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新生儿川崎病伴多发动脉动脉瘤:病例报告。

Neonatal Kawasaki disease with multiple arterial aneurysms: a case report.

机构信息

Pediatric Heart Center, Children's Hospital of Fudan University, 399 Wan Yuan Road, Shanghai, 201102, China.

出版信息

Pediatr Rheumatol Online J. 2020 Jun 15;18(1):50. doi: 10.1186/s12969-020-00440-x.

Abstract

BACKGROUND

Kawasaki disease (KD) is a medium vessel vasculitis that typically occurs in children aged between 6 months and 5 years. It is extraordinarily rare in the neonatal period. KD-related systemic artery aneurysms (SAAs) have never been reported in neonates.

CASE PRESENTATION

A male infant was transferred to our institution for persistent high-grade fever lasting 16 days. Symptoms started at day 14 of life, and he was admitted to a children's hospital on the second day of fever. Physical examination at the time found no signs suggestive of KD. The only laboratory parameters which were of significance were values suggestive of systemic inflammation. However, his fever persisted and inflammatory markers continued to rise despite 2 weeks of antibiotic therapy. KD as a noninfectious cause of fever was considered when he came to our institution, and echocardiographic findings of left and right medium coronary artery aneurysms (CAAs) confirmed our suspicions. Full-body magnetic resonance angiography also revealed bilateral axillary artery aneurysms. Administration of intravenous gamma globulin resulted in rapid improvement. His fever resolved on the next day and CAAs and SAAs regressed to normal at 6 months and 3 months after diagnosis, respectively.

CONCLUSION

This unique case of incomplete KD highlights the importance of considering KD in neonates with unexplained prolonged fever and reinforces the need to remain vigilant for SAAs in KD.

摘要

背景

川崎病(KD)是一种中等大小血管炎,通常发生在 6 个月至 5 岁的儿童中。在新生儿期非常罕见。新生儿从未有过与 KD 相关的体动脉动脉瘤(SAA)的报道。

病例介绍

一名男性婴儿因持续高热 16 天转入我院。症状于出生后第 14 天开始,发热第二天即被收入儿童医院。当时的体格检查没有提示 KD 的迹象。唯一有意义的实验室参数是提示全身炎症的数值。然而,尽管他已经接受了 2 周的抗生素治疗,但他的发热仍持续存在,炎症标志物持续升高。当他来到我院时,我们考虑发热是由非感染性川崎病引起的,左、右冠状动脉中动脉瘤(CAA)的超声心动图表现证实了我们的怀疑。全身磁共振血管造影也显示双侧腋动脉动脉瘤。静脉注射丙种球蛋白后迅速改善。他的发热于次日消退,CAA 和 SAA 在诊断后 6 个月和 3 个月分别恢复正常。

结论

本例不完全川崎病的独特之处在于强调了在不明原因持续高热的新生儿中考虑川崎病的重要性,并强调了在川崎病中保持警惕 SAA 的必要性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/4d7b/7296651/3808d6415eb9/12969_2020_440_Fig1_HTML.jpg

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