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Sleep - related rhythmic movement disorder (RMD) is characterized by repetitive,stereotyped and rhythmic motor behaviors (not tremors) that occur predominantly during drowsiness or sleep and involve large muscle groups. The onset of RMD is typically in early childhood, rarely continue into adolescence and adulthood. This paper retrospectively analyzed the diagnosis and treatment in a case of adult-onset RMD. A 76 -year-old male mainly presented rhythmic forehead banging during sleep. The number of forehead banging ranged from dozens to tens of seconds, pause seconds after the second attack, lasting 1 to 2 h and can not be recalled after waking up. Polysomnography (PSG) showed sleep efficiency decreased, awakening time prolonged, times of wakefulness increased, sleep structure disordered, non-rapid eye movement (NREM) 1 and 2 sleep increased, rapid eye movement (REM) sleep decreased, sleep latency of REM prolonged. Apnea hypopnea index (AHI) was 8.40. Several abnormal behavior attacks were observed, each attack lasting 3 to 18 s, 0.60 to 1.20 Hz, total time lasting 6 min. The diagnosis was RMD, clonazepam 0.50 mg/night and pramipexole 0.0625 mg/night were given and the result of 33 months follow-up showed effective. By literature review, we know the clinical features, diagnosis and treatment of RMD. We also analyze the reasons of misdiagnosis for reducing diagnostic errors and mistreatments. DOI: 10.3969/j.issn.1672-6731.2017.09.008