Study Objectives Rhythmic movements (RMs) during sleep are frequent and often considered as benign in children. Disabling forms are diagnosed as RM disorder and may persist in adulthood. Whether RMs severely impact sleep architecture in patients with RM disorder remain unclear. We performed a case–control study to characterize the clinical and polysomnographic patterns of children and adults with a diagnosis of RM disorder in comparison to controls, and to assess the associations between the RMs and the sleep architecture. Methods All consecutive patients (n = 50; 27 children, 35 males) with RM disorder from a single sleep clinic (from 2006 to 2019) underwent a comprehensive clinical evaluation and a polysomnographic recording in comparison to 75 controls (42 children and 53 males). Results About 82% of children and adult patients had a complaint of disturbed nighttime sleep. Comorbid neurodevelopmental, affective or sleep disorders were found in 92% of patients. While RM sequences defined by video polysomnographic criteria were observed in 82% of patients (in wakefulness and in all sleep stages), no similar sequences were observed in controls. Patients had altered sleep continuity, with low sleep efficiency, increased wake time after sleep onset, and frequent periodic leg movements and apnea events. The severity of RMs was associated with disrupted nighttime sleep, even after controlling for comorbid motor and respiratory events. Conclusions RM disorder is a rare, highly comorbid and disabling condition both in children and adults with frequent disturbed nighttime sleep that may contribute to the burden of the disease.