Benign metastasizing leiomyoma from uterine myoma; report of a case

A case, 53-year-old female, of benign metastasizing leiomyoma was reported. The patient had undergone a hysterectomy for uterine myoma at the age of 40. The computed tomography (CT) scan film on health screening revealed 4 nodular lesions on right lung field and 2 nodular lesions on left lung field. Two tumors of the left side were resected under thoracoscopic surgery. Pathologically 2 lesions were consisted of benign spindle cells and diagnosed as benign metastasizing leiomyoma. Postoperative course was uneventful. Right lung nodules have been meticulously followed up.     

Pulmonary benign metastasizing leiomyoma

Pulmonary benign metastasizing leiomyoma (BML) is a rare disorder affecting women with a history of uterine leiomyoma metastasizing to lung. These lesions present either as a multiple benign synchronous or metachronous nodules. We report a case of 53-year-old postmenopausal women who presented with multiple bilateral pulmonary nodules on a chest X-ray during an evaluation for dyspnea, 11 years after a hysterectomy due to uterine fibroids and excision of abdominal wall tumor. Chest computed tomography (CT) revealed multiple well-defined soft tissue enhancing mass lesions in both lungs, with the largest one in the right lower lobe. She underwent right side bilobectomy by thoracotomy. Pathologically, the resected lesion consisted of benign spindle cells and was diagnosed as smooth muscle tumor of unknown potential which includes BML [1]. Other lung nodules have been meticulously monitored at follow-up, and repeat X-ray after 1 year later showed that these nodules had not increased at all in size and that no new lobe nodules had appeared. She did not receive any adjuvant hormonal therapy because of her postmenopausal status.     

Benign metastasizing leiomyoma of the lung: report of a case

A 35-year-old female presented with multiple bilateral pulmonary nodules on a chest X-ray during a regular health checkup. Chest computed tomography revealed multiple well-defined nodular shadows in the lung. She had undergone a myomectomy 7 years previously for leiomyoma of the uterus. Thoracoscopic resection of one of the nodules was performed to establish a pathological diagnosis. Pathological findings were consistent with benign metastasizing leiomyoma. Estrogen and progesterone receptors showed strong positives by immunohistostaining of the tumor. The patient is currently receiving outpatient treatment for a benign metastasizing leiomyoma by administration of a GnRH analog drug.     

Multiple lung tumors found 17 years after hysterectomy —A case of benign metastasizing leiomyoma—

The patient is a 50-year-old female who presented with cough. A chest X-ray showed bilateral multiple nodular shadows. Even though a diagnosis of metastatic lung tumors was strongly suspected, no primary lesion could be detected on close examination. Therefore, an exploratory thoracotomy, and histopathological examination of a specimen obtained from a resected tumor were carried out. The resected tumor revealed pathological proliferation of spindle cells without high cellularity or mitosis with nuclear atypism. The patient had a history of surgery for uterine leiomyoma at the age of 33. The histopathological characteristics of the resected lung tumors were remarkably similar to those of the previously resected uterine leiomyoma. In addition, the serum levels of sex hormones, including LH, measured after lung surgery, were within normal range suggesting the possibility of the lung tumors being metastatic. The residual lung tumors did not show any increase in size 26 months after surgery. Therefore, these lung tumors could be considered as lung metastases from a low grade malignant uterine leiomyoma (benign metastasizing leiomyoma). (benign metastasizing leiomyoma).     

Benign metastasizing leiomyoma of the lung

A 44-year-old woman was found to have an abnormal shadow on a chest X-ray during a regular health checkup, and visited our department. Chest computed tomography showed multiple nodular shadows in both lungs. The patient had no history of neoplasm except for myomectomy for uterine leiomyoma 6 years previously. Eighteen months later, the nodules showed a gradual increase in size, and video-assisted thoracoscopic biopsy of a nodule was performed. Histopathologically, the pulmonary nodule was composed of benign smooth muscle cells proliferating in fascicles, consistent with the diagnosis of benign metastasizing leiomyoma. Benign metastasizing leiomyoma is defined as a histologically benign uterine smooth muscle tumor that acts in a somewhat malignant fashion and produces benign metastases. Although it is a rare condition, it should be considered in asymptomatic women of reproductive age with a history of uterine leiomyoma, who present with solitary or multiple pulmonary nodules. Herein, we report a case of pulmonary benign metastasizing leiomyoma.     

Benign metastasizing leiomyoma: metastasis to rib and vertebra

Benign metastasizing leiomyoma is very rare and characterized by the presence of pelvic, peritoneal, nodal, or pulmonary nodules in women with a history of uterine leiomyomas. We report a case of benign metastasizing leiomyoma in a 30-year-old woman who had undergone a prior myomectomy due to uterine cellular leiomyoma 3 years earlier. The patient had a mass on the right sixth rib and 2 masses in the sixth thoracic vertebra. Pathologically, these masses were diagnosed as cellular leiomyomas. Estrogen and progesterone receptors were both positive in the metastatic tumors as well as in the uterine leiomyomas. The diagnosis of benign metastasizing leiomyoma can only be made after careful examination of the primary tumor to exclude small foci of malignant change.     

Pulmonary Benign Metastasizing Leiomyoma from the Uterus in a Postmenopausal Woman: Report of a Case

We report a case of pulmonary benign metastasizing leiomyoma (BML) from the uterus in a 77-year-old woman. The patient presented for investigation of multiple pulmonary nodules on a routine chest roentgenogram. Because a preoperative diagnosis could not be made, the largest tumor, 3.5cm in diameter, was resected from the right lower lobe, and histological examination confirmed BML. She had undergone hysterectomy with oophorectomy for uterine leiomyomas 12 years earlier, at the age of 65. The microscopic findings of the lung tumor were similar to those of the uterine leiomyoma, and both lesions were histologically benign. Although this disease is considered to be hormone-dependent, metastasis was found in this elderly postmenopausal woman whose tumor was negative for estrogen receptor.     

Multiple benign metastasizing leiomyomas in the pelvic lymph nodes and biceps muscle: Report of a case

A 47-year-old woman with an earlier history of uterine leiomyoma suffered from multiple recurrent tumors in the retroperitoneal lymph nodes and biceps muscle of the right upper arm. The woman with a right lower abdominal tumor was referred to our hospital. An abdominal computed tomography scan revealed two round nodules with well-defined margins in the retroperitoneum in the pelvis, and echography revealed a similar nodule in the biceps of the right upper arm. A biopsy of the abdominal retroperitoneal tumor demonstrated benign metastasizing leiomyoma (BML). An extirpation of the abdominal tumors was therefore performed. After the operation, false climacteric medical treatment was performed for 3 years and no recurrence has since been observed. This is the first reported case of multiple BML in the lymph nodes and muscle occurring simultaneously.     

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 benign lung tumors

Multiple benign pulmonary nodules are rare and are from a variety of etiologies. Infectious causes, such as histoplasmosis, tuberculosis, or parasitic infections, usually require biopsy for confirmation. An interesting entity, benign metastasizing leiomyoma, is rare but occurs from a low-grade leiomyoma that most commonly spreads from the uterus.     

The effectiveness of medroxyprogesterone in the treatment of multiple metastasizing leiomyosarcomas: Report of a case

A 51-year-old woman was admitted to our hospital for further investigation of chest X-ray films which showed multiple shadows that had been growing slowly over 2 years. Her only symptom was hemosputa. The lesions were suspected of being metastasizing leiomyoma due to her past history of uterine leiomyoma. Just 1 week before undergoing scheduled open lung biopsy, the lung lesions increased remarkably in size and number. A thoracotomy was performed and six of the numerous nodules were removed. The resected specimens were pathologically diagnosed as metastasizing leimyosarcoma which was positive for the progesterone and estrogen receptors. Thus, 1 month postoperatively, a course of medroxyprogesterone (MPA), 600 mg daily, was commenced. The residual lesions in her chest started to diminish, shortly afterward. She has remained well on this MPA regimen for 45 months. The prognosis of patients with metastasizing leiomyosarcoma is poor because of its low sensitivity to chemotherapy; however, some types of leiomyosarcoma are hormone-sensitive. It is therefore important to examine the hormone receptors of excised tumors from patients suspected of having metastasizing leiomyoma or leimyosarcoma.     

Distinctive cytogenetic profile in benign metastasizing leiomyoma: pathogenetic implications

"Benign metastasizing leiomyoma" is the terminology used to describe a controversial entity characterized by a proliferation of bland-appearing smooth muscle in lung or abdominopelvic lymph nodes. In this report, we describe 5 cases of pulmonary-based smooth muscle tumors that are clinically and histologically consistent with this entity, and in which we identified consistent chromosomal aberrations (19q and 22q terminal deletion in all cases). This cytogenetic profile is found in approximately 3% of uterine leiomyoma, but has not been described in other types of benign or malignant neoplasia. These findings suggest that the nodular pulmonary smooth muscle proliferations termed "benign metastasizing leiomyoma," are a genetically distinct entity, which likely originate from a biologically distinctive subset of uterine leiomyoma.     

Benign metastasizing leiomyoma

The cases of 2 patients with benign metastasizing leiomyofibroma are reviewed. Although extremely rare both in our experience and in the literature, benign metastasizing leiomyofibroma is an important differential diagnosis in young women seen with multiple lung nodules. Our patients underwent staged thoracotomies for diagnosis and treatment, and have experienced no evidence of recurrence over a six-year period.     

A case of multiple leiomyomatous lesions of the lung: An analysis of flowcytometry and hormone receptors

A 36 year old woman was admitted to our department because of a chest X-ray which showed multiple developing shadows. She underwent bilateral exploratory thoracotomies and a total 5 tumors were resected and pathologically diagnosed as benign metastasizing leiomyoma, the largest of which was positive for the progesterone receptor and negative for the estrogen receptor. A histogram of this tumor using a flow cytometer showed a diploid pattern and 4.6 percent of the S phase which was not more than that of a leiomyoma of the uterus from another patient. Two months later, she underwent a hysterectomy and bilateral salpingo-oophorectomy for treatment of the positive progesterone receptor in the pulmonary lesions. The resected uterine myoma and normal myometrium showed positive estrogen and progesterone receptors. For the subsequent 28 months she has been free of any further symptoms. Benign metastasizing leiomyoma of the uterus is a rare disease and very interesting because of its histological benignity and hormonal dependency. However, according to the literature, it is often confused in entity due to the fact that normal lung tissue also possesses hormone receptors. Considering our data on hormone receptors, it is rational to think that multiple leiomyomatous lesions in the lung should only be diagnosed as benign metastasizing leiomyomas when they possess positive estrogen and progesterone receptors.     

Benign metastasizing leiomyoma; report of a case

We report very rare case of benign mestasizing leiomyoma (BML). A 42-year-old female was referred to our hospital presenting with abnormal shadows revealed by a chest X-ray film. Chest computed tomography (CT) scan revealed multiple well-defined nodules in bilateral lung field, suggesting metastatic lung tumor. Further examination could not detect any suspicious primary lesion. Several tumor markers were all within normal limits. Surgical resection was performed to establish diagnosis and treatment. Pathological diagnosis was BML from the uterus myoma which had been resected 5 years before.     

Intravenous leiomyomatosis: Case series and review of the literature

IntroductionIntravenous leiomyomatosis (ILV) is a rare pathology, part of leiomyoma beyond the uterus (LBU), characterized by benign smooth muscle cell tumor outside of the uterus and mainly affecting premenopausal woman with a medical history of leiomyoma or gynecologic surgical treatment. The treatment depends on the localization of the tumor, age of the patient, initial size, symptoms and the suitability for surgery but should always aims in toto surgical resection.Case presentationRetrospective case series and review of literature.Clinical discussionSymptoms presented by the patient were aspecific and only localized in the pelvic area. All cases were fortuitous histopathological diagnosis. No relapse was. Two out of 5 patients have pulmonary nodules, only one was biopsied and diagnosed with PBML (pulmonary benign metastasizing leiomyoma).ConclusionIVL and BML are rare disease that can co-exist. Because of tumoral hormonal receptors, hormonotherapy could be an optional treatment but to date no clear efficacy is demonstrated. In case of high recurrence risk such as voluminous initial mass, impairment of broad ligament, failure of total surgical resection, adjuvant hormonotherapy could be useful. Recurrence rate is about 16.6-30% and can occur even dozen years later and even after radical surgery, justifying a regular follow up.     

Leiomyomatosis peritonealis disseminata: A case report and meticulous review of the literature

IntroductionLeiomyomatosis peritonealis disseminata (LPD) is a peculiar benign clinical disorder characterized by proliferation of peritoneal and subperitoneal nodules. LPD is a difficultly diagnosed benign disease that rarely degenerates into malignancy.Presentation of caseA 40-year-old Caucasian female with vaginal bleeding proceeded to our institution for elective excision of abdominal and pelvic masses which were firstly considered as leiomyosarcomas. The histologic diagnosis of the mass lesions revealed smooth muscle benign cells. This is the first case of LPD reported in Greece. A meticulous review of the literature was conducted as well.DiscussionThe differential diagnosis of LPD is difficult due to its clinical resemblance with peritoneal carcinomatosis or metastatic lesions and with benign metastasizing leiomyoma (BML) as well. Etiological factors, pathophysiology and clinical manifestations which lead to a safe diagnosis of LPD are adequately described.ConclusionSurgeons’ thorough knowledge concerning this rare clinical condition is fundamental and crucial in order to establish a correct diagnosis and assert the appropriate treatment and the minimization of the probability of malignant transformation of LPD.     

Endometrial stromal sarcomas with unusual histologic features: a report of 24 primary and metastatic tumors emphasizing fibroblastic and smooth muscle differentiation

We report the clinicopathologic features of 24 uterine primary and metastatic endometrial stromal sarcomas with fibromyxoid features (ESS-F) and smooth muscle differentiation (ESS-SM) (endometrial stromal sarcoma variants). Two groups of tumors were retrieved from the surgical pathology files at Memorial Sloan-Kettering Cancer Center: 1) gynecologic mesenchymal neoplasms with striking smooth muscle or fibroblastic differentiation that did not meet the clinical or histologic criteria for leiomyosarcoma or other established neoplasms containing smooth muscle; and 2) metastatic lesions showing ovoid and spindle cell morphology, involving lung, originally diagnosed as low-grade leiomyosarcoma, low-grade smooth muscle neoplasm, intravenous leiomyomatosis, fibrous hamartoma, and benign metastasizing leiomyoma. We identified 12 patients with 30 tumors; 24 were available for review. The mean age was 51 years (range 21-74 years). Follow-up >1 year was available for eight patients, with a mean time of 8.5 years. Each patient had a uterine primary and 10 experienced metastases. Mean time to recurrence was 6.8 years. Sites of metastasis included lung, retroperitoneum, right atrium/inferior vena cava, colon, and ovaries. No patient died of disease, but in many cases the follow-up period ended with the discovery of a metastasis. Four patients were originally diagnosed with endometrial stromal sarcoma, but other presenting diagnoses included benign metastasizing leiomyoma, fibroleiomyomatous tumor of lung, smooth muscle tumor of uncertain or low malignant potential, and intravascular leiomyomatosis. On review each patient had at least one tumor (primary and/or metastasis) that was determined to be an endometrial stromal sarcoma variant. Review diagnoses were as follows: endometrial stromal sarcoma (nonvariant), ESS-F, and ESS-SM. Eight of 10 primary tumors with available slides were endometrial stomal sarcoma variants (six ESS-F and two ESS-SM). When these variant features were present, they comprised between 50% and 100% of the neoplasm. The variant histology tumors exhibited prominent spiral arterioles, perivascular edema, and stromal cell condensation around blood vessels. All metastases but one were variant tumors; eight were ESS-F and five were ESS-SM. Four metastases did not resemble the uterine primary. Desmin marked smooth muscle mostly but not specifically. h-Caldesmon marked smooth muscle exclusively. Endometrial stromal cells as well as some fibroblasts and smooth muscle cells expressed CD10. We conclude that the presence of even focal endometrial stromal differentiation in an invasive uterine mesenchymal lesion with a predominant low-grade smooth muscle, fibroblastic, and/or myxoid phenotype should permit classification as low-grade sarcoma-they should be considered endometrial stromal sarcomas.     

Leiomyoma of the lung: a case report

An asymptomatic 43-year-old female was admitted to the Hospital because of an abnormal nodule in the left lung field on screening chest X-ray. Chest CT showed a tumor shadow mass (4 x 3 cm) in the left lower lobe. Bronchofiberscopy revealed an endobronchial polypoid mass obstructing the lower lobe bronchus at the orifice of left B10. Lt lower lobectomy was performed. Histopathological examination of the specimen showed the tumor (4.5 x 3.6 x 3.6 cm) developing not only within the tracheal lumen, but also in the lung tissue. Because immunohistochemical staining for desmin and alpha-smooth muscle actin was positive, we diagnosed leiomyoma. As MRI revealed multiple uterine masses after operation, we discussed the diagnostic problems in the relation to metastasizing leiomyoma of the uterus.     

Multiple pulmonary hamartomas; a case report and review of the literature.

Bilateral multiple hamartomas were found in a woman suspected of having metastatic malignancy of the lung. As extensive investigation for the primary tumour was unrevealing, a left exploratory thoracotomy and histological examination established the diagnosis. In view of the benign character of the tumours, local excision alone was performed. No surgical intervention was performed on the right side. Repeat chest films 12 months after surgery did not show the appearance of new lesions in the left lung or any increase in the size of the nodules in the right lung. This is the 12th case so far reported. The clinical characteristics and surgical management of these tumours are discussed.