Two X-linked agammaglobulinemia patients develop pneumonia as COVID-19 manifestation but recover
Two X-linked agammaglobulinemia patients develop pneumonia as COVID-19 manifestation but recover
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DOI：
10.1111/pai.13263
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发表时间：
2020-05-19
影响因子：
4.4
通讯作者：
Badolato, Raffaele
中科院分区：
文献类型：
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作者：
Soresina, Annarosa;Moratto, Daniele;Badolato, Raffaele
背景最近的SARS-CoV-2大流行最近于2月21日开始影响意大利，它对正常人群构成了威胁，因为新冠肺炎可以表现为从无症状病例到肺炎甚至死亡的广泛的临床表型。有证据表明，高龄和几种合并症可能会影响感染SARS-CoV-2的患者发展为严重肺炎的风险，并可能影响对机械通风的需求。方法当SARS-CoV-2流行到意大利时，我们启动了先天性免疫缺陷患者的监测程序，对出现发烧、咳嗽、腹泻或呕吐等可能表现为新冠肺炎症状的患者进行鼻咽拭子搜索。结果我们描述了2例X-连锁无丙种球蛋白血症(XLA)患者，年龄分别为34岁和26岁，外周血中B细胞完全缺失，发展为COVID-19。通过鼻咽拭子检测SARS-CoV-2确诊，同时接受免疫球蛋白输注。两名患者均出现以发热、咳嗽和厌食为特征的间质性肺炎，并伴有C反应蛋白和铁蛋白升高，但从不需要氧气或重症监护。结论我们的报告提示XLA患者在SARS-CoV-2感染后出现肺炎的风险很高，但可以从感染中恢复，提示B细胞反应可能是重要的，但不是克服疾病的严格要求。然而，有必要进行更大规模的观察性研究，将这些结论推广到其他有类似遗传免疫缺陷的患者。
Background The recent SARS-CoV-2 pandemic, which has recently affected Italy since February 21, constitutes a threat to normal subjects, as the coronavirus disease-19 (COVID-19) can manifest with a broad spectrum of clinical phenotypes ranging from asymptomatic cases to pneumonia or even death. There is evidence that older age and several comorbidities can affect the risk to develop severe pneumonia and possibly the need of mechanic ventilation in subjects infected with SARS-CoV-2. Therefore, we evaluated the outcome of SARS-CoV-2 infection in patients with inborn errors of immunity (IEI) such as X-linked agammaglobulinemia (XLA).Methods When the SARS-CoV-2 epidemic has reached Italy, we have activated a surveillance protocol of patients with IEI, to perform SARS-CoV-2 search by nasopharyngeal swab in patients presenting with symptoms that could be a manifestation of COVID-19, such as fever, cough, diarrhea, or vomiting.Results We describe two patients with X-linked agammaglobulinemia (XLA) aged 34 and 26 years with complete absence of B cells from peripheral blood who developed COVID-19, as diagnosed by SARS-CoV-2 detection by nasopharyngeal swab, while receiving immunoglobulin infusions. Both patients developed interstitial pneumonia characterized by fever, cough, and anorexia and associated with elevation of CRP and ferritin, but have never required oxygen ventilation or intensive care.Conclusion Our report suggests that XLA patients might present with high risk to develop pneumonia after SARS-CoV-2 infection, but can recover from infection, suggesting that B-cell response might be important, but is not strictly required to overcome the disease. However, there is a need for larger observational studies to extend these conclusions to other patients with similar genetic immune defects.