肾上腺血管淋巴管瘤8例诊治分析并文献复习

Clinical analysis of 8 cases of adrenal hemolymphangioma and literature review

摘要

目的 探讨肾上腺血管淋巴管瘤的临床病理特征和手术治疗效果，为临床认识该疾病提供参考。方法 回顾性分析2013年1月-2022年12月北京大学第三医院泌尿外科收治的8例肾上腺血管淋巴管瘤患者的临床、病理资料并复习相关文献。结果 患者中男性5例，女性3例，中位年龄54（25～75）岁。所有患者均经体检发现，无明显症状，其中4例有高血压病史。肾上腺功能学检查结果无明显异常，影像学检查共查出肿瘤9处，1例单侧多发2处瘤灶、7例单侧单发，中位肿瘤直径3.6（1.0～5.4）cm。7例行后腹腔镜肾上腺肿瘤切除术，1例行机器人辅助腹腔镜肾上腺肿瘤切除术，均完整切除肿瘤。中位手术时间77（53～115）min、术中中位失血量7.5（2.0～20.0）mL、术后中位住院时间4（1～7）d。术后病理显示：囊性肿瘤内畸形扩张的血管和淋巴管相互交织，内含大量淋巴液、淋巴细胞和红细胞，管壁间可见慢性淋巴细胞浸润；囊腔部分相通，内衬以扁平内皮细胞，病理诊断为肾上腺血管淋巴管瘤。8例中位随访时间53.5（12.0～106.8）个月，术后患者恢复良好，血压稳定，未见肿瘤复发或转移。结论 肾上腺血管淋巴管瘤多无特异性临床症状，肾上腺功能学检测亦无明显异常，确诊需依靠病理学检查。采用后腹腔镜/机器人辅助腹腔镜肾上腺肿瘤切除术疗效确切且预后良好。

Abstract

Objective To investigate the clinicopathological characteristics and surgical outcomes of adrenal hemolymphangioma, so as to enhance the understanding of this disease. Methods Clinical and pathological data of 8 patients with adrenal hemolymphangioma admitted to the Department of Urology of our hospital during Jan. 2013 and Dec. 2022 were retrospectively analyzed, and relevant literature was reviewed. Results The patients included 5 males and 3 female, median age 54 (25-75) years. Adrenal hemolymphangioma was detected in all patients in physical examinations without obvious symptoms. And 4 of the patients had a history of hypertension. Adrenal function test results showed no abnormalities. A total of 9 tumors were identified by imaging examination, including 1 unilateral multiple and 7 unilateral solitary tumors, with a median diameter of 3.6 (1.0-5.4) cm. Posterior laparoscopic adrenal tumor resection was performed in 7 cases and robot-assisted laparoscopic adrenal tumor resection in 1 case; all of the tumors were completely removed. The median operation time was 77 (53-115) min, median intraoperative blood loss 7.5 (2.0-20.0) mL, and median postoperative hospital stay 4 (1-7) d. Postoperative pathology showed interwoven deformed and dilated blood vessels and lymphatic vessels in the cystic tumors, with a large amount of lymphoid fluid, lymphocytes and red blood cells. Chronic lymphocyte infiltration was visible between the tube walls. The cystic cavity was partially connected, with flat endothelial cells lining. The pathological diagnosis was adrenal hemolymphangioma. During the median follow-up of 53.5 (12.0-106.8) months, all patients recovered well, with stable blood pressure and no tumor recurrence or metastasis. Conclusion Adrenal hemolymphangioma has no specific clinical symptoms. As adrenal functional tests show no obvious abnormalities, the diagnosis depends on pathological examinations. Posterior laparoscopic or robot-assisted laparoscopic resection has good efficacy and prognosis.

关键词

肾上腺肿瘤 / 血管淋巴管瘤 / 后腹腔镜肾上腺肿瘤切除术 / 机器人辅助腹腔镜肾上腺肿瘤切除术

Key words

adrenal tumors / hemolymphangioma / posterior laparoscopic adrenal tumor resection / robot-assisted laparoscopic adrenal tumor resection

参考文献

[1] MAO CP, JIN YF, YANG QX, et al. Radiographic findings of hemolymphangioma in four patients: a case report[J]. Oncol Lett, 2018, 15(1): 69-74.

[2] OHSAWA M, KOHASHI T, HIHARA J, et al. A rare case of retroperitoneal hemolymphangioma[J]. Int J Surg Case Rep, 2018, 51: 107-111.

[3] 马闰卓， 邱敏， 何为， 等. 原发性腹膜后畸胎瘤10例诊治分析并文献复习[J]. 北京大学学报（医学版）， 2019， 51（2）： 369-371.

[4] 吴芝莹， 刘承， 张树栋， 等. 机器人辅助腹腔镜治疗复杂嗜铬细胞瘤和副神经节瘤手术技巧及初步结果（附16例报道）[J]. 中国微创外科杂志， 2022， 22（10）： 783-787.

[5] 邱敏， 赵中和， 卢剑， 等. 肾上腺脉管瘤的诊治探讨[J]. 北京大学学报（医学版）， 2016， 48（5）： 923-925.

[6] 王建花， 马莉， 孙鹏飞. 左肾上腺血管淋巴管瘤1例[J]. 中国医学影像技术， 2017， 33（8）： 1245.

[7] 卢艳玉， 詹阿来. 血管淋巴管瘤的CT及MRI诊断[J]. 中国CT和MRI杂志， 2010， 8（6）： 51-53.

[8] 尤宗昊， 李庆文， 汪盛. 肾上腺血管淋巴管瘤1例并文献复习[J]. 临床泌尿外科杂志， 2015， 30（12）： 1089-1091， 1095.

[9] ZHANG DY, LU Z, MA X, et al. Multiple hemolymphangioma of the visceral organs: a case report and review of the literature[J]. Medicine (Baltimore), 2015, 94(27): e1126.

[10] 邹红群. 血管淋巴管瘤的影像征象及病理特征探讨[J]. 中外医疗， 2020， 39（8）： 193-195.

[11] 蒋健， 夏加增， 杜旭东， 等. 腹部血管淋巴管瘤7例及文献复习49例临床诊治分析[J]. 中国现代普通外科进展， 2014， 17（11）： 914-917.

[12] 王兆海， 韩宝三， 彭承宏. 腹部淋巴血管瘤九例诊治分析[J]. 中华外科杂志， 2007， 45（11）： 788-789.

[13] 王昆， 罗宇婷， 潘迪， 等. 腹膜后巨大混合性脉管瘤1例报告[J]. 现代泌尿外科杂志， 2023， 28（10）： 916-917.

[14] KALOF AN, COOPER K. D2-40 immunohistochemistry-so far! [J]. Adv Anat Pathol, 2009, 16(1): 62-64.

[15] ORDÓÑEZ NG. Immunohistochemical endothelial markers: a review[J]. Adv Anat Pathol, 2012, 19(5): 281-295.

[16] 王君， 刘勇. 腹膜后囊状脉管瘤1例报道[J]. 中国普外基础与临床杂志， 2010， 17（10）： 1113.

[17] WANG H, CAO J, HUANG H, et al. Contrast-enhanced ultrasound-based Bosniak classification for evaluating of a cystic renal mass: a rare case description of renal hemolymphangioma[J]. Quant Imaging Med Surg, 2023, 13(2): 1221-1226.

[18] 曹鹏， 姜永光， 邢念增， 等. 机器人辅助后腹腔镜肾上腺切除术与传统后腹腔镜肾上腺切除术的疗效对比分析[J]. 临床泌尿外科杂志， 2018， 33（10）： 780-784.

[19] 谢豪豪， 潘铁军， 涂忠， 等. 机器人辅助腹腔镜与传统腹腔镜治疗肾上腺肿瘤的疗效对比分析[J]. 联勤军事医学， 2024， 38（1）： 49-52.

[20] TOYOKI Y, HAKAMADA K, NARUMI S, et al. A case of invasive hemolymphangioma of the pancreas[J]. World J Gastroenterol, 2008, 14(18): 2932-2934.

[21] ZHANG Z, KE Q, XIA W, et al. An invasive hemolymphangioma of the pancreas in a young woman[J]. Comb Chem High Throughput Screen, 2018, 21(10): 798-800.