Abstract:
OBJECTIVE:Sleep disturbances are frequently reported in Mowat-Wilson Syndrome (MWS). The current study aimed to evaluate clinical and video-polysomnographic (VPSG) characteristics of the sleep architecture and abnormal electroencephalogram (EEG) patterns during sleep in MWS. METHODS:Sixteen individuals with MWS (range 16 months-25 years), attending the Department of Child Neurology and Psychiatry of the University of Bologna, were included. The "Sleep Disturbances Scale for Children (SDSC)" questionnaire was administered to all parents of MWS patients, and all patients underwent a VPSG recording. RESULTS:The analysis of the SDSC questionnaire revealed disturbances mainly at the sleep-wake transition and in initiating and maintaining sleep. Evaluation of sleep structure in MWS patients showed a significant reduction of total sleep time, an increase of wake after sleep onset and arousal index as compared to normal controls. An EEG pattern characterized by slowing of background activity and poverty of physiological sleep characterisitcs was observed in all patients. Moreover, in patients aged >7 years, anteriorly predominant spike and waves were observed, markedly activated by sleep configuring a sub-continuous or continuous activity. CONCLUSION:Our data (both clinical and VPSG) documented the presence of significant and clinically relevant sleep disturbances in MWS patients. Moreover, we identified a characteristic age-dependent sleep EEG pattern that could provide a new element to assist in the management of MWS.
journal_name
Sleep Medjournal_title
Sleep medicineauthors
Di Pisa V,Provini F,Ubertiello S,Bonetti S,Ricci E,Ivanovski I,Caraffi SG,Giordano L,Accorsi P,Savasta S,Raviglione F,Boni A,Grioni D,Graziano C,Garavelli L,Cordelli DMdoi
10.1016/j.sleep.2019.04.011subject
Has Abstractpub_date
2019-09-01 00:00:00pages
44-51eissn
1389-9457issn
1878-5506pii
S1389-9457(19)30126-1journal_volume
61pub_type
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