Division of Pediatric Hematology and Oncology.
Division of Pediatric Cardiology.
J Pediatr Hematol Oncol. 2023 May 1;45(4):e427-e432. doi: 10.1097/MPH.0000000000002583. Epub 2022 Oct 24.
Multisystem Inflammatory Syndrome in Children (MIS-C) is a late systemic inflammatory response to a recent mild or asymptomatic coronavirus disease of 2019 infection. The pathophysiology is incompletely understood but it often features significant coagulopathy along with cardiac and endothelial dysfunction. Endothelial inflammation has been primarily described in acute coronavirus disease of 2019 infection, with less characterization in MIS-C. Here we describe novel findings of nearly universal severe and prolonged factor VIII (FVIII) and von Willebrand factor antigen elevations in an institutional cohort of patients with MIS-C ages younger than or 21 years old (N=31). All patients had elevated acute phase reactants and D-dimer at presentation and met published criteria for MIS-C. FVIII was high at presentation in 97% of patients but continued to rise during the ensuing weeks of treatment to a mean 429%, peaking on median day 17 of illness as an outpatient. FVIII levels were >600% in multiple patients. von Willebrand factor antigen was measured less frequently but showed similar trends. These escalations occurred amidst resolving cardiac dysfunction and acute phase reactant normalization and despite patients receiving multimodal anti-inflammatory treatments and aspirin and enoxaparin thromboprophylaxis. No thrombotic events occurred. Endothelial dysfunction represented by very elevated FVIII levels may persist longer than other acute phase reactants may reflect.
儿童多系统炎症综合征(MIS-C)是对近期轻度或无症状 2019 年冠状病毒病感染的迟发性全身炎症反应。其病理生理学尚不完全清楚,但通常伴有明显的凝血功能障碍以及心脏和血管内皮功能障碍。血管内皮炎症主要在急性 2019 年冠状病毒病感染中描述,而在 MIS-C 中描述较少。在这里,我们描述了一项机构队列研究中的新发现,该队列研究中的 31 名年龄小于或等于 21 岁的 MIS-C 患者几乎普遍存在严重和持久的因子 VIII(FVIII)和血管性血友病因子抗原升高。所有患者在就诊时均存在急性反应物和 D-二聚体升高,并符合 MIS-C 的公布标准。97%的患者在就诊时 FVIII 升高,但在随后的几周治疗中持续升高,平均升高 429%,在疾病的第 17 天中位数达到峰值,作为门诊患者。多名患者的 FVIII 水平超过 600%。血管性血友病因子抗原的测量频率较低,但显示出类似的趋势。这些升高发生在心脏功能障碍和急性反应物正常化的同时,尽管患者接受了多种抗炎治疗以及阿司匹林和依诺肝素预防血栓形成。没有发生血栓事件。血管内皮功能障碍表现为非常高的 FVIII 水平可能比其他急性反应物持续时间更长。