Department of Pediatric Rheumatology, Immunology and Allergy, The First Hospital of Jilin University, Changchun, China.
Medicine (Baltimore). 2020 Dec 24;99(52):e23714. doi: 10.1097/MD.0000000000023714.
Kawasaki disease (KD) is the leading cause of acquired heart abnormalities during childhood. The infiltration of CD8+ T cells plays an essential role in the formation of coronary aneurysms. Follicular cytotoxic T (Tfc) cells are a newly defined subset of CD8+ T cells that express CXC-chemokine receptor 5. The role of Tfc cells in KD is unclear. However, in this report, we present 2 KD children with sustained coronary artery aneurysms (CAA), and we found that their peripheral C-X-C Chemokine Receptor 5+ T cells contained quite amounts of CD4 negative cells. Importantly, these cells have never been reported in KD.
Case 1 was a 3-year-old boy with a complaint of continuous fever for 6 days and conjunctival injection for 3 days. Case 2 was a 6-month-old boy who was hospitalized because of persistent fever for 5 days, rashes and conjunctival injection for 1 day.
Case 1 was diagnosed with KD according to typical symptoms and signs including fever over 5 days, conjunctival injection, rashes, swelling cervical lymph nodes and a strawberry tongue. Case 2 had atypical symptoms including persistent fever for 5 days, rashes and conjunctival injection, and he was diagnosed with KD based on the echocardiographic findings.
Both the 2 patients received intravenous immunoglobulin and oral aspirin. Besides, case 1 was given the second infusion of intravenous immunoglobulin, intravenous prednisolone and low-molecular-weight heparin.
The CAA of case 1 did not regress until the 12th month after disease onset. The CAA of patient 2 began to regress at the third month after disease onset. During the months from disease onset to the recent follow-up, no cardiovascular events had occurred.
We speculate that Tfc cells may be associated with the formation of CAA. Further studies with larger sample size and functional analysis of these cells are needed.
川崎病（KD）是儿童时期获得性心脏异常的主要原因。CD8+T 细胞的浸润在冠状动脉瘤的形成中起着至关重要的作用。滤泡细胞毒性 T（Tfc）细胞是 CD8+T 细胞的一个新定义亚群，它表达 CXC-趋化因子受体 5。Tfc 细胞在 KD 中的作用尚不清楚。然而，在本报告中，我们介绍了 2 例持续冠状动脉瘤（CAA）的 KD 患儿，我们发现他们的外周血 C-X-C 趋化因子受体 5+T 细胞中含有相当数量的 CD4 阴性细胞。重要的是，这些细胞在 KD 中从未被报道过。
病例 1 是一名 3 岁男孩，主诉持续发热 6 天，结膜充血 3 天。病例 2 是一名 6 个月大的男孩，因持续发热 5 天、皮疹和结膜充血 1 天住院。
病例 1 根据典型症状和体征，包括发热超过 5 天、结膜充血、皮疹、颈淋巴结肿大和草莓舌，被诊断为 KD。病例 2 表现为不典型症状，包括持续发热 5 天、皮疹和结膜充血，根据超声心动图检查结果被诊断为 KD。
两名患者均接受静脉注射免疫球蛋白和口服阿司匹林治疗。此外，病例 1 还接受了第二次静脉注射免疫球蛋白、静脉注射泼尼松龙和低分子肝素治疗。
病例 1 的 CAA 直到发病后 12 个月才消退。患者 2 的 CAA 在发病后 3 个月开始消退。在发病至最近随访的几个月里，没有发生心血管事件。
我们推测 Tfc 细胞可能与 CAA 的形成有关。需要进一步研究这些细胞的更大样本量和功能分析。
