Late recurrence of low-risk gastrointestinal stromal tumor of jejunum diagnosed 30 years after tumor resection: A case report and literature review

Gastrointestinal stromal tumors (GISTs) have a significant risk of metastasis, although the degree varies in each case. The present report describes a case of late recurrence of GIST that was diagnosed 30 years after the primary tumor resection. An 80-year-old man was transported to Sanjo General Hospital (Sanjo, Japan) with hemorrhagic shock from gastrointestinal bleeding. Abdominal contrast-enhanced computed tomography revealed an 11.7-cm heterogenous tumor in the retroperitoneum adjacent to the third portion of the duodenum. The patient had a medical history of resection of 'leiomyoma' of the upper jejunum when he was 50 years old. Pathological examination using archival pathological samples revealed that the previously excised tumor was GIST because the tumor cells showed positive immunoreactivity for KIT and DOG1. Treatment was started with imatinib, a selective KIT tyrosine inhibitor, even though endoscopy failed to provide biopsy specimens. Positron emission tomography conducted on the 28th treatment day revealed that imatinib completely shut down F-18-fluorodeoxyglucose uptake in the tumor, confirming that the tumor was imatinib-sensitive. A literature review yielded 12 GIST cases wherein metastases were diagnosed >10 years after primary tumor resection. Of the 12, four were originally diagnosed as benign. Clinicians should keep in mind that GISTs were formerly confused with non-GIST tumors and that there is a risk of relapse 10 years or later after curative surgery.