Document Detail

Temporal trends of cardiac and respiratory responses to ventilatory challenges in congenital central hypoventilation syndrome.
MedLine Citation:
PMID:  15028846     Owner:  NLM     Status:  MEDLINE    
Congenital central hypoventilation syndrome (CCHS) patients exhibit respiratory deficits to ventilatory challenges, diminished breathing drive during sleep, and reduction of respiratory-related heart rate variation, but at least partially preserved peripheral chemoreception. We hypothesized that integration of afferent activity with respiratory motor output is deficient in CCHS, rather than chemoreceptor failure, and that examination of trends in heart and breathing rates and variabilities following ventilatory challenges may clarify the deficient mechanisms. Twelve children with CCHS and 12 age- and gender-matched control cases were subjected to hyperoxic hypercapnic, poikylocapnic hypoxic, and hyperoxic challenges while supine. Heart and respiratory rates and variabilities during 60-s baseline and 120-s challenge periods were assessed. Hypoxia and hypercapnia enhanced breathing rate in control subjects; in CCHS cases, the rise differed during hypercapnia and did not occur to hypoxia. Hyperoxia showed initial transient patterns in breathing rate that differed between groups. A heart rate increase to hypoxia and late decline to hyperoxia were muted in CCHS patients. In hypercapnia, heart rate followed similar rising patterns in both groups. Overall CCHS heart rate variability was lower in baseline and challenge periods, principally due to diminished respiratory-related variation, especially during hypercapnia. No heart rate variability group differences emerged in hypoxia, and only a late increase for CCHS cases developed in hyperoxia. The findings indicate retention of aspects of chemoreceptor sensitivity in CCHS cases. The heart rate alterations to ventilatory challenges suggest specific compensatory responses of a slower nature remain intact in CCHS, whereas other rapidly changing components are deficient.
Paul M Macey; Claire Valderama; Amy H Kim; Mary A Woo; David Gozal; Thomas G Keens; Rebecca K Harper; Ronald M Harper
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Publication Detail:
Type:  Journal Article; Research Support, U.S. Gov't, P.H.S.     Date:  2004-03-17
Journal Detail:
Title:  Pediatric research     Volume:  55     ISSN:  0031-3998     ISO Abbreviation:  Pediatr. Res.     Publication Date:  2004 Jun 
Date Detail:
Created Date:  2004-05-24     Completed Date:  2004-12-06     Revised Date:  2007-11-14    
Medline Journal Info:
Nlm Unique ID:  0100714     Medline TA:  Pediatr Res     Country:  United States    
Other Details:
Languages:  eng     Pagination:  953-9     Citation Subset:  IM    
Department of Neurobiology, David Geffen School of Medicine at UCLA, University of California at Los Angeles, Los Angeles, CA 90095-1763, USA.
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MeSH Terms
Afferent Pathways / physiopathology
Analysis of Variance
Anoxia / physiopathology
Case-Control Studies
Heart Rate
Hypercapnia / physiopathology
Hyperoxia / physiopathology
Sleep Apnea, Central / congenital,  physiopathology*
Time Factors
Trigeminal Nerve / physiopathology
Grant Support

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