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Catathrenia in children: clinical presentation, polysomnographic features, natural history, and response to therapy
Eliot S Katz1, Carolyn D'Ambrosio2
1Division of Respiratory Diseases, Boston Children's Hospital, Harvard Medical School, Boston, Massachusetts.
Insights
Catathrenia in children often presents incidentally during sleep-disordered breathing evaluations. While treatment for obstructive sleep apnea can reduce groaning, it persists, indicating distinct underlying conditions.
Area of Science:
- Pediatric Sleep Medicine
- Respiratory Sleep Disorders
Background:
- Catathrenia, characterized by vocalizations during sleep, is a rare parasomnia.
- Understanding its clinical presentation and response to treatment in children is crucial for accurate diagnosis and management.
Purpose of the Study:
- To investigate the clinical features, polysomnographic findings, and therapeutic responses in pediatric catathrenia.
- To differentiate catathrenia from other sleep-related breathing disorders in children.
Main Methods:
- Retrospective analysis of 21 children diagnosed with catathrenia at Boston Children's Hospital Sleep Center (1998-2021).
- Evaluation of clinical presentation, polysomnography (PSG) data, and treatment outcomes, particularly for obstructive sleep apnea (OSA).
Main Results:
- Catathrenia was diagnosed incidentally in 76% of cases, often associated with bruxism (62%).
- Events occurred post-arousal (79%) and predominantly in the latter half of the night (73%), across REM and non-REM sleep.
- Treatment of OSA improved or resolved OSA but only reduced catathrenia.
Conclusions:
- Pediatric catathrenia is frequently diagnosed incidentally, with onset often preceding diagnosis by years.
- While OSA treatment can mitigate catathrenia, its persistence suggests distinct pathophysiology.
- Catathrenia can occur independently of sleep-disordered breathing and does not typically cause significant daytime sleepiness or behavioral issues in children.
Study Objectives:
To determine the clinical presentation, polysomnographic appearance, and the response to therapy of catathrenia in children.
Methods:
A retrospective analysis was performed in children referred or evaluated at the Boston Children's Hospital Sleep Center between 1998 and 2021 who were documented to have catathrenia either as a chief complaint or an incidental finding.
Results:
Catathrenia was identified in 21 children. The age of onset by parent report was 6.4 ± 4.4 years (range: birth-14 years), but the diagnosis was at 8.3 ± 3.8 years (range: 1.5-14 years). Catathrenia was the chief complaint for 24% of patients and was incidentally identified in 76% referred for breathing concerns. Bruxism was observed in 62% of patients. Catathrenia events most often occur following an electrocortical arousal (79%) and are most common in the second half of the night (73%). Catathrenia was observed exclusively in rapid eye movement (REM) sleep (16%), exclusively in non-REM sleep (32%), and in both sleep states (52%). Treatment of obstructive sleep apnea resulted in resolution or improvement in the obstructive sleep apnea in all patients, but the catathrenia persisted at a reduced level.
Conclusions:
Most catathrenia in children is diagnosed incidentally during evaluation for sleep-disordered breathing, although the groaning often started many years earlier. Catathrenia events decreased considerably after treatment of obstructive sleep apnea, but persisted in all patients. Catathrenia was also observed in children without signs or symptoms of sleep-disordered breathing, indicating that these are distinct conditions. The majority of children with catathrenia had no sleepiness or behavioral concerns.
Citation:
Katz ES, D'Ambrosio C. Catathrenia in children: clinical presentation, polysomnographic features, natural history, and response to therapy. J Clin Sleep Med. 2023;19(8):1505-1511.
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