Introduction: Recurrent hypersomnia (RH) is a rare disorder without established treatment. Methods: We report 2 RH medication-responsive cases with typical characteristics of Kleine–Levin syndrome (KLS). Case-reports: A 10 y.o. girl and a 14 y.o. boy presented with sudden sleepiness for 3–9 days (every 2–3 weeks). Physical examination, brain images and blood tests were normal. Polysomnographic findings were heterogenous, including disrupted sleep architecture. MSLTs revealed 2–3 SOREMPs and short sleep latency. Carbamazepine rendered girl׳s sleep normalization, while risperidone normalized boy׳s sleep cycles. Conclusions: Facing the absence of clinical trials in RH, reports of responsive cases are the available therapeutic evidence.
