Periodic limb movements (PLM) and restless leg syndrome (RLS) are involuntary common sleep-related movements which often hamper sleep onset; they are mostly idiopathic and bilateral but are seldom described secondary after a stroke. These cases are rare, often unilateral, and because of the usually transitory duration of symptoms, often under-recognized. When a treatment is required, it can be tricky and the drug choice not foregone. We report 2 patients with unilateral poststroke PLM with similar clinical pictures but different symptoms, therapy, and outcome. The first is a long-lasting unilateral PLM video case with chronic vascular lesions leading to insomnia even if with no urgence or any subjective symptoms as in RLS but well responding only to a definite RLS treatment. The second case is an acute, short-duration self-limiting PLM with positive brain MRI lesion imaging. Our cases suggest that unilateral poststroke PLM even if distinct in subjective and radiological features from secondary RLS can sometimes have a definite and effective dopaminergic treatment if long-lasting. Putative mechanism of chronic case 1 PLM could be due to a further stroke sparing sensory pathways and making the patient unaware of subjective RLS-like symptoms.