Case Report: Malignant perivascular epithelioid cell tumor with aggressive mediastinal invasion and pulmonary metastasis

Perivascular epithelioid cell tumors (PEComas) are rare, typically benign soft tissue tumors that can develop at various anatomic sites. Malignant PEComas are rarer entities but may present aggressively with metastasis to the lungs or local recurrence years after initial presentation. In unresectabl...

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Main Authors: Daniel F. Leach, Srivikram Margam S, Marissa Foster, Jarrod B. Adkison
Format: Article
Language:English
Published: Frontiers Media S.A. 2025-08-01
Series:Frontiers in Oncology
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Online Access:https://www.frontiersin.org/articles/10.3389/fonc.2025.1551663/full
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author Daniel F. Leach
Srivikram Margam S
Marissa Foster
Jarrod B. Adkison
author_facet Daniel F. Leach
Srivikram Margam S
Marissa Foster
Jarrod B. Adkison
author_sort Daniel F. Leach
collection DOAJ
description Perivascular epithelioid cell tumors (PEComas) are rare, typically benign soft tissue tumors that can develop at various anatomic sites. Malignant PEComas are rarer entities but may present aggressively with metastasis to the lungs or local recurrence years after initial presentation. In unresectable or metastatic cases, treatment options are limited due to the resistance of PEComas to chemotherapy and radiotherapy. The present case describes a 59-year-old man with a highly aggressive malignant PEComa, which ultimately invaded the mediastinum and replaced the right middle and lower lobes of the lung despite systemic therapy with oral sirolimus and definitive radiotherapy. As only three prior cases have described malignant PEComas invading the mediastinum, we highlight the clinical course of such an aggressive cancer and review current treatment paradigms.
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publisher Frontiers Media S.A.
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series Frontiers in Oncology
spelling doaj-art-e8e6bce9b719483aa2ed3c6f6000f8ad2025-08-20T03:07:01ZengFrontiers Media S.A.Frontiers in Oncology2234-943X2025-08-011510.3389/fonc.2025.15516631551663Case Report: Malignant perivascular epithelioid cell tumor with aggressive mediastinal invasion and pulmonary metastasisDaniel F. Leach0Srivikram Margam S1Marissa Foster2Jarrod B. Adkison3Department of Radiation Oncology, The Ohio State University, Columbus, OH, United StatesFamily Medicine, Ventura County Medical Center, Ventura, CA, United StatesDepartment of Radiation Oncology, Indiana University School of Medicine, Indianapolis, IN, United StatesRadiation Oncology, Southeast Health, Dothan, AL, United StatesPerivascular epithelioid cell tumors (PEComas) are rare, typically benign soft tissue tumors that can develop at various anatomic sites. Malignant PEComas are rarer entities but may present aggressively with metastasis to the lungs or local recurrence years after initial presentation. In unresectable or metastatic cases, treatment options are limited due to the resistance of PEComas to chemotherapy and radiotherapy. The present case describes a 59-year-old man with a highly aggressive malignant PEComa, which ultimately invaded the mediastinum and replaced the right middle and lower lobes of the lung despite systemic therapy with oral sirolimus and definitive radiotherapy. As only three prior cases have described malignant PEComas invading the mediastinum, we highlight the clinical course of such an aggressive cancer and review current treatment paradigms.https://www.frontiersin.org/articles/10.3389/fonc.2025.1551663/fullperivascular epithelioid cell tumorsintensity-modulated radiotherapy3D conformal radiotherapypalliative radiotherapymTOR inhibitorsVEGF inhibitors
spellingShingle Daniel F. Leach
Srivikram Margam S
Marissa Foster
Jarrod B. Adkison
Case Report: Malignant perivascular epithelioid cell tumor with aggressive mediastinal invasion and pulmonary metastasis
Frontiers in Oncology
perivascular epithelioid cell tumors
intensity-modulated radiotherapy
3D conformal radiotherapy
palliative radiotherapy
mTOR inhibitors
VEGF inhibitors
title Case Report: Malignant perivascular epithelioid cell tumor with aggressive mediastinal invasion and pulmonary metastasis
title_full Case Report: Malignant perivascular epithelioid cell tumor with aggressive mediastinal invasion and pulmonary metastasis
title_fullStr Case Report: Malignant perivascular epithelioid cell tumor with aggressive mediastinal invasion and pulmonary metastasis
title_full_unstemmed Case Report: Malignant perivascular epithelioid cell tumor with aggressive mediastinal invasion and pulmonary metastasis
title_short Case Report: Malignant perivascular epithelioid cell tumor with aggressive mediastinal invasion and pulmonary metastasis
title_sort case report malignant perivascular epithelioid cell tumor with aggressive mediastinal invasion and pulmonary metastasis
topic perivascular epithelioid cell tumors
intensity-modulated radiotherapy
3D conformal radiotherapy
palliative radiotherapy
mTOR inhibitors
VEGF inhibitors
url https://www.frontiersin.org/articles/10.3389/fonc.2025.1551663/full
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