Benign metastasizing leiomyomatosis (BML): A rare cause of cavitary and cystic pulmonary nodules

Jolissaint, Joshua S.; Kilbourne, Sarah; LaFortune, Kristen; Patel, Manojkumar; Lau, Christine L.

doi:10.1016/j.rmcr.2015.09.008

Cited by 9 publications

(9 citation statements)

References 7 publications

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“…IVL is pathologically classified as a benign tumour, but behaves like a malignant lesion. IVL can extend into the atrium, leading to fatality 16 , or metastasize to the pulmonary artery 17 . However, with complete excision, the probability of recurrence is close to zero 15,18 .…”

Section: Discussionmentioning

confidence: 99%

Identification of the molecular relationship between intravenous leiomyomatosis and uterine myoma using RNA sequencing

Zhang

et al. 2019

Sci Rep

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The purpose of this study was to explore the potential relationship between intravenous leiomyomatosis (IVL) and uterine myoma (UM) at the molecular level. RNA-sequencing was performed on IVL tumours, UM tumours, and adjacent normal uterine muscle. We compared the gene expression levels between IVL and normal uterine muscle, UM and normal uterine muscle, to identify differentially expressed genes (DEGs). Then we used Gene Ontology Enrichment Analysis to determine the functions of the DEGs and performed specimen cluster analysis. We obtained 98 DEGs between IVL and adjacent normal uterine muscle, and 61 DEGs between UM and adjacent normal uterine muscle. Functional enrichment of both IVL and UM DEGs showed that they are associated with hormone stimulus, extracellular matrix, and cell adhesion. Unsupervised clustering analysis showed that IVL and UM could not be separated completely. Among these dysregulated genes, we found that HOXA13 showed a distinct dysregulated status between IVL and UM. HOXA13 may therefore serves as a biomarker to distinguish IVL and UM. Our results showed that IVL and UM may have similar dysregulated gene networks. They may be closely related, and HOXA13 may serves as a biomarker to distinguish between IVL and UM.

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Section: Discussionmentioning

confidence: 99%

Identification of the molecular relationship between intravenous leiomyomatosis and uterine myoma using RNA sequencing

Zhang

et al. 2019

Sci Rep

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“…The appearance of emphysematous regions within the lung is also a rare finding in BML. There are occasional reports of cystic16 or cavitating17 18 nodules; however, we could only identify one previous report of bullous lesions 14. Even in this case, the patient had a handful of large bullae which could be resected, as opposed to the widespread, bilateral emphysematous change along the lung fissures.…”

Section: Discussionmentioning

confidence: 79%

Benign metastasising leiomyoma presenting with respiratory distress

Stabler¹,

Azim

Edwards

2019

BMJ Case Rep

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Benign metastasising leiomyoma (BML) is a rare gynaecological tumour which most commonly presents with incidental lung nodules. Here, we present a case of BML in which a 43-year-old woman was admitted in respiratory distress. She was found to have widespread lung involvement with numerous, moderately sized lesions and emphysematous lung regions. Imaging of this patient demonstrated more extensive respiratory disease than has previously been reported in BML, and rendered surgical resection of the nodules impossible. We describe the patient’s treatment with chemical castration and report some improvement in both lung imaging and function.

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“…Immunohistochemical markers expressed by BML include smooth muscle actin, estrogen receptor, progesterone receptor, desmin, and caldesmon. Several spindled lesions should be taken into consideration in the differential diagnosis of BML, including solitary brous tumor, gastrointestinal stromal tumor, in ammatory myo broblastic tumor, lymphangioleiomyomatosis, sarcoidosis, leiomyomatous hamartoma, spindle-cell carcinomas, spindle cell melanoma, and metastatic uterine leiomyosarcoma or endometrial stromal sarcoma [25]. The diagnostic process may be especially hampered by small biopsies, thus a useful additional immunohistochemical panel includes CD10, CD34, CD117, DOG1, ALK, STAT6, cytokeratins, HMB-45, and melanin.…”

Section: Discussionmentioning

confidence: 99%

Benign metastasizing leiomyoma of the lung: diagnostic process and treatment based on three case reports and a review of the literature

Wojtyś

Kacalska-Janssen

Ptaszyński

et al. 2021

Preprint

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Background: Benign metastasizing leiomyoma (BML) is a rare disorder in women that is most commonly an incidental finding on chest X-rays. Patients share a common history of uterine leiomyoma. The transformed cells metastasize to various locations by an unclear mechanism, especially the lungs (BPML: benign pulmonary metastasizing leiomyoma). The disease takes the form of either, more commonly, multiple nodules or a solitary mass. Despite this pulmonary involvement, patients seldom present with signs of respiratory insufficiency, making diagnosis even more problematic.Case presentations: The three cases described in this paper are not alike. Two patients presented with the more common multiple-nodule variant while the other had a single mass, but all were symptom-free. The first patient was diagnosed with BML at the age of 50, and 12 years prior to the diagnosis, underwent a supracervical hysterectomy. The second patient had a myomectomy at the age of 36, and BML diagnosed 17 years later at the age of 53. The third patient had a hysterectomy with bilateral salpingo-oophorectomy at the age of 46, with lung lesions present before the hysterectomy. All patients underwent a significant number of tests before the decision was made to conduct invasive diagnostic procedures and operate. Immunohistochemical studies of postoperative materials showed positive staining of spindle cells with antibodies against desmin and smooth muscle actin as well as estrogen and progesterone receptors. There were entrapped bronchoalveolar structures stained with the TTF1 antibody. The final histopathological diagnoses were BPML. All patients are stable and symptom-free: two of them at 2 years follow-up and one at 6 months follow-up.Conclusion: A "gold standard" therapy for BML has yet to be established, with authors proposing hormonal manipulation (e.g., anti-estrogen therapy, aromatase inhibitors, gonadotropin-releasing hormone agonists), oophorectomy, and classical resection. This paper adds to the surgical experience in successful BPML management.

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Benign metastasizing leiomyomatosis (BML): A rare cause of cavitary and cystic pulmonary nodules

Cited by 9 publications

References 7 publications

Identification of the molecular relationship between intravenous leiomyomatosis and uterine myoma using RNA sequencing

Identification of the molecular relationship between intravenous leiomyomatosis and uterine myoma using RNA sequencing

Benign metastasising leiomyoma presenting with respiratory distress

Benign metastasizing leiomyoma of the lung: diagnostic process and treatment based on three case reports and a review of the literature

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