Metastasizing Uterine Leiomyoma

Takemura, Genzou; Takatsu, Yukiko; Kaitani, Kazuaki; Ono, Masamichi; Ando, Fumihiko; Tanada, Shuji; Niwa, Hideo; Tankawa, Hidemitsu; Fujiwara, T.; Yamabe, Hirohiko

doi:10.1016/s0344-0338(96)80116-6

Cited by 51 publications

(6 citation statements)

References 9 publications

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“…One theory suggests that BMLs are primary pulmonary lesions that represent multifocal leiomyomatosis [14] while others contend that they represent metastases. Some have suggested hematogenous extension following uterine surgical procedures [16], but presence of BML in the absence of such invasive procedures, including in our case, makes this a less likely etiology. The possibility of BML representing a metastasis from a low grade uterine leiomyosarcoma has also been entertained, but the histology and the molecular studies in which distinct signatures are noted only for uterine leiomyomas and BMLs makes this unlikely.…”

Section: Discussionmentioning

confidence: 66%

Benign metastasizing leiomyoma presenting with miliary pattern and fatal outcome: Case report with molecular analysis & review of the literature

Ofori

Fernandes

Cummings

et al. 2019

Respiratory Medicine Case Reports

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Benign metastasizing leiomyoma (BML) is a rare benign smooth muscle neoplasm that originates in the uterus and metastasizes to distant sites—most commonly the lungs. BMLs are often found incidentally in patients with a history of uterine leiomyoma(s) and tend to be indolent. Occasionally they may be symptomatic and rarely follow an aggressive clinical course. We report an unusual case of BML presenting in a 46-year-old woman as a miliary nodular pattern bilaterally in the lungs and progressive respiratory failure. Her past medical history was significant for uterine “leiomyomas” of at least 9 years' duration. Post mortem histologic evaluation of the uterine and lung lesions revealed benign smooth muscle neoplasms (leiomyomas and BMLs, respectively), and molecular analyses demonstrated identical clonal MED12 mutation, though with greater mutant allelic frequency in the uterus. We document a rare aggressive clinical course in a patient with BML, which presented as a miliary radiologic pattern mimicking an infectious etiology or interstitial lung disease.

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Section: Discussionmentioning

confidence: 66%

Benign metastasizing leiomyoma presenting with miliary pattern and fatal outcome: Case report with molecular analysis & review of the literature

Ofori

Fernandes

Cummings

et al. 2019

Respiratory Medicine Case Reports

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“…In one patient, histologic diagnosis may have been associated with significant morbidity, given close proximity of lung nodules to the heart (Case 1). Another case demonstrates the reported close pathogenesis between IVL and BML, as the patient presented with an IVC/cardiac tumor consistent with intravenous leiomyomatosis as well as pulmonary lesions consistent with BML (5, 42, 43) (Case 4).…”

Section: Discussionmentioning

confidence: 88%

Novel hormone treatment of benign metastasizing leiomyoma: an analysis of five cases and literature review

Lewis

Chason

DeCherney

et al. 2013

Fertility and Sterility

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Objective To evaluate novel hormonal therapies in patients with unresectable Benign Metastasizing Leiomyoma (BML) disease. Design Case series. Setting National Institutes of Health (NIH). Patients 5 subjects with the diagnosis of BML based on imaging and/or histopathologic diagnosis. Interventions Four patients were treated with single or combination therapy of Leuprolide acetate and/or an aromatase inhibitor. One patient was treated with an antiprogestin (CDB-2914). Main Outcome Measures Response to therapy was measured by tumor burden on cross-sectional imaging employing RECIST (Response Evaluation Criteria in Solid Tumours) 1.1 guidelines. Results Four patients treated with single or combination therapy of Leuprolide acetate and/or an aromatase inhibitor demonstrated stable disease with reduction in tumor burden. The fifth patient treated with antiprogestin (CDB-2914) had degeneration of her tumor, progression of its size and an improvement in symptoms. Conclusions Hormonal treatment with GnRH agonism and/or aromatase inhibition may be a therapeutic option to reduce tumor burden in unresectable BML disease or for those patients who wish to avoid surgical intervention. RECIST 1.1 guidelines, while traditionally used to evaluate tumor response to cancer therapeutics, may be useful in evaluating BML tumor burden response to hormonal therapy.

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“…They also stated that the longest survival period was 36 years (one patient with extensive lung involvement). One of the important prognostic factors is thought to depend on the estrogen or progesterone status of the patient because BML is associated with hormonal receptors [39,40]. Horstmann et al [26] mentioned that the disease process is indolent in postmenopausal patients, while progressive respiratory compromise and even death occur in the premenopausal patients.…”

Section: Discussionmentioning

confidence: 99%

Benign metastasizing leiomyoma of the lung

Hwang

Lee

et al. 2013

World J Surg Onc

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Benign leiomyomas of the uterus are uncommonly found in association with benign smooth muscle tumors beyond the confines of the uterus. Benign metastasizing leiomyoma (BML) is a rare disease in which the lung is described to be the most afflicted extrauterine organ. We present a brief review of the literature, along with case reports for four patients who were followed up after resection of a pulmonary lesion or after pathological confirmation by biopsy. The clinical course of BML varies from chronic asymptomatic appearance to rapid progression, leading to respiratory failure and death. Our BML patients did not complain of pulmonary symptoms, such as cough, dyspnea, or chest tightness. Pathology revealed benign leiomyomas with no atypia and mitotic activity <5 per 10 high-power field. Immunohistochemical staining was positive for actin and desmin. A standard treatment for BML has not yet been established. Because of the hormone-sensitive characteristics of BML, treatments are based on hormonal manipulation along with either surgical or medical oophorectomy. Benign metastasizing leiomyoma can be observed in postmenopausal women. We observed four patients who did not receive adjuvant hormonal therapy because they were postmenopausal or perimenopausal. All patients are still healthy and show no evidence of recurrence or progression of the disease.

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Metastasizing Uterine Leiomyoma

Cited by 51 publications

References 9 publications

Benign metastasizing leiomyoma presenting with miliary pattern and fatal outcome: Case report with molecular analysis & review of the literature

Benign metastasizing leiomyoma presenting with miliary pattern and fatal outcome: Case report with molecular analysis & review of the literature

Novel hormone treatment of benign metastasizing leiomyoma: an analysis of five cases and literature review

Benign metastasizing leiomyoma of the lung

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