胸膜上皮样血管内皮细胞瘤一例并文献复习_Chinese Journal of Respiratory and Critical Care Medicine

Authors：

范瑛琪 ¹ , 王发平 ¹ , 叶成 ² , 英英 ³ ,  毛辉 ¹

1. ;
2. ;
3. ;

Corresponding author：

毛辉, Email: merrymh@126.com

Keywords：

胸膜肿瘤; 上皮样血管内皮细胞瘤; 临床特征

DOI：

10.7507/1671-6205.2015117

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目的探讨胸膜上皮样血管内皮细胞瘤(PEH)的临床表现、影像、病理学特征、诊断、鉴别诊断、治疗及预后, 提高对该病的诊治水平。方法以"胸膜上皮样血管内皮细胞瘤"为检索词检索了四川大学华西医院2007年1月至2014年12月的住院患者资料, 仅有1例呼吸内科收治的患者符合条件, 就此回顾性分析其临床特点, 并进行相关文献复习。以"胸膜上皮样血管内皮细胞瘤"为检索词检索万方数据库和中国期刊网全文数据库, 以"pleural epithelioid hemangioendothelioma"为检索词检索PubMed、Embase、Ovid及Cochrane数据库, 检索时间为1975年1月至2015年4月。结果 68岁女性患者, 临床表现胸背痛, 查体发现除左侧胸腔积液体征外, 有新近发生的脊柱侧弯畸形及杵状指。胸部增强CT示左侧胸膜弥漫增厚伴胸腔大量积液, 部分包裹, 左肺舌叶及下叶压迫性不张, 不张舌叶内见密度减低影, 右肺散在结节。经胸膜活检获得病理, 组织学上查见异型细胞巢, 进一步免疫组化染色血小板内皮细胞黏附分子(CD31)蛋白阳性, 至此明确诊断。本例患者拒绝化疗, 明确诊断11个月后死亡。文献复习显示, 在上述数据库中共检索到74篇文献, 经筛选后, 有26篇文献40例患者明确诊断为PEH且具备相应临床资料, 符合检索条件。此类患者主要的临床表现为胸痛及呼吸困难, 影像学主要表现为胸腔积液, 胸膜不规则增厚, 组织病理学呈现血管内皮源性免疫标志物阳性。结论 PEH为罕见的低度恶性血管源性肿瘤, 病因不清, 临床表现及影像学不具有特征性, 与肿瘤胸膜转移、胸膜间皮瘤等疾病易混淆, 其诊断主要依靠活检组织病理学检查。现有的治疗手段有限, 尚无有效的治疗措施。预后与性别、年龄及病变累计范围等多种因素有关。

Citation： 范瑛琪, 王发平, 叶成, 英英, 毛辉. 胸膜上皮样血管内皮细胞瘤一例并文献复习. Chinese Journal of Respiratory and Critical Care Medicine, 2015, 14(5): 482-486. doi: 10.7507/1671-6205.2015117 Copy

1.	Lee YJ, Chung MJ, Jeong KC, et al. Pleural epithelioid hemangioendothelioma. Yonsei Med J, 2008, 49:1036-1040.
2.	Dail DH, Liebow AA, Gmelich JT, et al. Intravascular, bronchiolar, and alveolar tumor of the lung (IVBAT):an analysis of twenty cases of a peculiar sclerosing endothelial tumor. Cancer, 1983, 51:452-464.
3.	Weiss SW, Enzinger FM. Epithelioid hemangioendothelioma:a vascular tumor often mistaken for a carcinoma. Cancer, 1982, 50:970-981.
4.	Cronin P, Arenberg D. Pulmonary epithelioid hemangioendothelioma:an unusual case and a review of the literature. Chest, 2004, 125:789-793.
5.	Crotty EJ, McAdams HP, Erasmus JJ, et al. Epithelioid hemangioendothelioma of the pleura:clinical and radiologic features. Am J Roentgenol, 2000, 175:1545-1549.
6.	Saqi A, Nisbet L, Gagneja P, et al. Primary pleural epithelioid hemangioendothelioma with rhabdoid phenotype:report and review of the literature. Diagn Cytopathol, 2007, 35:203-208.
7.	Chad A. Reade, Apar Kishor Ganti, Anne Kessinger. Clinicopathologic characteristics of primary pleural epithelioid hemangioendothelioma. Oncol Rev, 2010, 4:219-222.
8.	Ha SY, Choi IH, Han J, et al. Pleural epithelioid hemangioendothelioma harboring CAMTA1 rearrangement. Lung Cancer, 2014, 83:411-415.
9.	Antonescu CR, Le Loarer F, Mosquera JM, et al. Novel YAP1-TFE3 fusion defines a distinct subset of epithelioid hemangioendothelioma. Genes Chromosomes Cancer, 2013, 52:775-784.
10.	Tanas MR, Sboner A, Oliveira AM, et al. Identification of disease-defining gene fusion in epithelioid hemangioendothelioma. Sci Transl Med, 2011, 3:98ra82.
11.	Mitelman F, Johansson B, Mertens F. The impact of translocations and gene fusions on cancer causation. Nat Rev Cancer, 2007, 7:233-245.
12.	陈恩国, 洪武军, 赵晨, 等.肺及胸膜上皮样血管内皮瘤一例.中华肿瘤杂志, 2003, 5:7.
13.	Lazarus A, Fuhrer G, Malekiani C, et al. Primary pleural epithelioid hemangioendothelioma (EHE)-two cases and review of the literature. Clin Respir J, 2011, 5:e1-5.
14.	Oliveira A, Carvalho L. Epithelioid haemangioendothelioma of the pleura:29 months survival. Rev Port Pneumol, 2005, 12:455-461.
15.	Vitório PK, Coletta EM, Morrone N, et al. Hemangioendotelioma epitelióide de pleura. J Bras Pneumol, 2004, 30:60-65.
16.	Lin BT, Colby T, Gown AM, et al. Malignant vascular tumors of the serous membranes mimicking mesothelioma:a report of 14 cases. Am J Surg Pathol, 1996, 20:1431-1439.
17.	Yu L, Gu T, Xiu Z, et al. Primary pleural epithelioid hemangioendothelioma compressing the myocardium. J Card Surg, 2013, 28:266-268.
18.	Duysinx B, Nguyen D, Louis R, et al. Evaluation of pleural disease with 18-fluorodeoxyglucose positron emission tomography imaging. Chest, 2004, 125:489-493.
19.	Salahudeen HM, Hoey ET, Robertson RJ, et al. CT appearances of pleural tumours. Clin Radiol, 2009, 64:918-930.
20.	艾三喜, 毕娅兰, 张勤凤, 等.胸膜上皮样血管内皮瘤一例并文献复习.中华结核和呼吸杂志, 2015, 38.
21.	Chou YY, Hsieh YS, Wu CC, et al. Epithelioid hemangioendotheliomas of the lung and pleura:report of three cases. Formosan J Surg, 2011, 44:163-167.
22.	Pinet C, Magnan A, Garbe L, et al. Aggressive form of pleural epithelioid haemangioendothelioma:complete response after chemotherapy. Eur Respir J, 1999, 14:237-238.
23.	Bahrami A, Allen TC, Cagle PT. Pulmonary epithelioid hemangioendothelioma mimicking mesothelioma. Pathol Int, 2008, 58:730-734.
24.	陈骏, 丁洁, 贾支俊, 等.肝脏上皮样血管内皮瘤临床病理观察.临床与实验病理学杂志, 2011, 27:234-238.
25.	Fujii T, Zen Y, Sato Y, et al. Podoplanin is a useful diagnostic marker for epithelioid hemangioendothelioma of the liver. Mod Pathol, 2008, 21:125-130.
26.	Liu JX, Shiau MC, Nonaka D. An 80-year-old man with shortness of breath and large right-sided pleural effusion. Chest, 2010, 138:1247-1252.
27.	Martínez D, Iturbe J, Buzeki R, et al. Hemangioendotelioma epitelioide de pleura. Presentacion de un caso con hemotorax. Rev Argent Med Respir, 2008, 8:73-76.
28.	André ST, Valente C, Paiva B, et al. Epithelioid hemangioendothelioma of the pleura-A rare presentation of a clinical case. Rev Port Pneumol, 2009, 16:477-482.
29.	Bansal A, Chawla M, Cohen PJ, et al. Pleural epithelioid hemangioendothelioma. Lung, 2012, 190:469-470.
30.	Knuuttila A, Jee KJ, Taskinen E, et al. Spindle cell tumours of the pleura:a clinical, histological and comparative genomic hybridization analysis of 14 cases. Virchows Arch, 2006, 448:135-141.
31.	Yousem SA, Hochholzer L. Unusual thoracic manifestations of epithelioid hemangioendothelioma. Arch Pathol Lab Med, 1987, 111:459-463.
32.	Ledson MJ, Convery R, Carty A, et al. Epithelioid haemangioendothelioma. Thorax, 1999, 54:560-561.

1. Lee YJ, Chung MJ, Jeong KC, et al. Pleural epithelioid hemangioendothelioma. Yonsei Med J, 2008, 49:1036-1040.
2. Dail DH, Liebow AA, Gmelich JT, et al. Intravascular, bronchiolar, and alveolar tumor of the lung (IVBAT):an analysis of twenty cases of a peculiar sclerosing endothelial tumor. Cancer, 1983, 51:452-464.
3. Weiss SW, Enzinger FM. Epithelioid hemangioendothelioma:a vascular tumor often mistaken for a carcinoma. Cancer, 1982, 50:970-981.
4. Cronin P, Arenberg D. Pulmonary epithelioid hemangioendothelioma:an unusual case and a review of the literature. Chest, 2004, 125:789-793.
5. Crotty EJ, McAdams HP, Erasmus JJ, et al. Epithelioid hemangioendothelioma of the pleura:clinical and radiologic features. Am J Roentgenol, 2000, 175:1545-1549.
6. Saqi A, Nisbet L, Gagneja P, et al. Primary pleural epithelioid hemangioendothelioma with rhabdoid phenotype:report and review of the literature. Diagn Cytopathol, 2007, 35:203-208.
7. Chad A. Reade, Apar Kishor Ganti, Anne Kessinger. Clinicopathologic characteristics of primary pleural epithelioid hemangioendothelioma. Oncol Rev, 2010, 4:219-222.
8. Ha SY, Choi IH, Han J, et al. Pleural epithelioid hemangioendothelioma harboring CAMTA1 rearrangement. Lung Cancer, 2014, 83:411-415.
9. Antonescu CR, Le Loarer F, Mosquera JM, et al. Novel YAP1-TFE3 fusion defines a distinct subset of epithelioid hemangioendothelioma. Genes Chromosomes Cancer, 2013, 52:775-784.
10. Tanas MR, Sboner A, Oliveira AM, et al. Identification of disease-defining gene fusion in epithelioid hemangioendothelioma. Sci Transl Med, 2011, 3:98ra82.
11. Mitelman F, Johansson B, Mertens F. The impact of translocations and gene fusions on cancer causation. Nat Rev Cancer, 2007, 7:233-245.
12. 陈恩国, 洪武军, 赵晨, 等.肺及胸膜上皮样血管内皮瘤一例.中华肿瘤杂志, 2003, 5:7.
13. Lazarus A, Fuhrer G, Malekiani C, et al. Primary pleural epithelioid hemangioendothelioma (EHE)-two cases and review of the literature. Clin Respir J, 2011, 5:e1-5.
14. Oliveira A, Carvalho L. Epithelioid haemangioendothelioma of the pleura:29 months survival. Rev Port Pneumol, 2005, 12:455-461.
15. Vitório PK, Coletta EM, Morrone N, et al. Hemangioendotelioma epitelióide de pleura. J Bras Pneumol, 2004, 30:60-65.
16. Lin BT, Colby T, Gown AM, et al. Malignant vascular tumors of the serous membranes mimicking mesothelioma:a report of 14 cases. Am J Surg Pathol, 1996, 20:1431-1439.
17. Yu L, Gu T, Xiu Z, et al. Primary pleural epithelioid hemangioendothelioma compressing the myocardium. J Card Surg, 2013, 28:266-268.
18. Duysinx B, Nguyen D, Louis R, et al. Evaluation of pleural disease with 18-fluorodeoxyglucose positron emission tomography imaging. Chest, 2004, 125:489-493.
19. Salahudeen HM, Hoey ET, Robertson RJ, et al. CT appearances of pleural tumours. Clin Radiol, 2009, 64:918-930.
20. 艾三喜, 毕娅兰, 张勤凤, 等.胸膜上皮样血管内皮瘤一例并文献复习.中华结核和呼吸杂志, 2015, 38.
21. Chou YY, Hsieh YS, Wu CC, et al. Epithelioid hemangioendotheliomas of the lung and pleura:report of three cases. Formosan J Surg, 2011, 44:163-167.
22. Pinet C, Magnan A, Garbe L, et al. Aggressive form of pleural epithelioid haemangioendothelioma:complete response after chemotherapy. Eur Respir J, 1999, 14:237-238.
23. Bahrami A, Allen TC, Cagle PT. Pulmonary epithelioid hemangioendothelioma mimicking mesothelioma. Pathol Int, 2008, 58:730-734.
24. 陈骏, 丁洁, 贾支俊, 等.肝脏上皮样血管内皮瘤临床病理观察.临床与实验病理学杂志, 2011, 27:234-238.
25. Fujii T, Zen Y, Sato Y, et al. Podoplanin is a useful diagnostic marker for epithelioid hemangioendothelioma of the liver. Mod Pathol, 2008, 21:125-130.
26. Liu JX, Shiau MC, Nonaka D. An 80-year-old man with shortness of breath and large right-sided pleural effusion. Chest, 2010, 138:1247-1252.
27. Martínez D, Iturbe J, Buzeki R, et al. Hemangioendotelioma epitelioide de pleura. Presentacion de un caso con hemotorax. Rev Argent Med Respir, 2008, 8:73-76.
28. André ST, Valente C, Paiva B, et al. Epithelioid hemangioendothelioma of the pleura-A rare presentation of a clinical case. Rev Port Pneumol, 2009, 16:477-482.
29. Bansal A, Chawla M, Cohen PJ, et al. Pleural epithelioid hemangioendothelioma. Lung, 2012, 190:469-470.
30. Knuuttila A, Jee KJ, Taskinen E, et al. Spindle cell tumours of the pleura:a clinical, histological and comparative genomic hybridization analysis of 14 cases. Virchows Arch, 2006, 448:135-141.
31. Yousem SA, Hochholzer L. Unusual thoracic manifestations of epithelioid hemangioendothelioma. Arch Pathol Lab Med, 1987, 111:459-463.
32. Ledson MJ, Convery R, Carty A, et al. Epithelioid haemangioendothelioma. Thorax, 1999, 54:560-561.

Chinese Journal of Respiratory and Critical Care Medicine

胸膜上皮样血管内皮细胞瘤一例并文献复习

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