Evidence map›Paper›PMID 27839945›Full record

ReviewSleep medicine2017

Animal models of RLS phenotypes.

Richard P Allen, Nathan C Donelson, Byron C Jones, Yuqing Li, Mauro Manconi, David B Rye, Subhabrata Sanyal, Juliane Winkelmann

Abstract readReview
In one paragraph

Review in Sleep medicine, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 20 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
20citing papers in PubMed, 1 pooled it
3.8field-weighted citation impact, top 6% 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

20 citing papers in PubMed, 1 synthesis or guideline pooled it, 36 citations in OpenAlex.

  1. Putative Animal Models of Restless Legs Syndrome: A Systematic Review and Evaluation of Their Face and Construct Validity.Neurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics · 2023
    Pooled it
  2. Article
  3. Article
  4. Article
  5. Article
  6. Review
  7. Article
  8. Article
  9. Article
  10. Article
  11. Homeobox gene Meis1 modulates cardiovascular regeneration.Seminars in cell & developmental biology · 2020
    Review
  12. Review
  13. Article
  14. Article
  15. New Insights into the Neurobiology of Restless Legs Syndrome.The Neuroscientist : a review journal bringing neurobiology, neurology and psychiatry · 2019
    Review
  16. Frontiers in neurology · 2019
    Review
  17. Article
  18. Article
  19. Disease models & mechanisms · 2017
    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

8 authors at 6 institutions in 3 countries.

Richard P AllenJohns Hopkins Research Institute, Asthma& Allergy Bldg 1B76b, 5501 Hopkins Bayview Blvd, Baltimore, MD 21224, USA.
Nathan C DonelsonNeurology Research, 115 Broadway, Bio 6, Biogen, Cambridge, MA 02142, USA.
Byron C JonesDepartment of Genetics, Genomics and Informatics, University of Tennessee, Memphis, TN, USA. Electronic address: bjone129@uthsc.edu.
Yuqing LiDepartment of Neurology, College of Medicine, University of Florida, Gainesville, Florida 32610, USA.
Mauro ManconiSleep Center, Neurocenter of Southern Switzerland, Civic Hospital of Lugano, Lugano, Switzerland.
David B RyeProgram in Sleep, Department of Neurology, Emory University School of Medicine, Atlanta, GA, USA.
Subhabrata SanyalNeurology Research, 115 Broadway, Bio 6, Biogen, Cambridge, MA 02142, USA.
Juliane WinkelmannInstitute of Neurogenomics, Helmholtz Zentrum München, Munich, Germany.
Biogen (United States) · USEmory University · USHelmholtz Zentrum München · DEOspedale regionale di Lugano · CHUniversity of Florida · USUniversity of Tennessee Health Science Center · US

Funding

THE IRON-DOPAMINE CONNECTIONP01AG021190 · NIA · JOHNS HOPKINS UNIVERSITY · PI EARLEY, CHRISTOPHER J · 2004 to 2009
$10.0M
Restless Legs Syndrome: Pathophysiology using Btbd9 Conditional Knockout MiceR01NS082244 · NINDS · UNIVERSITY OF FLORIDA · PI LI, YUQING · 2014 to 2018
$1.6M
Pathophysiology of DYT1 Dystonia: Targeted Mouse ModelsR01NS054246 · NINDS · UNIVERSITY OF FLORIDA · PI LI, YUQING · 2007 to 2010
$1.4M
Glutamate, hyperarousal and restless legs syndromeR01NS075184 · NINDS · JOHNS HOPKINS UNIVERSITY · PI ALLEN, RICHARD PUTNAM · 2012 to 2014
$1.2M
Pathophysiology and Animal Model of Restless Legs Syndrome (RLS): Btbd9 Null MicR21NS065273 · NINDS · UNIVERSITY OF FLORIDA · PI LI, YUQING · 2009 to 2010
$401k
Rapid-onset Dystonia Parkinsonism (DYT12 Dystonia): Pathophysiology & Atp1a3 MiceR21NS072872 · NINDS · UNIVERSITY OF FLORIDA · PI LI, YUQING · 2010 to 2011
$399k
NIA NIH HHS P01 AG021190NINDS NIH HHS R01 NS054246NINDS NIH HHS R01 NS075184NINDS NIH HHS R01 NS082244NINDS NIH HHS R21 NS065273NINDS NIH HHS R21 NS072872
6 · The paper itself

Abstract

Restless legs syndrome (RLS) is a complex disorder that involves sensory and motor systems. The major pathophysiology of RLS is low iron concentration in the substantia nigra containing the cell bodies of dopamine neurons that project to the striatum, an area that is crucial for modulating movement. People who have RLS often present with normal iron values outside the brain; recent studies implicate several genes are involved in the syndrome. Like most complex diseases, animal models usually do not faithfully capture the full phenotypic spectrum of "disease," which is a uniquely human construct. Nonetheless, animal models have proven useful in helping to unravel the complex pathophysiology of diseases such as RLS and suggesting novel treatment paradigms. For example, hypothesis-independent genome-wide association studies (GWAS) have identified several genes as increasing the risk for RLS, including BTBD9. Independently, the murine homolog Btbd9 was identified as a candidate gene for iron regulation in the midbrain in mice. The relevance of the phenotype of another of the GWAS identified genes, MEIS1, has also been explored. The role of Btbd9 in iron regulation and RLS-like behaviors has been further evaluated in mice carrying a null mutation of the gene and in fruit flies when the BTBD9 protein is degraded. The BTBD9 and MEIS1 stories originate from human GWAS research, supported by work in a genetic reference population of mice (forward genetics) and further verified in mice, fish flies, and worms. Finally, the role of genetics is further supported by an inbred mouse strain that displays many of the phenotypic characteristics of RLS. The role of animal models of RLS phenotypes is also extended to include periodic limb movements.

Indexed as

Disease Models, AnimalAnimalsHumansPhenotypeRestless Legs SyndromeFishFliesForward geneticsMiceReverse geneticsWorms

Identifiers

PMID27839945
PMCPMC5349858
OpenAlexW2514509072

What OpenQuestion holds

Textmetadata
LicenceTDM
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.