Multiple Giant Aneurysms and Stenoses of the Coronary and Systemic Arteries in an Infant with Kawasaki Disease at the Early Stage of Convalescent Period

Ekici, Filiz; Varan, Birgül; Kocabaş, Abdullah; Erdoğan, İlkay; Eminoglu, Sancar; Aktaş, Doğukan

doi:10.1111/echo.12535

Cited by 9 publications

(5 citation statements)

References 8 publications

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“…The incidence of systemic artery aneurysms (SAAs) confirmed by angiography in untreated KD children has been reported to be approximately 2% [2], which was similar to a recent study conducted by our team in all the KD patients [3]. Previous literature [2][3][4][5][6][7][8][9][10], predominantly case reports, revealed that SAAs in KD patients were generally associated with coronary artery lesions, especially giant CAAs, and were more likely to occur in infants, in patients with untreated KD, intravenous immunoglobulin (IVIG) -resistant or severe refractory KD, which are all related to high risk of developing CAAs. In addition, thrombotic events related to SAAs are rarely reported.…”

Section: Introductionsupporting

confidence: 72%

Giant bilateral axillary artery aneurysms with left complete obstructive thrombus in intravenous immunoglobulin-sensitive Kawasaki disease: a case report

et al. 2021

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Background Kawasaki disease (KD) is a systemic vasculitis that predominantly affects medium-sized arteries. In addition to well-known coronary artery aneurysms (CAAs), peripheral systemic artery aneurysms (SAAs) have also been sporadically reported. In the literatures, SAAs occurred mainly in untreated, intravenous immunoglobin (IVIG)-resistant, or severe refractory KD, and thrombotic events in SAAs were rarely reported. Case presentation A 10-month-old boy with a history of KD was referred to our hospital for suspected pseudoaneurysm of the axillary arteries. Four months prior to presentation, he had persistent fever, conjunctival congestion, and rash. On the 10th day of fever echocardiogram showed biliteral CAAs. He was then diagnosed with KD and given IVIG 2 g/kg and aspirin at a local hospital. His fever and symptoms soon subsided and he was discharged with low dose aspirin and dipyridamole. One month prior to presentation, his parents incidentally palpated swellings in his bilateral axillae. On admission, physical examination revealed a pulsatile swelling in his right axilla and a non-pulsatile swelling in the left with impalpable left brachial and radial pulses, cooler and less active left upper limb than the right one. While the pulses of other three limbs were normal. Ultrasound examination revealed giant bilateral axillary artery aneurysms (AAAs) with massive thrombus in the left. Angiography confirmed giant bilateral AAAs with left AAAs completely occluded and fine collateral vessels connecting to the distal brachial artery, in addition to giant bilateral multiple CAAs without stenoses. The patient was given intravenous prostaglandin for 10 days to allow for formation of collateral circulation, as well as aspirin, low molecular weight heparin (which was switched to warfarin before discharge) and metoprolol. At discharge, the temperature and movement of his left upper limb improved significantly. On follow-up at 7 months, his left upper limb further improved and was similar to the right with no occurrence of cardiovascular events. The images of CAAs and AAAs on echocardiogram and computerized tomography remained the same. Conclusions This case highlights the importance of evaluating peripheral SAAs in KD patients with CAAs, even if their course of treatment appears smooth. For both large non-aortic SAAs and CAAs in KD patients, antithrombotic therapy is of utmost importance.

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Section: Introductionsupporting

confidence: 72%

Giant bilateral axillary artery aneurysms with left complete obstructive thrombus in intravenous immunoglobulin-sensitive Kawasaki disease: a case report

et al. 2021

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“…The child was treated with aspirin and warfarin with restriction of physical activities and referral to a specialist paediatric cardiology unit.Although KD-related arterial aneurysms affecting the aorta, subclavian, brachiocephalic, axillary, intercostal, celiac, mesenteric, hepatic, renal and iliac arteries have been reported, they are rare. [1][2][3][4] This case report demonstrates the importance of a timely diagnosis of KD and makes paediatricians aware of a rare extra-coronary complication.…”

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confidence: 76%

Multiple Systemic Aneurysms in a Case of Neglected Kawasaki Disease

Roy¹,

Biswas²

2019

J Paediatrics Child Health

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While coronary artery aneurysms are well recognised in Kawasaki Disease (KD), extra-coronary aneurysms are rare. We report a child with undiagnosed KD who presented with multiple coronary and extra-coronary aneurysms.A 9-month-old male infant presented with the sole complaint of a progressively increasing soft swelling in the left axillary region. On examination, it was soft, pulsatile, non-tender and had a bruit. The child was otherwise clinically normal. Ultrasonography with colour Doppler of the swelling showed a fusiform left axillary artery aneurysm 1 cm in diameter and 2.3 cm in length. On history, he had prolonged fever when he was 5 months old, associated with redness of his eyes (non-discharging), lips and tongue, and a diffuse reddish truncal rash. The mother also remembered the peeling of skin in the peri-anal area later on. However, the disease was undiagnosed at the time, and after approximately 4 weeks, the fever disappeared spontaneously.Echocardiography demonstrated giant aneurysms in the left main circumflex artery, right circumflex artery and left anterior descending artery, confirming our clinical diagnosis of KD. Computed tomography angiogram indicated fusiform aneurysm of the left brachial artery, 10.5 mm in diameter and 2.2 cm in length, and the right common iliac artery, 9.5 mm × 1.6 cm (Fig. 1). The child was treated with aspirin and warfarin with restriction of physical activities and referral to a specialist paediatric cardiology unit.Although KD-related arterial aneurysms affecting the aorta, subclavian, brachiocephalic, axillary, intercostal, celiac, mesenteric, hepatic, renal and iliac arteries have been reported, they are rare. [1][2][3][4] This case report demonstrates the importance of a timely diagnosis of KD and makes paediatricians aware of a rare extra-coronary complication.

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“…However, many case reports in the current era have also revealed that SAAs are almost always associated with giant CAAs. [5][6][7]11,14,16,22,23 With these data, it is reasonable to assume that even screening patients with giant CAAs may in part reflect the incidence of SAAs in all patients with KD.…”

Section: Discussionmentioning

confidence: 99%

Systemic Artery Aneurysms and Kawasaki Disease

Zhao

Chu

et al. 2019

Pediatrics

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BACKGROUND: Coronary artery aneurysms (CAAs) are a well-known complication of Kawasaki disease (KD), but there are no data on incidence or outcomes of systemic artery aneurysms (SAAs) in the current era. METHODS: From April 1, 2016, to March 31, 2019, we screened for SAAs in 162 patients with KD at risk for SAAs with magnetic resonance angiography or peripheral angiography and analyzed incidence and early outcomes of SAAs. RESULTS: Twenty-three patients had SAAs, demonstrating an incidence of 14.2% (23 of 162) in patients who were screened at 1 month after onset. The proportion of patients with SAAs was estimated to be 2% (23 of 1148) of all patients with KD. The median age at onset of KD with SAAs was 5 months. All patients with SAAs had CAAs, with z scores .8. Of patients with giant CAAs, 38.6% (17 of 44) had SAAs. A total of 129 SAAs occurred in 17 different named arteries. The most common sites for SAAs were the axillary (18.6%), common iliac (12.4%), and brachial (11.6%) arteries. During a median follow-up time of 6 months, 92.9% (79 of 85) of SAAs had some degree of regression, with 80% (68 of 85) of SAAs returning to normal. The overall regression rate was higher for medium to large SAAs than for medium to giant CAAs. CONCLUSIONS: Although the incidence of SAAs may not be as dramatically reduced as we expected compared with previous data, SAAs have a high regression rate during short-term follow-up. WHAT'S KNOWN ON THIS SUBJECT: Studies from the 1980s revealed that systemic artery aneurysms (SAAs) may occur in 0.8% to 2% of patients with Kawasaki disease at 3 months after onset. It is speculated that SAAs are now much less common than before. WHAT THIS STUDY ADDS: SAAs occur in 14.2% of patients at risk for SAAs at 1 month after onset, giving an estimated proportion of patients with SAAs as 2% of all patients with Kawasaki disease. SAAs have a high regression rate during short-term follow-up.

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Multiple Giant Aneurysms and Stenoses of the Coronary and Systemic Arteries in an Infant with Kawasaki Disease at the Early Stage of Convalescent Period

Cited by 9 publications

References 8 publications

Giant bilateral axillary artery aneurysms with left complete obstructive thrombus in intravenous immunoglobulin-sensitive Kawasaki disease: a case report

Giant bilateral axillary artery aneurysms with left complete obstructive thrombus in intravenous immunoglobulin-sensitive Kawasaki disease: a case report

Multiple Systemic Aneurysms in a Case of Neglected Kawasaki Disease

Systemic Artery Aneurysms and Kawasaki Disease

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