A rare case of Kawasaki disease with giant coronary artery aneurysm

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CASE REPORT

A rare case of Kawasaki disease with giant coronary artery aneurysm Prateek Vaswani 1

&

Yatin Arora 1 & Manoj Kumar Sahu 1 & Devagourou Velayoudam 1

Received: 26 May 2020 / Revised: 29 July 2020 / Accepted: 12 August 2020 # Indian Association of Cardiovascular-Thoracic Surgeons 2020

Abstract Kawasaki disease (KD) is an acute, self-limiting vasculitis that occurs in children of all ages. This was first described by Kawasaki in 1967. Spontaneous regression is observed; however, 25% of patients develop coronary artery aneurysm (CAA). These may result in ischaemic heart disease causing myocardial infarction, rupture leading to pericardial tamponade and distal embolization which culminate in sudden cardiac death. Diagnosis of KD relies on clinical suspicion with no gold standard diagnostic test. A case of KD with giant CAA in a 14-year-old female is described with emphasis on challenges pre- and perioperatively. The review provided post description of the case emphasizes on pathophysiology with clinical course of CAA in association with KD and justification of our approach with an insight into newer treatment modalities. Keywords Kawasaki disease . Coronary artery . Aneurysm . Internal thoracic artery . Coronary artery bypass grafting

Introduction Kawasaki disease (KD) is a systemic vasculitis syndrome of unknown aetiology wherein vasculitis is responsible for all manifestations. Although rare, coronary artery aneurysms (CAA) are seen in 25% cases of KD of which 50% either persist or progress with stenosis causing myocardial ischaemia accounting for most of the mortality in KD [1]. The aneurysms which persist beyond the first year are unlikely to regress [1]. Giant CAA can thrombose, calcify and rarely rupture. A case of a female child with giant CAA due to KD is described wherein coronary revascularization with bilateral Internal thoracic artery (ITA) grafting was done. This is followed by literature review focussing on pathophysiology of CAA with KD and treatment with an insight into newer modalities.

Case report A 14-year-old female child presented with complains of progressive dyspnoea and angina on exertion since 5 years of age. Cardiology evaluation confirmed the diagnosis of KD with * Prateek Vaswani [email protected] 1

Department of Cardiothoracic and Vascular Surgery, CTVS office, 7th floor, Cardiothoracic and Neurosciences Centre, All India Institute of Medical Sciences, Ansari Nagar, New Delhi 110029, India

thrombosed CAA. She was kept on medical follow-up with oral anticoagulants, antiplatelet, beta blocker and nitrates. There was an episode of fungal pneumonia 7 years back for which she required hospitalization, prolonged mechanical ventilation and tracheostomy. She was discharged after a month and subsequently decannulated. After the discontinuation of antifungals, she was tested for fungal infection by two fungal cultures serum galactomannan which were negative. Cardiology evaluation by echocardiography revealed no evidence of fungal endocarditis in subsequent follow-up. Steroids w