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.
Highlights
Benign 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 disease develops as a proliferation of multiple nodules composed of smooth muscle cells.The most frequent site of metastasis is the lungs, other areas may be affected as well, including some atypical locations, e.g. the heart or spinal cord.Steiner (1939) was first to describe this disease in detail.He published a report of a patient who died from the effects of extensive pulmonary metastases of benign-appearing leiomyomas, which were histologically identical to the multiple leiomyomas in the uterus [1]
We found 321 hits for a broad search string [metastasizingà OR leiomyomaÃ], and 214 hits were found when the searched term was reduced to “benign metastasizing leiomyoma”.The researched articles were published between 1965 and 2016
The analysis of the site of metastasis and mean age during the primary and BML surgery did not exhibit any significant differences between the groups (Table 1B)
Summary
To determine age before BML surgery, age during diagnosis of BML, type of prior surgery, and location of metastasis based on the literature
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