Additionally, serum neutralizing antibody to HAdV-3 was significantly elevated in both patients 2?weeks after admission, compared with undetectable levels upon admission. persistent fever for 3?days. His data on admission were as follows: WBC, 12,600/l; CRP, 5.54?mg/dl; IFN-, 105.0?pg/ml; and TNF-, 33.6?pg/ml. Although he developed all of the common KD symptoms by day 4, his fever subsided spontaneously on day 6 without IVIG or aspirin. However, he developed a CAY10471 Racemate dilation of the coronary artery in the region of the left circumflex artery bifurcation on day 10. His coronary artery dilation had resolved 3?months after onset. HAdV-3 DNA was detected with PCR in stool samples from both patients, and HAdV3 was isolated from the younger brothers stool sample. Serum neutralizing antibodies to AdV3 were also significantly elevated in both patients, suggesting seroconversion. Conclusions There have been few reports of the simultaneous development of KD in monozygotic twins. Notably, both twins had an acute HAdV-3 contamination immediately before they developed KD. These cases strongly suggest that KD was brought on by HAdV-3 contamination, and they indicate that specific immune responses to some pathogens (such as HAdV-3), arising from genetic susceptibility, play a critical role in the pathogenesis of KD. white blood cells, sodium, aspartate aminotransferase, alanine aminotransferase, C-reactive protein, granulocyte colony-stimulating factor, tumor necrosis factor, coronary artery abnormalities, a transient dilation It has recently been presumed that this etiology of KD combines genetic susceptibility and specific infection, both of which are essential for KD development. Patients with KD seem to have some genetic predisposition, and ethnic differences in the morbidity and familial aggregation of KD have been reported [11]. Some functional single-nucleotide polymorphisms (SNPs) of genes such as inositol 1,4,5-trisphosphate 3-kinase C (have all been reported as triggering pathogens of KD. In general, KD most commonly develops in infants, toddlers, and young children; CAY10471 Racemate adult patients are rarely reported [14]. Most children get infected with common pathogens, such as HAdV, during early childhood, so if these pathogens can trigger KD, this may partially explain the higher KD prevalence in children than that in adults. HAdV contamination itself could be Mouse monoclonal to EphA5 one of the differential diagnoses of KD. HAdV contamination also shows KD-like symptoms such as conjunctival injection, red cracked lips, and cervical lymphadenopathy. However, skin erythema and swollen red palms and soles followed by desquamation are unique features of KD. Additionally, a coronary lesion can allow the definitive diagnosis of KD. In our case, the younger brothers echocardiography revealed a dilation of the left circumflex artery, so we definitively diagnosed him with KD. Several previous reports show that HAdV was detected by PCR in samples from the respiratory tract of patients with KD [15C17]. However, it is difficult to distinguish between latent and acute HAdV contamination by this method. Coincidental isolation of HAdV by PCR may also occur in some patients with KD. In our case, monozygotic twins simultaneously developed KD after acute HAdV-3 contamination. HAdV-3 was detected in stool samples from both patients by PCR, and HAdV3 was directly isolated from the younger brothers stool sample. Additionally, serum neutralizing antibody to HAdV-3 was CAY10471 Racemate significantly elevated in both patients 2?weeks after admission, compared with undetectable levels upon admission. This sero-conversion suggests acute contamination. Furthermore, the elevated serum IFN- and IL-18 levels observed in these patients might also reflect a systemic inflammatory reaction against an acute viral infection. Therefore, we hypothesize that HAdV contamination in children with a genetic susceptibility to KD may abnormally stimulate their innate immunity and evoke a cytokine storm leading to the development of KD. As acute infection can trigger KD, KD may have self-limiting and acute-onset features. Conclusion Our report contributes further evidence that a CAY10471 Racemate specific.