1例复杂静脉畸形的靶向治疗

doi:10.3969/j.issn.1673-0364.2020.03.006

组织工程与重建外科杂志 ›› 2020, Vol. 16 ›› Issue (3): 198-200.doi: 10.3969/j.issn.1673-0364.2020.03.006

1例复杂静脉畸形的靶向治疗

刘泓源, 杨希, 顾豪, 胡丽, 孙怡, 陈辉(), 林晓曦()

2000011 上海市上海交通大学医学院附属第九人民医院整复外科

收稿日期:2020-04-16 修回日期:2020-04-29 出版日期:2020-07-27 发布日期:2020-06-26
基金资助:
上海市申康临床技能与临床创新三年行动计划(16CR1007A);上海交通大学医学院附属第九人民医院临床研究助推计划(“临+计划”JYLJ001);上海交通大学医学院附属第九人民医院生物样本库专项基金(YBKA201902);上海交通大学医学院附属第九人民医院临床研究型MDT项目(201701001)

Case Report: Clinical Attempt of Targeted Therapy for Complex Venous Malformations

Hongyuan LIU, Xi YANG, Hao GU, Li HU, Yi SUN, hui CHEN(), Xiaoxi LIN()

Department of Plastic and Reconstructive Surgery, Shanghai Ninth People's Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai 200011, China

Received:2020-04-16 Revised:2020-04-29 Online:2020-07-27 Published:2020-06-26

摘要/Abstract

摘要：

目前对于复杂静脉畸形的治疗尚未达成共识。近年来,应用西罗莫司成功治疗复杂的脉管畸形的个案报道较多。本文报道1例右上肢及躯干部复杂静脉畸形患者,口服西罗莫司治疗2年后病灶显著消退,停药后随访1年未见明显复发。

关键词: 静脉畸形, 西罗莫司, 脉管畸形, 靶向治疗

Abstract: The management of severe and complicated venous malformation is still not standardized currently. Sirolimus has been reported to successfully treat complicated vascular anomalies in many case reports and clinical trials recently. One case of extremely severe and complicated venous malformation of right upper limb and trunk was described. After 2 years of oral treatment with sirolimus, the lesion was significantly resolved, and no obvious recurrence was found after 1 year of follow-up.

Key words: Venous malformation, Sirolimus, Vascular malformation, Target therapy

中图分类号:

R732.2

刘泓源, 杨希, 顾豪, 胡丽, 孙怡, 陈辉, 林晓曦. 1例复杂静脉畸形的靶向治疗[J]. 组织工程与重建外科杂志, 2020, 16(3): 198-200.

Hongyuan LIU, Xi YANG, Hao GU, Li HU, Yi SUN, hui CHEN, Xiaoxi LIN. Case Report: Clinical Attempt of Targeted Therapy for Complex Venous Malformations[J]. Journal of Tissue Engineering and Reconstructive Surgery, 2020, 16(3): 198-200.

图/表 3

参考文献 13

[1]	中华医学会整形外科分会血管瘤和脉管畸形学组. 血管瘤和脉管畸形的诊断及治疗指南(2019版)[J]. 组织工程与重建外科杂志, 2019,15(5):277-317.
[2]	Freixo C, Ferreira V, Martins J , et al. Efficacy and safety of sirolimus in the treatment of vascular anomalies: A systematic review[J]. J Vasc Surg, 2020,71(1):318-327.
[3]	Soblet J, Kangas J, Natynki M , et al. Blue rubber bleb nevus (BRBN) syndrome is caused by somatic TEK (TIE2) mutations[J]. J Invest Dermatol, 2017,137(1):207-216.
[4]	Castel P, Carmona FJ, Grego-Bessa J , et al. Somatic PIK3CA mutations as a driver of sporadic venous malformations[J]. Sci Transl Med, 2016, 8(332):332ra42.
[5]	Adams DM, Trenor CC 3rd, Hammill AM, et al. Efficacy and safety of sirolimus in the treatment of complicated vascular anomalies[J]. Pediatrics, 2016,137(2):e20153257.
[6]	Hammer J, Seront E, Duez S , et al. Sirolimus is efficacious in treatment for extensive and/or complex slow-flow vascular malformations: a monocentric prospective phase II study[J]. Orphanet J Rare Dis, 2018,13(1):191.
[7]	Hammill AM, Wentzel M, Gupta A , et al. Sirolimus for the treatment of complicated vascular anomalies in children[J]. Pediatr Blood Cancer, 2011,57(6):1018-1024.
[8]	Lackner H, Karastaneva A, Schwinger W , et al. Sirolimus for the treatment of children with various complicated vascular anomalies[J]. Eur J Pediatr, 2015,174(12):1579-1584.
[9]	Morelon E, Stern M, Israel-Biet D , et al. Characteristics of sirolimus-associated interstitial pneumonitis in renal transplant patients[J]. Transplantation, 2001,72(5):787-790.
[10]	Russell TB, Rinker EK, Dillingham CS , et al. Pneumocystis jirovecii pneumonia during sirolimus therapy for kaposiform hemangioendo the lioma[J]. Pediatrics, 2018,141(Suppl 5):S421-S424.
[11]	Fujii T, Shimono R, Tanaka A , et al. A case of suspected adverse reactions to sirolimus in the treatment of generalized lymphatic anomaly[J]. Case Rep Pediatr, 2019,2019:3101357.
[12]	Ying H, Qiao C, Yang X , et al. A case report of 2 sirolimus-related deaths among infants with kaposiform hemangioendotheliomas[J]. Pediatrics, 2018,141(Suppl 5):S425-S429.
[13]	Goldenberg DC, Carvas M, Adams D , et al. Successful treatment of a complex vascular malformation with sirolimus and surgical resection[J]. J Pediatr Hematol Oncol, 2017,39(4):e191-e195.

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1例复杂静脉畸形的靶向治疗

Case Report: Clinical Attempt of Targeted Therapy for Complex Venous Malformations

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