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Predictive modeling of spread in adult-onset isolated dystonia: Key properties and effect of tremor inclusion.
Wang, Meng; Sajobi, Tolulope; Morgante, Francesca; Adler, Charles; Agarwal, Pinky; Bäumer, Tobias; Berardelli, Alfredo; Berman, Brian D; Blumin, Joel; Borsche, Max; Brashear, Allison; Deik, Andres; Duque, Kevin; Espay, Alberto J; Ferrazzano, Gina; Feuerstein, Jeanne; Fox, Susan; Frank, Samuel; Hallett, Mark; Jankovic, Joseph; LeDoux, Mark S; Leegwater-Kim, Julie; Mahajan, Abhimanyu; Malaty, Irene A; Ondo, William; Pantelyat, Alexander; Pirio-Richardson, Sarah; Roze, Emmanuel; Saunders-Pullman, Rachel; Suchowersky, Oksana; Truong, Daniel; Vidailhet, Marie; Shukla, Aparna Wagle; Perlmutter, Joel S; Jinnah, Hyder A; Martino, Davide.
Afiliación
  • Wang M; Department of Community Health Sciences, Department of Clinical Neurosciences and Hotchkiss Brain Institute, University of Calgary, Calgary, Alberta, Canada.
  • Sajobi T; Department of Community Health Sciences, Department of Clinical Neurosciences and Hotchkiss Brain Institute, University of Calgary, Calgary, Alberta, Canada.
  • Morgante F; Neurosciences Research Centre, Molecular and Clinical Sciences Research Institute, St. George's, University of London, London, UK.
  • Adler C; Department of Clinical and Experimental Medicine, University of Messina, Messina, Italy.
  • Agarwal P; Department of Neurology, Mayo Clinic College of Medicine, Mayo Clinic Arizona, Scottsdale, Arizona, USA.
  • Bäumer T; Booth Gardner Parkinson's Center, Evergreen Health, Kirkland, Washington, USA.
  • Berardelli A; Institute of Systems Motor Science, Center for Rare Diseases, University Medical Hospital Schleswig-Holstein, University of Lübeck, Lübeck, Germany.
  • Berman BD; Department of Human Neurosciences, University of Rome "La Sapienza", Rome, Italy.
  • Blumin J; IRCCS Neuromed, Pozzilli, Italy.
  • Borsche M; Department of Neurology, Virginia Commonwealth University, Richmond, Virginia, USA.
  • Brashear A; Department of Otolaryngology & Communication Sciences, Medical College of Wisconsin, Milwaukee, Wisconsin, USA.
  • Deik A; Institute of Neurogenetics, University of Lübeck, Lübeck, Germany.
  • Duque K; Department of Neurology, University of California, Davis, Sacramento, California, USA.
  • Espay AJ; Disease and Movement Disorders Center, Department of Neurology, University of Pennsylvania, Philadelphia, Pennsylvania, USA.
  • Ferrazzano G; Department of Neurology and Rehabilitation Medicine, Gardner Family Center for Parkinson's Disease and Movement Disorders, University of Cincinnati, Cincinnati, Ohio, USA.
  • Feuerstein J; Department of Neurology and Rehabilitation Medicine, Gardner Family Center for Parkinson's Disease and Movement Disorders, University of Cincinnati, Cincinnati, Ohio, USA.
  • Fox S; Department of Human Neurosciences, University of Rome "La Sapienza", Rome, Italy.
  • Frank S; Department of Neurology, University of Colorado, Aurora, Colorado, USA.
  • Hallett M; Movement Disorder Clinic, Edmond J Safra Program in Parkinson Disease, Toronto Western Hospital, and Division of Neurology, University of Toronto, Toronto, Ontario, Canada.
  • Jankovic J; Beth Israel Deaconess Medical Center, Boston, Massachusetts, USA.
  • LeDoux MS; Human Motor Control Section, National Institute of Neurological Disorders and Stroke, NIH, Bethesda, Maryland, USA.
  • Leegwater-Kim J; Parkinson's Disease Center and Movement Disorders Clinic, Department of Neurology, Baylor College of Medicine, Houston, Texas, USA.
  • Mahajan A; Department of Psychology and School of Health Sciences, University of Memphis, and Veracity Neuroscience, Memphis, Tennessee, USA.
  • Malaty IA; Lahey Hospital and Medical Center, Tufts University School of Medicine, Burlington, Massachusetts, USA.
  • Ondo W; Rush Parkinson's disease and movement disorders program, Department of Neurological Sciences, Rush University Medical Center, Chicago, Illinois, USA.
  • Pantelyat A; Department of Neurology, Fixel Institute for Neurological Diseases, University of Florida, Gainesville, Florida, USA.
  • Pirio-Richardson S; Houston Methodist Hospital, Houston, Texas, USA.
  • Roze E; Weill Cornell Medical School, New York, New York, USA.
  • Saunders-Pullman R; Department of Neurology, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA.
  • Suchowersky O; Department of Neurology, University of New Mexico Health Sciences Center, Albuquerque, New Mexico, USA.
  • Truong D; Sorbonne Université, Institut du Cerveau - Paris Brain Institute - ICM, Inserm, CNRS, AP-HP, Hôpital Salpetriere, Paris, France.
  • Vidailhet M; Department of Neurology, Icahn School of Medicine at Mount Sinai and Mount Sinai Beth Israel, New York, New York, USA.
  • Shukla AW; Department of Medicine, University of Alberta, Edmonton, Alberta, Canada.
  • Perlmutter JS; Department of Neurosciences, UC Riverside, Riverside, California, USA.
  • Jinnah HA; The Parkinson and Movement Disorder Institute, Fountain Valley, California, USA.
  • Martino D; Sorbonne Université, Institut du Cerveau - Paris Brain Institute - ICM, Inserm, CNRS, AP-HP, Hôpital Salpetriere, Paris, France.
Eur J Neurol ; 28(12): 3999-4009, 2021 12.
Article en En | MEDLINE | ID: mdl-34296504
BACKGROUND AND PURPOSE: Several clinical and demographic factors relate to anatomic spread of adult-onset isolated dystonia, but a predictive model is still lacking. The aims of this study were: (i) to develop and validate a predictive model of anatomic spread of adult-onset isolated dystonia; and (ii) to evaluate whether presence of tremor associated with dystonia influences model predictions of spread. METHODS: Adult-onset isolated dystonia participants with focal onset from the Dystonia Coalition Natural History Project database were included. We developed two prediction models, one with dystonia as sole disease manifestation ("dystonia-only") and one accepting dystonia OR tremor in any body part as disease manifestations ("dystonia OR tremor"). Demographic and clinical predictors were selected based on previous evidence, clinical plausibility of association with spread, or both. We used logistic regressions and evaluated model discrimination and calibration. Internal validation was carried out based on bootstrapping. RESULTS: Both predictive models showed an area under the curve of 0.65 (95% confidence intervals 0.62-0.70 and 0.62-0.69, respectively) and good calibration after internal validation. In both models, onset of dystonia in body regions other than the neck, older age, depression and history of neck trauma were predictors of spread. CONCLUSIONS: This predictive modeling of spread in adult-onset isolated dystonia based on accessible predictors (demographic and clinical) can be easily implemented to inform individuals' risk of spread. Because tremor did not influence prediction of spread, our results support the argument that tremor is a part of the dystonia syndrome, and not an independent or coincidental disorder.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Trastornos Distónicos / Distonía Tipo de estudio: Etiology_studies / Prognostic_studies / Risk_factors_studies Límite: Adult / Humans Idioma: En Revista: Eur J Neurol Asunto de la revista: NEUROLOGIA Año: 2021 Tipo del documento: Article País de afiliación: Canadá Pais de publicación: Reino Unido
Texto completo
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Trastornos Distónicos / Distonía Tipo de estudio: Etiology_studies / Prognostic_studies / Risk_factors_studies Límite: Adult / Humans Idioma: En Revista: Eur J Neurol Asunto de la revista: NEUROLOGIA Año: 2021 Tipo del documento: Article País de afiliación: Canadá Pais de publicación: Reino Unido