Auricular epithelioid hemangioendothelioma: Two cases first diagnosed as auricular pseudocyst

Wang,Yibei; Niu,Yiru; Mi,Zhiyu; Zhao,Jianhui; Zhang,Ruoqi; Liu,Jianfeng

doi:10.3892/ol.2024.14763

Auricular epithelioid hemangioendothelioma: Two cases first diagnosed as auricular pseudocyst

Authors:
- Yibei Wang
- Yiru Niu
- Zhiyu Mi
- Jianhui Zhao
- Ruoqi Zhang
- Jianfeng Liu
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Affiliations: Department of Otolaryngology‑Head and Neck Surgery, China‑Japan Friendship Hospital, Beijing 100029, P.R. China, Department of Pathology, China‑Japan Friendship Hospital, Beijing 100029, P.R. China, Department of Otolaryngology, Changping Maternal and Child Health Care Hospital, Beijing 102299, P.R. China
Published online on: October 21, 2024 https://doi.org/10.3892/ol.2024.14763
Article Number: 17
Copyright: © Wang et al. This is an open access article distributed under the terms of Creative Commons Attribution License.

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Abstract

Epithelioid hemangioendothelioma (EHE) is a rare soft‑tissue vascular neoplasm with a prevalence of one in one million. The present study firstly reports two cases of EHE occurring in the auricle. The clinical, histopathological and immunohistochemical features of two patients with auricular EHE are described, and the associated literature are reviewed. Two adult male patients each presented with an asymptomatic, unilateral soft skin‑colored noninflammatory swelling of the auricle. Based on their clinical manifestations, both patients were initially diagnosed with auricular pseudocysts. Auricular excision surgery was performed under general anesthesia. The resected specimens were sent for pathological examination. Immunohistochemical examination showed that the specimens were positive for CD31, CD34, friend leukemia integration 1 (FLI‑1), coagulation factor 8 and E26 transformation‑specific‑related gene, which was consistent with EHE. Follow‑up after surgery showed no evidence of tumor recurrence. It may be concluded EHEs of low malignancy should be included in the differential diagnosis of patients with auricular pseudocysts. EHEs can be diagnosed based on their morphological and histological characteristics, with immunohistochemical positivity for FLI‑1 and CD31 being suggestive of a diagnosis of EHE.

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1	Sardaro A, Bardoscia L, Petruzzelli MF and Portaluri M: Epithelioid hemangioendothelioma: An overview and update on a rare vascular tumor. Oncol Rev. 8:2592014.
2	Rosenberg A and Agulnik M: Epithelioid Hemangioendothelioma: Update on diagnosis and treatment. Curr Treat Options Oncol. 19:192018. View Article : Google Scholar
3	Bagan P, Hassan M, Barthes FL, Peyrard S, Souilamas R, Danel C and Riquet M: Prognostic factors and surgical indications of pulmonary epithelioid hemangioendothelioma: A review of the literature. Ann Thorac Surg. 82:2010–2013. 2006. View Article : Google Scholar
4	Cohen V, Fortier-Riberdy G, Saliba I and Davar S: A case of auricular pseudocyst. J Cutan Med Surg. 20:573–574. 2016. View Article : Google Scholar
5	Anderson T, Zhang L, Hameed M, Rusch V, Travis WD and Antonescu CR: Thoracic epithelioid malignant vascular tumors: A clinicopathologic study of 52 cases with emphasis on pathologic grading and molecular studies of WWTR1-CAMTA1 fusions. Am J Surg Pathol. 39:132–139. 2015. View Article : Google Scholar
6	Gill R, O'Donnell RJ and Horvai A: Utility of immunohistochemistry for endothelial markers in distinguishing epithelioid hemangioendothelioma from carcinoma metastatic to bone. Arch Pathol Lab Med. 133:967–972. 2009. View Article : Google Scholar
7	Tosta TA, de Faria PR, Neves LA and do Nascimento MZ: Computational normalization of H&E-stained histological images: Progress, challenges and future potential. Artif Intell Med. 95:118–132. 2019. View Article : Google Scholar
8	Ribeiro MB and Ibiapina JO: Immunohistochemical analysis by KI67 and IDH1 in patients with chondrosarcoma. Acta Ortop Bras. 31:e2672122023. View Article : Google Scholar
9	Weiss SW and Enzinger FM: Epithelioid hemangioendothelioma: A vascular tumor often mistaken for a carcinoma. Cancer. 50:970–981. 1982. View Article : Google Scholar
10	Kitaichi M, Nagai S, Nishimura K, Itoh H, Asamoto H, Izumi T and Dail DH: Pulmonary epithelioid haemangioendothelioma in 21 patients, including three with partial spontaneous regression. Eur Resp J. 12:89–96. 1998. View Article : Google Scholar
11	Abbas O, Chedraoui A, Baki JA, Kibbi AG and Ghosn S: Bilateral auricular pseudocysts as the presenting sign of hepatocellular carcinoma. Clin Exp Dermatol. 35:e34–e36. 2010. View Article : Google Scholar
12	Pereira FC, Chinelli PA, Takahashi MD and Nico MM: Bilateral pseudocyst of the auricle in a man with pruritus secondary to lymphoma. Int J Dermatol. 42:818–821. 2003. View Article : Google Scholar
13	Hoffmann TJ, Richardson TF, Jacobs RJ and Torres A: Pseudocyst of the auricle. J Dermatol Surg Oncol. 19:259–262. 1993. View Article : Google Scholar
14	Engel D: Pseudocysts of the auricle in Chinese. Arch Otolaryngol. 83:197–202. 1966. View Article : Google Scholar
15	Flucke U, Vogels RJ, de Saint Aubain Somerhausen N, Creytens DH, Riedl RG, van Gorp JM, Milne AN, Huysentruyt CJ, Verdijk MA, van Asseldonk MM, et al: Epithelioid hemangioendothelioma: Clinicopathologic, immunhistochemical, and molecular genetic analysis of 39 cases. Diagn Pathol. 9:1312014. View Article : Google Scholar
16	Guo W, Zhou D, Huang H, Chen H, Wu X, Yang X, Ye H and Hong C: Successful chemotherapy with continuous immunotherapy for primary pulmonary endovascular epithelioid hemangioendothelioma: A case report. Medicine (Baltimore). 102:e329142023. View Article : Google Scholar
17	Ye B, Li W, Feng J, Shi JX, Chen Y and Han BH: Treatment of pulmonary epithelioid hemangioendothelioma with combination chemotherapy: Report of three cases and review of the literature. Oncol Lett. 5:1491–1496. 2013. View Article : Google Scholar
18	Bansal A, Chawla M, Cohen PJ and Kwon JS: Pleural epithelioid hemangioendothelioma. Lung. 190:469–470. 2012. View Article : Google Scholar
19	Ma J, Wang L, Mo W, Yang X and Xiao J: Epithelioid hemangioendotheliomas of the spine: Clinical characters with middle and long-term follow-up under surgical treatments. Eur Spine J. 20:1371–1376. 2011. View Article : Google Scholar
20	Zheng Z, Wang H, Jiang H, Chen E, Zhang J and Xie X: Apatinib for the treatment of pulmonary epithelioid hemangioendothelioma: A case report and literature review. Medicine (Baltimore). 96:e85072017. View Article : Google Scholar
21	Kobayashi N, Shimamura T, Tokuhisa M, Goto A and Ichikawa Y: Sorafenib Monotherapy in a patient with unresectable hepatic epithelioid hemangioendothelioma. Case Rep Oncol. 9:134–137. 2016. View Article : Google Scholar
22	Semenisty V, Naroditsky I, Keidar Z and Bar-Sela G: Pazopanib for metastatic pulmonary epithelioid hemangioendothelioma-a suitable treatment option: Case report and review of anti-angiogenic treatment options. BMC Cancer. 15:4022015. View Article : Google Scholar