Refractory Kasabach-Merritt phenomenon successfully treated with sirolimus, and a mini-review of the published work

被引:15
作者
Wang, Zuopeng [1 ]
Li, Kai [1 ]
Dong, Kuiran [1 ]
Xiao, Xianmin [1 ]
Zheng, Shan [1 ]
机构
[1] Fudan Univ, Childrens Hosp, Dept Pediat Surg, Shanghai 201102, Peoples R China
关键词
case report; Kasabach-Merritt phenomenon; mini-review; sirolimus; treatment; KAPOSIFORM HEMANGIOENDOTHELIOMA; VINCRISTINE;
D O I
10.1111/1346-8138.12797
中图分类号
R75 [皮肤病学与性病学];
学科分类号
100206 ;
摘要
Kasabach-Merritt phenomenon (KMP) is a rare and life-threatening disease involving a vascular tumor combined with severe consumptive coagulopathy. We present for the first time a case of KMP with the vascular tumor involving two anatomical sites; the patient failed to respond to steroids and vincristine. Following sirolimus therapy at a dose of 0.8mg/m(2) twice daily, the lesions shrank and the platelet count improved and remained normal 4months after initial therapy. Current treatments for KMP are not particularly effective. Sirolimus at 0.8mg/m(2) per dose, administrated twice daily, appears to be a safe and effective management option. It appears to be an interesting therapeutic option in refractory KMP, but the time to response is variable.
引用
收藏
页码:401 / 404
页数:4
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