Background and purpose: The objective was to evaluate the presence of Restless Legs Syndrome (RLS) in acute stroke, its association with sleep disturbances and clinical outcome during long-term follow-up. Methods: This was a longitudinal study (N = 96, 59 men, mean age 64.0 ± 8.9) of cases with acute ischaemic stroke. Patients were asked about the occurrence of RLS symptoms before the cerebrovascular event. RLS was diagnosed using the criteria established by the International RLS Study Group. Stroke outcome was estimated by the Barthel Index and the modified Rankin Scale. Daytime somnolence (Epworth Sleepiness Scale -ESS > 10), poor sleep quality (Pittsburgh Sleep Quality Index -PSQI > 5) and risk of obstructive sleep apnea (OSA) (Berlin questionnaire) were evaluated. Results: Twelve patients (12.5%) met the diagnostic criteria for RLS. All cases had symptoms of RLS before stroke. However, none of the cases had a previous medical diagnosis of RLS or were on use of specific medication. In only one case, a family history of RLS was found. In all patients, RLS symptoms started after the age of 40 (mean age 64 ± 6.7). Daytime sleepiness (44.8%) and poor quality sleep (62.8%) were present. Patients with RLS (12.5%) presented greater neck circumference (P = 0.04) and worse sleep quality (P = 0.007). Risk of OSA (56.2%) was associated with hypertension [OR = 0.12; CI=0.03-0.42]. Stroke outcome was significantly worse at three and 12 months (ANCOVA, P < 0.005) in patients with RLS, remaining after adjustment for diabetes and body mass index (P < 0.05). Conclusions: Patients with acute stroke and RLS have worse clinical outcome, at three and 12 months of follow-up.