Evidence map›Paper›PMID 35715199›Full record

ArticleNeurology2022

Top 10 Research Themes for Dystonia in Cerebral Palsy: A Community-Driven Research Agenda.

Laura A Gilbert, Darcy L Fehlings, Paul Gross, Michael C Kruer, Wendy Kwan, Jonathan W Mink, Michele Shusterman, Bhooma R Aravamuthan, Cerebral Palsy Research Network Dystonia Study Group

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

Article in Neurology, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 12 papers.

0numbers the graph read from it
0cells of the map it votes in
12citing papers in PubMed
3.3field-weighted citation impact, top 7% 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

12 citing papers in PubMed, 22 citations in OpenAlex.

  1. Article
  2. Article
  3. Article
  4. Article
  5. Article
  6. Article
  7. Os odontoideum and craniovertebral junction instability secondary to dystonia: case series and review of the literature.European spine journal : official publication of the European Spine Society, the European Spinal Deformity Society, and the European Section of the Cervical Spine Research Society · 2024
    Review
  8. Article
  9. Caregiver descriptions of dystonia in cerebral palsy.Annals of clinical and translational neurology · 2024
    Article
  10. Article
  11. Article
  12. Determinants of gait dystonia severity in cerebral palsy.Developmental medicine and child neurology · 2023
    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

9 authors at 1 institution in 2 countries.

Laura A GilbertFrom the Department of Neurology (L.A.G., B.R.A.), Washington University School of Medicine and St. Louis Children's Hospital, MO; Department of Pediatrics (D.L.F.), University of Toronto and Holland Bloorview Kids Rehabilitation Hospital, Ontario, Canada; Department of Population Health Sciences (P.G., W.K.), University of Utah, Salt Lake City; Departments of Child Health, Neurology, Genetics, and Cellular and Molecular Medicine (M.C.K.), College of Medicine-Phoenix, University of Arizona and Cerebral Palsy and Pediatric Movement Disorders Program, Barrow Neurological Institute, Phoenix Children's Hospital; Department of Neurology (J.W.M.), University of Rochester School of Medicine and Dentistry, NY; and The Cerebral Palsy Research Network (P.G., M.S.), Salt Lake City, UT.
Darcy L FehlingsFrom the Department of Neurology (L.A.G., B.R.A.), Washington University School of Medicine and St. Louis Children's Hospital, MO; Department of Pediatrics (D.L.F.), University of Toronto and Holland Bloorview Kids Rehabilitation Hospital, Ontario, Canada; Department of Population Health Sciences (P.G., W.K.), University of Utah, Salt Lake City; Departments of Child Health, Neurology, Genetics, and Cellular and Molecular Medicine (M.C.K.), College of Medicine-Phoenix, University of Arizona and Cerebral Palsy and Pediatric Movement Disorders Program, Barrow Neurological Institute, Phoenix Children's Hospital; Department of Neurology (J.W.M.), University of Rochester School of Medicine and Dentistry, NY; and The Cerebral Palsy Research Network (P.G., M.S.), Salt Lake City, UT.
Paul GrossFrom the Department of Neurology (L.A.G., B.R.A.), Washington University School of Medicine and St. Louis Children's Hospital, MO; Department of Pediatrics (D.L.F.), University of Toronto and Holland Bloorview Kids Rehabilitation Hospital, Ontario, Canada; Department of Population Health Sciences (P.G., W.K.), University of Utah, Salt Lake City; Departments of Child Health, Neurology, Genetics, and Cellular and Molecular Medicine (M.C.K.), College of Medicine-Phoenix, University of Arizona and Cerebral Palsy and Pediatric Movement Disorders Program, Barrow Neurological Institute, Phoenix Children's Hospital; Department of Neurology (J.W.M.), University of Rochester School of Medicine and Dentistry, NY; and The Cerebral Palsy Research Network (P.G., M.S.), Salt Lake City, UT.ORCID 0000-0001-9833-169X
Michael C KruerFrom the Department of Neurology (L.A.G., B.R.A.), Washington University School of Medicine and St. Louis Children's Hospital, MO; Department of Pediatrics (D.L.F.), University of Toronto and Holland Bloorview Kids Rehabilitation Hospital, Ontario, Canada; Department of Population Health Sciences (P.G., W.K.), University of Utah, Salt Lake City; Departments of Child Health, Neurology, Genetics, and Cellular and Molecular Medicine (M.C.K.), College of Medicine-Phoenix, University of Arizona and Cerebral Palsy and Pediatric Movement Disorders Program, Barrow Neurological Institute, Phoenix Children's Hospital; Department of Neurology (J.W.M.), University of Rochester School of Medicine and Dentistry, NY; and The Cerebral Palsy Research Network (P.G., M.S.), Salt Lake City, UT.
Wendy KwanFrom the Department of Neurology (L.A.G., B.R.A.), Washington University School of Medicine and St. Louis Children's Hospital, MO; Department of Pediatrics (D.L.F.), University of Toronto and Holland Bloorview Kids Rehabilitation Hospital, Ontario, Canada; Department of Population Health Sciences (P.G., W.K.), University of Utah, Salt Lake City; Departments of Child Health, Neurology, Genetics, and Cellular and Molecular Medicine (M.C.K.), College of Medicine-Phoenix, University of Arizona and Cerebral Palsy and Pediatric Movement Disorders Program, Barrow Neurological Institute, Phoenix Children's Hospital; Department of Neurology (J.W.M.), University of Rochester School of Medicine and Dentistry, NY; and The Cerebral Palsy Research Network (P.G., M.S.), Salt Lake City, UT.
Jonathan W MinkFrom the Department of Neurology (L.A.G., B.R.A.), Washington University School of Medicine and St. Louis Children's Hospital, MO; Department of Pediatrics (D.L.F.), University of Toronto and Holland Bloorview Kids Rehabilitation Hospital, Ontario, Canada; Department of Population Health Sciences (P.G., W.K.), University of Utah, Salt Lake City; Departments of Child Health, Neurology, Genetics, and Cellular and Molecular Medicine (M.C.K.), College of Medicine-Phoenix, University of Arizona and Cerebral Palsy and Pediatric Movement Disorders Program, Barrow Neurological Institute, Phoenix Children's Hospital; Department of Neurology (J.W.M.), University of Rochester School of Medicine and Dentistry, NY; and The Cerebral Palsy Research Network (P.G., M.S.), Salt Lake City, UT.
Michele ShustermanFrom the Department of Neurology (L.A.G., B.R.A.), Washington University School of Medicine and St. Louis Children's Hospital, MO; Department of Pediatrics (D.L.F.), University of Toronto and Holland Bloorview Kids Rehabilitation Hospital, Ontario, Canada; Department of Population Health Sciences (P.G., W.K.), University of Utah, Salt Lake City; Departments of Child Health, Neurology, Genetics, and Cellular and Molecular Medicine (M.C.K.), College of Medicine-Phoenix, University of Arizona and Cerebral Palsy and Pediatric Movement Disorders Program, Barrow Neurological Institute, Phoenix Children's Hospital; Department of Neurology (J.W.M.), University of Rochester School of Medicine and Dentistry, NY; and The Cerebral Palsy Research Network (P.G., M.S.), Salt Lake City, UT.
Bhooma R AravamuthanFrom the Department of Neurology (L.A.G., B.R.A.), Washington University School of Medicine and St. Louis Children's Hospital, MO; Department of Pediatrics (D.L.F.), University of Toronto and Holland Bloorview Kids Rehabilitation Hospital, Ontario, Canada; Department of Population Health Sciences (P.G., W.K.), University of Utah, Salt Lake City; Departments of Child Health, Neurology, Genetics, and Cellular and Molecular Medicine (M.C.K.), College of Medicine-Phoenix, University of Arizona and Cerebral Palsy and Pediatric Movement Disorders Program, Barrow Neurological Institute, Phoenix Children's Hospital; Department of Neurology (J.W.M.), University of Rochester School of Medicine and Dentistry, NY; and The Cerebral Palsy Research Network (P.G., M.S.), Salt Lake City, UT. aravamuthanb@wustl.edu.ORCID 0000-0003-0205-2149
Cerebral Palsy Research Network Dystonia Study Group
United Cerebral Palsy · US

Funding

Genomic Insights into the Neurobiology of Cerebral PalsyR01NS106298 · NINDS · UNIVERSITY OF ARIZONA · PI KRUER, MICHAEL C · 2019 to 2023
$3.7M
Modulation of striatal cholinergic interneuron activity to prevent dystonic cerebral palsyK08NS117850 · NINDS · WASHINGTON UNIVERSITY · PI ARAVAMUTHAN, BHOOMA · 2021 to 2025
$927k
NINDS NIH HHS K08 NS117850NINDS NIH HHS R01 NS106298
6 · The paper itself

Abstract

Dystonia in cerebral palsy (DCP) is a common, debilitating, but understudied condition. The CP community (people with CP and caregivers) is uniquely equipped to help determine the research questions that best address their needs. We developed a community-driven DCP research agenda using the well-established James Lind Alliance methodology. CP community members, researchers, and clinicians were recruited through multiple advocacy, research, and professional organizations. To ensure shared baseline knowledge, participants watched webinars outlining our current knowledge on DCP prepared by a Steering Group of field experts (cprn.org/research-cp-dystonia-edition). Participants next submitted their remaining uncertainties about DCP. These were vetted by the Steering Group and consolidated to eliminate redundancy to generate a list of unique uncertainties, which were then prioritized by the participants. The top-prioritized uncertainties were aggregated into themes through iterative consensus-building discussions within the Steering Group. 166 webinar viewers generated 67 unique uncertainties. 29 uncertainties (17 generated by community members) were prioritized higher than their randomly matched pairs. These were coalesced into the following top 10 DCP research themes: (1) develop new treatments; (2) assess rehabilitation, psychological, and environmental management approaches; (3) compare effectiveness of current treatments; (4) improve diagnosis and severity assessments; (5) assess the effect of mixed tone (spasticity and dystonia) in outcomes and approaches; (6) assess predictors of treatment responsiveness; (7) identify pathophysiologic mechanisms; (8) characterize the natural history; (9) determine the best treatments for pain; and (10) increase family awareness. This community-driven research agenda reflects the concerns most important to the community, both in perception and in practice. We therefore encourage future DCP research to center around these themes. Furthermore, noting that community members (not clinicians or researchers) generated the majority of top-prioritized uncertainties, our results highlight the important contributions community members can make to research agendas, even beyond DCP.

Indexed as

Biomedical ResearchCerebral PalsyDystoniaDystonic DisordersCaregiversHumansResearch PersonnelUncertainty

Identifiers

PMID35715199
PMCPMC9442618
OpenAlexW4283066661

What OpenQuestion holds

Textmetadata
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.