Department of General Internal Medicine, Asahikawa City Hospital, 1-65, Kinseicho-1-chome, Asahikawa, Hokkaido, Japan.
Department of Hematology, Asahikawa City Hospital, 1-65, Kinseicho-1-chome, Asahikawa, Hokkaido, Japan.
J Med Case Rep. 2022 Aug 31;16(1):324. doi: 10.1186/s13256-022-03543-z.
Previous research has suggested that some autoimmune diseases develop after the occurrence of coronavirus disease 2019. Hypereosinophilic syndrome is a rare disease presenting with idiopathic eosinophilia and multiple organ involvement, including the skin, lungs, gastrointestinal tract, heart, and nervous system. The diagnosis of idiopathic hypereosinophilic syndrome poses a dilemma because clinical manifestation and serum biomarkers are similar to those of eosinophilic granulomatosis with polyangiitis. Only a few cases have been reported where coronavirus disease 2019 may have caused the new onset or exacerbation of eosinophilic granulomatosis with polyangiitis or idiopathic hypereosinophilic syndrome.
We present the case of a 48-year-old Japanese woman with history of asthma who developed deteriorating symptoms of insidiously developed idiopathic hypereosinophilic syndrome following asymptomatic coronavirus disease 2019. She developed acute-onset back pain, tachycardia, abdominal discomfort, loss of appetite, weight loss, skin rash on the back, and numbness of the extremities 3 days after the quarantine period. Extreme hypereosinophilia with multiple abnormal findings including pulmonary ground-glass opacity lesions and mononeuritis multiplex was consistent with hypereosinophilic syndrome. Normal cellularity with eosinophilic proliferation in the bone marrow and negative FIP1L1-PDGFRA raised the diagnosis of idiopathic hypereosinophilic syndrome. Although the patient tested negative for anti-neutrophilic cytoplasmic antibodies and skin biopsy was negative for vasculitis, eosinophilic granulomatosis with polyangiitis could not be excluded. Since glucocorticoids are a standard therapy for both idiopathic hypereosinophilic syndrome and eosinophilic granulomatosis with polyangiitis, we initiated glucocorticoids following a multidisciplinary discussion.
Although the relationship between asymptomatic coronavirus disease 2019 and acute idiopathic hypereosinophilic syndrome exacerbation was uncertain, the chronological order of the symptomatic development suggested a possible link. More clinical cases and population-based studies are needed to determine the potential effect of coronavirus disease 2019 on autoimmune diseases.
先前的研究表明，某些自身免疫性疾病是在 2019 年冠状病毒病（COVID-19）发生后发展而来的。嗜酸性粒细胞增多综合征是一种罕见的疾病，表现为特发性嗜酸性粒细胞增多和多个器官受累，包括皮肤、肺、胃肠道、心脏和神经系统。特发性嗜酸性粒细胞增多综合征的诊断存在困境，因为临床表现和血清生物标志物与嗜酸性粒细胞肉芽肿伴多血管炎相似。仅有少数病例报告 COVID-19 可能导致嗜酸性粒细胞肉芽肿伴多血管炎或特发性嗜酸性粒细胞增多综合征的新发或恶化。
我们报告了一例 48 岁日本女性的病例，该患者有哮喘病史，在无症状 COVID-19 后出现特发性嗜酸性粒细胞增多综合征症状逐渐加重。她在隔离期后 3 天出现急性背痛、心动过速、腹部不适、食欲不振、体重减轻、背部皮疹和四肢麻木。极度嗜酸性粒细胞增多伴有多个异常发现，包括肺部磨玻璃样混浊病变和单神经病多发性神经病，符合嗜酸性粒细胞增多综合征。骨髓细胞正常并有嗜酸性粒细胞增生，FIP1L1-PDGFRA 阴性，提示特发性嗜酸性粒细胞增多综合征。尽管患者抗中性粒细胞胞质抗体检测阴性，皮肤活检无血管炎，但不能排除嗜酸性粒细胞肉芽肿伴多血管炎。由于糖皮质激素是特发性嗜酸性粒细胞增多综合征和嗜酸性粒细胞肉芽肿伴多血管炎的标准治疗方法，因此我们在多学科讨论后开始使用糖皮质激素。
尽管无症状 COVID-19 与急性特发性嗜酸性粒细胞增多综合征恶化之间的关系尚不确定，但症状发作的时间顺序提示可能存在关联。需要更多的临床病例和基于人群的研究来确定 COVID-19 对自身免疫性疾病的潜在影响。
