SESSION TITLE: Lung Pathology Cases
SESSION TYPE: Affiliate Case Report Slide
PRESENTED ON: Tuesday, October 27, 2015 at 04:30 PM - 05:30 PM
INTRODUCTION: Benign metastasizing leiomyomatosis (BML) is a rare extra uterine presentation of common benign tumor. It is known to occur in reproductive women with a history of a uterine myomectomy or hysterectomy with a radiographic finding of a single to multiple pulmonary nodules. We report a rare patient with BML who presented with a chronic cough and a rarer radiographic finding of a diffuse reticular nodular pattern.
CASE PRESENTATION: 39 year old female with prior uterine myomectomy presented with an intermittent non-productive chronic cough. Pulmonary function testing (PFT) revealed mild reduction in diffusion capacity. Chest X-ray and chest CT showed a diffuse reticular nodular pattern. Wedge resection demonstrated benign tumor cells which stained positively for desmin and negatively for HMB-45 with negative tissue culture, leading to the diagnosis of BML. She was offered medical castration with gonadotropin-releasing hormone agonists plus an aromatase inhibitor and surgical castration via total abdominal hysterectomy with bilateral salpingo-oophorectomy versus observation. She had a desire for a potential pregnancy, and chose observation with 3 month follow-up CT scan and PFTs. At 3 month follow-up she had decreasing DLCO, and choose to undergo medical and surgical castration.
DISCUSSION: BML is a rare benign disease that can have invasive features on radiographic imaging. Tissue biopsy is required to exclude malignant or infectious etiologies. The best reported treatment is combine medical and surgical castration. Optimal timing of treatment is uncertain given the typically younger patient age and indolent nature of the tumor. If a patient with BML desires pregnancy or refuses treatment, observational monitoring with serial PFTs and radiographic imaging can be considered.
CONCLUSIONS: We describe a rare case of pulmonary BML with an unusual radiographic presentation of a diffuse reticular nodular pattern that suggested invasive disease. Medical and surgical castration are the therapy of choice, but close observation may be preferred by patients who are considering pregnancy. Our patient elected combined medical and surgical castration due to decreasing diffusion capacity after initial observation. BML is a disease that may be benign histologically, yet have an aggressive clinical course and radiographic features which mimic a metastatic malignancy.
Reference #1: Lewis EI. Novel hormone treatment of benign metastasizing leiomyoma: an analysis of five cases and literature review. Fertil Steril. 2013 Jun;99(7): 2017-24.
DISCLOSURE: The following authors have nothing to disclose: William Londeree, Sean McKay, Thomas Fitzpatrick
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