ArticleCurrent biology : CB2012
Sleep fragmentation and motor restlessness in a Drosophila model of Restless Legs Syndrome.
Article in Current biology : CB, 2012. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 64 papers, 4 of them syntheses that pooled it.
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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.
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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.
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Who cites it
64 citing papers in PubMed, 4 syntheses or guidelines pooled it, 126 citations in OpenAlex.
- 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 · 2023Pooled it
- Genome-wide association analysis of self-reported daytime sleepiness identifies 42 loci that suggest biological subtypes.Nature communications · 2019Pooled it
- Genome-wide analysis of insomnia in 1,331,010 individuals identifies new risk loci and functional pathways.Nature genetics · 2019Pooled it
- Identification of novel risk loci for restless legs syndrome in genome-wide association studies in individuals of European ancestry: a meta-analysis.The Lancet. Neurology · 2017Pooled it
- RNAi-based screen for pigmentation iniScience · 2026Article
- Article
- RNAi-based screen for pigmentation inbioRxiv : the preprint server for biology · 2025Article
- Advancements in Restless Leg Syndrome Management: A Review of Physiotherapeutic Modalities and Their Efficacy.Cureus · 2023Review
- Exploring the causes of augmentation in restless legs syndrome.Frontiers in neurology · 2023Review
- Further Studies on the Role of BTBD9 in the Cerebellum, Sleep-like Behaviors and the Restless Legs Syndrome.Neuroscience · 2022Article
- Brain-iron deficiency models of restless legs syndrome.Experimental neurology · 2022Review
- Consensus guidelines on the construct validity of rodent models of restless legs syndrome.Disease models & mechanisms · 2022Article
- BTBD9 attenuates manganese-induced oxidative stress and neurotoxicity by regulating insulin growth factor signaling pathway.Human molecular genetics · 2022Article
- Modulation of Metabolic Hormone Signaling via a Circadian Hormone and Biogenic Amine inInternational journal of molecular sciences · 2022Article
- Integrative Proteome and Ubiquitinome Analyses Reveal the Substrates of BTBD9 and Its Underlying Mechanism in Sleep Regulation.ACS omega · 2022Article
- Detection of Parent-of-Origin Effects for the Variants Associated With Behavioral Disinhibition in the MCTFR Data.Frontiers in genetics · 2022Article
- Restless legs syndrome.Nature reviews. Disease primers · 2021Review
- Periodic limb movements during sleep: a narrative review.Journal of thoracic disease · 2021Review
- Association of proton pump inhibitor and histamine H2-receptor antagonists with restless legs syndrome.Sleep · 2021Observational
- TheFrontiers in neural circuits · 2021Review
4 more citing papers are in PubMed but not listed here.
Corrections and comments
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Authors and funding
9 authors at 1 institution in 1 country.
Funding
Abstract
Restless Legs Syndrome (RLS), first chronicled by Willis in 1672 and described in more detail by Ekbom in 1945, is a prevalent sensorimotor neurological disorder (5%-10% in the population) with a circadian predilection for the evening and night. Characteristic clinical features also include a compelling urge to move during periods of rest, relief with movement, involuntary movements in sleep (viz., periodic leg movements of sleep), and fragmented sleep. Although the pathophysiology of RLS is unknown, dopaminergic neurotransmission and deficits in iron availability modulate expressivity. Genome-wide association studies have identified a polymorphism in an intronic region of the BTBD9 gene on chromosome 6 that confers substantial risk for RLS. Here, we report that loss of the Drosophila homolog CG1826 (dBTBD9) appreciably disrupts sleep with concomitant increases in waking and motor activity. We further show that BTBD9 regulates brain dopamine levels in flies and controls iron homeostasis through the iron regulatory protein-2 in human cell lines. To our knowledge, this represents the first reverse genetic analysis of a "novel" or heretofore poorly understood gene implicated in an exceedingly common and complex sleep disorder and the development of an RLS animal model that closely recapitulates all disease phenotypes.
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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.