Successful treatment with rituximab of refractory idiopathic thrombocytopenic purpura in a patient with Kabuki syndrome.
Title | Successful treatment with rituximab of refractory idiopathic thrombocytopenic purpura in a patient with Kabuki syndrome. |
Publication Type | Journal Article |
Year of Publication | 2009 |
Authors | Torii Y, Yagasaki H, Tanaka H, Mizuno S, Nishio N, Muramatsu H, Hama A, Takahashi Y, Kojima S |
Journal | International journal of hematology |
Volume | 90 |
Issue | 2 |
Pagination | 174-6 |
Date Published | 2009 Sep |
ISSN | 1865-3774 |
Keywords | Abnormalities, Multiple, Antibodies, Monoclonal, Antibodies, Monoclonal, Murine-Derived, Child, Preschool, Humans, Immunologic Factors, Male, Platelet Count, Purpura, Thrombocytopenic, Idiopathic, Syndrome |
Abstract | Kabuki syndrome (KS) is often associated with autoimmune abnormalities, such as idiopathici thrombocytopenic purpurai (ITPi), autoimmune hemolytic anemiai, leukoplakia and thyroiditis, as well as congenitali anomalies. We herein present a KS patient with refractory ITP who achieved durable and complete remission in response to a total of four once-monthly infusions of rituximab. KS patients are often more susceptible to infection, so splenectomy should be avoided. Therefore, rituximab therapy is an alternative option for KS patients with ITP who fail to respond to first-line therapy. |
Alternate Journal | Int. J. Hematol. |
Citation Key | 393 |
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