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Pulmonary Metastases of a Uterine Smooth Muscle Tumour with Undefined Malignancy Potential.

ABSTRACT: Smooth muscle neoplasms with atypical proliferative behaviour, but without clear histopathological malignancy represent a diagnostic and therapeutic challenge, as distinction from a sarcoma can be difficult and no guaranteed treatment recommendations are available due to the rarity of these changes. In the event of uncertain primary histology, even metastases cannot be assessed as malignancy criteria, but may contribute to the clarification of the histology. Similarities with other smooth muscle proliferations, such as lymphangioleiomyomatosis, are striking. The diagnostic difficulties and treatment options are explained based on the example of a 59-year-old patient, in whom a retroperitoneal mass and pulmonary lesion of such a tumour occurred 4 years after a hysterectomy. Even though the genesis and histological diagnostics have not been conclusively clarified, slow growth and a low recurrence rate for post-menopausal patients allow for a wait-and-see approach, whereby the option for anti-hormonal treatment exists in the event of positive evidence of hormone receptors.

SUBMITTER: Esch M

PROVIDER: S-EPMC4024611 | biostudies-literature | 2014 Mar

REPOSITORIES: biostudies-literature

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Pulmonary Metastases of a Uterine Smooth Muscle Tumour with Undefined Malignancy Potential.

Esch M M Teschner M M Braesen J-H JH

Geburtshilfe und Frauenheilkunde 20140301 3

Smooth muscle neoplasms with atypical proliferative behaviour, but without clear histopathological malignancy represent a diagnostic and therapeutic challenge, as distinction from a sarcoma can be difficult and no guaranteed treatment recommendations are available due to the rarity of these changes. In the event of uncertain primary histology, even metastases cannot be assessed as malignancy criteria, but may contribute to the clarification of the histology. Similarities with other smooth muscle ...[more]

PMID: 24882880

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Project description:The chronic phase of pulmonary arterial hypertension (PAH) is associated with vascular remodeling, especially thickening of the smooth muscle layer of large pulmonary arteries and muscularization of small pulmonary vessels, which normally have no associated smooth muscle. Serotonin (5-hydroxytryptamine, 5-HT) has been shown to induce proliferation and hypertrophy of pulmonary artery smooth muscle cells (PASMC), and may be important for in vivo pulmonary vascular remodeling. Here, we show that 5-HT stimulates migration of pulmonary artery PASMC. Treatment with 5-HT for 16h increased migration of PASMC up to four-fold as monitored in a modified Boyden chamber assay. Increased migratory responses were associated with cellular morphological changes and reorganization of the actin cytoskeleton. 5-HT-induced alterations in morphology were previously shown in our laboratory to require cAMP [Lee SL, Fanburg BL. Serotonin produces a configurational change of cultured smooth muscle cells that is associated with elevation of intracellular cAMP. J Cell Phys 1992;150(2):396-405], and the 5-HT4 receptor was pharmacologically determined to be the primary activator of cAMP in bovine PASMC [Becker BN, Gettys TW, Middleton JP, Olsen CL, Albers FJ, Lee SL, et al. 8-Hydroxy-2-(di-n-propylamino)tetralin-responsive 5-hydroxytryptamine4-like receptor expressed in bovine pulmonary artery smooth muscle cells. Mol Pharmacol 1992;42(5):817-25]. We examined the role of the 5-HT4 receptor and cAMP in 5-HT-induced bovine PASMC migration. PASMC express 5-HT4 receptor mRNA, and a 5-HT4 receptor antagonist and a cAMP antagonist completely blocked 5-HT-induced cellular migration. Consistent with our previous report that a cAMP-dependent Cl(-) channel is required for 5-HT-induced morphological changes in PASMC, phenylanthranilic acid, a Cl(-) channel blocker, inhibited actin cytoskeletal reorganization and migration produced by 5-HT. We conclude that 5-HT stimulates PASMC migration and associated cytoskeletal reorganization through the 5-HT4 receptor and cAMP activation of a chloride channel.

Project description:BackgroundPhyllodes tumours (PTs) are fibroepithelial breast tumours, which can be classified as benign, borderline or malignant, according to their histological characteristics. While various huge borderline or malignant PTs have been previously described, a benign PT with a pulmonary nodule mimicking malignancy has not yet been reported. In order that doctors may have a comprehensive understanding of super-giant benign PTs (≥ 20 cm), we also performed a literature review to summarize the clinical features, differential diagnosis, and treatment of this disease.Case summaryA 42-year-old woman with severe anaemia presented with a rapidly enlarging right breast mass, measuring approximately 30 cm × 25 cm × 20 cm that was first noticed 1 year previously. A region of skin ulceration and necrosis (20 cm × 15 cm) was observed on the lateral side of the mass. Computed tomography (CT) of the chest revealed a pulmonary nodule, which initially suggested a diagnosis of metastasis. CT showed that the boundaries between the pectoralis major and the mass were blurred, which was presumed to be due to tumour invasion. However, two core needle biopsies of the mass showed no evidence of malignancy. Following these results, the tumour was removed by mastectomy of the right breast. Interestingly, postoperative pathology finally proved the diagnosis of a benign PT. After 1 year of follow-up, wedge resection of the small pulmonary nodule was performed, and it was confirmed that the lung nodule was actually adenocarcinoma rather than metastatic breast cancer. The patient recovered very well without any postoperative treatment.ConclusionThis case is unique in that the giant breast mass initially mimicking a malignant clinical presentation was eventually pathologically confirmed to be a benign PT, which misled the diagnosis and complemented the atypical features of benign PTs. The pathological and immunohistochemical results were important in the differential diagnosis. In addition, total mastectomy should be recommended due to difficulty in the precise diagnosis of PTs, especially in large breast masses. In the literature, almost one-half of super-giant benign cases were thought to be malignant tumours before surgery. This finding is a reminder to consider all conditions in order to make an accurate diagnosis and avoid excessive treatment.

Dataset Information

Pulmonary Metastases of a Uterine Smooth Muscle Tumour with Undefined Malignancy Potential.

Publications

Pulmonary Metastases of a Uterine Smooth Muscle Tumour with Undefined Malignancy Potential.

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