Complete Regression of Giant Aneurysms in an Infant with Delayed Diagnosis and Refractory Kawasaki Disease via Combination Anticytokine Therapy: Case Report and Review of Similar Cases

Background. Kawasaki disease (KD) is an inflammatory vasculitis and is the most common cause of acquired childhood heart disease in developed countries. Current treatment with intravenous immunoglobulin (IVIG) is often ineffective in patients with delayed or refractory disease. We present a case of...

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Main Authors: Maegan Williams, Lakshmi Nagaraju, Mark Gorelik
Format: Article
Language:English
Published: Wiley 2020-01-01
Series:Case Reports in Rheumatology
Online Access:http://dx.doi.org/10.1155/2020/6249013
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author Maegan Williams
Lakshmi Nagaraju
Mark Gorelik
author_facet Maegan Williams
Lakshmi Nagaraju
Mark Gorelik
author_sort Maegan Williams
collection DOAJ
description Background. Kawasaki disease (KD) is an inflammatory vasculitis and is the most common cause of acquired childhood heart disease in developed countries. Current treatment with intravenous immunoglobulin (IVIG) is often ineffective in patients with delayed or refractory disease. We present a case of combination anticytokine therapy in an infant with delayed and refractory KD. Case Presentation. A 3-month-old infant presented with refractory KD with giant aneurysms after a delayed diagnosis of one month. Use of combination anticytokine therapy led to resolution of giant aneurysms over approximately 6 months. Conclusions. Our case is unique in effective use of anticytokine therapy in very delayed disease with giant aneurysms. Additionally, we review other cases for a broader perspective. Prospective study of anticytokine therapy for patients with giant aneurysms may be warranted.
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spelling doaj-art-ccac617b22aa4d0cbe12a794b9181e522025-02-03T00:59:44ZengWileyCase Reports in Rheumatology2090-68892090-68972020-01-01202010.1155/2020/62490136249013Complete Regression of Giant Aneurysms in an Infant with Delayed Diagnosis and Refractory Kawasaki Disease via Combination Anticytokine Therapy: Case Report and Review of Similar CasesMaegan Williams0Lakshmi Nagaraju1Mark Gorelik2Division of General Pediatrics, Children’s Hospital of San Antonio, Baylor College of Medicine, 333 N Santa Rosa St, San Antonio, TX, USADivision of Cardiology, Children’s Hospital of San Antonio, Baylor College of Medicine, 333 N Santa Rosa St, San Antonio, TX, USADivision of Allergy, Immunology and Rheumatology, Children’s Hospital of San Antonio, Baylor College of Medicine, 333 N Santa Rosa St, San Antonio, TX, USABackground. Kawasaki disease (KD) is an inflammatory vasculitis and is the most common cause of acquired childhood heart disease in developed countries. Current treatment with intravenous immunoglobulin (IVIG) is often ineffective in patients with delayed or refractory disease. We present a case of combination anticytokine therapy in an infant with delayed and refractory KD. Case Presentation. A 3-month-old infant presented with refractory KD with giant aneurysms after a delayed diagnosis of one month. Use of combination anticytokine therapy led to resolution of giant aneurysms over approximately 6 months. Conclusions. Our case is unique in effective use of anticytokine therapy in very delayed disease with giant aneurysms. Additionally, we review other cases for a broader perspective. Prospective study of anticytokine therapy for patients with giant aneurysms may be warranted.http://dx.doi.org/10.1155/2020/6249013
spellingShingle Maegan Williams
Lakshmi Nagaraju
Mark Gorelik
Complete Regression of Giant Aneurysms in an Infant with Delayed Diagnosis and Refractory Kawasaki Disease via Combination Anticytokine Therapy: Case Report and Review of Similar Cases
Case Reports in Rheumatology
title Complete Regression of Giant Aneurysms in an Infant with Delayed Diagnosis and Refractory Kawasaki Disease via Combination Anticytokine Therapy: Case Report and Review of Similar Cases
title_full Complete Regression of Giant Aneurysms in an Infant with Delayed Diagnosis and Refractory Kawasaki Disease via Combination Anticytokine Therapy: Case Report and Review of Similar Cases
title_fullStr Complete Regression of Giant Aneurysms in an Infant with Delayed Diagnosis and Refractory Kawasaki Disease via Combination Anticytokine Therapy: Case Report and Review of Similar Cases
title_full_unstemmed Complete Regression of Giant Aneurysms in an Infant with Delayed Diagnosis and Refractory Kawasaki Disease via Combination Anticytokine Therapy: Case Report and Review of Similar Cases
title_short Complete Regression of Giant Aneurysms in an Infant with Delayed Diagnosis and Refractory Kawasaki Disease via Combination Anticytokine Therapy: Case Report and Review of Similar Cases
title_sort complete regression of giant aneurysms in an infant with delayed diagnosis and refractory kawasaki disease via combination anticytokine therapy case report and review of similar cases
url http://dx.doi.org/10.1155/2020/6249013
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