Clinical Outcomes for Systemic Corticosteroids Versus Vincristine in Treating Kaposiform Hemangioendothelioma and Tufted Angioma

Abstract

A meta-analysis was performed to evaluate the efficacy and safety of systemic corticosteroids versus those of vincristine in the treatment of kaposiform hemangioendothelioma (KHE) and tufted angioma (TA).A literature search of PubMed, Embase, and Web of Science was performed for clinical studies on systemic corticosteroid versus vincristine therapies in treating KHE/TA. Pooled relative risks (RRs) and response rate with 95% confidence intervals (CIs) were used to measure outcomes. Heterogeneity, subgroup analysis, sensitivity analysis, and publication bias analysis were performed for result evaluation.Thirteen studies, comprising 344 participants, were used in the analysis. Vincristine therapy was found to be relatively more effective than systemic corticosteroids (RRs = 0.45, 95%CI: 0.35–0.58). The result of pooled adverse reactions response rate for systemic corticosteroids was 0.31 (95%CI, 0.18–0.43), significantly higher than that for vincristine, which was 0.12 (95%CI, 0.06–0.19). In subgroup analyses, factors including mean age and race of patients, and period of follow-up were examined as possible sources of heterogeneity.This is the first meta-analysis estimating the clinical outcomes of systemic corticosteroids in comparison with those of vincristine in the treatment of KHE/TA. The results showed that vincristine was considerably more effective with lower complication rates than systemic corticosteroids; thus, vincristine could be suggested as the first-line therapy for KHE/TA.

Kaposiform hemangioendothelioma: current knowledge and future perspectives

“…Haisley-Royster et al [103] reported encouraging findings regarding the successful use of vincristine in the management of KMP. Many studies have also demonstrated the satisfactory outcomes of vincristine in treating KHE with KMP, including steroid-resistant patients [5,[104][105][106].…”

Sirolimus for the treatment of progressive kaposiform hemangioendothelioma: A multicenter retrospective study

“…Medical treatments with corticosteroids and/or vincristine have been recommended for the management of KHE . However, first‐line treatment with corticosteroids is successful in only 10–27% of all cases, and treatment with vincristine is successful in 60–70% of patients . Moreover, vincristine monotherapy has not been confirmed to provide significant benefits in critically ill patients .…”

“…6,7 However, first-line treatment with corticosteroids is successful in only 10-27% of all cases, and treatment with vincristine is successful in 60-70% of patients. 8,9 Moreover, vincristine monotherapy has not been confirmed to provide significant benefits in critically ill patients. [10][11][12][13] Currently, the optimal therapeutic agent or combination of agents remains undetermined.…”

Low dose sirolimus treatment for refractory tufted angioma and congenital kaposiform hemangioendothelioma, both with Kasabach‐Merritt phenomenon

“…A 2013 consensus recommended steroids associated with vincristine for KMP. 6,9,10 Currently, sirolimus alone or combined with a short course of oral corticosteroids appears to be the best therapeutic option. [11][12][13][14][15][16][17][18] The optimal sirolimus dose in patients with KMF has not been established, but serum levels should be maintained between 5 and 15 ng/mL.…”

“…In this case, the tumor's invasion of the spinal canal was an aggravating factor concomitant with KMP‐related coagulopathy. A 2013 consensus recommended steroids associated with vincristine for KMP . Currently, sirolimus alone or combined with a short course of oral corticosteroids appears to be the best therapeutic option .…”