Evidence map›Paper›PMID 34291595›Full record

ArticleBrain and behavior2021

Non-motor phenotypic subgroups in adult-onset idiopathic, isolated, focal cervical dystonia.

Megan E Wadon, Grace A Bailey, Zehra Yilmaz, Emily Hubbard, Meshari AlSaeed, Amy Robinson, Duncan McLauchlan, Richard L Barbano, Laura Marsh, Stewart A Factor and 12 more

Open access · goldAbstract read
In one paragraph

Article in Brain and behavior, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 13 papers.

0numbers the graph read from it
0cells of the map it votes in
13citing papers in PubMed
1.5field-weighted citation impact, top 15% 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

13 citing papers in PubMed, 17 citations in OpenAlex.

  1. Article
  2. Polysomnographic characteristics in patients with idiopathic dystonia.Clinical parkinsonism & related disorders · 2026
    Article
  3. Article
  4. Article
  5. Review
  6. Review
  7. [Pain and cervical dystonia].Schmerz (Berlin, Germany) · 2024
    Review
  8. Article
  9. Article
  10. Article
  11. Article
  12. Cognitive and Neuropsychiatric Impairment in Dystonia.Current neurology and neuroscience reports · 2022
    Review
  13. 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

22 authors at 15 institutions in 3 countries.

Megan E WadonNeuroscience and Mental Health Research Institute, Division of Psychological Medicine and Clinical Neurosciences, Cardiff University, Maindy Road, Cardiff, CF24 4HQ, UK.ORCID 0000-0002-7694-6228
Grace A BaileyNeuroscience and Mental Health Research Institute, Division of Psychological Medicine and Clinical Neurosciences, Cardiff University, Maindy Road, Cardiff, CF24 4HQ, UK.
Zehra YilmazNeuroscience and Mental Health Research Institute, Division of Psychological Medicine and Clinical Neurosciences, Cardiff University, Maindy Road, Cardiff, CF24 4HQ, UK.
Emily HubbardSchool of Medicine, Cardiff University, Heath Park Campus, Cardiff, CF14 4YS, UK.
Meshari AlSaeedSchool of Medicine, Cardiff University, Heath Park Campus, Cardiff, CF14 4YS, UK.
Amy RobinsonSchool of Medicine, Cardiff University, Heath Park Campus, Cardiff, CF14 4YS, UK.
Duncan McLauchlanNeuroscience and Mental Health Research Institute, Division of Psychological Medicine and Clinical Neurosciences, Cardiff University, Maindy Road, Cardiff, CF24 4HQ, UK.
Richard L BarbanoDepartment of Neurology, University of Rochester, Elmwood Avenue, Rochester, New York, NY 14642, USA.
Laura MarshMenninger Department of Psychiatry, Baylor College of Medicine, Butler Boulevard, Houston, Texas, 77030, USA.
Stewart A FactorDepartments of Neurology & Human Genetics, Emory University, Woodruff Circle, Atlanta, Georgia, 30322, USA.
Susan H FoxEdmond J Safra Program in Parkinson Disease, Movement Disorder Clinic, Toronto Western Hospital, Bathurst Street, Toronto, Ontario, M5T 2S8, Canada.
Charles H AdlerThe Parkinson's Disease and Movement Disorders Center, Mayo Clinic, Department of Neurology, East Shea Boulevard, Scottsdale, Arizona, 85259, USA.
Ramon L RodriguezDepartment of Neurology, University of Florida, Newell Drive, Gainesville, Florida, 32611, USA.
Cynthia L ComellaDepartment of Neurological Sciences, Rush University Medical Center, West Harrison Street, Chicago, Illinois, 60612, USA.
Stephen G ReichDepartment of Neurology, University of Maryland School of Medicine, south Paca Street, Baltimore, Maryland, 21201, USA.
William L SevertBeth Israel Medical Center, First Avenue, New York, New York, 10003, USA.
Christopher G GoetzDepartment of Neurological Sciences, Rush University Medical Center, West Harrison Street, Chicago, Illinois, 60612, USA.
Joel S PerlmutterNeurology, Radiology, Neuroscience, Physical Therapy and Occupational Therapy, Washington University School of Medicine, South Euclid Avenue, St. Louis, Missouri, 63110, USA.
Hyder A JinnahDepartments of Neurology & Human Genetics, Emory University, Woodruff Circle, Atlanta, Georgia, 30322, USA.
Katharine E HardingDepartment of Neurology, Aneurin Bevan University Health Board, Corporation Road, Newport, NP19 0BH, UK.
Cynthia SandorUK Dementia Research Institute, Cardiff University, Maindy Road, Cardiff, CF24 4HQ, UK.
Kathryn J PeallNeuroscience and Mental Health Research Institute, Division of Psychological Medicine and Clinical Neurosciences, Cardiff University, Maindy Road, Cardiff, CF24 4HQ, UK.
Cardiff University · GBEmory University · USRush University Medical Center · USAneurin Bevan University Health Board · GBBaylor College of Medicine · USMayo Clinic in Arizona · USMount Sinai Beth Israel · USQueen Mary University of London · GBUK Dementia Research Institute · GBUniversity of British Columbia Hospital · CAUniversity of Florida · USUniversity of Maryland, Baltimore · USUniversity of Rochester · USUniversity of Toronto · CAWashington University in St. Louis · US

Funding

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
Medical Research Council MR/P008593/1NCATS NIH HHS U54 TR001456NINDS NIH HHS U54 NS065701NINDS NIH HHS U54 NS116025
6 · The paper itself

Abstract

backgroundNon-motor symptoms are well established phenotypic components of adult-onset idiopathic, isolated, focal cervical dystonia (AOIFCD). However, improved understanding of their clinical heterogeneity is needed to better target therapeutic intervention. Here, we examine non-motor phenotypic features to identify possible AOIFCD subgroups.

methodsParticipants diagnosed with AOIFCD were recruited via specialist neurology clinics (dystonia wales: n = 114, dystonia coalition: n = 183). Non-motor assessment included psychiatric symptoms, pain, sleep disturbance, and quality of life, assessed using self-completed questionnaires or face-to-face assessment. Both cohorts were analyzed independently using Cluster, and Bayesian multiple mixed model phenotype analyses to investigate the relationship between non-motor symptoms and determine evidence of phenotypic subgroups.

resultsIndependent cluster analysis of the two cohorts suggests two predominant phenotypic subgroups, one consisting of approximately a third of participants in both cohorts, experiencing increased levels of depression, anxiety, sleep impairment, and pain catastrophizing, as well as, decreased quality of life. The Bayesian approach reinforced this with the primary axis, which explained the majority of the variance, in each cohort being associated with psychiatric symptomology, and also sleep impairment and pain catastrophizing in the Dystonia Wales cohort.

conclusionsNon-motor symptoms accompanying AOIFCD parse into two predominant phenotypic sub-groups, with differences in psychiatric symptoms, pain catastrophizing, sleep quality, and quality of life. Improved understanding of these symptom groups will enable better targeted pathophysiological investigation and future therapeutic intervention.

Indexed as

Dystonic DisordersTorticollisAdultBayes TheoremHumansPhenotypeQuality of Lifedystonia disordersphenotypesurveys and questionnairestorticollis

Identifiers

PMID34291595
PMCPMC8413761
OpenAlexW3186692258

What OpenQuestion holds

Textmetadata
LicenceCC BY
Read underepoch 390

Registered trials

None linked

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.