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• W1986348823 abstract "Pulmonary benign metastatic leiomyoma is a rare entity with a debated pathogenesis. We report a case in which benign metastatic leiomyoma involving the lung had spontaneously regressed after debulking surgery. Although no adjuvant treatment was done, the remaining masses regressed significantly in the 1-year follow-up image. Pulmonary benign metastatic leiomyoma is a rare entity with a debated pathogenesis. We report a case in which benign metastatic leiomyoma involving the lung had spontaneously regressed after debulking surgery. Although no adjuvant treatment was done, the remaining masses regressed significantly in the 1-year follow-up image. Benign metastasizing leiomyoma (BML) is a rare disease, with approximately 100 cases reported since it was described for the first time in 1939 [1Steiner P.E. Metastasizing fibroleiomyoma of the uterus: report of a case and review of the literature.Am J Pathol. 1939; 15: 89-110.7PubMed Google Scholar]. Treatment options for symptomatic BML are tumorectomy, oophorectomy, and hormonal therapy. However, there are no definite guidelines. In this case, a palliative surgery caused an unexpected effect: spontaneous regression of other inoperable masses in the 1-year follow-up image progressed continuously before surgery. A 51-year-old woman was referred to us with a large mediastinal mass compressing the left main bronchus. The patient had a history of hysterectomy owing to uterine myoma 20 years earlier, and 8 years later a lung mass was incidentally found at the left upper lobe. After a left upper lobectomy, the mass was pathologically diagnosed as metastasis of BML. During follow-up, a right pulmonary mass was found 6 years ago, and a needle biopsy confirmed it to be also BML. Two years of hormonal therapy was unsuccessful, and eventually the patient stopped coming to the hospital 3 years ago. At the time of referral, the patient had moderate dyspnea of functional class II to III. Radiologic studies, including chest computed tomography, showed that she developed bilateral pulmonary masses (Figs 1A, 1D), grown in both size and number when compared with her previous images. She was postmenopausal, and the results of her blood chemistry work-up were nonspecific. The FEV1:FVC ratio (1.82:2.31) was tolerable for thoracotomy. For palliative purposes, we decided to remove the left side mass, which was compressing the left main bronchus and causing her dyspnea. Through a left thoracotomy, we resected the left mediastinal mass (Fig 2) and any palpable left lower lobe masses with no perioperative event. Histologic and pathologic analyses confirmed the same diagnosis as before; BML to the mediastinum and lung, which was positive for estrogen and progesterone. The patient was discharged 7 days later without complication, and no additional treatment was performed. Postoperative 3-month (Figs 1B, 1E) and 1-year (Figs 1C, 1F) follow-up computed tomographic scan showed no evidence of recurrence at the left lung and mediastinum. However, there were changes on the right thorax in the 1-year follow-up images. The multiple right lung masses, which were continuously growing before the surgery, had decreased in both size and number after the surgery. Currently, the patient has no symptoms except mild dyspnea of functional class I to II, and is subject to regular follow-up for the remaining right side lesions. We have debated the proper adjuvant management of the right lung lesions, such as palliative radiotherapy, hormonal therapy, or just merely observation to use. The fate of lung lesions in this case might be favorable after palliative resection at short term follow-up, but could not be predictable. We need more long term observation of this pulmonary BML showing unusual features against our expectations.Fig 2Photograph of resected left mediastinal mass with 12 cm of longitudinal diameter.View Large Image Figure ViewerDownload (PPT) Benign metastatic leiomyoma affects women with a history of uterine leiomyoma and is characterized by multiple histologically benign pulmonary smooth muscle tumors. It mostly affects the lungs. Speculations on its pathogenesis include a benign uterine leiomyoma colonizing the lung, a metastatic low-grade uterine leiomyosarcoma, and primary pulmonary leiomyomatosis [2Patton K.T. Cheng L. Papavero V. et al.Benign metastasizing leiomyoma: clonality, telomere length and clinicopathologic analysis.Mod Pathol. 2006; 19: 130-140Crossref PubMed Scopus (118) Google Scholar]. The current treatment of choice for BML remains controversial. BML has a long disease-free interval; therefore, observation can also be an option for masses that are not symptomatic. For symptomatic BML, several case reports have stated that oophorectomy can cause spontaneous regression [3Bastian A. Philippou S. Bollow M. et al.The benign metastasizing leiomyoma: case collection and therapeutical options.Pneumologie. 2007; 61: 568-572Crossref PubMed Scopus (2) Google Scholar]. Other reports show that some BMLs respond to hormonal therapy [4Mizuno M. Nawa A. Nakanishi T. Yatabe Y. Clinical benefit of endocrine therapy for benign metastasizing leiomyoma.Int J Clin Oncol. 2011; 16: 587-591Crossref PubMed Scopus (7) Google Scholar]. One case report shows that megestrol completely resolved residual BML masses [5Wentling G.K. Sevin B.U. Geiger X.J. Bridges M.D. Benign metastasizing leiomyoma responsive to megestrol: case report and review of the literature.Int J Gynecol Cancer. 2005; 15: 1213-1217Crossref PubMed Scopus (38) Google Scholar]. Our case shows that tumor mass debulking resection may have an additional effect in hormone responsive BML causing regression of the other masses that are not resectable. Although we could not remove the masses on the right lung, resecting the largest mass on the left lung also reduced the size and number of contralateral masses. However, the mechanism of spontaneous regression is still unclear. The most likely theory is that the resected left mass has been a dominant mass, which was stimulating the growth of other satellite mass. Cohen and colleagues [6Cohen J.D. Robins H.I. Response of ”benign” metastasizing leiomyoma to progestin withdrawal Case report.Eur J Gynaecol Oncol. 1993; 14: 44-45PubMed Google Scholar] reported that progestin promoted pulmonary metastases of benign metastasizing leiomyoma, and the withdrawal of progestin induced a marked tumor regression. We were not able to confirm it biochemically, but the primary mass may have been producing a hormone or antibody that provoked and maintained other metastatic masses. However with the dominant mass gone, hormone-dependent satellite masses that were not able to produce such a transmitter stopped growing and slowly regressed. Another suggested mechanism is that the primary mass may have been disrupting the patient's immune system [7Awonuga A.O. Shavell V.I. Imudia A.N. Rotas M. Diamond M.P. Puscheck E.E. Pathogenesis of Benign Metastasizing Leiomyoma: a review.Obstet Gynecol Surv. 2010; 65: 189-195Crossref PubMed Scopus (74) Google Scholar]. As a result, the leiomyoma cells were able to avoid the attack of the host's immune system and metastasize freely. After the surgery, the patient's immune system returned to normal and started to attack the leiomyoma cells resulting in tumor regression. In some cases of BML, tumorectomy may have a larger effect than we think. However, to identify the mechanism behind this unusual case, we will need further evaluation of the pathology along with the immunochemistry. In summary, pulmonary BML with indolent features in this case, tumor progression may be very slow or dynamic for long periods. Surgical palliation could be an option in highly selected patients, but the results are debatable. In addition, the effect of reducing the tumor burden through surgical palliation should be evaluated, which requires further observation in these unusual pulmonary tumors." @default.
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• W1986348823 title "Spontaneous Regression of Metastatic Pulmonary Leiomyoma After Resection of Contralateral Metastatic Mass" @default.
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