Benign metastasizing leiomyoma in triple location: lungs, parametria and appendix

Abstract

Benign metastasizing leiomyoma (BML) usually are situated in one organ, most often in lungs. BML patients typically have a history of uterine leiomyoma treated with hysterectomy, myomectomy or subtotal hysterectomy. The aim of the study was to present the case of a 53-year-old woman with triple location in the lungs, parametria and appendix. She had undergone a myomectomy 26 years earlier. In 2015, she was admitted to the surgical department because of abdominal pain, whereupon a cholecystectomy was performed. CT scans showed pelvic mass with pulmonary metastasis. Upon discharge the patient was referred to the Gynecology Clinic, where a laparotomy was performed. The intraoperative findings were: 1) uterus with multiple leiomyomas, 2) four tumors in the parametria, 3) tumor connected to the appendix. A subtotal hysterectomy, with a bilateral salpingo-oophorectomy, removal of the tumors from the parametria and appendectomy was performed. Pathology confirmed the diagnosis based on morphology and immunohistochemical staining (strongly positive for estrogen receptors and SMA, while Ki67 was very low, below 1%). Upon postoperative recovery, the patient was referred to the Thoracic Surgery Department. During the thoracotomy, multiple nodes, surrounded by lung parenchyma, were revealed. Wedge resection was performed, for localized pulmonary lesions, and sent for pathological examination. The final pathological diagnosis was benign metastasizing leiomyomatosis.In conclusion, the triple location of BML could possibly be a result of a parallel different metastasizing mechanism, although it is impossible to exclude one mechanism, which may be the cause of the metastases in three locations.

Benign metastasizing leiomyoma: A review of current literature in respect to the time and type of previous gynecological surgery

“…In review study, microscopic examination of haematoxylin and eosin slides has demonstrated the characteristic features of smooth muscle cell differentiation, which was also confirmed by immunohistochemistry smooth muscle actin positivity.Additional immunohistochemistry Ki67 showed a low tumour cell proliferation index, which favors a benign behavior [ 3 , 4 , 7 , 8 , 11 – 14 , 17 , 18 , 20 – 22 , 27 , 31 – 37 , 41 – 55 ].…”

Natural history of leiomyomas beyond the uterus

“…Our study did not provide a clear answer why some uterine leiomyomas cause metastases described in some articles as benign. [1–4]…”

“…Some reports describe only synchronous occurrence of different types of LBU. [3,4] The case reports describe different types of LBU as an individual event in the history of the disease. [5–8]…”

The Heart as a Site of Metastasis of Benign Metastasizing Leiomyoma: Case Report and Review of the Literature

“…Despite being histologically benign, leiomyoma has clinically malignant potential. Metastases have been reported to a number of different sites, including the spine [ 1 – 4 ], skull [ 1 ], rib and vertebra [ 5 ], retroperitoneum [ 6 ], parametria [ 7 ], appendix [ 7 ], lymph nodes [ 8 , 9 ], and most commonly the lung [ 10 ].…”