Benign Pulmonary Metastasizing Leiomyomatosis: Case Report and a Review of the Literature

Abstract

The authors report here on a case of a nearly asymptomatic 51-year-old Korean woman who was found to have diffuse, multiple nodules of the lungs on a routine chest radiograph. She had undergone hysterectomy 16 years previously for uterine myoma. An open lung biopsy revealed tumor that was composed of interlacing bundles of spindle cells with cigar shaped nucleus and eosinophilic myofibrils in the cytoplasm; consistent with multiple leiomyomas. The stains for SMA, desmin, MSA and Ki-67 were positive and the stain for c-kit was negative. The other stains for estrogen and progesterone receptor were positive. During the open lung biopsy procedure, all the nodules were excised.We report here on an interesting case of benign metastasizing leiomyoma (BML) in 51-year-old patient. To the best of our knowledge, this case showed the longest period of clinical progression in Korea. This is also one of a few cases in which curative excision was successfully performed.

A Rare Cause of Multiple Pulmonary Nodules: CT Features of Benign Metastasizing Leiomyoma

“…[6] Another point of view in this regard is the surgical removal of metastases where possible. [1,3] We found multiple lung metastases in our case, despite hysterectomy. We started aromatase treatment primarily because of multiple metastases, an asymptomatic case, and positive estrogen and progesterone receptors found after the biopsy.…”

“…BML is most commonly seen in the lung, but it may occur in many different locations, including the paraaortic lymph nodes, abdominal lymph nodes, heart, breast, liver, and esophagus. [3] The PBML pathogenesis has not yet been fully explained. However, various pathogenetic mechanisms such as the in situ proliferation of hormone-sensitive smooth muscle bun-dles, benign smooth muscle cells that are hematogenously transported from the uterine leiomyoma and colonized in the lung, and low-grade uterine leiomyosarcoma that metastasized to the lung have been suggested.…”

Leiomyomas beyond the uterus; benign metastasizing leiomyomatosis with paraaortic metastasizing endometriosis and intravenous leiomyomatosis: a case series and review of the literature

“…Metastatic foci of leiomyoma have been discovered up to 24 years after hysterectomy for benign leiomyoma of the uterus and the average age of presentation is 48 years [7,8]. Although they are most commonly found in the lungs [9,10] they have also been detected in lymph nodes, skin, bones, the retroperitoneum [11], heart [12], brain [13], and the major vessels [14] and even bones [15]. The pulmonary lesions tend to be bilateral, multiple, nodular, and well-circumscribed and do not display contrast enhancement on CT.…”

Obstructive Sleep Apnea and Hypertension

“…9 This spontaneous regression phenomenon is thought to be because of the hormonal responsiveness of the tumors themselves. 11 Our patient's history of uterine fibroids was discovered only after her current diagnosis was revealed; this history was not disclosed during her initial thoracic surgical evaluation. 9,10 In addition, rare patients with complete surgical resection of limited disease have been described.…”