Adult-onset Kawasaki disease (mucocutaneous lymph node syndrome) and concurrent Coxsackievirus A4 infection: a case report

Ueda, Yuki; Kenzaka, Tsuneaki; Noda, Ayako; Yamamoto, Yu; Matsumura, Masami

doi:10.2147/imcrj.s90685

Cited by 5 publications

(3 citation statements)

References 15 publications

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“…HAdV, group A streptococcus, Staphylococcus , Bacillus cereus , and Yersinia have all been reported as triggering pathogens of KD. In general, KD most commonly develops in infants, toddlers, and young children; 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.…”

Section: Discussionmentioning

confidence: 99%

Simultaneous development of Kawasaki disease following acute human adenovirus infection in monozygotic twins: A case report

et al. 2017

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BackgroundThe etiology of Kawasaki disease (KD) remains unknown. However, many studies have suggested that specific genetic factors and/or some infectious agents underlie the onset of KD. Previous studies have suggested that human adenovirus (HAdV) is one of the triggering pathogens of KD. Here, we report monozygotic twin boys who sequentially developed KD in conjunction with acute HAdV type 3 (HAdV-3) infection.Case presentationThe patients were four-year-old monozygotic twin boys. The elder brother developed a high fever and was diagnosed with HAdV infection with an immunochromatographic kit for HAdV (IC-kit). He was transferred to our institute after persistent fever for 7 days. On admission, he already fulfilled all the diagnostic criteria for KD. His laboratory data were as follows: WBC, 9700/μl; CRP, 2.42 mg/dl; IFN-γ, 99.8 pg/ml; and TNF-α, 10.9 pg/ml. He received intravenous immunoglobulin (IVIG) and aspirin and responded well, with no coronary artery abnormalities. The younger brother, who was also IC-kit-positive, was hospitalized on the same day as his elder brother after persistent fever for 3 days. His data on admission were as follows: WBC, 12,600/μl; CRP, 5.54 mg/dl; IFN-γ, 105.0 pg/ml; and TNF-α, 33.6 pg/ml. Although he developed all of the typical KD symptoms by day 4, his fever subsided spontaneously on day 6 without IVIG or aspirin. However, he developed a 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 brother’s stool sample. Serum neutralizing antibodies to AdV3 were also significantly elevated in both patients, suggesting seroconversion.ConclusionsThere have been few reports of the simultaneous development of KD in monozygotic twins. Notably, both twins had an acute HAdV-3 infection immediately before they developed KD. These cases strongly suggest that KD was triggered by HAdV-3 infection, 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.

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Section: Discussionmentioning

confidence: 99%

Simultaneous development of Kawasaki disease following acute human adenovirus infection in monozygotic twins: A case report

et al. 2017

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“…The coxsackie virus, which belongs to enteroviruses of small RNA viridine, has been identified as the main cause of viral myocarditis in humans since 1955 ( Dalldorf, 1955 ). Both coxsackie virus B3 (CVB3) and coxsackie virus A4 were identified to correlate with KD ( Rigante et al, 2012 ; Ueda et al, 2015 ), and this type of virus can induce neonatal symptoms similar to viral myocarditis observed in KD ( Verma et al, 2009 ).…”

Section: Involvement Of Pathogens During Kd Pathogenesismentioning

confidence: 99%

Emerging evidence of microbial infection in causing systematic immune vasculitis in Kawasaki disease

Wang,

Zhu,

et al. 2023

Front. Microbiol.

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Kawasaki disease (KD) is a systematic vasculitis that is often complicated by coronary artery lesions and is a leading cause of acquired heart disease in developed countries. Previous studies have suggested that genetic susceptibility, together with an inducing infectious agent, could be involved in KD pathogenesis; however, the precise causative agent of this disease remains unknown. Moreover, there are still debates concerning whether KD is an infectious disease or an autoimmune disease, although many studies have begun to show that various pathogens functioning as critical inducers could activate different kinds of immune cells, consequently leading to the dysfunction of endothelial cells and systematic vasculitis. Here in this review, we attempt to summarize all the available evidence concerning pathogen infections associated with KD pathogenesis. We also discuss the related mechanisms, present a future perspective, and identify the open questions that remain to be investigated, thereby providing a comprehensive description of pathogen infections and their correlations with the host immune system in leading to KD.

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“…Notably, CVA4 was also reported as pathogens in HFMD outbreaks (Hu et al, 2011). Associations between CVA4 infection and herpangina (Cai et al, 2015), mucocutaneous lymph node syndrome (Ueda et al, 2015), and bilateral idiopathic retinal vasculitis (Mine et al, 2017) have also been reported. These clinical findings highlight that CVA4 infections exert a disease burden to global public health, especially for neonates and young children which necessitates a greater understanding of the genetic diversity of this pathogen.…”

Section: Introductionmentioning

confidence: 99%

Genome Analysis of Coxsackievirus A4 Isolates From Hand, Foot, and Mouth Disease Cases in Shandong, China

Wang

Yao

et al. 2019

Front. Microbiol.

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Coxsackievirus A4 (CVA4) is one of the most prevalent pathogens associated with hand, foot and mouth disease (HFMD), an acute febrile illness in children, and is also associated with acute localized exanthema, myocarditis, hepatitis and pancreatitis. Despite this, limited CVA4 genome sequences are currently available. Herein, complete genome sequences from CVA4 strains ( n = 21), isolated from patients with HFMD in Shandong province, China between 2014 and 2016, were determined and phylogenetically characterized. Phylogenetic analysis of the VP1 gene from a larger CVA4 collection ( n = 175) showed that CVA4 has evolved into four separable genotypes: A, B, C, and D; and genotype D could be further classified in to two sub-genotypes: D1 and D2. Each of the 21 newly described genomes derived from isolates that segregated with sub-genotype D2. The CVA4 genomes displayed significant intra-genotypic genetic diversity with frequent synonymous substitutions occurring at the third codon positions, particularly within the P2 region. However, VP1 was relatively stable and therefore represents a potential target for molecular diagnostics assays and also for the rational design of vaccine epitopes. The substitution rate of VP1 was estimated to be 5.12 × 10 -3 substitutions/site/year, indicative of ongoing CVA4 evolution. Mutations at amino acid residue 169 in VP1 gene may be responsible for differing virulence of CVA4 strains. Bayesian skyline plot analysis showed that the population size of CVA4 has experienced several dynamic fluctuations since 1948. In summary, we describe the phylogenetic and molecular characterization of 21 complete genomes from CVA4 isolates which greatly enriches the known genomic diversity of CVA4 and underscores the need for further surveillance of CVA4 in China.

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Adult-onset Kawasaki disease (mucocutaneous lymph node syndrome) and concurrent Coxsackievirus A4 infection: a case report

Cited by 5 publications

References 15 publications

Simultaneous development of Kawasaki disease following acute human adenovirus infection in monozygotic twins: A case report

Simultaneous development of Kawasaki disease following acute human adenovirus infection in monozygotic twins: A case report

Emerging evidence of microbial infection in causing systematic immune vasculitis in Kawasaki disease

Genome Analysis of Coxsackievirus A4 Isolates From Hand, Foot, and Mouth Disease Cases in Shandong, China

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