Benign metastasizing leiomyoma in femur and thigh with a history of uterine leiomyoma: A case report and literature review

Abstract

Benign metastasizing leiomyoma (BML) is a rare disease that is characterized by well‐differentiated smooth muscle tumors occurring extrauterine site in women with a history of uterine leiomyoma. The lung is the most common metastatic site for BML. A 48‐year‐old woman, who had histories of laparoscopic myomectomy and transabdominal total hysterectomy, visited an orthopedics complaining of a mass in her left thigh and difficulty in walking. Magnetic resonance imaging (MRI) and fluorine 18 fluorodeoxyglucose positron emission tomography/computed tomography revealed multiple mass lesions in her both thighs and left femur as well as both lungs. She was referred to our hospital for further examination. We diagnosed her tumors as BML according to histopathological analysis of tumor specimen. The left thigh tumor was resected and the treatment with gonadotropin releasing hormone agonist regressed the size of the residual tumors by approximately 30%. BML should be considered when multiple soft tissue tumors are found in women with a history of leiomyomas.

Leiomioma metastatizante benigno: reporte de caso

“…El pulmón es el sitio mas frecuente de aparición en donde se manifiestan como múltiples nódulos bien delimitados que frecuentemente pueden ser confundidos con metástasis de algún proceso maligno, con aproximadamente 120 casos reportados en la literatura, sin embargo, se han descrito también casos de metástasis cardiaca, linfática, cerebral, ósea o cutánea 2 . Estos tumores suelen ser de crecimiento lento y de pronóstico favorable, sus manifestaciones clínicas están en función del sitio que involucren, el número y el tamaño de los tumores 2 .…”

Benign metastasizing leiomyoma – a case of benign metastasis

“…1 Benign metastatic leiomyoma (BML), first reported in 1939 by Steiner, is a rare disease characterized by welldifferentiated smooth muscle tumours that affect extra-uterine sites, namely lungs (79.5%), abdominal cavity, retroperitoneum, muscle tissue, lymph nodes, blood vessels or even the heart. 2,3 Pulmonary BML is characterized by the presence of multiple bilateral pulmonary nodules of different sizes that can be confused with metastatic cancer. 3 Studies suggest that metastatic leiomyoma derives from uterine leiomyomas.…”

“…5 The distinction between uterine leiomyoma and leiomyosarcoma is made by morphology, degree of cytological atypia, mitotic index and presence of tumour cell necrosis. 1,2 BML can also be confused with Pulmonary Lymphangioleiomyomatosis (LAM), which constitutes a progressive proliferation of perivascular epithelioid cells along the bronchial interstitium, instead of inert nodular aggregates of smooth muscle cells that involve the alveolar parenchyma. 1,2 The diagnosis of BML is based on the histological diagnosis of extra-uterine lesions with microscopic features of leiomyoma.…”

“…However, hormonal treatment constitute a valid alternative to unresectable tumours. 2 Hysterectomy may be necessary to exclude sarcoma if the patient has a mass or several uterine masses. 1…”

“…1,2 BML can also be confused with Pulmonary Lymphangioleiomyomatosis (LAM), which constitutes a progressive proliferation of perivascular epithelioid cells along the bronchial interstitium, instead of inert nodular aggregates of smooth muscle cells that involve the alveolar parenchyma. 1,2 The diagnosis of BML is based on the histological diagnosis of extra-uterine lesions with microscopic features of leiomyoma. 1 Expectant conduct is an option for asymptomatic disease.…”