Eur J Pediatr Surg 2019; 29(05): 401-407
DOI: 10.1055/s-0038-1673708
Review Article
Georg Thieme Verlag KG Stuttgart · New York

Comparison of Corticosteroid and Vincristine in Treating Kaposiform Hemangioendothelioma and Tufted Angioma: A Systematic Review and Meta-Analysis

Wei Yao
1   Department of Pediatric Surgery, Children's Hospital of Fudan University, Shanghai, People's Republic of China
,
Kai Li
1   Department of Pediatric Surgery, Children's Hospital of Fudan University, Shanghai, People's Republic of China
,
Zuopeng Wang
1   Department of Pediatric Surgery, Children's Hospital of Fudan University, Shanghai, People's Republic of China
,
Jiahao Pei
1   Department of Pediatric Surgery, Children's Hospital of Fudan University, Shanghai, People's Republic of China
,
Xiangqi Liu
1   Department of Pediatric Surgery, Children's Hospital of Fudan University, Shanghai, People's Republic of China
,
Shan Zheng
1   Department of Pediatric Surgery, Children's Hospital of Fudan University, Shanghai, People's Republic of China
,
Kuiran Dong
1   Department of Pediatric Surgery, Children's Hospital of Fudan University, Shanghai, People's Republic of China
› Author Affiliations
Further Information

Publication History

06 June 2018

30 August 2018

Publication Date:
29 October 2018 (online)

Abstract

Objective To evaluate the outcome and safety of corticosteroids and vincristine (VCR) in the treatment of kaposiform hemangioendothelioma (KHE) and tufted angioma (TA).

Materials and Methods Clinical studies involving corticosteroids and VCR therapies in treating KHE/TA were identified by using PubMed, Cochrane Library, OVID, EBSCO, CNKI, VIP, and Wanfang databases from their establishment date to December 2017. Randomized controlled trials, case–control, or case series with more than five cases were included. The following data were extracted: study sample, demographics, responses rate, recurrence rate, and adverse reactions. Two reviewers completed screening and extraction. Methodological quality was evaluated with quality appraisal tool.

Results A total of 266 studies were found, and 27 studies were finally included in this research; quality of all studies was low. Seven studies with a total of 123 participants, which compared the effect of systemic corticosteroids with that of VCR, were performed for the meta-analysis. The results indicated that the effect of VCR was significantly higher than that of corticosteroids (relative risk [RR] = 2.08, 95% confidence interval [CI]: 1.38–3.16). The recurrence rate of VCR (11.1%) was lower than that of corticosteroids (50%), but there was no statistical difference between the two therapies (p = 0.1312). The result of pooled adverse reactions response rate for VCR was 18.2%, significantly lower than that for corticosteroids, which was 52.0%.

Conclusion The present profile shows that VCR is relatively more effective and safer in treating KHE/TA than corticosteroids are. So, we believe VCR could be used as a first-line medication agent in the treatment of KHE/TA.

Note

Informed consent was obtained from all individual participants included in the study.


 
  • Reference

  • 1 Zukerberg LR, Nickoloff BJ, Weiss SW. Kaposiform hemangioendothelioma of infancy and childhood. An aggressive neoplasm associated with Kasabach-Merritt syndrome and lymphangiomatosis. Am J Surg Pathol 1993; 17 (04) 321-328
  • 2 Fernández Y, Bernabeu-Wittel M, García-Morillo JS. Kaposiform hemangioendothelioma. Eur J Intern Med 2009; 20 (02) 106-113
  • 3 Sarkar M, Mulliken JB, Kozakewich HP, Robertson RL, Burrows PE. Thrombocytopenic coagulopathy (Kasabach-Merritt phenomenon) is associated with kaposiform hemangioendothelioma and not with common infantile hemangioma. Plast Reconstr Surg 1997; 100 (06) 1377-1386
  • 4 Drolet BA, Trenor III CC, Brandão LR. , et al. Consensus-derived practice standards plan for complicated kaposiform hemangioendothelioma. J Pediatr 2013; 163 (01) 285-291
  • 5 Margolin JF, Soni HM, Pimpalwar S. Medical therapy for pediatric vascular anomalies. Semin Plast Surg 2014; 28 (02) 79-86
  • 6 Drucker AM, Pope E, Mahant S, Weinstein M. Vincristine and corticosteroids as first-line treatment of Kasabach-Merritt syndrome in kaposiform hemangioendothelioma. J Cutan Med Surg 2009; 13 (03) 155-159
  • 7 Fahrtash F, McCahon E, Arbuckle S. Successful treatment of kaposiform hemangioendothelioma and tufted angioma with vincristine. J Pediatr Hematol Oncol 2010; 32 (06) 506-510
  • 8 Moga C, Guo B, Schopflocher D. , et al. Development of a Quality Apraisal Tool for Case Series Studies Using a Modified Delphi Technique. Edmonton, Alberta: Institute of Health Economics; 2012
  • 9 Ryan C, Price V, John P. , et al. Kasabach-Merritt phenomenon: a single centre experience. Eur J Haematol 2010; 84 (02) 97-104
  • 10 Yasui N, Koh K, Kato M. , et al. Kasabach-Merritt phenomenon: a report of 11 cases from a single institution. J Pediatr Hematol Oncol 2013; 35 (07) 554-558
  • 11 Wang P, Zhou W, Tao L, Zhao N, Chen XW. Clinical analysis of Kasabach-Merritt syndrome in 17 neonates. BMC Pediatr 2014; 14: 146
  • 12 Boccara O, Fraitag S, Lasne D. , et al. Kaposiform haemangioendothelioma-spectrum lesions with Kasabach-Merritt phenomenon: retrospective analysis and long-term outcome. Acta Derm Venereol 2016; 96 (01) 77-81
  • 13 Yuan SM, Shen WM, Wang X. , et al. Clinical study on 11 cases of Kasabach-Merritt phenomenon in Chinese children. J Tissue Eng and Reconstr Surg 2010; 6 (03) 148-152
  • 14 Miao X, Tianxiang O, Yan X. , et al. [Comprehensive therapy for infant vascular tumor associated with Kasabach-Merritt phenomenon]. Zhonghua Zheng Xing Wai Ke Za Zhi 2015; 31 (04) 263-268
  • 15 Sun YC, Li K, Zheng S. , et al. The initial experience of treatment of Kasabach-Merritt syndrome with vincristine. Linchuang Xiaoer Waike Zazhi 2009; 8 (05) 3-5
  • 16 Haisley-Royster C, Enjolras O, Frieden IJ. , et al. Kasabach-merritt phenomenon: a retrospective study of treatment with vincristine. J Pediatr Hematol Oncol 2002; 24 (06) 459-462
  • 17 Rodriguez V, Lee A, Witman PM, Anderson PA. Kasabach-merritt phenomenon: case series and retrospective review of the mayo clinic experience. J Pediatr Hematol Oncol 2009; 31 (07) 522-526
  • 18 Larsen EC, Zinkham WH, Eggleston JC, Zitelli BJ. Kasabach-Merritt syndrome: therapeutic considerations. Pediatrics 1987; 79 (06) 971-980
  • 19 Fernandez-Pineda I, Lopez-Gutierrez JC, Chocarro G, Bernabeu-Wittel J, Ramirez-Villar GL. Long-term outcome of vincristine-aspirin-ticlopidine (VAT) therapy for vascular tumors associated with Kasabach-Merritt phenomenon. Pediatr Blood Cancer 2013; 60 (09) 1478-1481
  • 20 Shin HY, Ryu KH, Ahn HS. Stepwise multimodal approach in the treatment of Kasabach-Merritt syndrome. Pediatr Int 2000; 42 (06) 620-624
  • 21 Wang Z, Li K, Yao W, Dong K, Xiao X, Zheng S. Steroid-resistant kaposiform hemangioendothelioma: a retrospective study of 37 patients treated with vincristine and long-term follow-up. Pediatr Blood Cancer 2015; 62 (04) 577-580
  • 22 Wananukul S, Nuchprayoon I, Seksarn P. Treatment of Kasabach-Merritt syndrome: a stepwise regimen of prednisolone, dipyridamole, and interferon. Int J Dermatol 2003; 42 (09) 741-748
  • 23 Tan XY, Zhang J, Zhou SY. , et al. Successful treatment of steroid-resistant Kasabach-Merritt syndrome in infants with transcatheter arterial scleroembolization and vincristine. Zhonghua Fang She Xue Za Zhi 2014; 48 (11) 934-937
  • 24 Xia HP, He ZJ, Zhu JX. , et al. Kasabach-Merritt syndrome:report of ten cases. Zhongguo Shiyong Erke Zazhi 2011; 26 (02) 125-127
  • 25 Zhang YY, Zhang YH, Luo J, Xiang X. Kasabach-Merritt syndrome: eleven cases report. J China Pediatr Blood and Cancer. 2007; 12 (02) 64-67
  • 26 Jiang HX, Liu Y, Gao XM. Kasabach-Merritt syndrome: ten cases report. J Hainan Med. 2004; 15 (08) 104
  • 27 Qiu H. The treatment of Kasabach-Merritt syndrome: ten cases report. J Fujian Med. 2001; 23 (04) 93-94
  • 28 Li KL, Qin ZP, Liu XJ, Hu XQ. [Clinical analysis of 13 cases of hemangioma and vascular malformation associated with thrombopenia]. Shanghai Kou Qiang Yi Xue 2005; 14 (02) 108-112
  • 29 Qi HY, Ma L, Zhang JZ. Huge hemangioma combined with thrombocytopenia in infants: eight cases report. Linchuang Xiaoer Waike Zazhi 2008; 7 (06) 47-50
  • 30 Yan L, Xu Z, Sun YJ. , et al. Clinical analysis of 16 cases with infantile Kasabach-Merritt syndrome. J Pract Dermatol 2015; 8 (04) 257-259
  • 31 Lei HZ, Sun B, Liu DK. , et al. [Combined and sequential therapy for Kasabach-Merritt syndrome]. Zhonghua Yi Xue Za Zhi 2011; 91 (36) 2538-2541
  • 32 Wang X, Chen S, Qiao LJ. Clinical analysis of 5 cases of Kasabach-Merritt syndrome. J China Pediatr Blood and Cancer. 2014; 19 (06) 327-331
  • 33 Zhou B, Li WL, Shu Y. , et al. Clinical analysis on 13 cases with Kasabach-Merritt syndrome. Zhongguo Pifu Xingbingxue Zazhi 2009; 23 (04) 215-221
  • 34 Guo XK, Li P, Guo ZT. , et al. Retrospective analysis of 38 cases of Kasabach-Merritt syndrome. Zhongguo Pifu Xingbingxue Zazhi 2011; 25 (10) 775-777
  • 35 Hartman KR, Moncur JT, Minniti CP, Creamer KM. Mediastinal kaposiform hemangioendothelioma and Kasabach-Merritt phenomenon in an infant: treatment with interferon. J Pediatr Hematol Oncol 2009; 31 (09) 690-692
  • 36 Wang Z, Li K, Dong K, Xiao X, Zheng S. Variable response to propranolol treatment of kaposiform hemangioendothelioma, tufted angioma, and Kasabach-Merritt phenomenon. Pediatr Blood Cancer 2014; 61 (08) 1518-1519
  • 37 Wang Z, Li K, Dong K, Xiao X, Zheng S. Successful treatment of Kasabach-Merritt phenomenon arising from kaposiform hemangioendothelioma by sirolimus. J Pediatr Hematol Oncol 2015; 37 (01) 72-73
  • 38 Garcia-Monaco R, Giachetti A, Peralta O. , et al. Kaposiform hemangioendothelioma with Kasabach-Merritt phenomenon: successful treatment with embolization and vincristine in two newborns. J Vasc Interv Radiol 2012; 23 (03) 417-422
  • 39 Malhotra Y, Yang CS, McNamara J, Antaya RJ. Congenital kaposiform hemangioendothelioma with Kasabach-Merritt phenomenon successfully treated with low-dose radiation therapy. Pediatr Dermatol 2014; 31 (05) 595-598
  • 40 Munn SE, Jackson JE, Jones RR. Tufted haemangioma responding to high-dose systemic steroids: a case report and review of the literature. Clin Exp Dermatol 1994; 19 (06) 511-514