Etanercept treatment of cutaneous granulomas in common variable immunodeficiency

被引:58
作者
Lin, JH
Liebhaber, M
Roberts, RL
Dyer, Z
Stiehm, ER
机构
[1] Univ Calif Los Angeles, David Geffen Sch Med, Dept Pediat, Div Rheumatol Allergy & Immunol, Los Angeles, CA 90095 USA
[2] Sansum Santa Barbara Med Fdn Clin, Santa Barbara, CA USA
关键词
common variable immunodeficiency; granuloma; TNF-alpha inhibitors; etanercept;
D O I
10.1016/j.jaci.2006.01.034
中图分类号
R392 [医学免疫学];
学科分类号
100102 ;
摘要
Common variable immunodeficiency (CVID) is a primary immunodeficiency characterized by hypogammaglobulinemia, poor antibody responses, and recurrent bacterial infections, usually of the sinorespiratory tract. A not uncommon complication is granuloma of the lungs, spleen, liver, and/or skin. We report the case of an 18-year-old boy with CVID and chronic granulomas of the left arm (since 13 years of age) refractory to treatment with antibiotics, intravenous immunoglobulin, antifungal agents, systemic and intralesional steroids, IFN-gamma, cyclosporine, methotrexate, hydroxy-chloroquine, localized radiation therapy, and surgical excision. The lesions improved after treatment with the systemic administration of the TNF-alpha inhibitor etanercept for 1 year. Etanercept prevents soluble TNF from binding to its cell membrane receptor, leading to inhibition of its inflammatory cascade. We recommend further trials of etanercept in patients with CVID with noninfectious recalcitrant granulomas.
引用
收藏
页码:878 / 882
页数:5
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