腎上腺吻合狀血管瘤3例報告并文獻復習

Report and literature review of three cases of adrenal anastomotic hemangioma

ZHANG Changsheng1 ，HU Jing2，ZHANG Jinsong1 ，LIU Qi1 ，YUAN Huixing3，XU Hao2，QIN Baolong？ （1. Department of Urology，2. Department of Anesthesiology，Huanggang Central Hospital，Huanggang 438000；3. Department of Urology，Tongji Hospital，Tongji Medical Collge of Huazhong University of Science and Technology，Wuhan ，China）

ABSTRACT：ObjectiveTosummarizeandanalyze theclinical features，maging manifestations，pathologicalcharacteristics， treatmentapproaches，andprognosisofadrenalanastomotichemangioma（AH），soas toenhance theclinical understandingof this disease.MethodsAretrospective analysis was conductedon3adrenal AHpatients treatedin Tongji Hospitalduring Jan. 2012 and Dec.2024.Relevant literature was reviewed.ResultsAll patients were male，aged 34-56 years，with a maximum tumor diameter of 2.0-5.0cm. All tumors were solitary.No patients presented with special clinical manifestations.Biochemical tests indicated tumors withno endocrine function.Contrast-enhanced computed tomography showed circular mixed density shadows with unevenedge enhancement.Twopatientsunderwent laparoscopic partial adrenal resection，and one patient receivedrobot-asisted laparoscopicpartialadrenal resection.Postoperative pathologicalresultsofallthreecases idicated AH. Immunohistochemistry showed CD31 （+） ，CD34（+），and ERG （+） . No recurrence was found in a postoperative follow-up of 1-45 months.A totalof 22 cases of adrenal AH were reported in the literature and our center，with onset age ranging from 34 to 83 years.The patients included 17 males and 5 females，with a maximum tumor diameter of 0.6-8.8cm .Among them， 2 cases were multifocaland the tumors were located inthe kidneys，beside kidneys，orintheinferiorvenacava.Twocases showed clinical manifestatiosof lower back pain.During the follow-upof 156 months，norecurrence or metastasis was observed.ConclusionAdrenal AH is a rare benign tumor，which mainly occurs in midle-aged and elderly people with no clinical symptoms and endocrine function.Imaging caneasilymistakeitforpheochromocytoma or welldifferentiated vascular sarcoma，nd thediagnosis mainlyrelies onpathological examination.Itshould be distinguished from pheochromocytomaand well differentiated vascular sarcoma.Surgery is the main treatment method and the prognosis is good.

KEY WORDS：adrenal tumor; anastomotic hemangioma; resection of adrenal tumor

中圖分類號：R699.3 文獻標志碼：A DOI：10.3969/j.issn.1009-8291.2025.08.011

吻合狀血管瘤（anastomotichemangioma，AH）又稱交織狀血管瘤，由MONTGOMERY等[1］于2009年首次報道并命名，在2016年世界衛生組織的泌尿系統和男性生殖器官腫瘤分類中，AH已被正式確定為一種獨特的血管瘤亞型，其好發于泌尿生殖系統，其次是腹膜后和椎旁軟組織區域。組織學上AH呈邊界清楚的竇隙樣、篩狀、簇狀、互相吻合的血管網，局灶內襯鞋釘樣內皮細胞2。腎上腺AH于2012年被首次報道[3]，在影像學及病理學上易與嗜鉻細胞瘤、高分化血管肉瘤混淆。截至目前，腎上腺AH在英文文獻中僅有19例報道[4]，國內文獻有5例[5-7]。本研究對2012年1月—2024年12月華中科技大學同濟醫學院附屬同濟醫院（下文簡稱“本院\"收治的3例腎上腺AH患者的臨床資料進行總結報告并行文獻復習，以期提高臨床對腎上腺AH的認識。

1資料與方法

1.1病例資料患者1男性，56歲，身體質量指數（bodymassindex，BMI）27.7，因\"檢查發現右側腎上腺腫物1d\"收入本院?；颊邿o明顯臨床癥狀，增強計算機斷層掃描（computerizedtomography，CT）提示右側腎上腺有一直徑約 20mm 的結節，呈漸進性不均勻強化（圖1A）。初步考慮右側腎上腺腺瘤或其他腫瘤性病變可能。人院后查血促腎上腺皮質激素（adrenocorticotropic hormone，ACTH）、腎素/醛固酮（直立位、仰臥位）、皮質醇 （8am，4pm） 、間甲腎上腺素及去甲變腎上腺素未見異常； 24h 尿17-羥皮質類固醇（17-hydroxycorticosteroid，17-OH）、17-酮皮質類固醇（17-ketosteroide，17-KS）、香草扁桃酸（vanillymandelicacid，VMA）、腎上腺素、去甲腎上腺素及多巴胺未見異常。遂行腹腔鏡下右側腎上腺腫物切除術，手術順利，術后病理示腎上腺血管瘤（主呈AH構象）。免疫組化檢查示：CD31（十）、CD34（+） 、ERG（ + ）、 D2-40（+） 、SMA （+） 、Ki-67（LI約2%～3% ）、MPO（小灶 + ）（圖2）。術后12個月門診復查CT，未見腫瘤復發及轉移，之后失訪?！?br>