背景:嗜酸性粒细胞性胃炎(EoG)很少与谷蛋白敏感性肠病(GSE)结合报道。当这种情况发生时,患者通常表现为胃肠道症状。据我们所知,低蛋白血症尚未被报道为主要表现。抗IgE治疗,比如奥马珠单抗,降低血液中嗜酸性粒细胞计数,肺,和直觉。其治疗活性EoG的效率仍然未知。
方法:我们报告了一个33个月大的男孩,有食物过敏和特应性皮炎的病史,他反复出现水肿,低蛋白血症,和嗜酸性粒细胞增多在14个月大。EoG和GSE的诊断是根据临床表现和胃肠道活检和血清学检测结果确认的。虽然泼尼松和饮食干预最初是有效的,这个男孩出现了泼尼松相关的面部肿胀。停止泼尼松后,他的症状复发了.随后使用奥马珠单抗治疗,结合饮食干预,具有良好的疗效和安全性。
结论:据我们所知,这是首例并发EoG和GSE,主要表现为低蛋白血症.我们强调这两种疾病的罕见表现,以引起临床怀疑并防止错过和延迟诊断。EoG的发病机制具有异质性和复杂性。奥马珠单抗显示出良好的疗效,提示IgE介导的过程可能参与了该患者疾病的发病机制。
BACKGROUND: Eosinophilic gastritis (EoG) has rarely been reported in conjunction with gluten-sensitive enteropathy (GSE). When this does occur, patients typically present with gastrointestinal symptoms. To our knowledge, hypoproteinemia has not been reported as the primary manifestation. Anti-IgE therapy, such as omalizumab, lowers eosinophil counts in the blood, lungs, and gut. Its efficiency in treating active EoG remain unknown.
METHODS: We report a 33-month-old boy with a history of food allergy and atopic dermatitis who developed recurrent edema, hypoproteinemia, and eosinophilia at the age of 14 months. The diagnoses of EoG and GSE were confirmed based on the clinical presentation and results of gastrointestinal biopsies and serological testing. Although prednisone and dietary intervention were initially effective, the boy developed prednisone-related facial swelling. After stopping prednisone, his symptoms relapsed. Subsequent treatment with omalizumab, combined with dietary intervention, showed good efficacy and safety.
CONCLUSIONS: To our knowledge, this is the first case of concurrent EoG and GSE that presented primarily with hypoproteinemia. We highlight the rare manifestations of these two diseases to raise clinical suspicion and prevent missed and delayed diagnoses. The pathogenesis of EoG is heterogeneous and complex. Omalizumab showed good efficacy, indicating that IgE-mediated processes may be involved in the pathogenesis of this patient\'s diseases.