Benign Metastasizing Leiomyoma Research Articles

Benign metastasizing leiomyoma of the lung

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.

Sex-specific lung diseases: effect of oestrogen on cultured cells and in animal models.

Sex prevalence in lung disease suggests that sex-specific hormones may contribute to the pathogenesis and/or progression of at least some lung diseases, such as lung adenocarcinoma, lymphangioleiomyomatosis (LAM) and benign metastasising leiomyoma (BML). Oestrogen is an important hormone in normal lung development and in the pathogenesis of female predominant pulmonary diseases. In vivo and in vitro studies have facilitated our understanding of disease pathogenesis and discovery of potential therapeutic targets. Oestrogen promoted disease progression in cell and animal models of lung adenocarcinoma, LAM and BML. Specifically, oestrogen enhanced tumour growth and metastasis in animal models of these diseases. Furthermore, 17β-estradiol (E2), the most abundant form of oestrogen in humans, increased the size and proliferation of cultured cells of lung adenocarcinoma and LAM. Coupled with the known mechanisms of oestrogen metabolism and signalling, these model systems may provide insights into the diverse effects of oestrogen and other hormones on lung diseases. Anti-oestrogen treatments that target key events of oestrogen synthesis or signalling, such as aromatase activity, oestrogen receptors and signalling pathways, may offer additional opportunities for clinical trials.

Benign metastasizing leiomyoma

Benign metastasizing leiomyoma (BML) is a rare clinical condition predominantly occurring in reproductive women with a history of uterine leiomyoma resection or hysterectomy. Oftentimes, it is easy to be misdiagnosed as a malignant tumor when nodules are found in multiple tissues. In order to raise clinicians' awareness of BML, we present a short review of the literature in combination with an unusual case.

Benign metastasizing leiomyoma: ‘a sheep in wolf’s clothing’

Benign metastasizing leiomyoma (BML) is a rare entity, of which a small number of cases, about 75 to 120, have been described in the literature since the condition was first described in 1939. It usually affects women of reproductive age with a history of uterine leiomyomatosis who have undergone a hysterectomy. The lung is the most common site of involvement. The pathologic features of this entity are described as having originated from the metastasis of the histologically benign uterine leiomyoma and are an uncommon lesion characterized by the presence of multiple smooth-muscle nodules that are often located in the lung, lymph nodes, or abdomen. Although these lesions are slow growing and asymptomatic and are usually incidentally diagnosed, they may rarely cause debilitating symptoms. Optimal treatment is controversial, but careful follow-up of these patients is recommended because the lesions show a lowgrade clinical malignant behavior although their appearance is benign. We describe here a rare case of pulmonary BML.

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

To evaluate novel hormonal therapies in patients with unresectable benign metastasizing leiomyoma (BML) disease. Case series. National Institutes of Health (NIH). Five subjects with the diagnosis of BML based on imaging and/or histopathologic diagnosis. 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). Response to therapy was measured by tumor burden on cross-sectional imaging employing RECIST (Response Evaluation Criteria in Solid Tumors) 1.1 guidelines. 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. Hormone treatment with GnRH agonist 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 hormone therapy.

Benign Metastasizing Leiomyomas Involving Multiple Pelvic and Para-Aortic Lymph Nodes: A Case Report

Benign Metastasizing Leiomyomas Involving Multiple Pelvic and Para-Aortic Lymph Nodes: A Case Report A 45-year old lady presented with vague abdominal discomfort of 2 weeks duration. She gave a past history of hysterectomy for fibroid. Per vaginal examination detected a large mass in right pelvis. A radiological examination confirmed the mass. Multiple enlarged internal nodes were also detected radiologically. A guided biopsy was done which on histopathology showed bland spindle cells. The patient was taken up for surgery and resection of the masses were done. Bilateral ovaries along with fallopian tubes were also identified and resected. Histopathological examination showed the resected masses to be lymph nodes with benign spindle cell neoplasm. The spindle cells were positive for SMA, PR and were negative for HMB 45, Ckit and DOG 1. MIB-1 proliferation index was low. Based on histopathological and immunohistochemical findings and previous clinical history a diagnosis of benign metastasizing leiomyoma (BML) was made. BML is a relatively rare disease. There are few reports about lymph node metastasis in BML. However, multiple pelvic and para-aortic lymph node metastases such as in our case are quite rare and hence we are reporting this case.

CT, MRI, and &amp;lt;sup&amp;gt;18&amp;lt;/sup&amp;gt;F-FDG PET-CT Findings of Pulmonary Benign Metastasizing Leiomyoma: A Case Report

Here we report imaging studies of a patient with pulmonary benign metastasizing leiomyoma (BML). A 44-year-old woman who underwent a hysterectomy for uterine cellular leiomyoma presented with abnormal shadows on a chest X-ray. Chest computed tomography (CT) revealed multiple well-defined nodules in both lungs. Chest magnetic resonance imaging (MRI) indicated these nodules as T1-low/T2-high intensity lesions. Contrast-enhanced MRI indicated these nodules as well-enhanced lesions, while 18F-fluorodeoxyglucose positron emission tomography-CT revealed no abnormal accumulation in these nodules. Bilateral lung wedge resections were performed for the largest 2 lesions to confirm the diagnosis, and both nodules were histologically diagnosed as BML.

Benign Metastasizing Leiomyoma: A Very Rare Case Report

ABSTRACT We report a case of benign metastasizing leiomyoma (BML) recurring after 4 years of total abdominal hysterectomy with bilateral salpingo-oophorectomy for fibroid uterus. Probably in this case, recurrence was due to extragonadal source of estrogen and progesterone. Therefore, patient was put on antiestrogen and antiprogesterone therapy, after hysterectomy to prevent further recurrence. Through this case, importance of surgery combined with antiestrogen and antiprogesterone therapy is emphasized. How to cite this article Agarwal A, Bhattacharya AB, Dwivedi AD, Kumar A. Benign Metastasizing Leiomyoma: A Very Rare Case Report. J South Asian Feder Obst Gynae 2013;5(1): 35-36.

Epidural intracranial metastasis from benign leiomyoma: A case report with literature review

Uterine leiomyomas are benign tumors of smooth muscle oriin believed to result from a clonal proliferation of uterine smooth uscle tissue [1]. They are the most common gynecologic tumors iagnosed in women of reproductive age, and the most common ndication for hysterectomy. Rarely, a benign uterine leiomyoma an be found in sites of the body other than the uterus. It is called benign metastasizing leiomyoma (BML). BML is a slow growing etastatic tumor, usually associated with a current or prior hisory of a uterine leiomyoma. The lung is the most common site of ML, but extrauterine locations such as the pelvic cavity, retroperioneal space, and even the lymphatic and vascular channels have lso been reported. BML of the brain is extremely rare and very few ases have been reported [2]. It has similar radiologic features as meningioma, and consequentially has often been misdiagnosed. varian leiomyomas are rare as well, and constitute less than 1% f all benign ovarian tumors. The treatment of BML is surgical esection of the mass to exclude malignancy, and the outcome is enerally good. We present a case of BML of the brain and the ovary

Benign Pulmonary Metastasizing Leiomyoma Uteri. Case Report and Review of Literature

Benign metastasizing leiomyoma (BML) is a rare condition in middle-aged women with a history of uterine leiomyomata. It is characterized by the proliferation of, usually multiple, smooth muscle nodules. Approximately 100 cases have been reported in the literature, and the lungs were the most common site of metastases. We report a case of 52-year-old obese woman (BMI 31), hospital worker, smoker, admitted to the hospital with exertional dyspnoea, night sweats, loss of weight, and productive cough. Hysterectomy for a uterine leiomyoma was performed 9 years earlier. In addition, a history of two episodes of superficial vein thrombosis 3 and 2 years before admission was noted. Chest X-ray and subsequently CT chest examinations revealed multiple, non-calcified nodules within the middle and lower parts of both lungs. Specimens obtained by transbronchial biopsy (TBLB) and from open lung biopsy displayed benign muscle cell proliferation compatible with BML. The levels of sex hormones were characteristic for the menopause; therefore, observation was advised. Additionally, Streptococcus pneumoniae was cultured from bronchial washing, and bronchitis was diagnosed. Antibiotics, bronchodilators, and mucolytics were administered, and dyspnoea and cough with expectoration were diminished. Two years later pulmonary lesions have been stable; however, she has put on weight. Subsequently the patient has developed deep vein thrombosis with pulmonary embolism. Anticoagulant treatment was introduced, with some improvement.

Benign Metastasizing Leiomyoma: A Rare Cause of Multiple Pulmonary Nodules

Benign metastasizing leiomyoma (BML) is a rare cause of pulmonary nodules that occurs when uterine leiomyomas metastasize to the lung. The management of these lesions varies from resection and hysterectomy to nonsurgical treatments such as hormonal therapy. We report a case of a 45-year-old woman with multiple nodules of the right lung identified during preoperative imaging before her hysterectomy for uterine fibroids.

Multiple, Small Bilateral Pulmonary Nodules on Thoracic CT in a Patient With Chronic Cough

Patients with multiple, bilateral small pulmonary nodules are commonly encountered in practice. Typically such nodules result from metastatic disease or infections, however, rare disorders also present in this manner. Benign metastasizing leiomyoma (BML) is a rare entity, typically affecting middle-aged women with a history of uterine fibroids. BML often metastasizes to lungs, appearing as multiple small nodules. We report a woman with BML presenting with an unremitting, chronic cough and small pulmonary nodules on thoracic computed tomography, and review the literature surrounding this rare entity.

FDG-PET/CT finding of benign metastasizing leiomyoma of the lung

We report a case of multiple benign metastasizing leiomyoma (BML) lung nodules showing faint or non-avid uptake of F-18 fluorodeoxyglucose (FDG) (respective 1-hour early and 2-hour delayed maximum standardized uptake values; 1.3 or less and 1.2 or less) in a 50-year-old woman with a history of hysterectomy for uterine leiomyoma at the age of 38 years. When multiple lung nodules show faint or non-avid FDG uptake in a patient with a history of hysterectomy for uterine leiomyoma, BML should be included in the differential diagnosis.

Benign Metastasizing Leiomyoma

Pulmonary metastases from benign-appearing smooth muscle tumors of the uterus are rare, and are termed benign metastasizing leiomyoma (BML). Affected patients usually present with single or multiple lung nodules and are usually women who have undergone hysterectomy. Only a few cases of BML with lung cysts have been reported, with 2 patients presenting with spontaneous pneumothoraces. We report a case of BML in a 29-year-old woman with an abnormal preoperative chest radiograph who several years after hysterectomy developed spontaneous bilateral pneumothoraces.

Pneumonic benign metastasizing leiomyoma: a case report and clinical treatment

Benign metastasizing leiomyoma (BML) is a rare entity usually occuring in females with history of uterus leiomyoma. It appears as a histopathologic benign smooth muscle tumors, distant from the uterus. It is hormone dependent and progresses slowly. Operation is the main clinical treatment. The article was reported one case who was a thirty-eight years old female. She was found to have bilateral lung nodules after hysterectomy of uterine leiomyoma seven years ago. An open lung biopsy revealed that the nodules consist of proliferating smooth muscle cells with a histopathologic feature which was similar to her uterus leiomyoma. Positive staining of estrogen and progesterone receptors were detected, which proved the tumer originate from uterine. The patient did not accept operation, chemotherapy, radiotherapy or other treatment. We still follow up the development of the case.

Uterine sarcomas: a multidisciplinary challenge

With an incidence lower than 1/100,000/year, uterine sarcomas are a rare subgroup within the rare family of sarcomas, giving rise to 1% of female malignancies. However, they are a variegated set of malignancies, encompassing leiomyosarcomas, endometrial stromal sarcomas, undifferentiated endometrial sarcomas and adenosarcomas [1]. Another group, malignant mixed mesodermal tumours, are currently regarded as metaplastic (aggressive) endometrial carcinomas, i.e. epithelial tumours in nature, not sarcomas [2]. Both the low incidence and the heterogeneity of uterine sarcomas pose their first multidisciplinary challenge, inasmuch as a correct pathological diagnosis is crucial to proper management. Even the differential diagnosis between benign and malignant tumours can be problematic. One may recall the existence of a distinct entity among smooth muscle tumours, i.e. smooth muscle tumours of uncertain malignant potential (STUMP), whose behaviour is by definition unpredictable pathologically, and of benign metastasising leiomyoma and disseminated peritoneal leiomyomatosis, i.e. benign smooth muscle tumours presenting with metastatic dissemination [3]. Improvements in the pathological classification, and grading systems, for all these tumours are highly necessary. Endometrial stromal sarcomas are low-grade malignancies, although the relapse rate after surgery of localised disease is relatively high, possibly amounting to one third of cases, although over a long period. Pathologically, these tumours are marked by their positivity for oestrogen and progesterone receptors. This underlies the efficacy in the advanced disease setting of progestins, aromatase inhibitors and gondadotropin-releasing hormone analogues [4]. Responses are frequent and long-lasting, also owing to the natural history of these tumours, even when metastatic. For this reason, surgery of lung metastases is an option to consider within a multidisciplinary approach. The usefulness of adjuvant hormonal therapies is still an open issue, given the relapse rate and the hormonal sensitivity of these tumours [5]. Bilateral oophorectomy, in addition to total abdominal hysterectomy for local treatment, also has an adjuvant intent. Currently, endometrial stromal sarcomas are conceptually separated from undifferentiated endometrial sarcomas, although a de-differentiation process of some endometrial stromal sarcomas into high-grade sarcomas may occasionally take place [6]. Undifferentiated endometrial sarcomas are inherently high-grade malignancies (pathologically marked by a high mitotic rate, cellular pleomorphism and necrosis), which are insensitive to hormonal therapy and clinically aggressive. This considered, an aggressive, multidisciplinary approach to this rare entity is logical. Medical therapy must resort to standard agents for high-grade softtissue sarcomas. On the contrary, adenosarcomas are low-grade malignancies encompassing a benign epithelial component and a low-malignant mesenchymal one. Their good prognosis is unfavourably affected when a truly sarcomatous overgrowth or myometrial invasion is present [7]. Uterine leiomyosarcomas are cured in less than one half of cases by total abdominal hysterectomy, with size and pelvic extent as the main prognostic factors. Improvements in grading systems would be required, in order to refine the prognostic value of pathological assessment. Radiation therapy did not add to surgery alone in a large randomised trial [8], in spite of positive evidence provided by uncontrolled and retrospective studies as far as the local regional relapse rate is concerned [9]. This may leave room for individualised decision-making on radiation therapy in some cases, depending on the surgical outcome and anatomical considerations. Of course, radiation therapy can be useful in local regional relapses. The efficacy of adjuvant chemotherapy is an open issue in soft-tissue sarcomas, and therefore in uterine leiomyosarcomas as well. These are essentially insensitive to hormonal therapies, although hormonal receptors are found in the tumour tissue in a proportion of cases and there is anecdotal evidence of hormonal sensitivity in occasional patients. Medical therapy in the advanced disease resorts to agents active in

Benign metastasizing leiomyoma of the lung with malignant transformation mimicking mediastinal tumor

We report a rare case of benign metastasizing leiomyomas (BMLs) showing malignant transformation in one of them. A 65-year-old woman had a fever. Computed tomography (CT) revealed a mass on the left side of the anterior mediastinum and multiple nodules in bilateral lungs. The masses proved to be leiomyomas and a leiomyosarcoma. The latter showed heterogeneous contrast enhancement on CT and high heterogeneous uptake of 18F-fluorodeoxyglucose (FDG) on positron emission tomography (PET)-CT. This case suggested the potential usefulness of PET-CT for differentiating these entities.

Serial spread of benign metastasizing leiomyoma to the thoracic spine

Benign metastasizing leiomyoma (BML) is a rare condition due to a histologically benign smooth muscle tumour originating from a uterine leiomyoma. It rarely affects the spine to cause spinal cord compression. Here we report a patient with serial metastases to the thoracic spine causing neurological compromise. The rapid spread of BML in this patient illustrates an aggressive manifestation of a usually benign indolent tumour.

Pulmonary and retroperitoneal benign metastasizing leiomyoma

Benign metastasizing leiomyoma (BML) is a rare disease, which usually occurs in women with a history of a prior hysterectomy or myomectomy for benign uterine leiomyoma, and has the potential to metastasize to distant sites, such as the lung, lymph nodes, muscular tissue, heart, or retroperitoneum. These lesions are slow-growing, asymptomatic, and usually found incidentally. The prognosis of BML is also excellent. However, there has been debate on the origin and the correct classification of BML, and there are no guidelines for the treatment of BML. We report here on a rare case of BML in both the retroperitoneal cavity and lung in a 48-year-old woman with a history of hysterectomy due to histologically benign uterine leiomyoma. The patient underwent retroperitoneal mass excision and bilateral salpingo-oophorectomy, and then wedge biopsy of two pulmonary nodules was performed additionally 9 days later. Until now, there has been no sign of recurrence and the patient remains asymptomatic. To our knowledge, pulmonary BML is rare and the co-existence of the retroperitoneal metastases after previous hysterectomy is even rarer.

Clinical benefit of endocrine therapy for benign metastasizing leiomyoma

Benign metastasizing leiomyoma (BML) is a rare disease that is caused by metastasis of a histologically benign leiomyoma that spreads to distant organs. Metastasis commonly follows surgical treatment of myomectomy or hysterectomy for benign uterine leiomyoma. Since the work of Steiner in 1939, several other studies have been published, but a unified view has not emerged regarding the biological nature, pathogenesis, and prognosis of this disease. In particular, a standard treatment has not been established. Here we report a case of BML of the lung and peritoneum in a premenopausal 45-year-old woman who underwent hysterectomy for uterine leiomyoma 10years earlier. A computed tomography (CT) scan showed multiple pulmonary nodules (maximum size 48mm) and a solitary intra-peritoneal nodule. The lung nodules were diagnosed as BML following a CT-guided needle biopsy. Histologically, the biopsied tumor was composed of spindle cells without significant atypia, and mitosis was not observed. Immunohistochemically, the tumor cells were strongly positive for smooth muscle actin, desmin, and estrogen receptor, suggesting a uterine leiomyomatous lineage. The patient was treated with a gonadotropin-releasing hormone agonist for 36months, during which time the nodules reduced in size slightly. No new lesions appeared, and the tumors were stable at 15months after the cessation of treatment. In conclusion, endocrine therapy can serve as one of the conservative treatments for premenopausal women with BML, rather than surgery.