Multisystemic Benign Metastasizing Leiomyoma: An Unusual Condition with an Atypical Clinical Presentation

Abstract

Benign metastasizing leiomyoma (BML) is a rarely found entity with few documented cases in the literature, usually occurring in women of reproductive age with a history of myomectomy or hysterectomy. The leiomyomas can metastasize to several organs, the lungs being the most commonly affected. We report a case of a 40-year-old female patient who presented at our institution with low back pain. She underwent a lumbar MR that revealed the presence of an expansive and compressive mass in the body of L4. This mass was biopsied, corresponding to a metastasizing leiomyoma with no malignant characteristics. Subsequently, a CT examination showed several soft tissue density round masses in both lungs, but the most striking feature was a 12 cm mass located in the left iliac crest. There was asymmetrical uterus enlargement, caused by the presence of several leiomyomas. Since the lesions were estrogen and progesterone positive, hormone suppression consisting of oophorectomy followed by anastrozole was the chosen treatment. No signs of progression were observed at the 6-month follow-up. This case is one of the very few that occurred in a woman with no previous uterine intervention, adding further evidence that surgery is not an essential condition for this entity to develop.

Spontaneous versus morcellator‐related benign metastasizing leiomyoma—A retrospective study

“…However, in the present study, in 12 of the 20 cases, BML had developed without any previous surgery. This was rarely previously reported 10,11 . One of the theories concerning the growth mechanism of spontaneous BML is that BML arises from vascular walls within the myometrium, possibly as a result of an unusually wide invasion of the myometrium itself by a leiomyoma, 13 and is spread by the lymphatic system and/or blood vessels to sites located remote from the uterus.…”

“…This was rarely previously reported. 10,11 One of the theories concerning the growth mechanism of spontaneous BML is that BML arises from vascular walls within the myometrium, possibly as a result of an unusually wide invasion of the myometrium itself by a leiomyoma, 13 and is spread by the lymphatic system and/or blood vessels to sites located remote from the uterus. In rare cases, myoma cells might infiltrate into the right heart chamber and the lungs, 13 a phenomenon termed intravascular leiomyomatosis and is considered to be a subtype of BML.…”

“…[5][6][7][8][9] Nevertheless, BML has also been found to occur among women without a previous leiomyoma cut-through, and even without uterine leiomyomas at all. 10,11 Importantly, a comparison between spontaneous and iatrogenic BML is underreported, possibly because of the rarity of this phenomenon.…”

Intravenous Leiomyomatosis: An Uncommon Cause of Pulmonary Embolism

“…Furthermore, IVL is known to cause benign metastases in the lungs, liver, skin, lymph nodes, esophagus, bladder, or even spine, with the lung being the most common metastatic location [ 12 , 13 ]. Although most cases of benign pulmonary metastases described in the literature presented as multiple pulmonary nodules, this etiology could not be excluded in our patient with a small 6-mm pulmonary nodule.…”

Benign metastasizing leiomyoma of the lung: diagnostic process and treatment based on three case reports and a review of the literature

“…In case of quali cation for lung metastasectomy VATS uniportal approach is recommended if possible. Similarly as with uterine leiomyoma, not only surgical treatment, but also conservative hormonal treatment could be an option for some cases of BML [38,39].…”