肾上腺旁吻合状血管瘤1例并文献复习

doi:10.16150/j.1671-2870.2022.06.12

摘要/Abstract

摘要：

目的：报道1例肾上腺旁吻合状血管瘤（anastomosing hemangioma, AH）患者，并复习相关文献，对AH的临床病理特征、诊断及鉴别诊断要点进行总结。方法：收集本例AH患者的临床资料，观察其组织学形态及免疫组化特点，并结合相关文献进行综合分析。结果：大体检查见肿瘤位于肾上腺旁脂肪组织内，边界清楚，切面呈灰白、灰红色，质地中等。显微镜下可见肿瘤由相互吻合的窦隙样管腔构成，管腔内衬靴钉样内皮细胞，少数上皮伴有轻度不典型性，可见纤维蛋白性血栓，个别细胞质内可见嗜酸性小体。免疫组化检测示，肿瘤细胞表达CD31和CD34，不表达D2-40、CK5/6、calretinin、CKpan、EMA、Melan-A和HMB45，Ki-67增殖指数约为5%。最终该患者经病理诊断为（肾上腺旁）AH。结果：本文报道1例AH患者，其为罕见的良性血管源性肿瘤，因其血管高度吻合、管腔衬覆的靴钉样内皮细胞常伴不典型的特点，需要与高分化血管肉瘤相区别；泌尿生殖系统的AH还需与血管平滑肌脂肪瘤、腺瘤样瘤、继发于肾细胞癌的血管瘤样增生等疾病相鉴别。

关键词: 肾上腺, 吻合状血管瘤, 免疫表型

Abstract:

Objective: This paper reported 1 case of paradrenal anastomosing hemangioma (AH), and to summarize the clinicopathological features, diagnosis and differential diagnosis of AH. Methods: The clinical data of this patient was collected, the histological morphology and immunohistochemical characteristics were observed and analyzed in combination with relevant literature. Results: The tumor was located in the adipose tissue around the adrenal gland, it was well circumscribed, gray and red colored. Microscopically, the tumor was composed of irregularly anastomosing sinusoidal-like spaces lined by hobnail endothelial cells, focal mild atypia was observed. Intravascular fibrin thrombi and hyaline globules could be seen. Immunohistochemical results showed diffuse staining for endothelial markers, including CD31 and CD34. D2-40, CK5/6, calretinin, CKpan, EMA, Melan-A and HMB45 immunostains were negative. Ki-67 staining indicated a low proliferative activity. Eventually this patient was diagnosed with AH. Conclusions: AH is a rare but distinct type of vascular neoplasm. Owing to its striking anastomosing appearance, it needs to be distinguished from well differentiated angiosarcoma. In addition, the differential diagnosis of urogenital AH also includes angiomyolipoma, adenomatoid tumor, and hemangiomatoid hyperplasia secondary to renal cell carcinoma.

Key words: Adrenal gland, Anastomosing hemangioma, Immunohistochemical feature

中图分类号:

R692.39

闫化茹, 刘坤, 韩志宏, 房振, 吴丽莉. 肾上腺旁吻合状血管瘤1例并文献复习[J]. 诊断学理论与实践, 2022, 21(06): 735-740.

YAN Huaru, LIU Kun, HAN Zhihong, FANG Zhen, WU Lili. Paradrenal anastomosing hemangioma: a case report with literature review[J]. Journal of Diagnostics Concepts & Practice, 2022, 21(06): 735-740.

图/表 4

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图2

表1

94例AH临床特征

文献	例数（n）	性别	年龄（岁）	部位	最大径（cm）	术前诊断	处理	随访
O′Neill AC et al^[15]	32	18男 14女	17~81	腹膜后；肾脏；主动脉旁；肾旁；卵巢；肝脏；睾丸；肾上腺；肠系膜；精索	不详	肾上腺嗜铬细胞瘤；肠系膜脂肪肉瘤；肾癌；腹膜后脂肪肉瘤；淋巴结伴癌转移；其余未知	肿块切除术；粗针穿刺活检	4例2~26个月无复发或转移。其余失访。
John I，et al^[14]	16	10男 6女	2~85	主动脉旁；椎体；腰大肌；胸椎旁；脊椎旁；腹膜后；纵隔漏斗状骨盆韧带；子宫角；上臂；下腔静脉旁和右主动脉旁	1.5~6.7 4例不详	肉瘤；周围神经鞘瘤和淋巴瘤；其余未知	9例肿块切除术；7例粗针穿刺活检	10例1~46个月无复发转移或疾病进展。其余失访。
Bean GR, et al^[19]	13	9男 4女	39~70	肾；肾上腺；脊柱；椎旁；下腔静脉旁；腹膜；卵巢；附件旁；子宫肌层；肝脏	0.7~8.0	未知	肿块切除术	10例1~107个月无复发或转移。其余失访。
Kryvenko ON， et al^[17]	7	1男 7女	39~77	肾（其中一例为双侧）；卵巢	0.6~5.0	卵巢子宫内膜样癌；卵巢浆液囊肿；卵巢浆液性囊腺瘤；其余因其它疾病行CT检查发现肾脏肿块	肿块切除术	10例随访3~122个月，无复发或转移。其余失访。
Montgomery E, et al^[1]	6	4男 2女	49~75	肾；睾丸	1.3~2.0	未知	3例肾切除术，1例肿块切除术，2例睾丸切除术	5例8~36个月无复发或转移。1例失访。
Lin J, et al^[4]	6	2男 4女	46~71	肝；结肠小肠	0.2~6.0	3血管瘤；3血管肉瘤	肿块切除术	5例8~96个月无复发或转移。1例失访。
赵明等^[9]	5	3男 2女	47~77	肾上腺；肾	1.6~2.5	肝癌肾转移；肾错构瘤；肾上腺嗜铬细胞瘤或皮质腺瘤	1例部分肾切除术，1例根治性肾切除术；3例肾上腺肿瘤切除术	5例6~52个月无复发或转移。
Burton KR, et al^[16]	1	男	68	多发：肾旁、下腔静脉旁、髂内、双侧肾上腺	1.5~3.7	肾恶性肿瘤	左肾上腺切除术、左肾周和下腔静脉旁结节切除术	未知
Huang ZY, et al^[10]	1	男	37	鼻腔	2.3	鼻息肉、乳头状瘤	肿块切除术	22个月无复发转移。
Ross M, et al^[2]	1	男	49	肾上腺	2.0	肾上腺肿瘤	肾上腺切除术	未知
Rathore K, et al^[11]	1	男	64	左心房	4.0	左心房壁肿瘤	肿块切除术	未知
姚建国等^[6]	1	男	71	肾上腺	1.7	右肾上腺肿瘤	右肾上腺切除术	10个月无复发转移。
虞义建等^[7]	1	男	50	肾上腺	1.1	嗜铬细胞瘤	右肾上腺切除术	未知
王小刚等^[8]	1	男	55	肾	4.0	左肾占位	根治性左肾切除术	12个月无复发转移。
党琳琳等^[5]	1	女	44	肝脏	5.0	富于血供的肿瘤	肿块切除术	2个月无复发转移。

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参考文献 22

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第一作者	例数（n）	边界	吻合的小血管	靴钉样内皮细胞	细胞不典型性	核分裂	CD31/ CD34	微小血栓	嗜酸性小体	髓外造血	变性
John I, et al^[14]	16	1/16局灶不清	16/16+	16/16+	轻	少-无	+	16/16+	未知	7/16+	未知
Bean GR, et al^[15]	13	2/13局灶不清	13/13+	6/13+	轻-无	少-无	未知	13/13+	12/13+	7/13+	7/13+
Kryvenko ON, et al^[17]	7	清楚	7/7+	3/7+	无	无	+	未知	6/7+	3/17+	4/7+
Montgomery E, et al^[1]	6	3/6局灶浸润	6/6+	6/6+	轻-无	少-无	+	5/6+	2/6+	2/6+	5/6+
Lin J, et al^[4]	6	清楚	6/6+	6/6+	轻	无	3/3+	4/6+	1/6+	1/6+	未知
赵明等^[9]	5	清楚	5/5+	5/5+	无	少-无	+	+	1/5+	2/5+	5/5+
Huang ZY, et al^[10]	1	清楚	+	+	轻	无	+	未知	未知	无	未知
姚建国等^[6]	1	清楚	+	+	轻	未知	+	+	未知	未知+	+
虞义建等^[7]	1	清楚	+	+	无	无	+	+	未知	未知	未知
党琳琳等^[5]	1	欠清楚	+	+	轻	无	+	+	未知	未知	+