Orlane Felix1, Alessandro Amaddeo2, Jorge Olmo Arroyo1, Michel Zerah3, Stephanie Puget3, Valerie Cormier-Daire4, Genevieve Baujat5, Graziella Pinto6, Marta Fernandez-Bolanos1, Brigitte Fauroux7. 1. Pediatric Noninvasive Ventilation and Sleep Unit, AP-HP, Hôpital Necker Enfants-Malades, Paris, France. 2. Pediatric Noninvasive Ventilation and Sleep Unit, AP-HP, Hôpital Necker Enfants-Malades, Paris, France; Paris Descartes University, Paris, France. Electronic address: alessandro.amaddeo@aphp.fr. 3. Paris Descartes University, Paris, France; Pediatric Neurosurgery, AP-HP, Hôpital Necker Enfants-Malades, Paris, France. 4. Paris Descartes University, Paris, France; Genetic Department, Imagine Institute, Paris, France. 5. Genetic Department, Imagine Institute, Paris, France. 6. Pediatric Endocrinology, AP-HP, Hôpital Necker Enfants-Malades, Paris, France. 7. Pediatric Noninvasive Ventilation and Sleep Unit, AP-HP, Hôpital Necker Enfants-Malades, Paris, France; Paris Descartes University, Paris, France; Inserm U955, Team 13, Créteil Université, Paris XII, Créteil, France.
Abstract
OBJECTIVE: Central sleep apnea (CSA) syndromes are rare in children and data in children over one year of age are scarce. The aim of the study was to describe the sleep characteristics, underlying disorders, management, and outcome of children with CSA. PATIENTS/ METHODS: A retrospective chart review of all children >1 year of age, diagnosed with CSA on a laboratory sleep study during a 20-month period, was performed. CSA was defined by a central apnea index (CAI) >5 events/h. The clinical management and the patient's outcome were analyzed. RESULTS: Eighteen of 441 (4.1%) patients recorded during the study period had CSA. The median CAI, pulse oximetry, and oxygen desaturation index were 13/h (range 6-146), 96% (93-98%), and 18/h (6-98), respectively. Neurosurgical pathologies represented the most common underlying disorders with Arnold-Chiari malformation in four and ganglioglioma in three patients. Other underlying disorders were Prader-Willi syndrome (N = 3), achondroplasia (N = 2), and Down syndrome, with one patient having an achondroplasia and a Down syndrome. The remaining six patients had other genetic diseases. The most common investigation was brain magnetic resonance imaging (MRI). Individualized management with neurosurgery and/or chemotherapy, continuous positive airway pressure (in two patients having associated obstructive events), or noninvasive ventilation resulted in an improvement in CSA and the clinical presentation in 11 patients. CONCLUSION: CSA is rare in children >1 year of age. Underlying disorders are dominated by neurosurgical disorders. Individualized management is able to improve CSA and the clinical condition in most patients.
OBJECTIVE:Central sleep apnea (CSA) syndromes are rare in children and data in children over one year of age are scarce. The aim of the study was to describe the sleep characteristics, underlying disorders, management, and outcome of children with CSA. PATIENTS/ METHODS: A retrospective chart review of all children >1 year of age, diagnosed with CSA on a laboratory sleep study during a 20-month period, was performed. CSA was defined by a central apnea index (CAI) >5 events/h. The clinical management and the patient's outcome were analyzed. RESULTS: Eighteen of 441 (4.1%) patients recorded during the study period had CSA. The median CAI, pulse oximetry, and oxygen desaturation index were 13/h (range 6-146), 96% (93-98%), and 18/h (6-98), respectively. Neurosurgical pathologies represented the most common underlying disorders with Arnold-Chiari malformation in four and ganglioglioma in three patients. Other underlying disorders were Prader-Willi syndrome (N = 3), achondroplasia (N = 2), and Down syndrome, with one patient having an achondroplasia and a Down syndrome. The remaining six patients had other genetic diseases. The most common investigation was brain magnetic resonance imaging (MRI). Individualized management with neurosurgery and/or chemotherapy, continuous positive airway pressure (in two patients having associated obstructive events), or noninvasive ventilation resulted in an improvement in CSA and the clinical presentation in 11 patients. CONCLUSION: CSA is rare in children >1 year of age. Underlying disorders are dominated by neurosurgical disorders. Individualized management is able to improve CSA and the clinical condition in most patients.
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