Resistant kawasaki disease in an infant causing giant coronary aneurysms with thrombosis

A. Anass, Hasbaoui Brahim, S. Salah, Yajouri Abdelhakim, A. Rachid, Hassani Amale, A. Aomar, Faid Soumia
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Abstract

A 13 month-old boy male infant admitted for fever, generalized polymorphous exanthema, hyperemic conjunctivae, and diarrhea. The onset of the disease was approximately 2 weeks before the admission with a generalized polymorphous exanthema for which he received antihistaminic treatment, but after 7 days he associated fever, hyperemic conjunctivae, and diarrhea being admitted in the regional hospital. The fever persisted despite the antibiotic and symptomatic treatment, and therefore he was transferred in our hospital. The clinical exam at the time of admission revealed influenced general status, generalized polymorphous exanthema, hyperemic pharynx, bilateral conjunctivitis, and approximately five diarrheic stools per day. The laboratory tests revealed leukocytosis (27,050/mL), with neutrophilia (19,900/mL), anemia (Hb 6.9 g/dL, Htc 19.7%, MEV 68.6 fL, MEH 24 pg), thrombocytosis (1,111,000/ mL), elevated inflammatory biomarkers (CRP An Abstract Giant coronary artery aneurysms that occur in 0.5 to 1% of patients with Kawasaki disease can be fatal if associated with thrombosis. Some patients may show persistent inflammation and fever despite treatment with repeated doses of intravenous immunoglobulin (IVIG), steroids, and aspirin. This report describes an infant boy with resistant Kawasaki disease who presented with extensive coronary artery involvement and coronary thrombosis. His inflammation was not controlled with multiple doses of IVIG, parenteral and oral steroids, or high-dose aspirin, and he finally needed infliximab, a monoclonal antibody against tumor necrosis factor alpha.
一例婴儿的耐药性川崎病导致巨大冠状动脉瘤伴血栓形成
一例13个月大的男婴因发热、全身性多形疹、结膜充血和腹泻入院。患者在入院前约2周发病,伴有广泛性多形性皮疹,并接受了抗组胺药治疗,但7天后,患者出现发热、结膜充血和腹泻,住进了地区医院。尽管抗生素和对症治疗,发烧仍持续,因此他被转移到我们医院。入院时的临床检查显示全身状态受影响,全身性多形样疹,咽部充血,双侧结膜炎,每天约5次腹泻。实验室检查显示白细胞增多(27,050/mL),中性粒细胞增多(19,900/mL),贫血(Hb 6.9 g/dL, Htc 19.7%, MEV 68.6 fL, MEH 24 pg),血小板增多(1,111,000/ mL),炎症生物标志物(CRP)升高。在0.5%至1%的川崎病患者中发生的巨大冠状动脉动脉瘤如果与血栓形成相关,可能是致命的。尽管多次静脉注射免疫球蛋白(IVIG)、类固醇和阿司匹林,一些患者仍可能出现持续的炎症和发烧。本报告描述了一个顽固性川崎病的男婴,他表现出广泛的冠状动脉受累和冠状动脉血栓形成。多剂量IVIG、肠外和口服类固醇或大剂量阿司匹林都无法控制他的炎症,最后他需要英夫利昔单抗,一种抗肿瘤坏死因子α的单克隆抗体。
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