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小儿下肢疣状静脉畸形罕见病例:治疗方法及注意事项

Rare Case of Pediatric Lower Limb Verrucous Venous Malformation: Treatment Approach and Considerations.

作者信息

Chen-Carrington Annie, Goodreau Adam, Strife Brian J, Shinn Laurie, Rhodes Jennifer

机构信息

Virginia Commonwealth University School of Medicine, Richmond, Virginia.

Commonwealth Dermatology, Richmond, Virginia.

出版信息

Eplasty. 2025 Jan 21;25:e2. eCollection 2025.

PMID:40469488
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12132595/
Abstract

BACKGROUND

Verrucous venous malformation (VVM) is a rare, congenital, low-flow vascular anomaly composed of capillaries and veins in the dermis and subcutaneous tissues. Diagnosis is difficult because knowledge of cases is rare and typically identified with sonographic imaging and elevated GLUT-1. Treatment is mired by high rates of recurrence and residual pain, with further enlargement upon resection or injury. Current management strategies include a combination of laser therapy, surgery, cryotherapy, and topical steroids, each with varying rates of success.

METHODS

The authors examine a case of confirmed VVM, which was initially treated with a candela pulsed dye laser and propranolol to no avail. A combination of surgical approaches were chosen.

CONCLUSIONS

We now look to topical sirolimus 1% as an adjunct for this patient with residual lesions. Questions remain as to the effectiveness, optimal dose, and duration of topical sirolimus and whether this combination of surgical and topical intervention will be effective in the treatment for VVM.

摘要

背景

疣状静脉畸形(VVM)是一种罕见的先天性低流量血管异常,由真皮和皮下组织中的毛细血管和静脉组成。由于病例少见,诊断困难,通常通过超声成像和GLUT-1升高来确诊。治疗因复发率高、残留疼痛以及切除或损伤后进一步增大而陷入困境。目前的管理策略包括激光治疗、手术、冷冻治疗和外用类固醇的联合应用,每种方法的成功率各不相同。

方法

作者检查了一例确诊的VVM病例,该病例最初用坎德拉脉冲染料激光和普萘洛尔治疗无效。选择了多种手术方法联合治疗。

结论

我们现在考虑将1%的外用西罗莫司作为该残留病变患者的辅助治疗。关于外用西罗莫司的有效性、最佳剂量和持续时间,以及这种手术和外用干预的联合治疗对VVM是否有效,仍存在疑问。

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Eplasty. 2025 Jan 21;25:e2. eCollection 2025.
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本文引用的文献

1
Sirolimus for the Management of Verrucous Venous Malformation: A Case Report and Literature Review.西罗莫司治疗疣状静脉畸形:病例报告及文献综述
Case Rep Dermatol. 2021 Jun 17;13(2):298-303. doi: 10.1159/000515984. eCollection 2021 May-Aug.
2
Verrucous Venous Malformation-Subcutaneous Variant.疣状静脉畸形-皮下型
Am J Dermatopathol. 2021 Dec 1;43(12):e181-e184. doi: 10.1097/DAD.0000000000001963.
3
Verrucous venous malformation.疣状静脉畸形
Dermatol Online J. 2019 Dec 15;25(12):13030/qt9dw4w7bd.
4
Treatment of superficial vascular anomalies with topical sirolimus: A multicenter case series.局部应用西罗莫司治疗表浅血管异常:多中心病例系列。
Pediatr Dermatol. 2020 Mar;37(2):272-277. doi: 10.1111/pde.14104. Epub 2020 Jan 19.
5
Comment on "Sirolimus for the treatment of verrucous venous malformation: A retrospective cohort study": Are we missing the lymphatic malformation component?对《西罗莫司治疗疣状静脉畸形:一项回顾性队列研究》的评论:我们是否遗漏了淋巴管畸形成分?
J Am Acad Dermatol. 2023 Mar;88(3):e133-e134. doi: 10.1016/j.jaad.2018.08.050. Epub 2018 Sep 18.
6
Verrucous hemangioma (also known as verrucous venous malformation): A vascular anomaly frequently misdiagnosed as a lymphatic malformation.疣状血管瘤(也称为疣状静脉畸形):一种常被误诊为淋巴管畸形的血管异常。
Pediatr Dermatol. 2018 Nov;35(6):e378-e381. doi: 10.1111/pde.13671. Epub 2018 Sep 14.
7
Sirolimus for treatment of verrucous venous malformation: A retrospective cohort study.西罗莫司治疗疣状静脉畸形:一项回顾性队列研究。
J Am Acad Dermatol. 2019 Feb;80(2):556-558. doi: 10.1016/j.jaad.2018.07.014. Epub 2018 Jul 24.
8
A Case Report of 2 Sirolimus-Related Deaths Among Infants With Kaposiform Hemangioendotheliomas.婴儿 Kaposiform 血管内皮细胞瘤中发生的 2 例与西罗莫司相关的死亡病例报告。
Pediatrics. 2018 Apr;141(Suppl 5):S425-S429. doi: 10.1542/peds.2016-2919.
9
The utility of GLUT1 as a diagnostic marker in cutaneous vascular anomalies: A review of literature and recommendations for daily practice.葡萄糖转运蛋白1(GLUT1)作为皮肤血管异常诊断标志物的效用:文献综述及日常实践建议
Pathol Res Pract. 2017 Jun;213(6):591-597. doi: 10.1016/j.prp.2017.04.023. Epub 2017 Apr 27.
10
Favorable Response to Sirolimus in a Child With Blue Rubber Bleb Nevus Syndrome in the Gastrointestinal Tract.西罗莫司对一名患有胃肠道蓝色橡皮疱痣综合征儿童的良好疗效。
J Pediatr Hematol Oncol. 2017 Mar;39(2):147-149. doi: 10.1097/MPH.0000000000000681.