Delayed Development of Coronary Artery Dilitation in Suspected Severe Acute Respiratory Syndrome Coronavirus 2 Multisystem Inflammatory Syndrome: More Research Needed.

Delayed Development of Coronary Artery Dilitation in Suspected Severe Acute Respiratory Syndrome Coronavirus 2 Multisystem Inflammatory Syndrome: More Research Needed.
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DOI:
10.1097/cce.0000000000000236
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发表时间:
2020-10
影响因子:
--
通讯作者:
Remy KE
Remy KE
中科院分区:
其他
文献类型:
--
作者:
Orr WB;Elward AM;Lin JC;Reich PJ;Scheel JN;Hayes EV;Remy KE

文献摘要

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尽管严重急性呼吸综合征冠状病毒2型大流行的重大疾病负担在儿童中相对罕见,但世界各地都出现了儿童感染后多系统炎症综合征的病例,以及可能由严重急性呼吸综合征冠状病毒2型感染引起的非典型川崎病。冠状病毒疾病-19疾病的最初想法是压倒性的促炎反应驱动疾病的发病。新出现的报告表明,强有力的免疫抑制可能更相关,也更具优势。最近报道的关于儿童多系统炎症综合征的数据表明,这些患者的免疫表型具有异质性,并担心初始时有强烈的促炎状态;然而,缺乏数据支持这一点。同样,缺乏对某些临床表现与免疫系统变化的发展的了解。我们报告一位12岁的多种族男性,冠状病毒病-19鼻咽RNA聚合酶链式反应试验阴性,但严重急性呼吸综合征冠状病毒2型血清学阳性,随后发展为血管扩张性休克并伴有心肌抑制,随后在心肌抑制消失后延迟发展冠状动脉扩张。与以前报道的儿童多系统炎症综合征病例不同,他表现出严重的淋巴细胞减少,而没有特异性炎症细胞因子的升高,而非特异性标志物(铁蛋白和C-反应蛋白)增加。他随后于住院第12天出院,完全康复。与已报道的病例系列相比,我们的代表性病例是一名患有冠状病毒病-19相关的多系统炎症综合征的儿童患者,与已报道的病例系列相比,患者没有强烈的炎症反应,冠状动脉扩张的发现延迟,这突显了进一步从机制上理解冠状病毒病-19疾病以及随后的儿童多系统炎症综合征或川崎病发展的必要性。这份报告提供了一些疾病机制和临床演变考虑,以进一步阐明,以指导潜在的治疗方法的发展。
Although significant disease burden in the severe acute respiratory syndrome coronavirus 2 pandemic has been relatively uncommon in children, worldwide cases of a postinfectious multisystem inflammatory syndrome in children and possible atypical Kawasaki-like disease attributing to severe acute respiratory syndrome coronavirus 2 infection have arisen. Original thinking for coronavirus disease-19 disease was that an overwhelming proinflammatory response drove disease pathogenesis. Emerging reports suggest that a robust immune suppression may be more relevant and predominant. Recently reported data on children with multisystem inflammatory syndrome in children have demonstrated a heterogeneity of immune phenotypes among these patients, with concern for a strong initial proinflammatory state; however, data are lacking to support this. Likewise, understanding development of certain clinical findings to changes in the immune system is lacking. We report a 12-year-old multiracial male with negative coronavirus disease-19 nasopharyngeal RNA polymerase chain reaction testing but positive severe acute respiratory syndrome coronavirus 2 serology, subsequent development of vasodilatory shock with myocardial depression, and subsequent delayed development of coronary artery dilatation after resolution of myocardial depression. Unlike previous reported cases of multisystem inflammatory syndrome in children, he exhibited profound lymphopenia without specific inflammatory cytokines elevations, whereas nonspecific markers (ferritin and C-reactive protein) were increased. He subsequently was discharged on day 12 of hospitalization with complete recovery. Our representative case of a patient with coronavirus disease-19-associated multisystem inflammatory syndrome in children without robust hyperinflammation and a delayed finding of coronary artery dilatation compared with reported case series highlights the need for further mechanistic understanding of coronavirus disease-19 disease and subsequent multisystem inflammatory syndrome in children or Kawasaki disease development. This report offers a number of disease mechanisms and clinical evolution considerations for further elucidation to guide development of potential therapies.