Evidence map›Paper›PMID 34296504›Full record

ArticleEuropean journal of neurology2021

Predictive modeling of spread in adult-onset isolated dystonia: Key properties and effect of tremor inclusion.

Meng Wang, Tolulope Sajobi, Francesca Morgante, Charles Adler, Pinky Agarwal, Tobias Bäumer, Alfredo Berardelli, Brian D Berman, Joel Blumin, Max Borsche and 26 more

Open access · bronzeAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in European journal of neurology, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
0.3field-weighted citation impact, top 38% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

1 citing paper in PubMed, 4 citations in OpenAlex.

  1. Trial
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

36 authors at 20 institutions in 6 countries.

Meng WangDepartment of Community Health Sciences, Department of Clinical Neurosciences and Hotchkiss Brain Institute, University of Calgary, Calgary, Alberta, Canada.
Tolulope SajobiDepartment of Community Health Sciences, Department of Clinical Neurosciences and Hotchkiss Brain Institute, University of Calgary, Calgary, Alberta, Canada.
Francesca MorganteNeurosciences Research Centre, Molecular and Clinical Sciences Research Institute, St. George's, University of London, London, UK.ORCID 0000-0002-9834-3639
Charles AdlerDepartment of Neurology, Mayo Clinic College of Medicine, Mayo Clinic Arizona, Scottsdale, Arizona, USA.ORCID 0000-0002-1045-2743
Pinky AgarwalBooth Gardner Parkinson's Center, Evergreen Health, Kirkland, Washington, USA.
Tobias BäumerInstitute of Systems Motor Science, Center for Rare Diseases, University Medical Hospital Schleswig-Holstein, University of Lübeck, Lübeck, Germany.
Alfredo BerardelliDepartment of Human Neurosciences, University of Rome "La Sapienza", Rome, Italy.ORCID 0000-0003-3598-3142
Brian D BermanDepartment of Neurology, Virginia Commonwealth University, Richmond, Virginia, USA.
Joel BluminDepartment of Otolaryngology & Communication Sciences, Medical College of Wisconsin, Milwaukee, Wisconsin, USA.
Max BorscheInstitute of Neurogenetics, University of Lübeck, Lübeck, Germany.ORCID 0000-0002-9651-5986
Allison BrashearDepartment of Neurology, University of California, Davis, Sacramento, California, USA.
Andres DeikDisease and Movement Disorders Center, Department of Neurology, University of Pennsylvania, Philadelphia, Pennsylvania, USA.
Kevin DuqueDepartment of Neurology and Rehabilitation Medicine, Gardner Family Center for Parkinson's Disease and Movement Disorders, University of Cincinnati, Cincinnati, Ohio, USA.
Alberto J EspayDepartment of Neurology and Rehabilitation Medicine, Gardner Family Center for Parkinson's Disease and Movement Disorders, University of Cincinnati, Cincinnati, Ohio, USA.
Gina FerrazzanoDepartment of Human Neurosciences, University of Rome "La Sapienza", Rome, Italy.
Jeanne FeuersteinDepartment of Neurology, University of Colorado, Aurora, Colorado, USA.
Susan FoxMovement Disorder Clinic, Edmond J Safra Program in Parkinson Disease, Toronto Western Hospital, and Division of Neurology, University of Toronto, Toronto, Ontario, Canada.
Samuel FrankBeth Israel Deaconess Medical Center, Boston, Massachusetts, USA.
Mark HallettHuman Motor Control Section, National Institute of Neurological Disorders and Stroke, NIH, Bethesda, Maryland, USA.
Joseph JankovicParkinson's Disease Center and Movement Disorders Clinic, Department of Neurology, Baylor College of Medicine, Houston, Texas, USA.
Mark S LeDouxDepartment of Psychology and School of Health Sciences, University of Memphis, and Veracity Neuroscience, Memphis, Tennessee, USA.
Julie Leegwater-KimLahey Hospital and Medical Center, Tufts University School of Medicine, Burlington, Massachusetts, USA.
Abhimanyu MahajanRush Parkinson's disease and movement disorders program, Department of Neurological Sciences, Rush University Medical Center, Chicago, Illinois, USA.ORCID 0000-0001-8807-6672
Irene A MalatyDepartment of Neurology, Fixel Institute for Neurological Diseases, University of Florida, Gainesville, Florida, USA.
William OndoHouston Methodist Hospital, Houston, Texas, USA.
Alexander PantelyatDepartment of Neurology, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA.
Sarah Pirio-RichardsonDepartment of Neurology, University of New Mexico Health Sciences Center, Albuquerque, New Mexico, USA.
Emmanuel RozeSorbonne Université, Institut du Cerveau - Paris Brain Institute - ICM, Inserm, CNRS, AP-HP, Hôpital Salpetriere, Paris, France.
Rachel Saunders-PullmanDepartment of Neurology, Icahn School of Medicine at Mount Sinai and Mount Sinai Beth Israel, New York, New York, USA.
Oksana SuchowerskyDepartment of Medicine, University of Alberta, Edmonton, Alberta, Canada.
Daniel TruongDepartment of Neurosciences, UC Riverside, Riverside, California, USA.
Marie VidailhetSorbonne Université, Institut du Cerveau - Paris Brain Institute - ICM, Inserm, CNRS, AP-HP, Hôpital Salpetriere, Paris, France.
Aparna Wagle ShuklaDepartment of Neurology, Fixel Institute for Neurological Diseases, University of Florida, Gainesville, Florida, USA.
Joel S PerlmutterDepartments of Neurology, Psychiatry, Radiology, Neurobiology, Physical Therapy and Occupational Therapy, Washington University School of Medicine, St. Louis, Missouri, USA.
Hyder A JinnahDepartments of Neurology, Human Genetics, and Pediatrics, Emory University School of Medicine, Atlanta, Georgia, USA.
Davide MartinoDepartment of Clinical Neurosciences & Hotchkiss Brain Institute, University of Calgary, Calgary, Alberta, Canada.ORCID 0000-0002-2217-0487
University of Calgary · CACentre National de la Recherche Scientifique · FRUniversity of Cincinnati · USUniversity of Florida · USUniversity of Lübeck · DEBaylor College of Medicine · USBeth Israel Deaconess Medical Center · USCornell University · USEmory University · USEvergreen Health Medical Center · USIstituto Neurologico Mediterraneo · ITJohns Hopkins University · USMayo Clinic in Arizona · USMedical College of Wisconsin · USMount Sinai Beth Israel · USNational Institute of Neurological Disorders and Stroke · USRush University Medical Center · USSapienza University of Rome · ITTufts University · USUniversity of Alberta · CA

Funding

Pathophysiology of Basal Ganglia DisordersZIANS003031 · NINDS · NATIONAL INSTITUTE OF NEUROLOGICAL DISORDERS AND STROKE · PI HALLETT, MARK · 2009 to 2023
$22.7M
Dystonia Coalition- Pilot ProjectU54NS116025 · NINDS · EMORY UNIVERSITY · PI JINNAH, HYDER A · 2019 to 2023
$7.1M
Dystonia CoalitionU54NS065701 · NINDS · EMORY UNIVERSITY · PI JINNAH, HYDER A · 2009 to 2014
$7.0M
Dystonia CoalitionU54TR001456 · NCATS · EMORY UNIVERSITY · PI JINNAH, HYDER A · 2015 to 2016
$2.5M
NCATS NIH HHS U54 TR001456NINDS NIH HHS U54 NS065701NINDS NIH HHS U54 NS116025
6 · The paper itself

Abstract

background and purposeSeveral 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.

methodsAdult-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.

resultsBoth 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.

conclusionsThis 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.

Indexed as

DystoniaDystonic DisordersAdultDatabases, FactualHumansTremorisolated dystonianeurological diseasespredictive modelsspreadtremor

Identifiers

PMID34296504
PMCPMC9100858
OpenAlexW3185811795

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