Skip to main content
Top
Published in: Pediatric Rheumatology 1/2020

Open Access 01-12-2020 | Aneurysm | Case Report

Neonatal Kawasaki disease with multiple arterial aneurysms: a case report

Authors: Qu-ming Zhao, Xue-cun Liang, Lin Wu, Fang Liu

Published in: Pediatric Rheumatology | Issue 1/2020

Login to get access

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.
Literature
1.
go back to reference Group JCSJW. Guidelines for diagnosis and management of cardiovascular sequelae in Kawasaki disease (JCS 2013). Digest version. Circ J. 2014;78(10):2521–62.CrossRef Group JCSJW. Guidelines for diagnosis and management of cardiovascular sequelae in Kawasaki disease (JCS 2013). Digest version. Circ J. 2014;78(10):2521–62.CrossRef
2.
go back to reference Hangai M, Kubota Y, Kagawa J, Yashiro M, Uehara R, Nakamura Y, et al. Neonatal Kawasaki disease: case report and data from nationwide survey in Japan. Eur J Pediatr. 2014;173(11):1533–6.CrossRef Hangai M, Kubota Y, Kagawa J, Yashiro M, Uehara R, Nakamura Y, et al. Neonatal Kawasaki disease: case report and data from nationwide survey in Japan. Eur J Pediatr. 2014;173(11):1533–6.CrossRef
3.
go back to reference Lee EJ, Park YW, Hong YM, Lee JS, Han JW. Epidemiology of Kawasaki disease in infants 3 months of age and younger. Korean J Pediatr. 2012;55(6):202–5.CrossRef Lee EJ, Park YW, Hong YM, Lee JS, Han JW. Epidemiology of Kawasaki disease in infants 3 months of age and younger. Korean J Pediatr. 2012;55(6):202–5.CrossRef
4.
go back to reference Altammar F, Lang B. Kawasaki disease in the neonate: case report and literature review. Pediatr Rheumatol Online J. 2018;16(1):43.CrossRef Altammar F, Lang B. Kawasaki disease in the neonate: case report and literature review. Pediatr Rheumatol Online J. 2018;16(1):43.CrossRef
5.
go back to reference Karia VR, Hescock GC, Gedalia A, Ross-Ascuitto N. Successful emergent coronary thrombolysis in a neonate with Kawasaki's disease. Pediatr Cardiol. 2010;31(8):1239–42.CrossRef Karia VR, Hescock GC, Gedalia A, Ross-Ascuitto N. Successful emergent coronary thrombolysis in a neonate with Kawasaki's disease. Pediatr Cardiol. 2010;31(8):1239–42.CrossRef
6.
go back to reference Nakagawa N, Yoshida M, Narahara K, Kunitomi T. Kawasaki disease in an 8-day-old neonate. Pediatr Cardiol. 2009;30(4):527–9.CrossRef Nakagawa N, Yoshida M, Narahara K, Kunitomi T. Kawasaki disease in an 8-day-old neonate. Pediatr Cardiol. 2009;30(4):527–9.CrossRef
7.
go back to reference Thapa R, Pramanik S, Dhar S, Kundu R. Neonatal Kawasaki disease with multiple coronary aneurysms and thrombocytopenia. Pediatr Dermatol. 2007;24(6):662–3.CrossRef Thapa R, Pramanik S, Dhar S, Kundu R. Neonatal Kawasaki disease with multiple coronary aneurysms and thrombocytopenia. Pediatr Dermatol. 2007;24(6):662–3.CrossRef
8.
go back to reference Stanley TV, Grimwood K. Classical Kawasaki disease in a neonate. Arch Dis Child Fetal Neonatal Ed. 2002;86(2):F135–6.CrossRef Stanley TV, Grimwood K. Classical Kawasaki disease in a neonate. Arch Dis Child Fetal Neonatal Ed. 2002;86(2):F135–6.CrossRef
9.
go back to reference Manlhiot C, Yeung RS, Clarizia NA, Chahal N, McCrindle BW. Kawasaki disease at the extremes of the age spectrum. Pediatrics. 2009;124(3):e410–5.CrossRef Manlhiot C, Yeung RS, Clarizia NA, Chahal N, McCrindle BW. Kawasaki disease at the extremes of the age spectrum. Pediatrics. 2009;124(3):e410–5.CrossRef
10.
go back to reference Satoh K, Wakejima Y, Gau M, Kiguchi T, Matsuda N, Takasawa R, et al. Risk of coronary artery lesions in young infants with Kawasaki disease: need for a new diagnostic method. Int J Rheum Dis. 2018;21(3):746–54.CrossRef Satoh K, Wakejima Y, Gau M, Kiguchi T, Matsuda N, Takasawa R, et al. Risk of coronary artery lesions in young infants with Kawasaki disease: need for a new diagnostic method. Int J Rheum Dis. 2018;21(3):746–54.CrossRef
11.
go back to reference Zhao QM, Chu C, Wu L, Liang XC, Sun SN, He L, et al. Systemic artery aneurysms and Kawasaki disease. Pediatrics. 2019;144(6):e20192254. Zhao QM, Chu C, Wu L, Liang XC, Sun SN, He L, et al. Systemic artery aneurysms and Kawasaki disease. Pediatrics. 2019;144(6):e20192254.
12.
go back to reference McCrindle BW, Rowley AH, Newburger JW, Burns JC, Bolger AF, Gewitz M, et al. Diagnosis, treatment, and long-term Management of Kawasaki Disease: a scientific statement for health professionals from the American Heart Association. Circulation. 2017;135(17):e927–99.CrossRef McCrindle BW, Rowley AH, Newburger JW, Burns JC, Bolger AF, Gewitz M, et al. Diagnosis, treatment, and long-term Management of Kawasaki Disease: a scientific statement for health professionals from the American Heart Association. Circulation. 2017;135(17):e927–99.CrossRef
13.
go back to reference Roy S, Biswas MK. Multiple systemic aneurysms in a case of neglected Kawasaki disease. J Paediatr Child Health. 2019;55(1):117.CrossRef Roy S, Biswas MK. Multiple systemic aneurysms in a case of neglected Kawasaki disease. J Paediatr Child Health. 2019;55(1):117.CrossRef
14.
go back to reference Ozdemir E, Peterson RE. Systemic arterial aneurysm complicated by thrombosis in an infant with resistant Kawasaki disease. Ann Pediatr Cardiol. 2019;12(2):147–9.CrossRef Ozdemir E, Peterson RE. Systemic arterial aneurysm complicated by thrombosis in an infant with resistant Kawasaki disease. Ann Pediatr Cardiol. 2019;12(2):147–9.CrossRef
15.
go back to reference Hsu B, Soriano B, Salamet MSB, Reeder S, Ralphe JC. Systemic arterial aneurysms in a 5 month old male with severe refractory Kawasaki's disease. Pediatr Dimens. 2016;1(4):102-4. Hsu B, Soriano B, Salamet MSB, Reeder S, Ralphe JC. Systemic arterial aneurysms in a 5 month old male with severe refractory Kawasaki's disease. Pediatr Dimens. 2016;1(4):102-4.
16.
go back to reference Briceno-Medina M, Perez M, Waller BR, Sathanandam S. Systemic and pulmonary artery aneurysms in incomplete Kawasaki disease. J Cardiol Cases. 2016;13(6):185–8.CrossRef Briceno-Medina M, Perez M, Waller BR, Sathanandam S. Systemic and pulmonary artery aneurysms in incomplete Kawasaki disease. J Cardiol Cases. 2016;13(6):185–8.CrossRef
17.
go back to reference Dionne A, Burns JC, Dahdah N, Tremoulet AH, Gauvreau K, de Ferranti SD, et al. Treatment intensification in patients with Kawasaki disease and coronary aneurysm at diagnosis. Pediatrics. 2019;143(6):e20183341.CrossRef Dionne A, Burns JC, Dahdah N, Tremoulet AH, Gauvreau K, de Ferranti SD, et al. Treatment intensification in patients with Kawasaki disease and coronary aneurysm at diagnosis. Pediatrics. 2019;143(6):e20183341.CrossRef
Metadata
Title
Neonatal Kawasaki disease with multiple arterial aneurysms: a case report
Authors
Qu-ming Zhao
Xue-cun Liang
Lin Wu
Fang Liu
Publication date
01-12-2020
Publisher
BioMed Central
Published in
Pediatric Rheumatology / Issue 1/2020
Electronic ISSN: 1546-0096
DOI
https://doi.org/10.1186/s12969-020-00440-x

Other articles of this Issue 1/2020

Pediatric Rheumatology 1/2020 Go to the issue