Taliha Öner, Barış Güven, Timur Meşe, Vedide Tavlı, Mehmet Küçük, Nuri Bayram, Rahmi Özdemir, İlker Devrim, Murat Muhtar Yılmazer

Kawasaki disease in Turkish children: a single center experience with emphasis on intravenous immunoglobulin resistance and giant coronary aneurysms

Prompt diagnosis and the administration of intravenous immunoglobulin(IVIG) has reduced the incidence of coronary artery abnormalities (CAA)in Kawasaki Disease (KD). The resistance to treatment and development ofthe coronary sequelae remain the most important problems in KD. We aimedto determine the predicting factors of nonresponse to initial IVIG therapyand to analyze the cases who had giant coronary aneurysms. A total of 120KD cases, including 61 children fulfilling the criteria for KD and 59 withincomplete KD were enrolled into this study. Demographic, laboratory, clinical,echocardiographic characteristics, and treatment regimens were reviewed,retrospectively. The median age of the patients was 33.5 months (range: 3-168months). Coronary artery aneurysms were detected in 35 patients (29%) atthe time of diagnosis. Twenty-eight patients had coronary aneurysms smallor medium in size, one had a large, and seven had giant coronary aneurysms.CAA persisted in 8 cases in the follow-up, all of which were large or giantaneurysms. A ten month-old girl with a giant coronary aneurysm wasreferred to coronary bypass surgery in the subacute phase of follow-up, dueto myocardial ischemia. Eighteen patients were unresponsive to the initialIVIG therapy (%15), of whom 10 were diagnosed as cKD and 8 were iKD.Patients who did not respond to initial IVIG therapy, had higher white bloodcell (WBC) count, higher C-reactive protein (CRP) and lower albumin levelsthan those who did (P

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1. McCrindle BW, Rowley AH, Newburger JW, et al; American Heart Association Rheumatic Fever, Endocarditis, and Kawasaki Disease Committee of the Council on Cardiovascular Disease in the Young; Council on Cardiovascular and Stroke Nursing; Council on Cardiovascular Surgery and Anesthesia; and Council on Epidemiology and Prevention. Diagnosis, Treatment, and Long-Term Management of Kawasaki Disease: A Scientific Statement for Health Professionals From the American Heart Association. Circulation 2017; 135: e927-e999.
2. Kim DS. Kawasaki disease.Yonsei Med J 2006; 47: 759-772.
3. Ogata S, Tremoulet AH, Sato Y, et al. Coronary artery outcomes among children with Kawasaki disease in the United States and Japan. Int J Cardiol 2013; 168: 3825-3828.
4. Newburger JW, Takahashi M, Beiser AS, et al. A single intravenous infusion of gamma globulin as compared with four infusions in the treatment of acute Kawasaki syndrome. N Engl J Med 1991; 324: 1633-1639.
5. Wallace CA, French JW, Kahn SJ, Sherry DD. Initial intravenous gammaglobulin treatment failure in Kawasaki disease. Pediatrics 2000; 105: E78.
6. Xie T, Wang Y, Fu S, et al. Predictors for intravenous immunoglobulin resistance and coronary artery lesions in Kawasaki disease. Pediatr Rheumatol Online J 2017; 15: 17.
7. Ashouri N, Takahashi M, Dorey F, Mason W. Risk factors for nonresponse to therapy in Kawasaki disease. J Pediatr 2008; 153: 365-368.
8. Kobayashi T, Inoue Y, Takeuchi K, et al. Prediction of intravenous immunoglobulin unresponsiveness in patients with Kawasaki disease. Circulation 2006; 113: 2606-2612.
9. Fukazawa R, Kobayashi T, Mikami M. et al. Nationwide survey of patients with giant coronary aneurysm secondary to Kawasaki disease 1999-2010 in Japan. Circ J 2018; 82: 239-246.
10. Lyskina GA, Bokeriya OL, Shirinskaya OG, et al. Giant aneurisms of coronary arteries in the Kawasaki syndrome. Kardiologiia 2017; 57: 76-84.
11. Newburger JW, Takahashi M, Gerber MA, et al; Committee on Rheumatic Fever, Endocarditis, and Kawasaki Disease, Council on Cardiovascular Disease in the Young, American Heart Association. Diagnosis, treatment, and long-term management of Kawasaki disease: a statement for health professionals from the Committee on Rheumatic Fever, Endocarditis, and Kawasaki Disease, Council on Cardiovascular Disease in the Young, American Heart Association. Pediatrics 2004; 114: 1708-1733.
12. Tremoulet AH, Best BM, Song S, et al. Resistance to intravenous immunoglobulin in children with Kawasaki disease. J Pediatr 2008; 153: 117-121.
13. Newburger JW, Sleeper LA, McCrindle BW, et al; Pediatric Heart Network Investigators. Randomized trial of pulsed corticosteroid therapy for primary treatment of Kawasaki disease. N Engl J Med 2007; 356: 663-675.
14. Do YS, Kim KW, Chun JK, Cha BH, Namgoong MK, Lee HY. Predicting factors for refractory kawasaki disease. Korean Circ J 2010; 40: 239-242.
15. Durongpisitkul K, Soongswang J, Laohaprasitiporn D, Nana A, Prachuabmoh C, Kangkagate C. Immunoglobulin failure and retreatment in Kawasaki disease. Pediatr Cardiol 2003; 24: 145-148.
16. Tavli V, Yilmazer MM, Güven B, Meşe T, Oner T, Demirpençe S. Kawasaki hastalığında standart yüksek doz intravenöz gama globulin tedavisine yanıtsızlığın değerlendirilmesi. Türk Kardiyoloji Derneği Arşivi 2010; 38: 20-24.
17. Burns JC, Capparelli EV, Brown JA, Newburger JW, Glode MP. Intravenous gamma-globulin treatment and retreatment in Kawasaki disease. US/Canadian Kawasaki Syndrome Study Group. Pediatr Infect Dis J 1998; 17: 1144-1148.
18. Terai M, Honda T, Yasukawa K, Higashi K, Hamada H, Kohno Y. Prognostic impact of vascular leakage in acute Kawasaki disease.Circulation 2003; 108: 325- 330.
19. Nakano M. Predictive factors of coronary aneurysm in Kawasaki disease correlation between coronary arterial lesions and serum albumin, cholinesterase activity, prealbumin, retinol-binding protein and immature neutrophils. Prog Clin Biol Res 1987; 250: 535-537.
20. Harada K. Intravenous gamma-globulin treatment in Kawasaki disease. Acta Paediatr Jpn 1991; 33: 805- 810.
21. Matsubara T, Ichiyama T, Furukawa S. Immunological profile of peripheral blood lymphocytes and monocytes/macrophages in Kawasaki disease. Clin Exp Immunol 2005; 141: 381-387.
22. Shin J, Lee H, Eun L.Verification current risk scores for Kawasaki disease in Korean children. J Korean Med Sci 2017; 32: 1991-1996.
23. Davies S, Sutton N, Blackstock S, et al. Predicting IVIG resistance in UK Kawasaki disease. Arch Dis Child 2015; 100: 366-368.
24. Ben Chehida A, Ben Messaoud S, Ben Abdelaziz R, et al. High frequency of cardiovascular complications in Tunisian Kawasaki disease patients: need for a further awareness. J Trop Pediatr 2019; 65: 217-223.
25. Suda K, Iemura M, Nishiono H, et al. Longterm prognosis of patients with Kawasaki disease complicated by giant coronary aneurysms: a singleinstitution experience. Circulation 2011; 123: 1836- 1842.