Benign metastasizing leiomyoma: A review of current literature in respect to the time and type of previous gynecological surgery

Abstract

IntroductionBenign metastasizing leiomyoma (BML) is a rare disorder that affects women with a history of uterine leiomyoma, which is found to metastasise within extrauterine sites. The aetiology of BML remains unexplained. Because BML is rare, and most publications contain descriptions of single cases, no statistically determined time relations were found between the primary and secondary surgeries, which may have aetiological implications.ObjectivesTo determine age before BML surgery, age during diagnosis of BML, type of prior surgery, and location of metastasis based on the literature.MethodsA systematic review of four databases (Medline/PubMed, Embase, Web of Science, and Cochrane) covering articles published from 1 January 1965 to 10 April 2016. The inclusion criteria were full-text articles in English and articles containing case reports. Articles in languages other than English (39), articles containing incomplete data (14), i.e. no information regarding the time of surgery and/or the site of metastasis, articles bereft of case studies (25), and articles with access only to summaries, without access to the complete text (10) were excluded. Of 321 titles identified, only 126 articles met the aforementioned criteria.Results and conclusionsThe mean age during primary surgery and BML diagnosis was 38.5 years and 47.3 years, respectively. The most common surgery was total hysterectomy. The most frequent site of metastasis was the lungs; other organs were affected less frequently.The site of metastases and their number were not related to the longer time span between the patient’s initial surgery and occurrence of metastasis. The analysed data, such as the age during primary surgery, age during BML diagnosis, site and type of metastasis, do not provide us a clear answer. Thus, BML pathogenesis is most probably complex in nature and requires further multidirectional research.

Multiple organ benign metastasizing leiomyoma: A case report and literature review

“…It occurs in women whom have undergone myomectomy or hysterectomy for uterine leiomyomas. The mean age of primary surgery and BML confirmation was 38.5 and 47.3 years, respectively . It is characterized by the presence of multiple nodules which most frequently located in the lung .…”

“…It is characterized by the presence of multiple nodules which most frequently located in the lung . Other metastasizing sites including lymph nodes, blood vessels (venous leiomyomatosis), pelvic, arm, bone (skull and spine), even pancreas and heart . The lung is the most frequently affected organ by BML.…”

“…The mean age of primary surgery and BML confirmation was 38.5 and 47.3 years, respectively. 4 It is characterized by the presence of multiple nodules which most frequently located in the lung. 2 Other metastasizing sites including lymph nodes, blood vessels (venous leiomyomatosis), pelvic, arm, bone (skull and spine), even pancreas and heart.…”

“…2 Other metastasizing sites including lymph nodes, blood vessels (venous leiomyomatosis), pelvic, arm, bone (skull and spine), even pancreas and heart. 4 The lung is the most frequently affected organ by BML. However, coexistence of BML in lung, thigh, ilium and pelvis have not been reported yet.…”

Loss of ATRX/DAXX expression and alternative lengthening of telomeres in uterine leiomyomas

“…18 The clinical course of BML is typically indolent, although rare malignant transformation has been reported. Pulmonary and uterine tumors displayed similar molecular alterations, and this suggested possible extrauterine dissemination of ULs and thus a condition known as benign metastasizing leiomyoma (BML) in these patients.…”