Pleural epithelioid angiosarcoma with lymphatic differentiation arisen after radiometabolic therapy for thyroid carcinoma: immunohistochemical findings and review of the literature

Our plain-English summary of Pleural epithelioid angiosarcoma with lymphatic differentiation arisen after radiometabolic therapy for thyroid carcinoma: immunohistochemical findings and review of the literature, by D. Cabibi, G. Pipitone, R. Porcasi, et al (Diagnostic Pathology, 2017).

The study

• Authors: D. Cabibi, G. Pipitone, R. Porcasi, S. Ingrao, I. Benza, C. Porrello, M. Cajozzo, A.G. Giannone
• Journal: Diagnostic Pathology
• Published: 2017;12(1):60
• Type of article: Review
• Cited by: 5 articles, as counted by NIH iCite
• Read it: PubMed · Publisher · Free full text

Abstract

Background: Pleural angiosarcoma is a rare tumor that causes diffuse pleural thickening and effusion, mimicking mesothelioma. Immunohistochemistry is needed to highlight endothelial differentiation. We describe the first case of pleural angiosarcoma with lymphatic differentiation following radiometabolic therapy for thyroid carcinoma.

Case Presentation: A 50-year-old man showed diffuse pleural thickening and effusion. Nine years earlier, he underwent thyroidectomy and radiometabolic therapy for thyroid carcinoma with lymph node metastases. Histologically, the tumor consisted of a solid proliferation of atypical epithelioid cells and anastomosed vascular spaces, lacking of red blood cells and containing Alcian blue positive material. The tumor showed positive immunostaining for Vimentin, CD31, CK7, D2-40, c-MYC, Ki67, focal positivity for PanCK, and negative immunostaining for Factor VIII, CD34, WT1, CK5/6, Calretinin, EMA, HBME-1, CEA, p63, EpCAM, Bcl-2, TTF1 and Thyroglobulin. CD99 showed a granular/paranuclear pattern of positivity. The histological and immunohistochemical features were consistent with "pleural angiosarcoma with lymphatic differentiation, epithelioid variant".

Discussion And Conclusions: Epithelioid angiosarcoma with lymphatic differentiation is very rare and aggressive. Moreover, the positivity for c-MYC suggests the relationship with radiometabolic therapy. To our knowledge, this is the first case of pleural c-MYC-positive angiosarcoma with lymphatic differentiation reported in the literature and the first one arisen after radiometabolic therapy for thyroid carcinoma.

In plain English

This article, labelled a review, described a single patient: a 50-year-old man admitted to the authors' hospital with breathlessness. Nine years earlier his thyroid had been removed for papillary thyroid cancer that had spread to neck lymph nodes. He was then treated with radioactive iodine-131, receiving a cumulative dose of 400 mCi. Scans now showed thickening of the pleura (the lining around the lung), which clinically suggested pleural mesothelioma. Biopsies instead showed a tumor of cells that line blood vessels. The tumor stained positive for CD31, D2-40 and c-MYC. It stained negative for markers usually positive in mesothelioma and for thyroid markers, which made spread of the old thyroid cancer unlikely. The authors diagnosed epithelioid angiosarcoma of the pleura with lymphatic differentiation. The patient died four months after diagnosis. The authors concluded that a link between the tumor and the cancer-causing effects of the radiation could be hypothesized. To their knowledge, it was the first such pleural tumor reported after this therapy for thyroid cancer. The article mentioned asbestos only when describing earlier cases reported by others. It did not report this patient's asbestos history.

We wrote this plain-English summary from the full article with the help of AI, and a second AI model checked each sentence against the article. It is not the authors' wording; the abstract and the article are their own account.

What the study did and found

• Design: case report of one patient, with a brief discussion of the literature (the article is headed 'REVIEW')
• Who was studied: one 50-year-old man with breathlessness, admitted to the authors' hospital (the authors' affiliations are in Palermo, Italy); dates not given
• Exposure: radiometabolic therapy with iodine-131 for metastatic papillary thyroid carcinoma about nine years earlier, cumulative dose 400 mCi; no asbestos exposure is reported for this patient
• Main results:
  • CT showed irregular circumferential pleural thickening with nodular masses, which clinically suggested pleural mesothelioma
  • Many tumor cell nuclei stained positive for c-MYC
  • Mesothelioma-associated markers and thyroid markers stained negative in the tumor
  • Ki67 expression was high, at about 30% of tumor nuclei
  • The findings supported a diagnosis of epithelioid angiosarcoma with lymphatic differentiation
  • The negative TTF1 and thyroglobulin staining made metastasis from the earlier thyroid carcinoma unlikely
  • The patient died four months after diagnosis
• The authors' conclusion: The authors concluded that a link between the pleural angiosarcoma and the cancer-causing effects of ionizing radiation could be hypothesized. They reported it as, to their knowledge, the first pleural angiosarcoma with lymphatic differentiation after radiometabolic therapy for metastatic thyroid papillary carcinoma.

Topics

Medical terms in this study, explained in our Lexicon: Epithelioid, Carcinoma, Diffuse Pleural Thickening, Mesothelioma, Immunohistochemistry, Podoplanin (D2-40), CD34, Calretinin.

Related studies

• Primary pleural epithelioid angiosarcoma. A case report and review of the literature — E. Dainese, B. Pozzi, et al. (2010)
• Solid cell nests of the thyroid gland: morphological, immunohistochemical and genetic features — M. Manzoni, G. Roversi, et al. (2016)
• Epithelioid angiosarcoma of the pleura: a case report — M.S. Roh, J.Y. Seo, et al. (2001)
• Pseudomesotheliomatous angiosarcoma: a pleuropulmonary lesion simulating malignant pleural mesothelioma — G. Falconieri, R. Bussani, et al. (1997)
• Primary pleural angiosarcoma as a mimicker of mesothelioma: a case report **VS** — Y.C. Kao, J.M. Chow, et al. (2011)
• Malignant epithelioid vascular tumors of the pleura: report of a series and literature review — P.J. Zhang, V.A. Livolsi, et al. (2000)

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