Reference - Detail
| RRC ID | 89879 |
|---|---|
| Author | Joo W, Choi JW, Schier AF. |
| Title | Disinhibition of cerebellar output by loss of restless legs syndrome-associated gene MEIS1. |
| Journal | Curr Biol |
| Abstract |
Genome-wide association studies have identified risk variants for restless legs syndrome (RLS),1,2,3,4,5,6 but the behavioral functions and sites of action of the corresponding genes remain unknown. Here, we analyzed zebrafish mutants for candidate RLS genes and found that meis1b is required for normal locomotion behavior and cerebellar development. Neuronal manipulation experiments indicated that loss of meis1b perturbs locomotor behavior by generating abnormal cerebellar output-a mechanism reminiscent of movement disorders such as ataxia and dystonia. |
| Volume | 36(13) |
| Pages | 3380-3387.e3 |
| Published | 2026-7-6 |
| DOI | 10.1016/j.cub.2026.05.043 |
| PII | S0960-9822(26)00648-2 |
| PMID | 42309051 |
| PMC | PMC13392613 |
| MeSH | Animals Cerebellum* / growth & development Locomotion* / genetics Myeloid Ecotropic Viral Integration Site 1 Protein* / genetics Myeloid Ecotropic Viral Integration Site 1 Protein* / metabolism Restless Legs Syndrome* / genetics Restless Legs Syndrome* / physiopathology Zebrafish* / genetics Zebrafish* / physiology Zebrafish Proteins* / genetics Zebrafish Proteins* / metabolism |
| Resource | |
| Zebrafish | gSA2AzGFF152B, hspGFFDMC90A, gSAIGFF23C, hspGFFDMC28C, hspzGFFgDMC156A, |