Department of Microbiology and Immunology, Experimental Laboratory Immunology, KU Leuven, Herestraat 49, 3000, Leuven, Belgium.
Department of Microbiology and Immunology, Laboratory of Clinical Immunology, KU Leuven, Herestraat 49, 3000, Leuven, Belgium.
J Clin Immunol. 2015 Nov;35(8):739-44. doi: 10.1007/s10875-015-0205-x. Epub 2015 Oct 15.
Autosomal recessive IL-1R-associated kinase 4 (IRAK-4) deficiency is a rare cause of recurrent pyogenic infections with limited inflammatory responses. We describe an adult female patient with severe lung disease who was phenotypically diagnosed as suffering from autosomal dominant Hyper IgE syndrome (AD HIES) because of recurrent skin infections with Staphylococcus aureus, recurrent pneumonia and elevated serum IgE levels. In contrast to findings in AD HIES patients, no abnormalities were found in the Th17 and circulating follicular helper T cell subsets. A panel-based sequencing approach led to the identification of a homozygous IRAK4 stop mutation (c.877C > T, p.Gln293*).
常染色体隐性 IL-1R 相关激酶 4 (IRAK-4) 缺陷是复发性化脓性感染伴有限炎症反应的罕见原因。我们描述了一名成年女性患者,她患有严重的肺部疾病,由于金黄色葡萄球菌反复皮肤感染、反复肺炎和血清 IgE 水平升高,表型诊断为常染色体显性高免疫球蛋白 E 综合征 (AD HIES)。与 AD HIES 患者的发现相反,Th17 和循环滤泡辅助 T 细胞亚群没有异常。基于面板的测序方法导致鉴定出一个纯合 IRAK4 终止突变 (c.877C > T, p.Gln293*)。