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Background: Intravenous leiomyomatosis is a special type of uterine leiomyoma and features formation and growth of benign leiomyoma tissue within vascular wall. Benign metastatic leiomyoma refers to benign leiomyoma metastasizing to extra-uterine sites, dominantly lung. Solitary or multiple small nodules in the lung can be seen in image scans., Methods: We report 2 cases of intravenous leiomyomatosis with inferior vena cava or intracardiac extension and concurrent multiple nodules in bilateral lungs., Results: Case 1 was a 40-year-old woman with a large mass in pelvic cavity, masses in heart chambers, and disseminates pulmonary nodules detected at preoperative image scans. Masses in pelvic cavity and heart were resected in a 2-stage surgery. Histology examination confirmed the diagnosis of intravenous leiomyomatosis. Pulmonary nodules stayed stable during follow-up. Case 2 was a 37-year-old woman with 3 times of uterine-related surgeries. A pelvic mass appeared again and filling defect was observed in left ovarian vein, right renal vein, right common iliac vein, and inferior vena cava. Tumors in pelvic cavity and within vessels were removed in a 1-stage surgery. Histology examination confirmed the diagnosis of intravenous leiomyomatosis. Pulmonary nodules remained stable during follow-up., Conclusion: The incidence of benign metastatic leiomyoma in patients with intravenous leiomyomatosis might be relatively high. Metastasis of intravenous leiomyomatosis lesions was a possible source of benign metastatic leiomyoma in these cases., Competing Interests: The authors have no funding and conflicts of interest to disclose.

Pulmonary benign metastasizing leiomyoma (PBML) is a rare entity usually occurring in females with history of uterine leiomyoma, and it is preferential to metastasize to the lung and appears as a histopathologic benign tumor of smooth muscle origin. In this article, the clinical and pathological data from 1 patient with PBML were analyzed. Chest CT scan showed that multiple well-defined nodules in the both lobes of the lungs. The tumor cells in the lung were well differentiated, and the pattern of tumor was similar to the original tumor. IHC identified it originated from smooth muscle cells, consistent with the diagnosis of PBML. Positive staining of estrogen and progestogen receptors was detected in both the leiomyoma and the metastasizing lesions. During two years of observation, pulmonary function parameters were within normal limits and there was no evidence of tumor recurrence.

We report a rare, simultaneous occurrence of benign metastasizing leiomyoma in the lung and retroperitoneum in a 49-year-old woman who had previously undergone myomectomy at 35 years of age and hysterectomy at 45 years of age for multiple recurrences of histologically benign uterine leiomyomas. At 49 years of age, computed tomography-guided biopsy indicated benign metastasizing leiomyomas in the lung. In addition, a retroperitoneal leiomyoma was found that was resected along with both the ovaries via laparotomy. No sign or symptom of recurrence was observed 5 years later. The coexistence of benign metastasizing leiomyoma in the lung and retroperitoneum following surgery for conventional leiomyomas has rarely been reported. Further, the nature and etiology of benign metastasizing leiomyoma are still not well understood. This case is therefore worth reporting, and exploring its etiology is important., (© 2014 The Authors. Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology.)

The authors report a rare case of leiomyomatosis of the lung diagnosed in a 43-year-old woman, with uterine intravenous leiomyomatosis. Benign metastasizing leiomyoma (BML) is an extremely rare lesion characterized by usually multiple, benign-appearing smooth muscle tumors of the lung in females with coexisting uterine leiomyoma. On the basis of their histological and immunohistological features, a unified histogenetic view of leiomyomas with vascular invasion (LWVI) and BML of the uterus is proposed. LWVI and BML may be the same pathological entity and microscopic vascular invasion may represent the metastatic mechanism of BML. LWVI seems to be the precursor of BML.

Benign metastasizing leiomyoma (BML) is a rare condition in middle-aged women with a history of uterine leiomyomata. It is characterized by the proliferation of, usually multiple, smooth muscle nodules. Approximately 100 cases have been reported in the literature, and the lungs were the most common site of metastases. We report a case of 52-year-old obese woman (BMI 31), hospital worker, smoker, admitted to the hospital with exertional dyspnoea, night sweats, loss of weight, and productive cough. Hysterectomy for a uterine leiomyoma was performed 9 years earlier. In addition, a history of two episodes of superficial vein thrombosis 3 and 2 years before admission was noted. Chest X-ray and subsequently CT chest examinations revealed multiple, non-calcified nodules within the middle and lower parts of both lungs. Specimens obtained by transbronchial biopsy (TBLB) and from open lung biopsy displayed benign muscle cell proliferation compatible with BML. The levels of sex hormones were characteristic for the menopause; therefore, observation was advised. Additionally, Streptococcus pneumoniae was cultured from bronchial washing, and bronchitis was diagnosed. Antibiotics, bronchodilators, and mucolytics were administered, and dyspnoea and cough with expectoration were diminished. Two years later pulmonary lesions have been stable; however, she has put on weight. Subsequently the patient has developed deep vein thrombosis with pulmonary embolism. Anticoagulant treatment was introduced, with some improvement.