Isolated congenital megacystis without intestinal obstruction: a mild variant of chronic intestinal pseudoobstruction syndrome?

Abstract Megacystis is frequently involved with chronic intestinal pseudoobstruction syndrome; however, isolated megacystis without intestinal obstruction is extremely rare. We present the case of a female patient with isolated congenital megacystis without severe intestinal obstruction. In this cas...

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Bibliographic Details
Published in:Journal of pediatric surgery 2011-11, Vol.46 (11), p.e29-e32
Main Authors: Shimizu, Masaki, Nishio, Sayaka, Ueno, Kazuyuki, Yokoyama, Tadafumi, Sakai, Seisho, Nagaoki, Shuya, Sugimoto, Naotoshi, Ohta, Kazuhide, Miyamoto, Masatoshi, Yachie, Akihiro
Format: Article
Language:English
Subjects:
Barium Sulfate
Chronic intestinal pseudoobstruction syndrome
Duodenum - abnormalities
Duodenum - diagnostic imaging
Enema
Female
Fetal Diseases - classification
Fetal Diseases - diagnostic imaging
Hirschsprung Disease - diagnosis
Hirschsprung Disease - pathology
Humans
Intestinal Pseudo-Obstruction - classification
Intestinal Pseudo-Obstruction - etiology
Magnetic Resonance Imaging
Megacystis
Megacystis microcolon intestinal hypoperistalsis syndrome
Myenteric Plexus - abnormalities
Pediatrics
Radionuclide Imaging
Submucous Plexus - abnormalities
Surgery
Ultrasonography, Prenatal
Urinary Bladder - abnormalities
Urinary Bladder - diagnostic imaging
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Summary:Abstract Megacystis is frequently involved with chronic intestinal pseudoobstruction syndrome; however, isolated megacystis without intestinal obstruction is extremely rare. We present the case of a female patient with isolated congenital megacystis without severe intestinal obstruction. In this case, barium enema did not reveal any significant findings; however, histologic evaluation of her rectum showed hypoganglionosis of the submucous and myenteric plexuses. These findings indicate that this case may be a mild variant of chronic intestinal pseudoobstruction syndrome. The presence of megacystis should alert the physician to the possibility of chronic intestinal pseudoobstruction syndrome.
ISSN:0022-3468
1531-5037
DOI:10.1016/j.jpedsurg.2011.08.012