12세 남아의 알레르기비염과 IgG3 결핍증을 동반한 기무라병에서 두필루맙 치료
Dupilumab therapy in a 12-year-old boy with Kimura disease, allergic rhinitis, and IgG3 deficiency
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Kimura disease is characterized by painless subcutaneous masses, usually in the head and neck area as well as serum/tissue eosinophilia and elevated serum IgE levels. Various treatment methods, such as surgical excision, corticosteroids, and radiotherapy, have been suggested, but none of these are considered standard therapy due to high recurrence rates. Dupilumab is a human monoclonal antibody that binds to the alpha subunit of IL-4 receptor for IL-4 and IL-13 and inhibits T helper 2 (Th2) response, which plays an important role in the pathogenesis of allergic diseases, such as atopic dermatitis, asthma, and nasal polyps. Kimura disease has also been shown to increase CD4+Th2 cells and Th2 cytokines. We report a case of a 12-year-old boy who had subcutaneous neck and orbital masses of Kimura disease concurrent with allergic rhinitis, decreased lung function, and IgG subclass deficiency. The masses did not improve with omalizumab or immunomodulatory agents, but resolved with dupilumab. Considering potential side effects of steroids or immunosuppressant agents, dupilumab may be an option for initial treatment with Kimura diseases in children.
Abstract
Kimura disease is characterized by painless subcutaneous masses, usually in the head and neck area as well as serum/tissue eosinophilia and elevated serum IgE levels. Various treatment methods, such as surgical excision, corticosteroids, and radiotherapy, have been suggested, but none of these are considered standard therapy due to high recurrence rates. Dupilumab is a human monoclonal antibody that binds to the alpha subunit of IL-4 receptor for IL-4 and IL-13 and inhibits T helper 2 (Th2) response, which plays an important role in the pathogenesis of allergic diseases, such as atopic dermatitis, asthma, and nasal polyps. Kimura disease has also been shown to increase CD4+Th2 cells and Th2 cytokines. We report a case of a 12-year-old boy who had subcutaneous neck and orbital masses of Kimura disease concurrent with allergic rhinitis, decreased lung function, and IgG subclass deficiency. The masses did not improve with omalizumab or immunomodulatory agents, but resolved with dupilumab. Considering potential side effects of steroids or immunosuppressant agents, dupilumab may be an option for initial treatment with Kimura diseases in children.
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