Systemic arterial aneurysms in a 5 month old male with severe refractory Kawasaki’s disease

Hsu, Benson; Soriano, Ben; Salamet, M. Shahirar; Reeder, Scott B.; Ralphe, J. Carter

doi:10.15761/pd.1000123

Cited by 7 publications

(6 citation statements)

References 8 publications

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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: 77%

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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confidence: 77%

Multiple Systemic Aneurysms in a Case of Neglected Kawasaki Disease

Roy¹,

Biswas²

2019

J Paediatrics Child Health

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“…[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%

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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“…Both neonatal KD and KD-related SAAs are not well recognized due to their rarity, and thus there are only sporadic reports of a few cases in the English language literature concerning either of these issues [13][14][15][16]. To the best of our knowledge, this is the first report of KD in a newborn with both CAAs and SAAs.…”

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confidence: 99%

Neonatal Kawasaki disease with multiple arterial aneurysms: a case report

Zhao

Liang

et al. 2020

Pediatr Rheumatol

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Background: Kawasaki disease (KD) is a medium vessel vasculitis that typically occurs in children aged between 6 months and 5 years. It is extraordinarily rare in the neonatal period. KD-related systemic artery aneurysms (SAAs) have never been reported in neonates. Case presentation: A male infant was transferred to our institution for persistent high-grade fever lasting 16 days. Symptoms started at day 14 of life, and he was admitted to a children's hospital on the second day of fever. Physical examination at the time found no signs suggestive of KD. The only laboratory parameters which were of significance were values suggestive of systemic inflammation. However, his fever persisted and inflammatory markers continued to rise despite 2 weeks of antibiotic therapy. KD as a noninfectious cause of fever was considered when he came to our institution, and echocardiographic findings of left and right medium coronary artery aneurysms (CAAs) confirmed our suspicions. Full-body magnetic resonance angiography also revealed bilateral axillary artery aneurysms. Administration of intravenous gamma globulin resulted in rapid improvement. His fever resolved on the next day and CAAs and SAAs regressed to normal at 6 months and 3 months after diagnosis, respectively. Conclusion: This unique case of incomplete KD highlights the importance of considering KD in neonates with unexplained prolonged fever and reinforces the need to remain vigilant for SAAs in KD.

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Systemic arterial aneurysms in a 5 month old male with severe refractory Kawasaki’s disease

Cited by 7 publications

References 8 publications

Multiple Systemic Aneurysms in a Case of Neglected Kawasaki Disease

Multiple Systemic Aneurysms in a Case of Neglected Kawasaki Disease

Systemic Artery Aneurysms and Kawasaki Disease

Neonatal Kawasaki disease with multiple arterial aneurysms: a case report

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