肝淋巴管瘤1例报道

doi:10.3969/j.issn.1009-976X.2022.03.015

摘要/Abstract

摘要： 目的探究此罕见肝脏肿瘤的特征,提高对本病的认识,提高诊断准确率,探索本疾病的规范诊疗方法。方法回顾性分析我中心1例采用解剖性肝段切除的肝脏淋巴管瘤的术前临床资料,术后免疫组化标记物Ki67、CD31、CD34、CK19等评判此例肿瘤良恶性,术后复查血清学标记物、肝脏彩超、CT、MRI等评估患者的预后及肿瘤复发情况。结果此例患者入院血常规、生化检查及肿瘤学标志物均在正常范围,无乙肝病史,术前诊断为肝S6段囊腺瘤,采用肝S6段肝肿瘤切除+胆囊切除术,术后免疫组化显示CK（-）,CD34（+）,CD31（+）,Ki67（<1%）,考虑为淋巴管瘤可能性大;术后快速康复,无手术相关并发症的发生。截止2022.3.17,规律随访13个月,未见肿瘤复发。结论术后病理结合免疫组化结果显示此例肝淋巴管瘤为良性肿瘤,整个肝段切除为治疗有效手段,近期预后良好,患者的中远期疗效需增加样本量并长期随访。

关键词: 肝淋巴管瘤, 诊断, 治疗, 体层摄影术, X线计算机, 超声检查, 病理学

Abstract: Objective To explore the characteristics of this rare liver tumor, improve the understanding of this disease, improve the diagnostic accuracy, and explore the standardized diagnosis and treatment methods of this disease. Methods The preoperative clinical data of one case of hepatic lymphangioma with anatomical segmentectomy in our center were retrospectively analyzed. The postoperative immunohistochemical markers Ki67, CD31, CD34 and CK19 were used to evaluate the benign or malignant tumor of this case. The postoperative serological markers, liver ultrasound, CT and MRI were reviewed to evaluate the prognosis and tumor recurrence of the patient, and to explore the curative effect of the treatment method. Results The blood routine examination, biochemical examination and tumor markers of this patient were all within the normal range, and there was no history of hepatitis B. The patient was diagnosed as hepatic S6 cystadenoma before operation, and was treated with hepatic S6 tumor resection + cholecystectomy. The postoperative immunohistochemistry showed CK （-）, CD34 （+）, CD31 （+）, Ki67, which were considered as lymphangioma. The patient recovered quickly after surgery, and no surgery-related complications occurred. As of 2022.3.17 regular follow-up was 13 months, no tumor recurrence. Conclusion Postoperative pathology combined with immunohistochemical results show that this case of hepatic lymphangioma is a benign tumor, and total resection of hepatic segment is an effective treatment. The short-term prognosis is good. The medium and long-term efficacy of the patient needs to increase the sample size and long-term follow-up.

Key words: hepatic lymphangioma, diagnosis, treatment, body photography, X-ray computer, ultrasonic examination, pathology

中图分类号:

R735.7

汤永生, 鲁旭, 李光辉, 贾昌昌, 李华. 肝淋巴管瘤1例报道[J]. 岭南现代临床外科, 2022, 22(03): 292-296.

TANG Yong-sheng, LU Xu, LI Guang-hui, JIA Chang-chang, LI Hua. A case report of hepatic lymphangioma[J]. Lingnan Modern Clinics In Surgery, 2022, 22(03): 292-296.

参考文献

[1] Godart S.Embryological significance of lymphangioma[J]. Arch Dis Child, 1966, 41(216): 204-206.
[2] Wigle JT, Oliver G.Prox1 function is required for the development of the murine lymphatic system[J]. Cell, 1999, 98(6): 769-778.
[3] Zhang YZ, Ye YS, Tian L, Li W.Rare case of a solitary huge hepatic cystic lymphangioma[J]. World J Clin Cases, 2013, 1(4): 152-154.
[4] Matsumoto T, Ojima H, Akishima-Fukasawa Y, et al.Solitary hepatic lymphangioma: report of a case[J]. Surg Today, 2010, 40(9): 883-889.
[5] Long X, Zhang L, Cheng Q, et al.Solitary hepatic lymphangioma mimicking liver malignancy: A case report and literature review[J]. World J Clin Cases, 2020. 8(19): 4633-4643.
[6] Huang L, Li J, Zhou F, et al.Giant cystic lymphangioma of the liver[J]. Hepatol Int, 2010, 4(4): 784-787.
[7] Liu Q, Sui CJ, Li BS, et al.Solitary hepatic lymphangioma: a one-case report[J]. Springerplus, 2014, 3: 314.
[8] Koh CC, Sheu JC.Hepatic lymphangioma--a case report[J]. Pediatr Surg Int, 2000, 16(7): 515-516.
[9] Jiménez-Rivera C, Avitzur Y, Fecteau AH, et al.Sirolimus for pediatric liver transplant recipients with post-transplant lymphoproliferative disease and hepatoblastoma[J]. Pediatr Transplant, 2004, 8(3): 243-248.
[10] Ooi CY, Brody D, Wong R, et al., Liver transplantation for massive hepatic lymphangiomatosis in a child[J]. J Pediatr Gastroenterol Nutr, 2011, 52(3): 366-369.
[11] Alqahtani A, Nguyen LT, Flageole H, et al.25 years′ experience with lymphangiomas in children[J]. J Pediatr Surg, 1999, 34(7): 1164-1168.
[12] 陈创奇, 詹文华, 汪建平, 等.腹部淋巴管瘤的临床特点及其诊断与治疗[J]. 中国实用外科杂志, 2000, 20(7): 421-422.
[13] Stavropoulos M, Vagianos C, Scopa CD, et al.Solitary hepatic lymphangioma. A rare benign tumour: a case report[J]. HPB Surg, 1994, 8(1): 33-336.
[14] Nakano T, Hara Y, Shirokawa M, et al. Hemorrhagic giant cystic lymphangioma of the liver in an adult female [J]. J Surg Case Rep, 2015, 2015(4): rjv033.
[15] Nzegwu MA, Ekenze SO, Okafor OC, et al.Solitary hepatic lymphangioma in an infant[J]. J Perinat Med, 2007, 35(2): 164-165.
[16] Zeidan S, Delarue A.Uncommon etiology of a cystic hepatic tumor[J]. Dig Surg, 2008, 25(1): 10-11.
[17] Shahi KS, Geeta B, Rajput P.Rajput, Solitary hepatic lymphangioma in a 22-day-old infant[J]. J Pediatr Surg, 2009, 44(8): E9-11.
[18] Zouari M, Ben Dhaou M.Solitary hepatic lymphangioma in an 8-month-old child[J]. Pan Afr Med J, 2015, 20: 440.
[19] Chan SC, Huang SF, Lee WC, Wan YL.Solitary hepatic lymphangioma--a case report[J]. Int J Clin Pract Suppl, 2005, 147: 100-102.
[20] 陈玉芳, 刘红, 程红岩, 杨永波.肝脏淋巴管瘤的MRI表现并文献复习[J]. 中华放射学杂志, 2018, 52(4): 300-313.
[21] 陈玉芳, 刘红, 程红岩,杨永波. 单发肝脏海绵状淋巴管瘤1例[J]. 中国介入影像与治疗学, 2017, 14(11): 714.
[22] Chung JH, Suh YL, Park IA, et al.A pathologic study of abdominal lymphangiomas[J]. J Korean Med Sci, 1999, 14(3): 257-62.
[23] Galambos C, Nodit L.Identification of lymphatic endothelium in pediatric vascular tumors and malformations[J]. Pediatr Dev Pathol, 2005, 8(2): 181-189.
[24] 杨永光, 刘丽娟, 谌力群, 等.肝囊性淋巴管瘤2例报告并国内文献回顾[J]. 中国普通外科杂志, 2018, 27(1): 101-106.
[25] Lee A, Rajanayagam J, Sarikwal A, et al.Hepatobiliary and pancreatic: liver transplantation for massive hepatic lymphangiomatosis[J]. J Gastroenterol Hepatol, 2014, 29(2): 233.
[26] Miller C, Mazzaferro V, Makowka L, et al.Orthotopic liver transplantation for massive hepatic lymphangiomatosis[J]. Surgery, 1988, 103(4): 490-495.
[27] Stunell H, Ridgway PF, Torreggiani WC, et al.Hepatic lymphangioma: a rare cause of abdominal pain in a 30-year-old female[J]. Ir J Med Sci, 2009, 178(1): 93-96.
[28] Kochin IN, Miloh TA, Arnon R, et al.Benign liver masses and lesions in children: 53 cases over 12 years[J]. Isr Med Assoc J, 2011, 13(9): 542-547.
[29] Kohashi T, Itamoto T, Matsugu Y, et al.An adult case of lymphangioma of the hepatoduodenal ligament mimicking a hepatic cyst[J]. Surg Case Rep, 2017, 3(1): 2.
[30] Datz C, Graziadei IW, Dietze O, et al.Massive progression of diffuse hepatic lymphangiomatosis after liver resection and rapid deterioration after liver transplantation[J]. Am J Gastroenterol, 2001, 96(4): 1278-1281.
[31] Ra SH, Bradley RF, Fishbein MC, et al.Recurrent hepatic lymphangiomatosis after orthotopic liver transplantation[J]. Liver Transpl, 2007, 13(11): 1593-1597.
[32] 孙启鑫, 张颖, 曾军, 朱志刚. 肝淋巴管瘤病合并血性乳糜胸1例[J]. 广东医学, 2009, 30(3): 496.
[33] 吴少平,许文萍,张新,等. 肝囊肿发病机制的研究进展[J].国际消化病杂志,2020,40(1):12-15.
[34] 石建果,丁雄.单纯性肝囊肿的诊断与治疗现状[J].世界最新医学信息文摘(连续型电子期刊),2021,21(15):107-108,111.
[35] 童心,邓剑波,沈莉. 先天性多囊肝、多囊肾CT影像学特征及病理对比分析[J]. 中国CT和MRI杂志,2019,17(9):107-109.