Pulmonary benign metastasising leiomyoma: a single-institution case series

Karishma Chandarana ,Edward Caruana ,Cathy Richards ,Sridhar Rathinam,Apostolos Nakas

doi:10.1186/s43057-021-00047-2

Abstract

BackgroundBenign metastasising leiomyoma (BML) is a rare and often asymptomatic presentation of smooth muscle tumour of uterine origin, occurring outside the uterus. We present the first case series of pulmonary BML in a contemporary UK population.Patients were identified from prospective histopathological databases spanning from 2003 to 2019. Data was collected retrospectively from physical and electronic data sources.ResultsSix post-menopausal females were identified with an average age of 44±8 years (mean±SD). Fifty percent of our cohort was asymptomatic with an incidental finding of pulmonary nodules, whilst the others complained of non-specific respiratory symptoms. Five patients (88%) had multiple lesions (median 9, range 2 to 12)—with bilateral distribution, measuring an average of 11 mm (range 7 to 27) in size on cross-sectional imaging. All patients underwent diagnostic surgical pulmonary wedge resection, with no perioperative morbidity.Immunohistochemistry confirmed the presence of ‘spindle cells’, staining positive for smooth muscle actin, desmin and progesterone receptors for all patients (100%), and oestrogen receptors in five patients (87%). Four patients (67%) underwent a hysterectomy prior to diagnosis of BML and one (17%) due to the diagnosis itself. Four patients (67%) underwent oophorectomy, whilst one patient required hormonal suppression therapy. Survival was 100% at a median follow-up of 59.5 months (range 29 to 166).ConclusionsBML is a rare clinical entity accounting for a small proportion of patients presenting with pulmonary nodules. Following successful tissue diagnosis, outcomes with conservative or medical management are excellent.

Highlights

Benign metastasising leiomyoma (BML) is a rare and often asymptomatic presentation of smooth muscle tumour of uterine origin, occurring outside the uterus
Patient demographics Our study identified six patients, all post-menopausal females who were diagnosed with pulmonary BML at a median age of 45 years
Three patients had undergone hysterectomy for symptomatic uterine leiomyoma prior to diagnosis (50%), with the diagnosis of pulmonary BML prompting subsequent hysterectomy and oophorectomy in one patient (17%)

Summary

Introduction

Benign metastasising leiomyoma (BML) is a rare and often asymptomatic presentation of smooth muscle tumour of uterine origin, occurring outside the uterus. We present the first case series of pulmonary BML in a contemporary UK population. Multiple pulmonary nodules remain a challenging prospect with regards to management. Benign metastasising leiomyoma (BML), first described by Steiner et al in 1939 [1], is a smooth muscle tumour of primary uterine origin, metastasising to a distant location within the body. Extra-uterine metastases are commonly pulmonary; there have been numerous reports of BML in other organs [4]. The mode of dissemination remains uncertain; BML is more common in post-menopausal females who have undergone uterine surgery, suggesting surgery creates a haematogenous route for tumour spread [5, 6]

Results

Discussion

Conclusion