Hostname: page-component-586b7cd67f-l7hp2 Total loading time: 0 Render date: 2024-11-27T00:02:04.887Z Has data issue: false hasContentIssue false

A girl with extremely refractory Kawasaki disease: an instructive case with unusual course and outcome

Published online by Cambridge University Press:  21 September 2011

Kossiva Lydia*
Affiliation:
Second Department of Pediatrics, ‘P&A Kyriakou’ Children's Hospital, University of Athens, Athens, Greece
Karanassios Evangelos
Affiliation:
Department of Pediatric Cardiology, ‘Aghia Sophia’ Children's Hospital, Athens, Greece
Papadopoulos George
Affiliation:
Department of Pediatric Cardiology, ‘P&A Kyriakou’ Children's Hospital, Athens, Greece
Karavanaki Kyriaki
Affiliation:
Second Department of Pediatrics, ‘P&A Kyriakou’ Children's Hospital, University of Athens, Athens, Greece
*
Correspondence to: Dr K. Lydia, Second Department of Pediatrics, ‘P&A Kyriakou’ Children's Hospital, University of Athens, 12, Kotieou str 11521, Athens, Greece. Tel: +30 210 6429289; Fax: +30 210 6469212; E-mail: [email protected]

Abstract

Kawasaki disease constitutes an acute febrile vasculitis of unknown aetiology. It is considered the most common cause of acquired cardiac failure in children. Although standard treatment comprises intravenous immunoglobulin and aspirin, some children exhibit refractory disease, necessitating the use of alternative therapies such as corticosteroids and anti-tumour necrosis factor-alpha. For these cases, few controlled data are available. This report focuses on an extremely refractory classical Kawasaki disease with coronary artery aneurysms and ongoing inflammation. We discuss the therapeutic approaches and the potential pitfalls undertaken, which led to an unfavourable clinical outcome.

Type
Brief Report
Copyright
Copyright © Cambridge University Press 2012

Access options

Get access to the full version of this content by using one of the access options below. (Log in options will check for institutional or personal access. Content may require purchase if you do not have access.)

References

1. Brogan, PA, Bose, A, Burgner, D, et al. Kawasaki disease: an evidence passed approach to diagnosis, treatment, and proposals for future research. Arch Dis Child 2002; 86: 286290.CrossRefGoogle Scholar
2. Kossiva, L, Papadopoulos, M, Lagona, E, Papadopoulos, G, Athanassaki, C. Myocardial infarction in a 35-day-old infant with incomplete Kawasaki disease and chickenpox. Cardiol Young 2010; 20: 567570.CrossRefGoogle Scholar
3. Rowley, AH, Shulman, ST. Pathogenesis and management of Kawasaki disease. Expert Rev Anti Infect Ther 2010; 8: 197203.Google Scholar
4. Wood, LE, Tulloh, RMR. Kawasaki disease in children. Heart 2009; 95: 787792.Google Scholar
5. Zhang, T, Yanagawa, H, Oki, I, et al. Factors relating to the cardiac sequelae of Kawasaki disease one month after initial onset. Acta Paediatr 2002; 91: 517520.CrossRefGoogle Scholar
6. Manhiot, C, Millar, K, Golding, F, McCrindle, BW. Improved classification of coronary artery abnormalities based only on coronary artery z-scores after Kawasaki disease. Pediatr Cardiol 2010; 31: 242249.CrossRefGoogle Scholar
7. Burns, JC, Mason, WH, Hauger, SB, et al. Infliximab treatment for refractory Kawasaki syndrome. J Pediatr 2005; 146: 662667.CrossRefGoogle ScholarPubMed
8. Hirono, K, Kemmotsu, Y, Wittkowski, H, et al. Infliximab reduces the cytokine-mediated inflammation but does not suppress cellular infiltration of the vessel wall in refractory Kawasaki disease. Pediatr Res 2009; 65: 696701.Google Scholar
9. McCandless, RT, Minich, LL, Tani, LY, Williams, RV. Does abciximab promote coronary artery remodelling in patients with Kawasaki disease? Am J Cardiol 2010; 105: 16251628.CrossRefGoogle ScholarPubMed
10. Rigante, D, Valentini, P, Onesimo, R, et al. Incomplete Kawasaki syndrome followed by systemic onset-juvenile idiopathic arthritis mimicking Kawasaki syndrome. Rheumatol Int 2010; 30: 535539.CrossRefGoogle ScholarPubMed