Yu Sugai1, Yutaka Hirayama2, Yasushi Iinuma2, Kengo Nakaya2, Takato Aikou2,3, Shotaro Taki2, Hideki Hashidate4, Yoshiaki Kinoshita5. 1. Department of Pediatric Surgery, Niigata-City General Hospital, 463-7 Shumoku, Chuo-ku, Niigata City, Niigata, 950-1197, Japan. hiiiiichan.1011@gmail.com. 2. Department of Pediatric Surgery, Niigata-City General Hospital, 463-7 Shumoku, Chuo-ku, Niigata City, Niigata, 950-1197, Japan. 3. Department of Pediatric Surgery, Nagaoka Red Cross Hospital, 2-297-1 Senshu, Nagaoka City, Niigata, 940-2085, Japan. 4. Department of Pathology, Niigata-City General Hospital, 463-7 Shumoku, Chuo-ku, Niigata City, Niigata, 950-1197, Japan. 5. Division of Pediatric Surgery, Niigata University Graduate School of Medical and Dental Sciences, 1-757 Asahimachi-dori, Chuo-ku, Niigata City, Niigata, 951-8510, Japan.
Abstract
BACKGROUND: Intestinal obstruction caused by a tumor is very rare in newborns, and the preoperative diagnosis is difficult. We herein report a rare case of neonatal colonic obstruction due to solitary intestinal myofibroma with characteristic findings on gastrografin enema and the surgical strategy. CASE PRESENTATION: A 4-day-old female infant presented to our neonatal intensive-care unit with abdominal distention and bilious vomiting after feeding. A gastrografin enema showed that the transverse colon near the hepatic flexure was not delineated at the oral side. When pressure was applied, a small amount of contrast material moved into the mouth in the form of threads. Microcolon was not observed, and stenosis of the transverse colon was found 9 cm from the Bauhin valve. Partial resection and end-to-end anastomosis were performed. A pathological examination of the resected specimen suggested gastrointestinal stromal tumor (GIST). After obtaining a second opinion, the histology and immunohistological markers were deemed characteristic of infantile myofibroma. CONCLUSION: If string sign and a napkin ring appearance are found in a case of neonatal intestinal obstruction, surgery should be performed with a tumor in mind. In cases of neonatal intestinal obstruction caused by a tumor, the lesion should be resected with a sufficient surgical margin before the pathological examination.
BACKGROUND:Intestinal obstruction caused by a tumor is very rare in newborns, and the preoperative diagnosis is difficult. We herein report a rare case of neonatal colonic obstruction due to solitary intestinal myofibroma with characteristic findings on gastrografin enema and the surgical strategy. CASE PRESENTATION: A 4-day-old female infant presented to our neonatal intensive-care unit with abdominal distention and bilious vomiting after feeding. A gastrografin enema showed that the transverse colon near the hepatic flexure was not delineated at the oral side. When pressure was applied, a small amount of contrast material moved into the mouth in the form of threads. Microcolon was not observed, and stenosis of the transverse colon was found 9 cm from the Bauhin valve. Partial resection and end-to-end anastomosis were performed. A pathological examination of the resected specimen suggested gastrointestinal stromal tumor (GIST). After obtaining a second opinion, the histology and immunohistological markers were deemed characteristic of infantile myofibroma. CONCLUSION: If string sign and a napkin ring appearance are found in a case of neonatal intestinal obstruction, surgery should be performed with a tumor in mind. In cases of neonatal intestinal obstruction caused by a tumor, the lesion should be resected with a sufficient surgical margin before the pathological examination.