Department of Pediatrics, Fujita Health University School of Medicine, 1-98 Kutsukake-cho, Dengakugakubo, Toyoake, Aichi, 47-01192, Japan.
Department of Pathology, National Institute of Infectious Diseases, 1-23-1 Toyama, Shinjuku-ku, Tokyo, 162-8640, Japan.
BMC Pediatr. 2022 Mar 14;22(1):132. doi: 10.1186/s12887-022-03203-7.
Kawasaki disease (KD) is an acute, febrile, systemic vasculitis of unknown etiology that primarily affects the coronary arteries and generally occurs at around 1 year of age. Although the diagnosis of KD is generally not difficult, it is challenging in cases of incomplete KD lacking characteristic clinical manifestations. The incidence of incomplete KD is higher in infants younger than 6 months of age. Pneumonia is an extremely rare complication of KD and can be misinterpreted as atypical pneumonia rather than KD. Herein, we report a neonate with atypical KD and severe pneumonia who required mechanical ventilation.
Japanese one-month-old infant had only fever and rash on admission (day 1), and he was transferred to the intensive care unit for severe pneumonia on day 2. Although pneumonia improved following intensive care, he was diagnosed with KD on day 14 because of emerging typical clinical manifestations such as fever, bulbar nonexudative conjunctival injection, desquamation of the fingers, and coronary artery aneurysm. KD symptoms improved after three doses of intravenous immunoglobulin plus cyclosporine. However, small coronary aneurysms were present at the time of discharge. In a retrospective analysis, no pathogens were detected by multiplex real-time PCR in samples collected at admission, and the serum cytokine profile demonstrated prominent elevation of IL-6 as well as elevation of neopterin, sTNF-RI, and sTNF-RII, which suggested KD.
The patient's entire clinical course, including the severe pneumonia, was caused by KD. As in this case, neonatal KD may exhibit atypical manifestations such as severe pneumonia requiring mechanical ventilation.
川崎病(KD)是一种病因不明的急性、发热性全身血管炎,主要影响冠状动脉,通常发生在 1 岁左右。尽管 KD 的诊断通常并不困难,但在缺乏特征性临床表现的不完全 KD 病例中,诊断具有挑战性。6 个月以下婴儿的不完全 KD 发病率较高。肺炎是 KD 的一种极其罕见的并发症,可能被误诊为非典型肺炎而非 KD。在此,我们报告了一例伴有严重肺炎且需要机械通气的非典型 KD 新生儿病例。
一名 1 月龄日本婴儿入院时仅表现为发热和皮疹(第 1 天),第 2 天因严重肺炎转入重症监护病房。尽管重症监护后肺炎有所改善,但由于出现典型临床表现,如发热、球结膜非渗出性充血、手指脱皮和冠状动脉瘤,第 14 天被诊断为 KD。静脉注射免疫球蛋白加环孢素治疗后 KD 症状改善。然而,出院时仍存在小的冠状动脉瘤。回顾性分析显示,入院时采集的样本经多重实时 PCR 检测未发现病原体,血清细胞因子谱显示 IL-6 显著升高,同时新蝶呤、sTNF-RI 和 sTNF-RII 升高,提示为 KD。
患者的整个临床过程,包括严重肺炎,均由 KD 引起。与本例一样,新生儿 KD 可能表现为需要机械通气的非典型肺炎。