Metastasizing leiomyoma of the uterus and hormonal manipulations. Case report

Evans, Alun; Wiltshaw, E.; Kochanowski, S. J.; Macfarlane, Aidan; Sears, R. T.

doi:10.1111/j.1471-0528.1986.tb08043.x

Cited by 34 publications

(18 citation statements)

References 7 publications

(4 reference statements)

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“…The rationale for the use of hormonal therapy is based on the presence of estrogen and progesterone receptors in both primary tumors as well as in the metastatic tumors [26]. Despite the presence of positive estrogen and progesterone receptors on smooth muscle cells, there was no significant change in size of BML after 6-12 months treatment of tamoxifen, progesterone, and an aromatase inhibitor [27,28]. Tamoxifen has proven to be effective in vitro for decreasing cell numbers and for stopping cell proliferation [29], but its role remains uncertain in vivo [5,15].…”

Section: Discussionmentioning

confidence: 98%

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

Mahmoud

Desai

Nezhat

2014

Arch Gynecol Obstet

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Uterine leiomyomas affect 20-30 % of women 35 years and older. Extrauterine leiomyomas are rare and present a greater diagnostic challenge. Those unusual growth patterns occur more often in women of reproductive age with a history of hysterectomy or surgery for uterine leiomyomas. They have been reported in the literature in case reports and small case series and include benign metastasizing leiomyoma (BML), disseminated peritoneal leiomyomatosis, intravenous leiomyomatosis (IVL), parasitic leiomyomas, and retroperitoneal growth. In this case series we present a case of BML with a first report of concomitant endometriosis metastasis to paraaortic lymphnodes, and a case of IVL. The findings and surgical management of those cases, as well as a review of the literature pertinent to those entities, are also presented.

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Section: Discussionmentioning

confidence: 98%

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

Mahmoud

Desai

Nezhat

2014

Arch Gynecol Obstet

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“…The first attempts at medical treatment with hormonal agents utilized progesterone, which resulted in increased lesions as well as worsening symptoms (12,22). Since then, progesterone therapy has resulted in varied responses, some with regression of disease and symptomatic relief (4,23) (Table 1).…”

Section: Discussionmentioning

confidence: 99%

“…While traditionally surgical resection of metastatic lesions was the mainstay of treatment with variable long-term results, more recent management has revolved around surgical or medical castration (10,11,12). Clinical evidence of hormonal influence is suggested by reports of BML lesion regression during menopause (13) and following pregnancy (14,15).…”

Section: Introductionmentioning

confidence: 99%

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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“…8 Tietze et al reported a case of BML in which tumors from both the uterus and lung were characterize as being monoclonal smooth muscle proliferations with identical Xchromosome inactivation and a balanced karyotype, most consistent with a clonal metastasizing process. 7 Cases of BML usually present after a long follow-up period as multiple pulmonary nodules in sexually mature women with a history of uterine leiomyoma. Case reports of BML cover a wide range and various follow-up intervals both before and after diagnosis.…”

Section: Discussionmentioning

confidence: 99%

“…1 Some investigators have classified the tumor as a low grade leiomyosarcoma with malignant potential, others as an implantation and proliferation of benign smooth muscle tissue, with embolization by an intravenous leiomyomatosis or mechanical means, and still others as a result of a systemic leiomyomatosis with multifocal but independent smooth muscle profileration. [3][4][5][6][7] The chromosomal abnormalities associated with BML have been difficult to characterize. Approximately 25% of the uterine leiomyomas may have a balance translocation, the most common of which are t(12;14)(q13-15;q23-240,del(70q21.2q31.2), trisomy 12, and rearrangements of 6p.…”

Section: Discussionmentioning

confidence: 99%

Benign Metastasizing Leiomyomatosis

Goyle¹,

Moore²,

Garrett³

et al. 2003

American Journal of Clinical Oncology

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The authors report an interesting case of a minimally symptomatic 23-year-old African American woman who was found to have extensive diffuse reticulonodular opacities of the lungs on a routine chest radiograph. She had a hysterectomy 5 years previously for multiple leiomyomas of the uterus. She had no history of any prior exposure to dusts or toxins. Collagen vasculitides and bacterial, mycobacterial, and fungal infectious causes were excluded through standard testing, and a bronchoscopic lung biopsy was nonspecific. An open lung biopsy revealed multiple nodules of proliferating smooth muscle cells intermixed with irregular areas of epithelial-lined spaces. Histologically, the muscle cells appear benign with a very low mitotic rate, and the pathologic findings were consistent with benign metastasizing leiomyomatosis (BML). Staining for estrogen and progesterone receptors, actin, and c-kit were performed. This case and the review of the medical literature support the concept that BML originates from an antecedent leiomyoma of the uterus in virtually all cases with rare exceptions. It appears that tumor metastasizes to lungs or other extrauterine tissues via hematogenous spread. However, the origin of the tumor remains controversial. BML is a rare entity, with only a handful of reports in the medical literature. The authors report an interesting case of BML in a 23-year-old patient who, to their knowledge, is the youngest such patient described and who, at 13 years, has the longest period of clinical follow-up. In this article, the authors review the pathogenesis, cytogenetics, histologic markers, and management options of this rare entity.

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Metastasizing leiomyoma of the uterus and hormonal manipulations. Case report

Cited by 34 publications

References 7 publications

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

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

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

Benign Metastasizing Leiomyomatosis

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