Chest imaging
Case TypeClinical Cases
Authors
C Ní Leidhin, R Heaney, P Beddy
Patient58 years, female
A 58-year-old woman was referred to our institution for investigation of pulmonary nodules identified on external chest radiograph performed at the time of a road traffic accident.
Repeated chest radiograph demonstrated unchanged multiple, bilateral pulmonary nodules, the largest in the right upper zone. A contrast-enhanced CT of the thorax, abdomen and pelvis was performed, which demonstrated multiple, bilateral, well-circumscribed, rounded pulmonary nodules, measuring up to 1.5 cm. A well-circumscribed, rounded, enhancing 2.3 cm mass was also noted in the posterior uterine myometrium. The patient underwent PET/CT, which demonstrated bilateral pulmonary nodules, at least one of which showed increased radiotracer uptake and an FDG-avid nodule in the posterior fundal myometrium.
The combination of CT TAP and PET/CT findings was suspicious for a primary endometrial tumour with haematogenous metastases to lung. A gynaecology opinion was recommended and the patient underwent hysteroscopy and curettage, which revealed multiple benign post-menopausal polyps only, with no evidence of hyperplasia, dysplasia or malignancy. Following multidisciplinary discussion, transbronchial biopsy of one of the pulmonary nodules was attempted but was non-diagnostic. Percutaneous CT-guided lung biopsy was subsequently performed, the histology of which revealed a bland smooth muscle tumour, suggestive of metastasising leiomyoma. The patient subsequently underwent total hysterectomy and bilateral salpingo-oophorectomy. The surgical specimen demonstrated multiple benign leiomyomata. No further intervention was required. Follow-up imaging demonstrates slow interval growth of the pulmonary lesions. The patient remains asymptomatic, 5 years later.
Benign metastasising leiomyoma (BML) is a rare condition, with only approximately 214 published research articles relating to the subject. [1] It was first described by Steiner in 1939. [2] It refers to extra-uterine metastases of benign smooth muscle tumours. As in our case, the lungs are the most frequent site of metastases. [1] BML is most common in asymptomatic, perimenopausal women [1], like our patient. They usually have a history of uterine fibroids. Imaging typically demonstrates well-circumscribed, non-calcified, non-enhancing pulmonary nodule(s), ranging in size from a few millimetres to a few centimetres. Endobronchial and pleural sparing are typical. Rarely, miliary/cavitating nodules, cysts and interstitial disease have been described. [3] The accumulation of 18-FDG in pulmonary BML varies significantly, with tumours more likely to be non-avid than FDG-avid. [4] Histopathology confirms the presence of smooth muscle cells with low mitotic activity, limited vascularisation, no anaplasia/necrosis and positive immunohistochemical staining for smooth muscle markers, i.e. actin, desmin and hormonal receptors, i.e. oestrogen/progesterone. [5] Although hormone-dependent, BML has a favourable long-term prognosis. Targeted hormonal therapies and/or surgery can be used in disease management, with disease control or regression in 79% of patients treated with hormonal manipulation. [6]
BML, although rare, should be considered in the differential diagnosis for women presenting with multiple pulmonary nodules, especially those with a history of uterine fibroids.
[1] Barnaś E, Książek M, Raś R, Skręt A, Skręt-Magierło J, Dmoch-Gajzlerska E. (2017) Benign metastasizing leiomyoma: A review of current literature in respect to the time and type of previous gynecological surgery. PLoS One;12(4):e0175875. (PMID: 28426767).
[2] Steiner PE. (1939) Metastasizing fibroleiomyoma of the uterus. Am J Pathol. 1939;15(1):89–110.7. (PMID: 19970436).
[3] Abramson S, Gilkeson RC, Goldstein JD, Woodard PK, Eisenberg R, Abramson N. (2001) Benign metastasizing leiomyoma: clinical, imaging, and pathologic correlation. AJR Am J Roentgenol;176(6):1409–1413. (PMID: 11373202).
[4] Sawai Y, Shimizu T, Yamanaka Y, Niki M, Nomura S. (2017) Benign metastasizing leiomyoma and 18-FDG-PET/CT: A case report and literature review. Oncol Lett.;14(3):3641–3646. (PMID: 28927125).
[5] Raposo MI, Meireles C, Cardoso M, Ormonde M, Ramalho C, Pires M, Afonso M, Petiz A. (2018) Benign Metastasizing Leiomyoma of the Uterus: Rare Manifestation of a Frequent Pathology. Case Rep Obstet Gynecol.;2018:5067276. (PMID: 30510824).
[6] Miller J, Shoni M, Siegert C, Lebenthal A, Godleski J, McNamee C. (2016) Benign Metastasizing Leiomyomas to the Lungs: An Institutional Case Series and a Review of the Recent Literature. Ann Thorac Surg.;101(1):253–258. (PMID: 26321441).
URL: | https://www.eurorad.org/case/16768 |
DOI: | 10.35100/eurorad/case.16768 |
ISSN: | 1563-4086 |
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