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Peculiar respiratory response observed during sleep-onset REM sleep of an infant with Ondine's curse.
Neuropediatrics. 2000 Oct; 31(5):269-72.N

Abstract

We treated an infant with congenital central hypoventilation syndrome ("Ondine's curse"). She was cyanotic and given ventilatory support at the first hour after birth. An investigation of sleep state and respiration performed at the age of 3 months led to this diagnosis. Hypoventilation persisted in all sleep stages, with the most severely reduced tidal volumes occurring during delta-wave sleep (stages 3 and 4). In addition, severe secondary reduction in tidal volumes occurred in sleep-onset REM sleep. This phenomenon was absent in non sleep-onset REM sleep. At 4 months of age, her respiratory treatment was successfully converted to positive-pressure ventilation via a nasal mask, thus avoiding tracheotomy. This transition to noninvasive ventilatory support dramatically improved her quality of life during wakefulness. This report may be a clue to discuss the function of sleep-onset REM sleep seen in the early stage of life and suggests that nasal mask ventilation is a viable option in selected cases with congenital central hypoventilation syndrome (CCHS).

Authors+Show Affiliations

Takahashi R
Department of Pediatrics, Nagasaki University School of Medicine, Japan. ritsu@net.nagasaki-u.ac.jp
Kakizawa H
No affiliation info available
Itou T
No affiliation info available
Yamada M
No affiliation info available
Nakae S
No affiliation info available

MeSH

FemaleHumansInfantInfant, NewbornIntermittent Positive-Pressure VentilationPolysomnographyRespirationSleep Apnea, CentralSleep Disorders, Circadian RhythmSleep, REMTreatment Outcome

Pub Type(s)

Case Reports
Journal Article

Language

eng

PubMed ID

11204285

Citation

Takahashi, R, et al. "Peculiar Respiratory Response Observed During Sleep-onset REM Sleep of an Infant With Ondine's Curse." Neuropediatrics, vol. 31, no. 5, 2000, pp. 269-72.
Takahashi R, Kakizawa H, Itou T, et al. Peculiar respiratory response observed during sleep-onset REM sleep of an infant with Ondine's curse. Neuropediatrics. 2000;31(5):269-72.
Takahashi, R., Kakizawa, H., Itou, T., Yamada, M., & Nakae, S. (2000). Peculiar respiratory response observed during sleep-onset REM sleep of an infant with Ondine's curse. Neuropediatrics, 31(5), 269-72.
Takahashi R, et al. Peculiar Respiratory Response Observed During Sleep-onset REM Sleep of an Infant With Ondine's Curse. Neuropediatrics. 2000;31(5):269-72. PubMed PMID: 11204285.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Peculiar respiratory response observed during sleep-onset REM sleep of an infant with Ondine's curse. AU - Takahashi,R, AU - Kakizawa,H, AU - Itou,T, AU - Yamada,M, AU - Nakae,S, PY - 2001/2/24/pubmed PY - 2001/4/6/medline PY - 2001/2/24/entrez SP - 269 EP - 72 JF - Neuropediatrics JO - Neuropediatrics VL - 31 IS - 5 N2 - We treated an infant with congenital central hypoventilation syndrome ("Ondine's curse"). She was cyanotic and given ventilatory support at the first hour after birth. An investigation of sleep state and respiration performed at the age of 3 months led to this diagnosis. Hypoventilation persisted in all sleep stages, with the most severely reduced tidal volumes occurring during delta-wave sleep (stages 3 and 4). In addition, severe secondary reduction in tidal volumes occurred in sleep-onset REM sleep. This phenomenon was absent in non sleep-onset REM sleep. At 4 months of age, her respiratory treatment was successfully converted to positive-pressure ventilation via a nasal mask, thus avoiding tracheotomy. This transition to noninvasive ventilatory support dramatically improved her quality of life during wakefulness. This report may be a clue to discuss the function of sleep-onset REM sleep seen in the early stage of life and suggests that nasal mask ventilation is a viable option in selected cases with congenital central hypoventilation syndrome (CCHS). SN - 0174-304X UR - https://www.unboundmedicine.com/medline/citation/11204285/Peculiar_respiratory_response_observed_during_sleep_onset_REM_sleep_of_an_infant_with_Ondine's_curse_ DB - PRIME DP - Unbound Medicine ER -