Diagnosis, follow-up and management of sleep-disordered breathing in children with osteogenesis imperfecta / Antoine Léotard in Annals of physical and rehabilitation medicine, Vol. 61, n°3 (Mai 2018)

Public ISBD [article]  inAnnals of physical and rehabilitation medicine > Vol. 61, n°3 (Mai 2018) . - p. 135-139 Titre : Diagnosis, follow-up and management of sleep-disordered breathing in children with osteogenesis imperfecta Type de document : texte imprimé Auteurs : Antoine Léotard ; J. Taytard ; Marion Aouate ; Michèle Boule ; V. Forin ; P. Lallemant-Dudek Année de publication : 2018 Article en page(s) : p. 135-139 Note générale : Doi : 10.1016/j.rehab.2018.02.001 Langues : Anglais (eng) Mots-clés : Osteogenesis imperfecta  Polysomnography  Sleep-disordered breathing  Motor function  Walking autonomy Résumé : Objectives Osteogenesis imperfecta (OI) is the most common genetic skeletal disorder. Extraskeletal findings are common but an association with sleep-disordered breathing (SDB) has never been described. The aim of this study was to investigate clinical features of children with OI and suspected SDB. Methods A retrospective study of clinical records, signs of SDB and polysomnographic recordings of children with OI was performed. We paid particular attention to symptoms that could be associated with SDB in this population – scoliosis, kyphosis, vertebral arthrodesis, chest wall deformities, basilar impression, autonomy – as well as data already known to be associated with obstructive sleep apnea such as body mass index and upper-airway impairment. Results We reviewed the clinical charts of 188 patients referred to our genetic skeletal disorders reference center for OI. Among the 15 patients (8%) with polysomnographic recordings, 12 (6.4%) had sleep-disordered breathing. We found a negative correlation between the Brief Assessment of Motor Function score and Apnea Hypopnea Index (r=−0.68; p=0.01) and Desaturation Index (r=−0.62; p=0.02). The Apnea Hypopnea Index was higher for non-walkers than walkers (mean [SD]: 6.5 [3.6] vs. 2.4 [1.5]; p=0.02) and with type III versus IV OI. Two patients were started on continuous positive airway pressure ventilation, with clinical improvement. Conclusion For OI children, symptoms suggesting obstructive sleep disorders should be searched for systematically, especially in children with compromised autonomy, high body mass index, trunk deformations, and severe OI type. Permalink : ./index.php?lvl=notice_display&id=80460 [article] Diagnosis, follow-up and management of sleep-disordered breathing in children with osteogenesis imperfecta [texte imprimé] / Antoine Léotard ; J. Taytard ; Marion Aouate ; Michèle Boule ; V. Forin ; P. Lallemant-Dudek . - 2018 . - p. 135-139. Doi : 10.1016/j.rehab.2018.02.001 Langues : Anglais (eng) in Annals of physical and rehabilitation medicine > Vol. 61, n°3 (Mai 2018) . - p. 135-139 Mots-clés : Osteogenesis imperfecta  Polysomnography  Sleep-disordered breathing  Motor function  Walking autonomy Résumé : Objectives Osteogenesis imperfecta (OI) is the most common genetic skeletal disorder. Extraskeletal findings are common but an association with sleep-disordered breathing (SDB) has never been described. The aim of this study was to investigate clinical features of children with OI and suspected SDB. Methods A retrospective study of clinical records, signs of SDB and polysomnographic recordings of children with OI was performed. We paid particular attention to symptoms that could be associated with SDB in this population – scoliosis, kyphosis, vertebral arthrodesis, chest wall deformities, basilar impression, autonomy – as well as data already known to be associated with obstructive sleep apnea such as body mass index and upper-airway impairment. Results We reviewed the clinical charts of 188 patients referred to our genetic skeletal disorders reference center for OI. Among the 15 patients (8%) with polysomnographic recordings, 12 (6.4%) had sleep-disordered breathing. We found a negative correlation between the Brief Assessment of Motor Function score and Apnea Hypopnea Index (r=−0.68; p=0.01) and Desaturation Index (r=−0.62; p=0.02). The Apnea Hypopnea Index was higher for non-walkers than walkers (mean [SD]: 6.5 [3.6] vs. 2.4 [1.5]; p=0.02) and with type III versus IV OI. Two patients were started on continuous positive airway pressure ventilation, with clinical improvement. Conclusion For OI children, symptoms suggesting obstructive sleep disorders should be searched for systematically, especially in children with compromised autonomy, high body mass index, trunk deformations, and severe OI type. Permalink : ./index.php?lvl=notice_display&id=80460

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