Evidence map›Paper›PMID 28397002›Full record

ArticleJournal of neurology2017

The UK Myotonic Dystrophy Patient Registry: facilitating and accelerating clinical research.

Libby Wood, Isabell Cordts, Antonio Atalaia, Chiara Marini-Bettolo, Paul Maddison, Margaret Phillips, Mark Roberts, Mark Rogers, Simon Hammans, Volker Straub and 9 more

Registry-linked trialOpen access · hybridAbstract read
In one paragraph

Article in Journal of neurology, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT04003363 (The UK National Registry for Myotonic Dystrophy), which is not on this map. Cited by 13 papers.

0numbers the graph read from it
0cells of the map it votes in
13citing papers in PubMed
2.0field-weighted citation impact, top 14% 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.

NCT04003363 recruitingnot on this map

The UK National Registry for Myotonic Dystrophy

Typeobservational_patient_registrySponsorNewcastle UniversityRan2013 to 2030Enrolled900ConditionsMyotonic DystrophyArmsPatient Registry
3 · Its place in the literature

Who cites it

13 citing papers in PubMed, 28 citations in OpenAlex.

  1. Article
  2. Article
  3. Article
  4. Article
  5. Review
  6. Article
  7. Benign tumors in myotonic dystrophy type I target disease-related cancer sites.Annals of clinical and translational neurology · 2019
    Article
  8. Article
  9. Article
  10. Article
  11. Article
  12. Article
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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

19 authors at 11 institutions in 2 countries.

Libby WoodJohn Walton Muscular Dystrophy Research Centre, Institute of Genetic Medicine, Newcastle University, Newcastle upon Tyne, UK. elizabeth.wood2@ncl.ac.uk.ORCID http://orcid.org/0000-0003-3299-4321
Isabell CordtsJohn Walton Muscular Dystrophy Research Centre, Institute of Genetic Medicine, Newcastle University, Newcastle upon Tyne, UK.
Antonio AtalaiaJohn Walton Muscular Dystrophy Research Centre, Institute of Genetic Medicine, Newcastle University, Newcastle upon Tyne, UK.
Chiara Marini-BettoloJohn Walton Muscular Dystrophy Research Centre, Institute of Genetic Medicine, Newcastle University, Newcastle upon Tyne, UK.
Paul MaddisonDepartment of Neurology, Queen's Medical Centre, Nottingham, UK.
Margaret PhillipsDepartment of Rehabilitation Medicine, Derby Teaching Hospitals NHS Foundation Trust, Derby, UK.
Mark RobertsDepartment of Neurology, Salford Royal NHS Foundation Trust, Salford, UK.
Mark RogersInstitute of Medical Genetics, University Hospital of Wales, Cardiff, UK.
Simon HammansWessex Neurological Centre, University Hospital of Southampton, Southampton, UK.
Volker StraubJohn Walton Muscular Dystrophy Research Centre, Institute of Genetic Medicine, Newcastle University, Newcastle upon Tyne, UK.
Richard PettyDepartment of Neurology, NHS Greater Glasgow and Clyde, Southern General Hospital, Glasgow, UK.
Richard OrrellDepartment of Neurology, Royal Free Hospital, London, UK.
Darren G MoncktonInstitute of Molecular, Cell and Systems Biology, College of Medical, Veterinary and Life Sciences, University of Glasgow, Glasgow, Scotland, UK.
Nikoletta NikolenkoJohn Walton Muscular Dystrophy Research Centre, Institute of Genetic Medicine, Newcastle University, Newcastle upon Tyne, UK.
Aura Cecilia Jimenez-MorenoJohn Walton Muscular Dystrophy Research Centre, Institute of Genetic Medicine, Newcastle University, Newcastle upon Tyne, UK.
Rachel ThompsonJohn Walton Muscular Dystrophy Research Centre, Institute of Genetic Medicine, Newcastle University, Newcastle upon Tyne, UK.
David Hilton-JonesDepartment of Clinical Neurology, John Radcliffe Hospital, Oxford, UK.
Chris TurnerUCL MRC Centre for Neuromuscular Diseases, Institute of Neurology, London, UK.
Hanns LochmüllerJohn Walton Muscular Dystrophy Research Centre, Institute of Genetic Medicine, Newcastle University, Newcastle upon Tyne, UK.
Newcastle University · GBEssex Cardiothoracic Centre · GBJohn Radcliffe Hospital · GBMRC Prion Unit · GBMuscular Dystrophy UK · GBNHS Greater Glasgow and Clyde · GBQueen's Medical Centre · GBSalford Royal NHS Foundation Trust · GBThe Royal Free Hospital · GBUniversity Hospital of Wales · GBUniversity of Glasgow · GB

Funding

Medical Research Council G1002274
6 · The paper itself

Abstract

Myotonic dystrophy type 1 (DM1) is the most frequent muscular dystrophy worldwide with complex, multi-systemic, and progressively worsening symptoms. There is currently no treatment for this inherited disorder and research can be challenging due to the rarity and variability of the disease. The UK Myotonic Dystrophy Patient Registry is a patient self-enrolling online database collecting clinical and genetic information. For this cross-sectional "snapshot" analysis, 556 patients with a confirmed diagnosis of DM1 registered between May 2012 and July 2016 were included. An almost even distribution was seen between genders and a broad range of ages was present from 8 months to 78 years, with the largest proportion between 30 and 59 years. The two most frequent symptoms were fatigue and myotonia, reported by 79 and 78% of patients, respectively. The severity of myotonia correlated with the severity of fatigue as well as mobility impairment, and dysphagia occurred mostly in patients also reporting myotonia. Men reported significantly more frequent severe myotonia, whereas severe fatigue was more frequently reported by women. Cardiac abnormalities were diagnosed in 48% of patients and more than one-third of them needed a cardiac implant. Fifteen percent of patients used a non-invasive ventilation and cataracts were removed in 26% of patients, 65% of which before the age of 50 years. The registry's primary aim was to facilitate and accelerate clinical research. However, these data also allow us to formulate questions for hypothesis-driven research that may lead to improvements in care and treatment.

Indexed as

RegistriesAdolescentAdultAgedAge DistributionArrhythmias, CardiacCataractChildChild, PreschoolCross-Sectional StudiesElectrocardiographyFatigueFemaleHumansInfantMaleMyotonin-Protein KinaseClinical trialsMyotonic dystrophyPatient RegistriesTrial readiness

Identifiers

PMID28397002
PMCPMC5413526
OpenAlexW2606591036

What OpenQuestion holds

Textmetadata
LicenceCC BY
Read underepoch 390

Registered trials

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.