학술논문

A Case of Infantile Myofibromatosis (Solitary Type) With Small Bowel Stenosis / 小腸狭窄をきたした乳幼児筋線維腫症(infantile myofibromatosis, solitary type)の1例
Document Type
Journal Article
Source
日本小児外科学会雑誌 / Journal of the Japanese Society of Pediatric Surgeons. 2022, 58(5):799
Subject
bilious vomiting
myofibromatosis
neonate
small intestinal tumor
小腸腫瘍
新生児
筋線維腫
胆汁性嘔吐
Language
Japanese
ISSN
0288-609X
2187-4247
Abstract
The patient was an 11-day-old female who presented with bilious vomiting. Low-allergen formula was given for the putative diagnosis of milk allergy, but the bilious vomiting persisted. She was transferred to our hospital and an upper gastrointestinal contrast study demonstrated a jejunal stenosis 50 cm on the anal side from the ligament of Treitz. She was diagnosed as having congenital jejunal stenosis and laparotomy was performed. A stenotic lesion was found in the jejunum at 45 cm from the ligament of Treitz. The lesion was resected, and the jejunum was repaired by an end-to-end anastomosis. The lumen of the specimen was occupied by a mass, that caused intestinal obstruction. The patient was started on oral feeding on postoperative day 4 and was discharged on postoperative day 25 after an uneventful course. Microscopic examination of the resected mass revealed a bundle-like proliferation of spindle-shaped cells with ellipse nuclei, and the histopathological diagnosis was infantile myofibromatosis (solitary type). Systemic exploration demonstrated no mass lesions elsewhere, and no postoperative treatment was given. She remains healthy without recurrence after six years.