A benign metastasizing leiomyoma involving a nodule in the pulmonary artery: case and literature review

Abstract

Benign metastasizing leiomyoma (BML) is a rare disease defined as a primary benign uterine tumor with "metastatic" lesions preferentially occurring in the lung, pelvis and lymph nodes. There are few reports about local recurrence after initial surgery. We report a case of a BML with local recurrence and metastasis growing into the wall of the left pulmonary artery, diagnosed 11 years after initial hysterectomy. A 55-year-old woman complaining of abdominal discomfort, heaviness and asthenia was admitted to our hospital for investigation of a voluminous uterine mass with high vascularization and three pulmonary nodules. The resection of the mass by laparotomy was complicated by uncontrolled severe hemorrhage due to vascular proliferation, requiring multiple transfusions, packing the cavity and postoperative uterine artery embolization. Three months later the patient underwent a left upper lobe lobectomy with the aim of removing the largest pulmonary nodule, a nodule a located in the lingular branch of the left pulmonary artery. The comparison of hysterectomy and lobectomy pieces showed a similar aspect, leading thus to the diagnosis of BML. Awareness of this rare entity should potentially avoid under-diagnosis and difficulties due to hemorrhage during surgery.