Thought this might be of interest, Judy Ritchie



http://www.medscape.com/viewarticle/434259?srcmp=gas-053102
<http://www.medscape.com/viewarticle/434259?srcmp=gas-053102&WebLogicSes
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&WebLogicSession=PP2MMjEHn5DEl82cH9iHzCLXDJMwtyoqlZeAn2cPbXaAAKyvv6Eq|17
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Esophageal Dysfunction Identified in Children With Rare Breathing
Disorder





NEW YORK (Reuters Health) May 28 - Esophageal dysmotility appears to be
a universal finding in children with congenital central hypoventilation
syndrome (CCHS), according to a report published in the May issue of
Gastroenterology.

CCHS is a rare disorder with only about 200 cases reported in the
literature, study author Dr. Christophe Faure, from the University of
Montreal, and colleagues note. While the etiology and pathophysiology of
the syndrome are unknown, about 20% of CCHS patients have Hirschsprung's
disease, suggesting a link between central breathing mechanisms and
neural crest-derived innervation of the gastrointestinal tract.

Because such innervation is not limited to the colon, Dr. Faure's team
decided to screen CCHS patients for esophageal dysmotility. Esophageal
manometric testing was performed in seven children, 11 to 18 years of
age, with CCHS and in seven age- and sex-matched control children. None
of the children had any upper gastrointestinal tract symptoms.

All of the CCHS patients demonstrated abnormal pressure wave
propagation, and the median percentage of propagated swallows in the
CCHS group was significantly lower than in the control group (p <
0.001).

In addition, lower esophageal sphincter (LES) relaxation was
significantly reduced in the CCHS group (p = 0.01). The median baseline
LES pressures of the two groups were not significantly different, but
two CCHS patients had baseline pressures above the 95th percentile of
control subject values.

The esophageal dysfunction observed "may reflect central swallowing
dysfunction," the authors postulate. "The clinical significance of
esophageal dysmotility in CCHS patients is unknown, underlining the need
for lifelong multidisciplinary followup in patients with this disease."

Gastroenterology 2002;122:1258-1263.




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