Extensive coronary and systemic arterial aneurysm development in severe refractory Kawasaki disease

Johnston, Niall; Coleman, David; McMahon, Colin J.

doi:10.1017/s1047951116001967

Cited by 6 publications

(6 citation statements)

References 2 publications

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“…Although only 23 patients with SAAs were included in this study (because of the rarity of the disease), it is still the largest sample size we are aware of, and may, in part, reflect the current incidence and early outcome of SAAs. Early screening of SAAs by full-body MRA in patients with medium or giant CAAs can detect clinically significant SAAs more promptly than PA, 6,8,24 which is essential for the management of patients with KD. Careful physical examination for pulsatile masses by palpation of the axillary, brachial, abdominal, and inguinal regions is also important.…”

Section: Discussionmentioning

confidence: 99%

“…[2][3][4] Since the advent of intravenous immunoglobulin (IVIg) therapy, almost all English-language literature on KD-related SAAs consist of case reports revealing that axillary, subclavian, brachial, iliac, and femoral arteries are common sites of involvement. [5][6][7][8][9][10][11][12][13][14][15][16][17][18][19] To date, there are only 2 reports from the same Japanese medical center in which authors reported the incidence of SAAs in patients with KD, but, in fact, less than half of their patients with KD were screened for SAAs; it was during their study that they found that only patients with giant CAAs developed SAAs. 3,4 Although it is a reasonable assumption that there is a concomitant decrease in SAA formation with early IVIg therapy, as well as a decrease in CAAs, there are no published data to support this.…”

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confidence: 99%

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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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Section: Discussionmentioning

confidence: 99%

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confidence: 99%

Systemic Artery Aneurysms and Kawasaki Disease

Zhao

Chu

et al. 2019

Pediatrics

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“…After excluding unrelated studies and duplicates, 60 articles were included. After cross-referencing from the included studies, a total of 61 articles describing CAA with multiple coexisting aneurysms were finally included, [ 3 – 63 ] with a total of 76 patients (age: 37.4 ± 26.5 years; male: 58 [76.3%]). The patients’ ages ranged from 7 weeks to 81 years, and 24 patients were less than or equal to 18 years of age.…”

Section: Methodsmentioning

confidence: 99%

Coronary artery aneurysm combined with other multiple aneurysms at multiple locations

2017

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Background:Coronary artery aneurysm (CAA) with concomitant aneurysms at multiple sites is quite unusual and rare. The characteristics and the etiology of this phenomenon are unknown.Methods:Herein, we present a case with right coronary aneurysm with concomitant abdominal aorta as well as right renal artery aneurysm. A systematic review of the literatures regarding CAA with other coexisting aneurysms at multiple locations was also conducted on Medline and Embase databases.Results:A total of 76 patients (male gender: 58; age: 37.4 ± 26.5) including the present case were included in the final study. The most common etiology of CAA with multiple aneurysms was Kawasaki (43.3%) and atherosclerotic disease (16.4%). CAA was the most frequently found at the right coronary artery (62.7%), following, left anterior descending (51%), left main (43.1%), and left circumflex (35.3%). The most common concomitant aneurysms were abdominal aorta (52.6%) and iliac artery (50%). In addition, 60.5% of the patients had an involved bilateral peripheral artery.Conclusion:CAA with coexisting systemic aneurysms in multiple sites is quite rare. And it usually involves multiple aneurysms at the coronary and bilateral peripheral arteries simultaneously. Currently, there are no general consensus regarding the clinical characteristics, diagnostic method, and treatment of these cases.

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“…In addition to CAAs commonly seen in KD, non-coronary systemic artery aneurysms can also be a sequela of KD. In previous reports on SAAs in KD [ 3 – 5 , 7 , 8 , 10 ], most of SAAs occured in infants, and in patients with untreated KD, IVIG-resistant KD or severe refractory KD, which are all at a higher risk of developing CAAs. In this case, although there was somewhat of a delay in the diagnosis and treatment of the patient, on the 10th day of onset, he responded to one dose of IVIG.…”

Section: Discussionmentioning

confidence: 99%

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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Extensive coronary and systemic arterial aneurysm development in severe refractory Kawasaki disease

Abstract: We describe the case of an 8-week-old infant with late presentation of severe refractory atypical Kawasaki disease. In addition to developing giant coronary arterial aneurysms and coronary thrombosis, she formed extensive bilateral arterial aneurysms throughout her systemic circulation.

Cited by 6 publications

References 2 publications

Systemic Artery Aneurysms and Kawasaki Disease

Systemic Artery Aneurysms and Kawasaki Disease

Coronary artery aneurysm combined with other multiple aneurysms at multiple locations

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

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