></affiliation><affiliation wicri:level="1"><inist:fA14 i1="08"><s1>INSERM U827, Institut Universitaire de Recherche Clinique</s1><s2>Montpellier</s2><s3>FRA</s3><sZ>12 aut.</sZ></inist:fA14><country>France</country><placeName><settlement type="city">Montpellier</settlement></placeName></affiliation></author><author><name sortKey="Mishra, Devendra" sort="Mishra, Devendra" uniqKey="Mishra D" first="Devendra" last="Mishra">Devendra Mishra</name><affiliation wicri:level="1"><inist:fA14 i1="09"><s1>Department of Pediatrics, Maulana Azad Medical College and LN Hospital, University of Delhi</s1><s2>Delhi</s2><s3>IND</s3><sZ>13 aut.</sZ></inist:fA14><country>Inde</country><wicri:noRegion>Delhi</wicri:noRegion></affiliation></author><author><name sortKey="Mariotti, Paolo" sort="Mariotti, Paolo" uniqKey="Mariotti P" first="Paolo" last="Mariotti">Paolo Mariotti</name><affiliation wicri:level="3"><inist:fA14 i1="10"><s1>Istituto di Neuropsichiatria Infantile, Università Cattolica del Sacro Cuore</s1><s2>Rome</s2><s3>ITA</s3><sZ>14 aut.</sZ></inist:fA14><country>Italie</country><placeName><settlement type="city">Rome</settlement><region nuts="2">Latium</region></placeName></affiliation></author><author><name sortKey="Temudo, Teresa" sort="Temudo, Teresa" uniqKey="Temudo T" first="Teresa" last="Temudo">Teresa Temudo</name><affiliation wicri:level="1"><inist:fA14 i1="11"><s1>Unidade de Neuropediatria, Serviço de Pediatria, Hospital Geral de Santo Antonio</s1><s2>Porto</s2><s3>PRT</s3><sZ>15 aut.</sZ></inist:fA14><country>Portugal</country><wicri:noRegion>Porto</wicri:noRegion></affiliation></author><author><name sortKey="Nardocci, Nardo" sort="Nardocci, Nardo" uniqKey="Nardocci N" first="Nardo" last="Nardocci">Nardo Nardocci</name><affiliation wicri:level="3"><inist:fA14 i1="04"><s1>Neurological Institute "Carlo Besta</s1><s2>Milan</s2><s3>ITA</s3><sZ>4 aut.</sZ><sZ>5 aut.</sZ><sZ>16 aut.</sZ></inist:fA14><country>Italie</country><placeName><settlement type="city">Milan</settlement><region nuts="2">Lombardie</region></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="2012">2012</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>Dystonia</term><term>Midazolam</term><term>Nervous system diseases</term><term>Pantothenate kinase</term><term>Prognosis</term></keywords><keywords scheme="Pascal" xml:lang="fr"><term>Dystonie</term><term>Pathologie du système nerveux</term><term>Pronostic</term><term>Midazolam</term><term>Pantothenate kinase</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Background: Status dystonicus (SD) is a rare, lifethreatening disorder characterized by acute worsening of generalized dystonia. Methods: This study was conducted to characterize the pathogenesis, clinical course, and prognosis of SD. We reviewed the records of six centers and analyzed them together with all the cases previously reported in the literature. Results: Eighty-nine episodes occurring in 68 patients were studied. The majority of patients were males (64.7%), were <15 years of age (58.8%), and had secondary dystonia as the underlying condition (37.8%). The episodes were mainly characterized by tonic muscle spasms (68.5%), with phasic forms more common in secondary forms and among females. Almost all cases needed a multistaged approach, with surgery being the most successful strategy. Neurological conditions preceding the episode worsened in 16.2% of cases (ending in death in 10.3%). Conclusions: The course and outcome of SD is highly variable; male gender and prevalent tonic phenotype predict a poor outcome.</div></front></TEI><inist><standard h6="B"><pA><fA01 i1="01" i2="1"><s0>0885-3185</s0></fA01><fA03 i2="1"><s0>Mov. disord.</s0></fA03><fA05><s2>27</s2></fA05><fA06><s2>6</s2></fA06><fA08 i1="01" i2="1" l="ENG"><s1>Status Dystonicus: Predictors of Outcome and Progression Patterns of Underlying Disease</s1></fA08><fA11 i1="01" i2="1"><s1>FASANO (Alfonso)</s1></fA11><fA11 i1="02" i2="1"><s1>RICCIARDI (Lucia)</s1></fA11><fA11 i1="03" i2="1"><s1>BENTIVOGLIO (Anna Rita)</s1></fA11><fA11 i1="04" i2="1"><s1>CANAVESE (Carlotta)</s1></fA11><fA11 i1="05" i2="1"><s1>ZORZI (Giovanna)</s1></fA11><fA11 i1="06" i2="1"><s1>PETROVIC (Igor)</s1></fA11><fA11 i1="07" i2="1"><s1>KRESOJEVIC (Nikola)</s1></fA11><fA11 i1="08" i2="1"><s1>KOSTIC (Vladimir S.)</s1></fA11><fA11 i1="09" i2="1"><s1>SVETEL (Marina)</s1></fA11><fA11 i1="10" i2="1"><s1>KOVACS (Norbert)</s1></fA11><fA11 i1="11" i2="1"><s1>BALAS (Istvan)</s1></fA11><fA11 i1="12" i2="1"><s1>ROUBERTIE (Agathe)</s1></fA11><fA11 i1="13" i2="1"><s1>MISHRA (Devendra)</s1></fA11><fA11 i1="14" i2="1"><s1>MARIOTTI (Paolo)</s1></fA11><fA11 i1="15" i2="1"><s1>TEMUDO (Teresa)</s1></fA11><fA11 i1="16" i2="1"><s1>NARDOCCI (Nardo)</s1></fA11><fA14 i1="01"><s1>Istituto di Neurologia, Università Cattolica del Sacro Cuore</s1><s2>Rome</s2><s3>ITA</s3><sZ>1 aut.</sZ><sZ>2 aut.</sZ><sZ>3 aut.</sZ></fA14><fA14 i1="02"><s1>AFaR-Fatebenefratelli Association for Biomedical Research, Neuromed Institute</s1><s2>Rome</s2><s3>ITA</s3><sZ>1 aut.</sZ></fA14><fA14 i1="03"><s1>Department of Neurosciences, Psychiatry and Anaesthesiological Science, University of Messina</s1><s2>Messina</s2><s3>ITA</s3><sZ>2 aut.</sZ></fA14><fA14 i1="04"><s1>Neurological Institute "Carlo Besta</s1><s2>Milan</s2><s3>ITA</s3><sZ>4 aut.</sZ><sZ>5 aut.</sZ><sZ>16 aut.</sZ></fA14><fA14 i1="05"><s1>Institute of Neurology, Clinical Center of Serbia, School of Medicine</s1><s2>Belgrade</s2><s3>SRB</s3><sZ>6 aut.</sZ><sZ>7 aut.</sZ><sZ>8 aut.</sZ><sZ>9 aut.</sZ></fA14><fA14 i1="06"><s1>Department of Neurology, University of Pecs</s1><s2>Pecs</s2><s3>HUN</s3><sZ>10 aut.</sZ><sZ>11 aut.</sZ></fA14><fA14 i1="07"><s1>CHU Montpellier, Hôpital Gui de Chauliac, Service de Neuropédiatrie</s1><s2>Montpellier</s2><s3>FRA</s3><sZ>12 aut.</sZ></fA14><fA14 i1="08"><s1>INSERM U827, Institut Universitaire de Recherche Clinique</s1><s2>Montpellier</s2><s3>FRA</s3><sZ>12 aut.</sZ></fA14><fA14 i1="09"><s1>Department of Pediatrics, Maulana Azad Medical College and LN Hospital, University of Delhi</s1><s2>Delhi</s2><s3>IND</s3><sZ>13 aut.</sZ></fA14><fA14 i1="10"><s1>Istituto di Neuropsichiatria Infantile, Università Cattolica del Sacro Cuore</s1><s2>Rome</s2><s3>ITA</s3><sZ>14 aut.</sZ></fA14><fA14 i1="11"><s1>Unidade de Neuropediatria, Serviço de Pediatria, Hospital Geral de Santo Antonio</s1><s2>Porto</s2><s3>PRT</s3><sZ>15 aut.</sZ></fA14><fA20><s1>783-788</s1></fA20><fA21><s1>2012</s1></fA21><fA23 i1="01"><s0>ENG</s0></fA23><fA43 i1="01"><s1>INIST</s1><s2>20953</s2><s5>354000507771510220</s5></fA43><fA44><s0>0000</s0><s1>© 2012 INIST-CNRS. All rights reserved.</s1></fA44><fA45><s0>31 ref.</s0></fA45><fA47 i1="01" i2="1"><s0>12-0248755</s0></fA47><fA60><s1>P</s1><s3>CC</s3></fA60><fA61><s0>A</s0></fA61><fA64 i1="01" i2="1"><s0>Movement disorders</s0></fA64><fA66 i1="01"><s0>USA</s0></fA66><fC01 i1="01" l="ENG"><s0>Background: Status dystonicus (SD) is a rare, lifethreatening disorder characterized by acute worsening of generalized dystonia. Methods: This study was conducted to characterize the pathogenesis, clinical course, and prognosis of SD. We reviewed the records of six centers and analyzed them together with all the cases previously reported in the literature. Results: Eighty-nine episodes occurring in 68 patients were studied. The majority of patients were males (64.7%), were <15 years of age (58.8%), and had secondary dystonia as the underlying condition (37.8%). The episodes were mainly characterized by tonic muscle spasms (68.5%), with phasic forms more common in secondary forms and among females. Almost all cases needed a multistaged approach, with surgery being the most successful strategy. Neurological conditions preceding the episode worsened in 16.2% of cases (ending in death in 10.3%). Conclusions: The course and outcome of SD is highly variable; male gender and prevalent tonic phenotype predict a poor outcome.</s0></fC01><fC02 i1="01" i2="X"><s0>002B17</s0></fC02><fC02 i1="02" i2="X"><s0>002B17G</s0></fC02><fC03 i1="01" i2="X" l="FRE"><s0>Dystonie</s0><s5>01</s5></fC03><fC03 i1="01" i2="X" l="ENG"><s0>Dystonia</s0><s5>01</s5></fC03><fC03 i1="01" i2="X" l="SPA"><s0>Distonía</s0><s5>01</s5></fC03><fC03 i1="02" i2="X" l="FRE"><s0>Pathologie du système nerveux</s0><s5>02</s5></fC03><fC03 i1="02" i2="X" l="ENG"><s0>Nervous system diseases</s0><s5>02</s5></fC03><fC03 i1="02" i2="X" l="SPA"><s0>Sistema nervioso patología</s0><s5>02</s5></fC03><fC03 i1="03" i2="X" l="FRE"><s0>Pronostic</s0><s5>09</s5></fC03><fC03 i1="03" i2="X" l="ENG"><s0>Prognosis</s0><s5>09</s5></fC03><fC03 i1="03" i2="X" l="SPA"><s0>Pronóstico</s0><s5>09</s5></fC03><fC03 i1="04" i2="X" l="FRE"><s0>Midazolam</s0><s2>NK</s2><s2>FR</s2><s5>10</s5></fC03><fC03 i1="04" i2="X" l="ENG"><s0>Midazolam</s0><s2>NK</s2><s2>FR</s2><s5>10</s5></fC03><fC03 i1="04" i2="X" l="SPA"><s0>Midazolam</s0><s2>NK</s2><s2>FR</s2><s5>10</s5></fC03><fC03 i1="05" i2="X" l="FRE"><s0>Pantothenate kinase</s0><s2>FE</s2><s5>11</s5></fC03><fC03 i1="05" i2="X" l="ENG"><s0>Pantothenate kinase</s0><s2>FE</s2><s5>11</s5></fC03><fC03 i1="05" i2="X" l="SPA"><s0>Pantothenate kinase</s0><s2>FE</s2><s5>11</s5></fC03><fC07 i1="01" i2="X" l="FRE"><s0>Transferases</s0><s2>FE</s2></fC07><fC07 i1="01" i2="X" l="ENG"><s0>Transferases</s0><s2>FE</s2></fC07><fC07 i1="01" i2="X" l="SPA"><s0>Transferases</s0><s2>FE</s2></fC07><fC07 i1="02" i2="X" l="FRE"><s0>Enzyme</s0><s2>FE</s2></fC07><fC07 i1="02" i2="X" l="ENG"><s0>Enzyme</s0><s2>FE</s2></fC07><fC07 i1="02" i2="X" l="SPA"><s0>Enzima</s0><s2>FE</s2></fC07><fC07 i1="03" i2="X" l="FRE"><s0>Syndrome extrapyramidal</s0><s5>37</s5></fC07><fC07 i1="03" i2="X" l="ENG"><s0>Extrapyramidal syndrome</s0><s5>37</s5></fC07><fC07 i1="03" i2="X" l="SPA"><s0>Extrapiramidal síndrome</s0><s5>37</s5></fC07><fC07 i1="04" i2="X" l="FRE"><s0>Mouvement involontaire</s0><s5>38</s5></fC07><fC07 i1="04" i2="X" l="ENG"><s0>Involuntary movement</s0><s5>38</s5></fC07><fC07 i1="04" i2="X" l="SPA"><s0>Movimiento involuntario</s0><s5>38</s5></fC07><fC07 i1="05" i2="X" l="FRE"><s0>Pathologie du muscle strié</s0><s5>39</s5></fC07><fC07 i1="05" i2="X" l="ENG"><s0>Striated muscle disease</s0><s5>39</s5></fC07><fC07 i1="05" i2="X" l="SPA"><s0>Músculo estriado patología</s0><s5>39</s5></fC07><fC07 i1="06" i2="X" l="FRE"><s0>Trouble neurologique</s0><s5>41</s5></fC07><fC07 i1="06" i2="X" l="ENG"><s0>Neurological disorder</s0><s5>41</s5></fC07><fC07 i1="06" i2="X" l="SPA"><s0>Trastorno neurológico</s0><s5>41</s5></fC07><fC07 i1="07" i2="X" l="FRE"><s0>Pathologie de l'encéphale</s0><s5>42</s5></fC07><fC07 i1="07" i2="X" l="ENG"><s0>Cerebral disorder</s0><s5>42</s5></fC07><fC07 i1="07" i2="X" l="SPA"><s0>Encéfalo patología</s0><s5>42</s5></fC07><fC07 i1="08" i2="X" l="FRE"><s0>Pathologie du système nerveux central</s0><s5>43</s5></fC07><fC07 i1="08" i2="X" l="ENG"><s0>Central nervous system disease</s0><s5>43</s5></fC07><fC07 i1="08" i2="X" l="SPA"><s0>Sistema nervosio central patología</s0><s5>43</s5></fC07><fN21><s1>191</s1></fN21><fN44 i1="01"><s1>OTO</s1></fN44><fN82><s1>OTO</s1></fN82></pA></standard></inist><affiliations><list><country><li>France</li><li>Hongrie</li><li>Inde</li><li>Italie</li><li>Portugal</li><li>Serbie</li></country><region><li>Latium</li><li>Lombardie</li></region><settlement><li>Milan</li><li>Montpellier</li><li>Rome</li></settlement></list><tree><country name="Italie"><region name="Latium"><name sortKey="Fasano, Alfonso" sort="Fasano, Alfonso" uniqKey="Fasano A" first="Alfonso" last="Fasano">Alfonso Fasano</name></region><name sortKey="Bentivoglio, Anna Rita" sort="Bentivoglio, Anna Rita" uniqKey="Bentivoglio A" first="Anna Rita" last="Bentivoglio">Anna Rita Bentivoglio</name><name sortKey="Canavese, Carlotta" sort="Canavese, Carlotta" uniqKey="Canavese C" first="Carlotta" last="Canavese">Carlotta Canavese</name><name sortKey="Fasano, Alfonso" sort="Fasano, Alfonso" uniqKey="Fasano A" first="Alfonso" last="Fasano">Alfonso Fasano</name><name sortKey="Mariotti, Paolo" sort="Mariotti, Paolo" uniqKey="Mariotti P" first="Paolo" last="Mariotti">Paolo Mariotti</name><name sortKey="Nardocci, Nardo" sort="Nardocci, Nardo" uniqKey="Nardocci N" first="Nardo" last="Nardocci">Nardo Nardocci</name><name sortKey="Ricciardi, Lucia" sort="Ricciardi, Lucia" uniqKey="Ricciardi L" first="Lucia" last="Ricciardi">Lucia Ricciardi</name><name sortKey="Ricciardi, Lucia" sort="Ricciardi, Lucia" uniqKey="Ricciardi L" first="Lucia" last="Ricciardi">Lucia Ricciardi</name><name sortKey="Zorzi, Giovanna" sort="Zorzi, Giovanna" uniqKey="Zorzi G" first="Giovanna" last="Zorzi">Giovanna Zorzi</name></country><country name="Serbie"><noRegion><name sortKey="Petrovic, Igor" sort="Petrovic, Igor" uniqKey="Petrovic I" first="Igor" last="Petrovic">Igor Petrovic</name></noRegion><name sortKey="Kostic, Vladimir S" sort="Kostic, Vladimir S" uniqKey="Kostic V" first="Vladimir S." last="Kostic">Vladimir S. Kostic</name><name sortKey="Kresojevic, Nikola" sort="Kresojevic, Nikola" uniqKey="Kresojevic N" first="Nikola" last="Kresojevic">Nikola Kresojevic</name><name sortKey="Svetel, Marina" sort="Svetel, Marina" uniqKey="Svetel M" first="Marina" last="Svetel">Marina Svetel</name></country><country name="Hongrie"><noRegion><name sortKey="Kovacs, Norbert" sort="Kovacs, Norbert" uniqKey="Kovacs N" first="Norbert" last="Kovacs">Norbert Kovacs</name></noRegion><name sortKey="Balas, Istvan" sort="Balas, Istvan" uniqKey="Balas I" first="Istvan" last="Balas">Istvan Balas</name></country><country name="France"><noRegion><name sortKey="Roubertie, Agathe" sort="Roubertie, Agathe" uniqKey="Roubertie A" first="Agathe" last="Roubertie">Agathe Roubertie</name></noRegion><name sortKey="Roubertie, Agathe" sort="Roubertie, Agathe" uniqKey="Roubertie A" first="Agathe" last="Roubertie">Agathe Roubertie</name></country><country name="Inde"><noRegion><name sortKey="Mishra, Devendra" sort="Mishra, Devendra" uniqKey="Mishra D" first="Devendra" last="Mishra">Devendra Mishra</name></noRegion></country><country name="Portugal"><noRegion><name sortKey="Temudo, Teresa" sort="Temudo, Teresa" uniqKey="Temudo T" first="Teresa" last="Temudo">Teresa Temudo</name></noRegion></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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