症例報告小児の上行結腸に発生したGIST関連腫瘍の1例

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A rare case of colonic leiomyoblastoma in a girl 2 years of age is reported. The patient complained of abdominal pain, mucous bloody stool and presented with a palpable mass at the right flank. She was found to have a hematocrit of 28.5%. Computed tomography demonstrated a well defined homogenous ovoid mass of soft tissue density. Air-contrast barium enema findings revealed a round and constant filling defect of the proximal ascending colon. At surgery, a firm, rubbery mass measuring 4 cm in diameter was identified arising from the posterior wall of the ascending colon. The colonic mass was removed using primary colocolic anastomosis, and the ileocecal valve was preserved. Macroscopically, the mass was a 4.0×3.5×3.0 cm submucosal tumor. Histopathologically, the tumor was diagnosed to be a leiomyoblastoma. After an extended follow up of 20 years, the patient has remained healthy with no recurrence.To our knowledge, sixteen cases of colonic gastrointestinal stromal tumors (GIST) and related lesions in children have been reported in the literature during the past 25 years. A colonic leiomyoblastoma is especially uncommon. Despite the rarity in children, GIST shoud be considered in differential diagnosis of abdominal pain, masses and hematochezia after excluding common causes. The surgical approach need not always be aggressive, but immunohistochemical diagnosis and prolonged follow-up are essential.
学校法人昭和大学・昭和医学会の論文