ABSTRACT
Background: Brain metastases with hematoma are clinically important as they indicate the potential for rapid neurological deterioration. Non-uterine leiomyosarcoma-derived brain metastases are particularly rare, and their clinical features, including the bleeding rate, are unclear. Herein, we present a rare case of thigh leiomyosarcoma-derived brain metastasis with intratumoral hematoma and review previous case reports. Case Description: A 68-year-old man with a right thigh leiomyosarcoma presented with multiple brain metastases. The patient received stereotactic radiotherapy; however, he reported sudden right-sided hemiparesis. We found a right frontal irradiated lesion with intratumoral hemorrhage and performed gross total tumor resection. Histopathological examination showed highly atypical cells with prominent necrosis and hemorrhage. Abnormal thin-walled vessels were prominent within the brain tumor, and vascular endothelial growth factor was diffusely expressed immunohistopathologically. To date, 11 cases of brain metastasis from non-uterine leiomyosarcoma, including the present case, have been reported. Of note, six patients had hemorrhage. Three out of six patients presented with hemorrhage before therapeutic intervention, three cases were from residual sites after surgery or radiation. Conclusion: More than half the patients with non-uterine leiomyosarcoma-derived brain metastases presented with intracerebral hemorrhage. Furthermore, these patients are at risk of developing rapid neurological deterioration due to intracerebral hemorrhage.
ABSTRACT
BACKGROUND: Leiomyosarcoma is a subtype of soft tissue sarcoma with adverse outcomes. Leiomyosarcoma accounts for nearly 70% of all uterine sarcomas and is responsible for a considerable proportion of deaths because of uterine cancer. Clinical characteristics and relevant diagnosis of pelvic leiomyosarcoma should be further explored. AIM: To identify the outcome and relevant perioperative evaluation of patients with pelvic leiomyosarcoma. METHODS: The Kaplan-Meier method was used to determine progression-free survival and overall survival rates. Factors predictive of outcomes were identified using univariate and multivariate Cox proportional hazards models. RESULTS: Fifty-one patients with pelvic leiomyosarcoma were enrolled and divided into two groups including uterine leiomyosarcoma and non-uterine leiomyosarcoma. Overall, 28.6% and 45.5% of uterine leiomyosarcoma and non-uterine leiomyosarcoma patients, respectively, had elevated carbohydrate antigen 125 levels, whereas 45.7% and 68.8%, respectively, underwent ultrasonography. Although 68.8% of uterine leiomyosarcoma patients were initially diagnosed with hysteromyoma, 72.7% of non-uterine leiomyosarcoma patients had pelvic and abdominal masses. Moreover, 93.3% of the recurrent lesions were detected using ultrasonography. Patients with International Federation of Gynaecology and Obstetrics (FIGO) stages III-IV disease had poorer progression-free survival values than those with FIGO stages I-II (P = 0.027) disease. FIGO stage was significantly associated with poor progression-free survival in the univariate (hazard ratio = 2.64, P = 0.03) and multivariate (hazard ratio = 2.49, P = 0.048) analyses. CONCLUSION: Serum tumour biomarkers cannot be used for pelvic leiomyosarcoma diagnosis. FIGO stage is critical to predict the outcome of uterine leiomyosarcoma. Ultrasonography is more reliable for postoperative follow-up than preoperative diagnosis.