Treatment of a patient with Kawasaki disease associated with selective IgA deficiency by continuous infusion of cyclosporine A without intravenous immunoglobulin

Intravenous immunoglobulin therapy is standard for Kawasaki disease (KD) treatment; however, anaphylactic reactions to immunoglobulins are a risk in KD patients with selective IgA deficiency (sIgAD). The therapy for KD associated with sIgAD has not been established. The IgA immune response is...

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Main Authors: Tatsuya Anzai, Takaomi Minami, Tomoyuki Sato, Sadahiro Furui, Takanori Yamagata
Format: Article
Language:English
Published: Hacettepe University Institute of Child Health 2016-12-01
Series:The Turkish Journal of Pediatrics
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Online Access:https://turkjpediatr.org/article/view/1178
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author Tatsuya Anzai
Takaomi Minami
Tomoyuki Sato
Sadahiro Furui
Takanori Yamagata
author_facet Tatsuya Anzai
Takaomi Minami
Tomoyuki Sato
Sadahiro Furui
Takanori Yamagata
author_sort Tatsuya Anzai
collection DOAJ
description Intravenous immunoglobulin therapy is standard for Kawasaki disease (KD) treatment; however, anaphylactic reactions to immunoglobulins are a risk in KD patients with selective IgA deficiency (sIgAD). The therapy for KD associated with sIgAD has not been established. The IgA immune response is believed to play an important role in KD vasculitis. We report the case of a 5-year-old boy with KD and sIgAD treated with intravenous cyclosporine A (CsA, 3.0 mg/kg/day) instead of intravenous immunoglobulin (IVIG). The fever and inflammation immediately resolved without a coronary artery lesion. In KD patients with sIgAD, we believe that an IgA immune response is lacking, which is the reason for milder KD symptoms than in those without sIgAD. This case report aids in clarifying the role of IgA antibodies in KD and provides evidence that CsA is a potential candidate for first-line therapy for patients with KD with contraindications to IVIG.
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id doaj-art-9c65e5edff7b4b00a243c569814a7d93
institution OA Journals
issn 0041-4301
2791-6421
language English
publishDate 2016-12-01
publisher Hacettepe University Institute of Child Health
record_format Article
series The Turkish Journal of Pediatrics
spelling doaj-art-9c65e5edff7b4b00a243c569814a7d932025-08-20T02:01:47ZengHacettepe University Institute of Child HealthThe Turkish Journal of Pediatrics0041-43012791-64212016-12-0158610.24953/turkjped.2016.06.015Treatment of a patient with Kawasaki disease associated with selective IgA deficiency by continuous infusion of cyclosporine A without intravenous immunoglobulinTatsuya Anzai0Takaomi Minami1Tomoyuki Sato2Sadahiro Furui3Takanori Yamagata4Department of Pediatrics, Jichi Medical University, Yakushiji, Japan.Department of Pediatrics, Jichi Medical University, Yakushiji, Japan.Department of Pediatrics, Jichi Medical University, Yakushiji, Japan.Department of Pediatrics, Jichi Medical University, Yakushiji, Japan.Department of Pediatrics, Jichi Medical University, Yakushiji, Japan. Intravenous immunoglobulin therapy is standard for Kawasaki disease (KD) treatment; however, anaphylactic reactions to immunoglobulins are a risk in KD patients with selective IgA deficiency (sIgAD). The therapy for KD associated with sIgAD has not been established. The IgA immune response is believed to play an important role in KD vasculitis. We report the case of a 5-year-old boy with KD and sIgAD treated with intravenous cyclosporine A (CsA, 3.0 mg/kg/day) instead of intravenous immunoglobulin (IVIG). The fever and inflammation immediately resolved without a coronary artery lesion. In KD patients with sIgAD, we believe that an IgA immune response is lacking, which is the reason for milder KD symptoms than in those without sIgAD. This case report aids in clarifying the role of IgA antibodies in KD and provides evidence that CsA is a potential candidate for first-line therapy for patients with KD with contraindications to IVIG. https://turkjpediatr.org/article/view/1178Kawasaki diseasecoronary artery lesioncyclosporine Aselective IgA deficiency
spellingShingle Tatsuya Anzai
Takaomi Minami
Tomoyuki Sato
Sadahiro Furui
Takanori Yamagata
Treatment of a patient with Kawasaki disease associated with selective IgA deficiency by continuous infusion of cyclosporine A without intravenous immunoglobulin
The Turkish Journal of Pediatrics
Kawasaki disease
coronary artery lesion
cyclosporine A
selective IgA deficiency
title Treatment of a patient with Kawasaki disease associated with selective IgA deficiency by continuous infusion of cyclosporine A without intravenous immunoglobulin
title_full Treatment of a patient with Kawasaki disease associated with selective IgA deficiency by continuous infusion of cyclosporine A without intravenous immunoglobulin
title_fullStr Treatment of a patient with Kawasaki disease associated with selective IgA deficiency by continuous infusion of cyclosporine A without intravenous immunoglobulin
title_full_unstemmed Treatment of a patient with Kawasaki disease associated with selective IgA deficiency by continuous infusion of cyclosporine A without intravenous immunoglobulin
title_short Treatment of a patient with Kawasaki disease associated with selective IgA deficiency by continuous infusion of cyclosporine A without intravenous immunoglobulin
title_sort treatment of a patient with kawasaki disease associated with selective iga deficiency by continuous infusion of cyclosporine a without intravenous immunoglobulin
topic Kawasaki disease
coronary artery lesion
cyclosporine A
selective IgA deficiency
url https://turkjpediatr.org/article/view/1178
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AT tomoyukisato treatmentofapatientwithkawasakidiseaseassociatedwithselectiveigadeficiencybycontinuousinfusionofcyclosporineawithoutintravenousimmunoglobulin
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