| Calcineurin inhibitor treatment of intravenous immunoglobulin-resistant Kawasaki disease. | |
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MedLine Citation:
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PMID: 22484354 Owner: NLM Status: MEDLINE |
Abstract/OtherAbstract:
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OBJECTIVE: To describe the clinical course and outcome of 10 patients with Kawasaki disease (KD) treated with a calcineurin inhibitor after failing to respond to multiple therapies. STUDY DESIGN: Demographic and clinical data were prospectively collected using standardized case report forms. T-cell phenotypes were determined by flow cytometry, and KD risk alleles in ITPKC (rs28493229), CASP3 (rs72689236), and FCGR2A (rs1801274) were genotyped. RESULTS: Intravenous followed by oral therapy with cyclosporine (CSA) or oral tacrolimus was well tolerated and resulted in defervescence and resolution of inflammation in all 10 patients. There were no serious adverse events, and a standardized treatment protocol was developed based on our experiences with this patient population. Analysis of T-cell phenotype by flow cytometry in 2 subjects showed a decrease in circulating activated CD8(+) and CD4(+) T effector memory cells after treatment with CSA. However, suppression of regulatory T-cells was not seen, suggesting targeting of specific, proinflammatory T-cell compartments by CSA. CONCLUSION: Treatment of refractory KD with a calcineurin inhibitor appears to be a safe and effective approach that achieves rapid control of inflammation associated with clinical improvement. |
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Authors:
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Adriana H Tremoulet; Paige Pancoast; Alessandra Franco; Matthew Bujold; Chisato Shimizu; Yoshihiro Onouchi; Alyson Tamamoto; Guliz Erdem; Debra Dodd; Jane C Burns |
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Publication Detail:
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Type: Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't Date: 2012-04-06 |
Journal Detail:
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Title: The Journal of pediatrics Volume: 161 ISSN: 1097-6833 ISO Abbreviation: J. Pediatr. Publication Date: 2012 Sep |
Date Detail:
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Created Date: 2012-08-24 Completed Date: 2013-01-02 Revised Date: 2013-04-26 |
Medline Journal Info:
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Nlm Unique ID: 0375410 Medline TA: J Pediatr Country: United States |
Other Details:
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Languages: eng Pagination: 506-512.e1 Citation Subset: AIM; IM |
Copyright Information:
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Copyright © 2012 Mosby, Inc. All rights reserved. |
Affiliation:
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Department of Pediatrics, University of California at San Diego and Rady Children's Hospital, La Jolla, CA, USA. |
Export Citation:
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APA/MLA Format Download EndNote Download BibTex |
| MeSH Terms | |
Descriptor/Qualifier:
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Child Child, Preschool Clinical Protocols Cyclosporine / therapeutic use* Female Humans Immunoglobulins, Intravenous / therapeutic use Immunologic Factors / therapeutic use Immunosuppressive Agents / therapeutic use* Infant Male Mucocutaneous Lymph Node Syndrome / drug therapy*, immunology T-Lymphocytes / immunology Tacrolimus / therapeutic use Treatment Failure |
| Grant Support | |
ID/Acronym/Agency:
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5UL1 RR031980/RR/NCRR NIH HHS; HL103536/HL/NHLBI NIH HHS; HL69413/HL/NHLBI NIH HHS |
| Chemical | |
Reg. No./Substance:
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0/Immunoglobulins, Intravenous; 0/Immunologic Factors; 0/Immunosuppressive Agents; 109581-93-3/Tacrolimus; 59865-13-3/Cyclosporine |
From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine
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