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The spectrum of <jats:italic>MECP2<\/jats:italic>\u2010related phenotypes is wide and it ranges from asymptomatic female carriers to severe neonatal\u2010onset encephalopathy in males. Abnormal breathing represents one of the leading features, but today little is known about polysomnographic features in RTT females; no data are available about males. We report the case of a male of Moroccan origins with a <jats:italic>MECP2<\/jats:italic> pathogenic variant and a history of encephalopathy and severe breathing disturbances in the absence of dysmorphic features. For the first time we describe in detail the polysomnographic characteristics of a <jats:italic>MECP2<\/jats:italic>\u2010mutated male and we show the relevance of severe central apneas, which may represent a new clinical clue to suggest the diagnosis. Moreover, we want to highlight the importance to maintain a high index of suspicion for <jats:italic>MECP2<\/jats:italic>\u2010related disorders in the presence of severe hypotonia, apneic crises, and respiratory insufficiency in males to permit an earlier diagnosis and the consequent definition of recurrence risk of the family and to avoid other useless and invasive exams.<\/jats:p>","DOI":"10.1002\/ajmg.a.61874","type":"journal-article","created":{"date-parts":[[2020,9,21]],"date-time":"2020-09-21T09:18:56Z","timestamp":1600679936000},"page":"2982-2987","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Sleep disordered breathing and daytime hypoventilation in a male with <scp>MECP2<\/scp> mutation"],"prefix":"10.1002","volume":"182","author":[{"given":"Elena","family":"Cacciatori","sequence":"first","affiliation":[{"name":"Fondazione IRCCS Ca' Granda  Milan 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