2019
DOI: 10.1002/pbc.27810
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Low dose sirolimus treatment for refractory tufted angioma and congenital kaposiform hemangioendothelioma, both with Kasabach‐Merritt phenomenon

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Cited by 17 publications
(17 citation statements)
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“…Many authors have reported maintaining the serum levels between 8 and 15 ng/ml [114,116,120]. However, low-dose sirolimus (2-3 ng/ml serum levels) may be associated with low toxicity and has been demonstrated to be effective for treating patients without KMP [127]. Interestingly, there is mounting evidence that low-dose sirolimus markedly ameliorates the development of inflammation and fibrogenesis in animal models, providing a theoretical basis for its use in KHE with musculoskeletal disorders [128,129].…”
Section: Sirolimusmentioning
confidence: 99%
“…Many authors have reported maintaining the serum levels between 8 and 15 ng/ml [114,116,120]. However, low-dose sirolimus (2-3 ng/ml serum levels) may be associated with low toxicity and has been demonstrated to be effective for treating patients without KMP [127]. Interestingly, there is mounting evidence that low-dose sirolimus markedly ameliorates the development of inflammation and fibrogenesis in animal models, providing a theoretical basis for its use in KHE with musculoskeletal disorders [128,129].…”
Section: Sirolimusmentioning
confidence: 99%
“…Sirolimus has shown excellent prospects for the treatment of hemangioma accompanied by KMS. [ 2 , 5 ] After 1-year treatment with sirolimus, the infant in this study showed stable vital signs, with all laboratory indicators as well as his pancreas size returning to normal. Therefore, sirolimus is effective in the treatment of hemangioma with KMS, but further prospective trials are needed to assess the efficacy of this drug and to determine the scientific therapeutic dose and duration of treatment.…”
mentioning
confidence: 66%
“…It is characterized by thrombocytopenia and intravascular coagulation that occasionally develops into disseminated intravascular coagulation. [ 1 , 2 ] A rare incidence of diffuse infiltrative pancreatic hemangioma with a high possibility of missed diagnosis of KMS makes it a fatal disease. Here, we described a case diagnosed with diffuse infiltrative hemangioma (DIH) of pancreas accompanied by KMS and provided ultrasonic data for early and accurate diagnosis, and successful treatment experiences with sirolimus.…”
mentioning
confidence: 99%
“…No hay reportes de afectos adversos graves. Aunque se obtuvo una respuesta positiva con toxicidad limitada en los casos publicados, no existe una estandarización de los criterios de respuesta ni de toxicidad (Tabla 1) [29][30][31][32][33][34][35][36][37] . Los HEK son tumores vasculares infrecuentes y localmente agresivos en la edad pediátrica 1 .…”
Section: Discussionunclassified