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A child with resistant Kawasaki disease successfully treated with anakinra: a case report

BACKGROUND: Kawasaki disease (KD) is an acute self-limited systemic vasculitis of unknown etiology. Intravenous immunoglobulin (IVIG) is an effective treatment and decreases the risk of cardiac complications to less than 5%. In spite of its effectiveness, some children do not respond to this therapy...

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Autores principales: Sánchez-Manubens, J., Gelman, A., Franch, N., Teodoro, S., Palacios, J. R., Rudi, N., Rivera, J., Antón, J.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2017
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5385011/
https://www.ncbi.nlm.nih.gov/pubmed/28390409
http://dx.doi.org/10.1186/s12887-017-0852-6
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author Sánchez-Manubens, J.
Gelman, A.
Franch, N.
Teodoro, S.
Palacios, J. R.
Rudi, N.
Rivera, J.
Antón, J.
author_facet Sánchez-Manubens, J.
Gelman, A.
Franch, N.
Teodoro, S.
Palacios, J. R.
Rudi, N.
Rivera, J.
Antón, J.
author_sort Sánchez-Manubens, J.
collection PubMed
description BACKGROUND: Kawasaki disease (KD) is an acute self-limited systemic vasculitis of unknown etiology. Intravenous immunoglobulin (IVIG) is an effective treatment and decreases the risk of cardiac complications to less than 5%. In spite of its effectiveness, some children do not respond to this therapy and still develop coronary aneurysms (CAA). The optimal treatment for IVIG non-responsive patients remains controversial although corticoids have been suggested to be an effective treatment in some patients. For those patients still resistant to IVIG and corticoids, interleukin-1 receptor antagonists (IL-1RA) such anakinra could be an alternative. CASE PRESENTATION: We present a 3 year-old Caucasian patient with KD without cardiac complications but with important resistance to treatment. After becoming resistant to IVIG and corticoids, anakinra proved to be an effective treatment. CONCLUSIONS: To our knowledge, this is the first report of the utility of IL-1RA in refractory KD without coronary impairment. The patient fulfilled the classical criteria for KD and, after becoming resistant to first and second line treatments, anakinra proved to be an effective treatment. Further studies are required to determine if this is an effective treatment option for other cases of resistant Kawasaki disease.
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spelling pubmed-53850112017-04-12 A child with resistant Kawasaki disease successfully treated with anakinra: a case report Sánchez-Manubens, J. Gelman, A. Franch, N. Teodoro, S. Palacios, J. R. Rudi, N. Rivera, J. Antón, J. BMC Pediatr Case Report BACKGROUND: Kawasaki disease (KD) is an acute self-limited systemic vasculitis of unknown etiology. Intravenous immunoglobulin (IVIG) is an effective treatment and decreases the risk of cardiac complications to less than 5%. In spite of its effectiveness, some children do not respond to this therapy and still develop coronary aneurysms (CAA). The optimal treatment for IVIG non-responsive patients remains controversial although corticoids have been suggested to be an effective treatment in some patients. For those patients still resistant to IVIG and corticoids, interleukin-1 receptor antagonists (IL-1RA) such anakinra could be an alternative. CASE PRESENTATION: We present a 3 year-old Caucasian patient with KD without cardiac complications but with important resistance to treatment. After becoming resistant to IVIG and corticoids, anakinra proved to be an effective treatment. CONCLUSIONS: To our knowledge, this is the first report of the utility of IL-1RA in refractory KD without coronary impairment. The patient fulfilled the classical criteria for KD and, after becoming resistant to first and second line treatments, anakinra proved to be an effective treatment. Further studies are required to determine if this is an effective treatment option for other cases of resistant Kawasaki disease. BioMed Central 2017-04-08 /pmc/articles/PMC5385011/ /pubmed/28390409 http://dx.doi.org/10.1186/s12887-017-0852-6 Text en © The Author(s). 2017 Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
spellingShingle Case Report
Sánchez-Manubens, J.
Gelman, A.
Franch, N.
Teodoro, S.
Palacios, J. R.
Rudi, N.
Rivera, J.
Antón, J.
A child with resistant Kawasaki disease successfully treated with anakinra: a case report
title A child with resistant Kawasaki disease successfully treated with anakinra: a case report
title_full A child with resistant Kawasaki disease successfully treated with anakinra: a case report
title_fullStr A child with resistant Kawasaki disease successfully treated with anakinra: a case report
title_full_unstemmed A child with resistant Kawasaki disease successfully treated with anakinra: a case report
title_short A child with resistant Kawasaki disease successfully treated with anakinra: a case report
title_sort child with resistant kawasaki disease successfully treated with anakinra: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5385011/
https://www.ncbi.nlm.nih.gov/pubmed/28390409
http://dx.doi.org/10.1186/s12887-017-0852-6
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