Multiple Systemic Aneurysms in a Case of Neglected Kawasaki Disease

Roy, Soumya; Biswas, Manoj K

doi:10.1111/jpc.14303

Cited by 5 publications

(6 citation statements)

References 4 publications

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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%

“…[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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“…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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“…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.…”

Section: Discussionmentioning

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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Multiple Systemic Aneurysms in a Case of Neglected Kawasaki Disease

Cited by 5 publications

References 4 publications

Systemic Artery Aneurysms and Kawasaki Disease

Systemic Artery Aneurysms and Kawasaki Disease

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

Neonatal Kawasaki disease with multiple arterial aneurysms: a case report

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