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dc.contributor.author
Merola, Aristide
dc.contributor.author
Dwivedi, Alok K.
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Shaikh, Aasef G.
dc.contributor.author
Tareen, Tamour Khan
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Da Prat, Gustavo A.
dc.contributor.author
Kauffman, Marcelo Andres
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Hampf, Jennie
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Mahajan, Abhimanyu
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Marsili, Luca
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Jankovic, Joseph
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Comella, Cynthia L.
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Berman, Brian D.
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Perlmutter, Joel S.
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Jinnah, Hyder A.
dc.contributor.author
Espay, Alberto J.
dc.date.available
2023-01-25T16:50:45Z
dc.date.issued
2019-08
dc.identifier.citation
Merola, Aristide; Dwivedi, Alok K.; Shaikh, Aasef G.; Tareen, Tamour Khan; Da Prat, Gustavo A.; et al.; Head tremor at disease onset: an ataxic phenotype of cervical dystonia; Dr. Dietrich Steinkopff Verlag; Journal of Neurology; 266; 8; 8-2019; 1844-1851
dc.identifier.issn
0340-5354
dc.identifier.uri
http://hdl.handle.net/11336/185565
dc.description.abstract
Background: Cervical dystonia (CD) can present with head tremor. It is unclear whether ataxic features are differentially associated with this phenotype at onset of CD. Objectives: We sought to evaluate: (1) the demographic features of CD patients with (Tr-CD) and without head tremor (nTr-CD) at onset, and (2) the differential ataxic features between these CD subtypes. Methods: For the first objective, we compared demographic data in Tr-CD versus nTr-CD subtypes in the entire cohort of CD subjects enrolled in the Dystonia Coalition Natural History and Biorepository studies (n = 1608). For the second objective, we rated the standardized videos from consecutively enrolled Tr-CD subjects (n = 50) and age-, gender-, and disease duration-matched nTr-CD subjects (n = 50) for ataxia severity scoring using the Scale for the Assessment and Rating of Ataxia (SARA) and the International Cooperative Ataxia Rating Scale (ICARS); and for dystonia severity using the Toronto Western Spasmodic Torticollis Rating Scale section-I (TWSTRS) and the Global Dystonia Rating Scale (GDRS). Results: Of 1,608 subjects, 18.1% (n = 291) were classified as Tr-CD and 81.9% (n = 1317) as nTr-CD. The Tr-CD cohort was older, predominantly female, and had longer disease duration than the nTr-CD cohort (p = 0.01). Compared to nTr-CD, Tr-CD subjects had worse generalized ataxia, speech, and gait and posture scores. High ataxia severity with low dystonia severity distinguished Tr-CD from nTr-CD with high accuracy (area under the curve, 0.91 (95% CI 0.85?0.97). Conclusions: Head tremor at disease onset represents a clinically distinguishable subtype of cervical dystonia affecting predominantly older women, with worse ataxia and milder dystonia than the non-tremulous dystonic phenotype.
dc.format
application/pdf
dc.language.iso
eng
dc.publisher
Dr. Dietrich Steinkopff Verlag
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dc.rights
info:eu-repo/semantics/openAccess
dc.rights.uri
https://creativecommons.org/licenses/by-nc-sa/2.5/ar/
dc.subject
ATAXIA
dc.subject
CEREBELLUM
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DYSTONIA
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HEAD TREMOR
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TREMOR
dc.subject.classification
Neurología Clínica
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dc.subject.classification
Medicina Clínica
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dc.subject.classification
CIENCIAS MÉDICAS Y DE LA SALUD
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dc.title
Head tremor at disease onset: an ataxic phenotype of cervical dystonia
dc.type
info:eu-repo/semantics/article
dc.type
info:ar-repo/semantics/artículo
dc.type
info:eu-repo/semantics/publishedVersion
dc.date.updated
2023-01-24T10:22:50Z
dc.journal.volume
266
dc.journal.number
8
dc.journal.pagination
1844-1851
dc.journal.pais
Alemania
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dc.description.fil
Fil: Merola, Aristide. University of Cincinnati; Estados Unidos
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Fil: Dwivedi, Alok K.. No especifíca;
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Fil: Shaikh, Aasef G.. No especifíca;
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Fil: Tareen, Tamour Khan. University of Cincinnati; Estados Unidos
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Fil: Da Prat, Gustavo A.. University of Cincinnati; Estados Unidos
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Fil: Kauffman, Marcelo Andres. Gobierno de la Ciudad de Buenos Aires. Hospital General de Agudos "Ramos Mejía"; Argentina. Universidad Austral. Facultad de Ciencias Biomédicas. Instituto de Investigaciones en Medicina Traslacional. Consejo Nacional de Investigaciones Científicas y Técnicas. Oficina de Coordinación Administrativa Parque Centenario. Instituto de Investigaciones en Medicina Traslacional; Argentina
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Fil: Hampf, Jennie. Universität Zu Lübeck; Alemania
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Fil: Mahajan, Abhimanyu. University of Cincinnati; Estados Unidos
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Fil: Marsili, Luca. University of Cincinnati; Estados Unidos
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Fil: Jankovic, Joseph. Baylor College Of Medicine; Estados Unidos
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Fil: Comella, Cynthia L.. No especifíca;
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Fil: Berman, Brian D.. University of Colorado; Estados Unidos
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Fil: Perlmutter, Joel S.. University of Washington; Estados Unidos
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Fil: Jinnah, Hyder A.. University of Emory; Estados Unidos
dc.description.fil
Fil: Espay, Alberto J.. University of Cincinnati; Estados Unidos
dc.journal.title
Journal of Neurology
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dc.relation.alternativeid
info:eu-repo/semantics/altIdentifier/doi/http://dx.doi.org/10.1007/s00415-019-09341-w
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