Abstract
BACKGROUND: Pleomorphic leiomyosarcomas (PLMSs) are extremely rare tumors. We present the first case of hepatic primary PLMS after surgery for gastric gastrointestinal stromal tumor (GIST). CASE PRESENTATION: The patient was a 62-year-old man who was referred to our hospital for resection of a hepatic tumor arising after gastric GIST surgery that was resistant to imatinib and sunitinib. A 40-mm tumor in the left lobe of the liver and three small nodules in the right lobe were detected. We performed hepatic left lobectomy and partial resections for three lesions. According to the histopathological and immunohistochemical findings and c-kit gene mutations analysis, the main tumor was diagnosed as a PLMS. CONCLUSION: It is necessary to consider the possibility that imatinib-resistant GIST recurrence lesions are a different kind of soft-tissue sarcoma. Accurate diagnosis is required to not miss the opportunity for radical excision of PLMS.