Two Cases of Epithelioid Angiosarcoma Involving the Thyroid and a Brief Review of Non-Alpine Epithelioid Angiosarcoma of the Thyroid

Goh, Seng Geok Nicholas; Chuah, Khoon Leong; Goh, Hood Keng Christopher; Chen, Yun Yin Cosmas

doi:10.5858/2003-127-e70-tcoeai

Cited by 51 publications

(7 citation statements)

References 6 publications

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“…[9,[20][21][22] The clinical symptoms of PPA are not specific and are commonly represented by chest pain, dyspnea, hemoptysis, pleural effusion, and recurring hemothorax. [8,16,23,24] Radiology provides multiparametric morphological and important help for the diagnosis of PPA, but the radiological signs are also not very specific. PET-CT was used in 11 cases to estimate the stage and primary site.…”

Section: Discussionmentioning

confidence: 99%

Characteristics and outcomes of primary pleural angiosarcoma

Wang

Luo

et al. 2022

Medicine

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Primary pleural angiosarcoma (PPA) is an extremely rare malignancy for which there is no consensus on treatment. The clinical course of PPA is usually quickly fatal, regardless of the treatment used.We summarized and evaluated a relatively large population of published PPA cases to assess prognostic factors, diagnostic approaches, treatment methods and clinical outcomes. Using the CNKI, Embase, and PubMed databases, literature published in English and Chinese from 1988 through 2020 was searched using the terms “primary pleural angiosarcoma,” “pleural angiosarcoma,” and “pleuropulmonary angiosarcoma.”A total of 43 patients with PPA were identified in retrospective case series and case reports. The median age at diagnosis was 64 years (range 24–87 years), and the median overall survival was 4 months (range 0.1–180 months). Approximately 80% of patients died from PPA within 10 months of diagnosis, and the 2-year survival rate was approximately 4.4%. In univariate analyses, the presence of pleural effusion and hemothorax were significant predictors of decreased survival, with hazard ratios (HRs) of 2.7 (P = .04) and 3.3 (P = .006), respectively. Sixteen patients received no therapy, and their prognosis was worse than patients who did receive therapy (P = .019). Radiation therapy improved survival more than no radiation therapy (P = .007). Patients appeared to derive clinical benefit from chemotherapy (P = .048). However, tumor resection did not seem to provide a survival benefit (P = .051). In multivariate analysis, tumor resection, and radiation were independent, statistically significant, positive predictors of better survival, with HRs of 0.3 (P = .017) and 0.1 (P = .006), respectively. The presence of hemothorax was an independent predictor of worse prognosis (P = .006).Primary angiosarcoma of the pleura is a rare, poorly understood malignancy with a poor prognosis; hence, the clinical spectrum of PPA is not completely defined. By multivariate analysis, this retrospective study showed a survival benefit of tumor resection or radiation therapy, and the presence of hemothorax was a significant prognostic factor for poor outcomes.

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Section: Discussionmentioning

confidence: 99%

Characteristics and outcomes of primary pleural angiosarcoma

Wang

Luo

et al. 2022

Medicine

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“…[9][10][11] The clinical symptoms of PPA are not specific and are commonly represented by chest pain, dyspnea, hemoptysis, pleural effusion and recurring hemothorax. [12][13][14][15] Radiology provides multiparametric morphological and important help for the diagnosis of PPA, but the radiological signs are also not very specific. The diagnosis of PPA is strongly dependent on pathological findings.…”

Section: Discussionmentioning

confidence: 99%

Primary pleural epithelioid angiosarcoma treated successfully with anti-PD-1 therapy

et al. 2021

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Rationale: Primary pleural angiosarcoma (PPA) is an extremely rare malignancy for which there is no consensus on treatment. The clinical course of PPA is usually quickly fatal, regardless of the treatment used. Patient concerns: We describe the rare case of a 52-year-old man who presented initially with hemoptysis and received emergency surgery for the primary. Diagnoses: He received a confirmed diagnosis of primary pleural angiosarcoma (PPA) by postoperative pathology and was subsequently treated with radiotherapy and chemotherapy, but had failed and was intolerant to chemotherapy. Interventions: The patient had 5% tumor PD-L1 positivity with 22C3 pharmDx and received pembrolizumab (200 mg every 21 days) for 13 cycles. Outcomes: The disease remained well controlled according to the RECIST 1.1. criteria. He is currently under observation and waiting to start the next cycle of immunotherapy. Lesson: Our case report suggests that the use of anti-PD-1 therapy does show efficacy in the treatment of PPA and may provide a viable treatment option for patients.

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“…The value of radiation therapy is also disputed due to limited published series: surgical treatment followed by radiotherapy appears to improve patient's survival in several but not all published cases [14][15][16][17]. Table 1 enlists the main published experiences.…”

Section: Discussionmentioning

confidence: 99%

Primary Epithelioid Angiosarcoma of the Thyroid: Long-Term Survival Case Report and Literature Review

Porcellini¹,

Alessandro²

2021

J Can Res Rev Rep

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Thyroid angiosarcoma is a rare neoplasm of debated clinical management. In addition, the epithelioid variant poses microscopic challenges due to its close resemblance to poorly differentiated carcinoma. Only 63 cases of thyroid angiosarcoma have been published in English literature. Due to its rarity, the limited clinical experience can make its management complex. We report a new case of primary thyroid angiosarcoma showing preponderant epithelioid features characterized by an exceptional long survival. We also provide a discussion on the role of radiotherapy in tumor treatment and a brief review of the pertinent literature.

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Two Cases of Epithelioid Angiosarcoma Involving the Thyroid and a Brief Review of Non-Alpine Epithelioid Angiosarcoma of the Thyroid

Cited by 51 publications

References 6 publications

Characteristics and outcomes of primary pleural angiosarcoma

Characteristics and outcomes of primary pleural angiosarcoma

Primary pleural epithelioid angiosarcoma treated successfully with anti-PD-1 therapy

Primary Epithelioid Angiosarcoma of the Thyroid: Long-Term Survival Case Report and Literature Review

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