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

Masaki Shimizu, Sayaka Nishio, Kazuyuki Ueno, Tadafumi Yokoyama, Seisho Sakai, Shuya Nagaoki, Naotoshi Sugimoto, Kazuhide Ohta, Masatoshi Miyamoto, Akihiro Yachie

Department of Pediatrics, School of Medicine, Institute of Medical, Pharmaceutical, and Health Sciences, Kanazawa University, 13-1 Takaramachi, Kanazawa 920-8641, Japan. shimizum@staff.kanazawa-u.ac.jp

Journal of pediatric surgery 2011 Nov

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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. Copyright © 2011 Elsevier Inc. All rights reserved.

Citation

Masaki Shimizu, Sayaka Nishio, Kazuyuki Ueno, Tadafumi Yokoyama, Seisho Sakai, Shuya Nagaoki, Naotoshi Sugimoto, Kazuhide Ohta, Masatoshi Miyamoto, Akihiro Yachie. Isolated congenital megacystis without intestinal obstruction: a mild variant of chronic intestinal pseudoobstruction syndrome? Journal of pediatric surgery. 2011 Nov;46(11):e29-32