Evidence map›Paper›PMID 36536233›Full record

SynthesisNeurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics2023

Putative Animal Models of Restless Legs Syndrome: A Systematic Review and Evaluation of Their Face and Construct Validity.

Alessandro Silvani, Imad Ghorayeb, Mauro Manconi, Yuqing Li, Stefan Clemens

Open access · bronzeAbstract readSystematic Review
In one paragraph

Synthesis in Neurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 9 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
9citing papers in PubMed, 1 pooled it
2.0field-weighted citation impact, top 13% 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

9 citing papers in PubMed, 1 synthesis or guideline pooled it, 13 citations in OpenAlex.

  1. Treatment of restless legs syndrome and periodic limb movement disorder: an American Academy of Sleep Medicine clinical practice guideline.Journal of clinical sleep medicine : JCSM : official publication of the American Academy of Sleep Medicine · 2025
    Guideline
  2. Review
  3. Article
  4. Article
  5. Article
  6. Article
  7. A New Look at Animal Models of Neurological Disorders.Neurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics · 2023
    Article
  8. Article
  9. Vitamin D and Restless Legs Syndrome: A Review of Current Literature.Tremor and other hyperkinetic movements (New York, N.Y.) · 2023
    Review
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

5 authors at 5 institutions in 4 countries.

Alessandro SilvaniDepartment of Biomedical and Neuromotor Sciences, Alma Mater Studiorum - University of Bologna, Ravenna Campus, Ravenna, Italy.
Imad GhorayebDépartement de Neurophysiologie Clinique, Pôle Neurosciences Cliniques, CHU de Bordeaux, Bordeaux, France.
Mauro ManconiSleep Medicine Unit, Neurocenter of Southern Switzerland, EOC, Ospedale Civico, Lugano, Switzerland.
Yuqing LiDepartment of Neurology, College of Medicine, Norman Fixel Institute for Neurological Diseases, University of Florida, Gainesville, FL, USA.
Stefan ClemensDepartment of Physiology, Brody School of Medicine, East Carolina University, Greenville, NC, USA. clemenss@ecu.edu.ORCID 0000-0003-2616-0558
Centre National de la Recherche Scientifique · FREast Carolina University · USUniversity Hospital of Bern · CHUniversity of Bologna · ITUniversity of Florida · US

Funding

Supplement: Pathophysiology of DYT1 dystonia: Targeted Mouse ModelsR01NS129873 · NINDS · UNIVERSITY OF FLORIDA · PI YUQING LI · 2022 to 2026
$1.9M
Restless Legs Syndrome: Pathophysiology using Btbd9 Conditional Knockout MiceR01NS082244 · NINDS · UNIVERSITY OF FLORIDA · PI LI, YUQING · 2014 to 2018
$1.6M
Characterization of Meis1 mutant mice and implications in restless legs syndrome and other sleep disordersR21NS111498 · NINDS · UNIVERSITY OF FLORIDA · PI LI, YUQING · 2020 to 2021
$419k
NINDS NIH HHS R01 NS082244NINDS NIH HHS R01 NS129873NINDS NIH HHS R21 NS111498
6 · The paper itself

Abstract

Restless legs syndrome (RLS) is a sensorimotor disorder that severely affects sleep. It is characterized by an urge to move the legs, which is often accompanied by periodic limb movements during sleep. RLS has a high prevalence in the population and is usually a life-long condition. While its origins remain unclear, RLS is initially highly responsive to treatment with dopaminergic agonists that target D2-like receptors, in particular D2 and D3, but the long-term response is often unsatisfactory. Over the years, several putative animal models for RLS have been developed, mainly based on the epidemiological and neurochemical link with iron deficiency, treatment efficacy of D2-like dopaminergic agonists, or genome-wide association studies that identified risk factors in the patient population. Here, we present the first systematic review of putative animal models of RLS, provide information about their face and construct validity, and report their role in deciphering the underlying pathophysiological mechanisms that may cause or contribute to RLS. We propose that identifying the causal links between genetic risk factors, altered organ functions, and changes to molecular pathways in neural circuitry will eventually lead to more effective new treatment options that bypass the side effects of the currently used therapeutics in RLS, especially for long-term therapy.

Indexed as

Dopamine AgonistsRestless Legs SyndromeAnimalsGenome-Wide Association StudyHumansModels, AnimalTreatment OutcomeDopamine AgonistsDopamineGeneticsIronSpinal cordStriatum

Identifiers

PMID36536233
PMCPMC10119375
OpenAlexW4311940806

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.