Altammar Fajer, Lang Bianca
Department of Pediatrics, IWK Health Centre, Dalhousie University, 5980 University Ave, Halifax, NS, B3K 6R8, Canada.
Division of Rheumatology, Department of Pediatrics, IWK Health Centre, Dalhousie University, 5980 University Ave, Halifax, NS, B3K 6R8, Canada.
Pediatr Rheumatol Online J. 2018 Jul 3;16(1):43. doi: 10.1186/s12969-018-0263-8.
Kawasaki Disease (KD), the leading cause of acquired heart disease in children in the developed world, is extremely rare in neonates. We present a case of incomplete KD in a neonate and a review of the literature on neonatal KD.
A previously healthy full term 15 day old Caucasian male with an unremarkable antenatal and perinatal history, presented on Day 2 of illness with fever, rash, irritability, and poor feeding. Examination revealed fever (39.6C), tachycardia, tachypnea, extreme irritability, and a generalized maculopapular rash, but was otherwise normal. His complete blood count, CRP and ESR were normal. Empiric intravenous antibiotics and acyclovir resulted in no improvement. On day 4, he had ongoing fever and developed recurrent apnea, required supplemental oxygen, and was transferred to the pediatric intensive care unit. On day 6, he developed bilateral non-purulent conjunctivitis, palmar erythema, bilateral non-pitting edema and erythema of his feet, and arthritis. His full septic work-up and viral studies were negative. On Day 7 he was treated with intravenous immunoglobulin, and over the next 48 h his symptoms including extremity edema resolved, he no longer required supplemental oxygen, and fever did not recur. On day 9 of illness he had marked thrombocytosis. Subsequently, he developed distal extremity desquamation. Repeated echocardiograms excluded the presence of coronary artery aneurysms (CAA).
We believe this to be a rare case of incomplete KD in a neonate, in which timely IVIG administration led to resolution of the acute illness and may have prevented CAA. A comprehensive English-language medical literature review of KD presenting in the neonatal period revealed only fifteen case reports. Cases often presented with incomplete KD, and a number had atypical laboratory features including a normal CRP in the acute phase, similar to what was seen in our patient. This case and our literature review should increase awareness that KD can rarely occur in neonates, often presenting atypically. Recognizing KD in a neonate enables appropriate treatment that can result in resolution of symptoms and may decrease the risk of cardiac complications.
川崎病（KD）是发达国家儿童后天性心脏病的主要病因，在新生儿中极为罕见。我们报告一例新生儿不完全性KD病例，并对新生儿KD的文献进行综述。
一名此前健康的足月15日龄白种男性，产前和围产期病史无异常，发病第2天出现发热、皮疹、易激惹和喂养困难。检查发现发热（39.6℃）、心动过速、呼吸急促、极度易激惹和全身性斑丘疹，但其他方面正常。他的全血细胞计数、CRP和ESR均正常。经验性静脉使用抗生素和阿昔洛韦后病情无改善。第4天，他持续发热并出现反复呼吸暂停，需要补充氧气，并被转入儿科重症监护病房。第6天，他出现双侧非脓性结膜炎、手掌红斑、双侧足部非凹陷性水肿和红斑以及关节炎。他的全面败血症检查和病毒学检查均为阴性。第7天，他接受了静脉注射免疫球蛋白治疗，在接下来的48小时内，他的症状包括肢体水肿消退，不再需要补充氧气，发热也未复发。发病第9天，他出现明显的血小板增多症。随后，他出现远端肢体脱皮。多次超声心动图检查排除了冠状动脉瘤（CAA）的存在。
我们认为这是一例罕见的新生儿不完全性KD病例，及时给予静脉注射免疫球蛋白导致急性疾病得到缓解，并可能预防了CAA。对新生儿期出现的KD进行的全面英文医学文献综述仅发现15例病例报告。病例常表现为不完全性KD，许多病例具有非典型实验室特征，包括急性期CRP正常，与我们患者的情况相似。该病例及我们的文献综述应提高人们对KD在新生儿中罕见发生且常表现为非典型的认识。在新生儿中识别KD可进行适当治疗，从而缓解症状并可能降低心脏并发症的风险。
