Evidence map›Paper›PMID 31848221›Full record

ArticleJournal of neurology, neurosurgery, and psychiatry2020

Risk of spread in adult-onset isolated focal dystonia: a prospective international cohort study.

Brian D Berman, Christopher L Groth, Stefan H Sillau, Sarah Pirio Richardson, Scott A Norris, Johanna Junker, Norbert Brüggemann, Pinky Agarwal, Richard L Barbano, Alberto J Espay and 10 more

Open access · hybridAbstract read
In one paragraph

Article in Journal of neurology, neurosurgery, and psychiatry, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 48 papers.

0numbers the graph read from it
0cells of the map it votes in
48citing papers in PubMed
5.3field-weighted citation impact, top 4% 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

48 citing papers in PubMed, 79 citations in OpenAlex.

  1. Article
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  5. Article
  6. The relationship between occupation and specific forms of idiopathic adult-onset dystonia.Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology · 2025
    Article
  7. Article
  8. Article
  9. Article
  10. Lesion-Induced Blepharospasm: Epidemiology and Clinical Characteristics.Tremor and other hyperkinetic movements (New York, N.Y.) · 2025
    Article
  11. Oromandibular dystonia: from onset to spread a multicenter italian study.Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology · 2024
    Article
  12. Article
  13. Article
  14. Article
  15. Article
  16. Article
  17. Genetic Update and Treatment for Dystonia.International journal of molecular sciences · 2024
    Review
  18. Risk of Spreading in Adult-onset Dystonia.Tremor and other hyperkinetic movements (New York, N.Y.) · 2024
    Article
  19. Article
  20. Article
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

20 authors at 11 institutions in 3 countries.

Brian D BermanDepartment of Neurology, University of Colorado Anschutz Medical Campus, Aurora, Colorado, USA brian.berman@ucdenver.edu.ORCID 0000-0002-0602-9942
Christopher L GrothDepartment of Neurology, University of Iowa, Iowa City, Iowa, USA.
Stefan H SillauDepartment of Neurology, University of Colorado Anschutz Medical Campus, Aurora, Colorado, USA.
Sarah Pirio RichardsonDepartment of Neurology, University of New Mexico, Albuquerque, New Mexico, USA.
Scott A NorrisDepartment of Neurology, Washington University in St. Louis, St. Louis, Missouri, USA.
Johanna JunkerDepartment of Neurology, University of Luebeck, Luebeck, Germany.
Norbert BrüggemannDepartment of Neurology, University of Luebeck, Luebeck, Germany.ORCID 0000-0001-5969-6899
Pinky AgarwalBooth Gardner Parkinson's Center, Evergreen Health, Kirkland, Washington, USA.
Richard L BarbanoDepartment of Neurology, University of Rochester, Rochester, New York, USA.
Alberto J EspayDepartment of Neurology, University of Cincinnati, Cincinnati, Ohio, USA.
Joaquin A VizcarraDepartment of Neurology and Rehabilitation Medicine, University of Cincinnati, Cincinnati, Ohio, USA.
Christine KleinInstitute of Neurogenetics, University of Luebeck, Luebeck, Germany.
Tobias BäumerInstitute of Neurogenetics, University of Luebeck, Luebeck, Germany.
Sebastian LoensInstitute of Neurogenetics, University of Luebeck, Luebeck, Germany.
Stephen G ReichDepartment of Neurology, University of Maryland Medical Centre, Baltimore, Maryland, USA.
Marie VidailhetDepartment of Neurology, Salpetriere Hospital, Paris, France.
Cecilia BonnetDepartment of Neurology, Salpetriere Hospital, Paris, France.
Emmanuel RozeDepartment of Neurology, Salpetriere Hospital, Paris, France.
Hyder A JinnahDepartment of Neurology, Emory University, Atlanta, Georgia, USA.
Joel S PerlmutterNeurology, Radiology, Neuroscience, Physical Therapy and Occupational Therapy, Washington University in St. Louis School of Medicine, St. Louis, Missouri, USA.
University of Lübeck · DESorbonne Université · FRUniversity of Cincinnati · USUniversity of Colorado Anschutz Medical Campus · USWashington University in St. Louis · USEmory University · USEvergreen Health Medical Center · USUniversity of Iowa · USUniversity of Maryland, Baltimore · USUniversity of New Mexico · USUniversity of Rochester · US

Funding

UNM HSC Clinical and Translational Science CenterUL1TR001449 · NCATS · UNIVERSITY OF NEW MEXICO HEALTH SCIS CTR · PI CAMPEN, MATTHEW J, PANDHI, NANCY · 2015 to 2024
$36.9M
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
University of New Mexico Clinical and Translational Science CenterKL2TR001448 · NCATS · UNIVERSITY OF NEW MEXICO HEALTH SCIS CTR · PI CANNON, JUDY LIN · 2015 to 2024
$3.5M
Colorado Clinical and Translational Sciences InstituteKL2TR001080 · NCATS · UNIVERSITY OF COLORADO DENVER · PI SOKOL, RONALD J. · 2013 to 2017
$2.6M
Dystonia CoalitionU54TR001456 · NCATS · EMORY UNIVERSITY · PI JINNAH, HYDER A · 2015 to 2016
$2.5M
NCATS NIH HHS KL2 TR001080NCATS NIH HHS KL2 TR001448NCATS NIH HHS U54 TR001456NCATS NIH HHS UL1 TR001449NINDS NIH HHS U54 NS065701NINDS NIH HHS U54 NS116025
6 · The paper itself

Abstract

objectiveIsolated focal dystonia can spread to muscles beyond the initially affected body region, but risk of spread has not been evaluated in a prospective manner. Furthermore, body regions at risk for spread and the clinical factors associated with spread risk are not well characterised. We sought here to prospectively characterise risk of spread in recently diagnosed adult-onset isolated focal dystonia patients.

methodsPatients enrolled in the Dystonia Coalition with isolated dystonia affecting only the neck, upper face, hand or larynx at onset of symptoms were included. Timing of follow-up visits was based on a sliding scale depending on symptom onset and ranged from 1 to 4 years. Descriptive statistics, Kaplan-Meier survival curves and Cox proportional hazard regression models were used to assess clinical characteristics associated with dystonia spread.

results487 enrolled participants (68.3% women; mean age: 55.6±12.2 years) met our inclusion/exclusion criteria. Spread was observed in 50% of blepharospasm, 8% of cervical dystonia, 17% of hand dystonia and 16% of laryngeal dystonia cases. Most common regions for first spread were the oromandibular region (42.2%) and neck (22.4%) for blepharospasm, hand (3.5%) for cervical dystonia and neck for hand (12.8%) and laryngeal (15.8%) dystonia. Increased spread risk was associated with a positive family history (HR=2.18, p=0.012) and self-reported alcohol responsiveness (HR=2.59, p=0.009).

conclusionsInitial body region affected in isolated focal dystonia has differential risk and patterns of spread. Genetic factors likely influence the risk of spread. These findings can aid clinical prognostication and inform future investigations into potential disease-modifying treatments.

Indexed as

AdultAgedAge of OnsetCohort StudiesDisease ProgressionDystonic DisordersFemaleHumansMaleMiddle AgedPrognosisRisk FactorsSurvival AnalysisSymptom Assessment

Identifiers

PMID31848221
PMCPMC7024047
OpenAlexW2995441916

What OpenQuestion holds

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.