Hoarseness as a Presenting Sign in Children With Kawasaki Disease

Leuin, Shelby; Shanbhag, S.; Lago, Denise; Sato, Yuichiro; Sun, Xiaoying; Jain, Sonia; Burns, Jane C.; Tremoulet, Adriana H.

doi:10.1097/inf.0b013e3182a0960b

Cited by 8 publications

(7 citation statements)

References 15 publications

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“…In this study of Latin American children with KD, we found a lower frequency of hoarseness during the acute phase than in the only previous report by Leuin et al 1 .We found a lower frequency of hoarseness during the KD acute phase in our study compared to the previous report in a single US population by Leuin et al . (11.6 % vs 30% respectively), where KD patients with hoarseness were younger than those without it (1.9 vs 3.1 years) 1 . Similarly, we observed that hoarse KD patients were younger (median age 18 vs 26 months).…”

Section: Discussioncontrasting

confidence: 61%

“…Thus, hoarseness could be present even in the absence of a viral co‐infection as a part of a systemic disease with inflammation in immunological diseases. In KD hoarseness patient association with earlier diagnosis and higher band count suggest that hoarseness may be a result of a more severe systemic inflammation involving the larynx 1 …”

Section: Discussionmentioning

confidence: 99%

“…Oropharyngeal manifestations of Kawasaki disease (KD) include erythematous cracked lips, erythematous oropharynx, and strawberry tongue. An increased incidence of hoarseness was first described among acute KD patients in a US cohort and may serve as a clinical sign that helps in the diagnosis of this illness 1 . The objective of this study was to determine the frequency of new‐onset hoarseness among Latin American patients with acute KD.…”

Section: Introductionmentioning

confidence: 99%

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Kawasaki disease presenting with hoarseness: A multinational study of the REKAMLATINA network

Gámez-González

Ulloa‐Gutiérrez

Murata

et al. 2021

Pediatrics International

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Background Recently, hoarseness affecting the supraglottic structure has been reported in Kawasaki disease (KD). The objective of this study was to characterize the frequency of hoarseness in acute KD patients in Latin America. Methods We used prospective data from the multinational Red de Enfermedad de Kawasaki en America Latina (REKAMLATINA) network. A total of 865 patients from 20 countries were enrolled during the 3 year study period. Data on hoarseness were available in 858 (99.2%) patients. The clinical and laboratory characteristics between hoarse and non‐hoarse KD were compared. Results Hoarseness was documented in 100 (11.6%) patients. Hoarse patients were younger than those with KD without hoarseness (median age 18 vs 26 months; P = 0.002) and presented with lower hemoglobin (10.7 g/dL vs 11.3 g/dL; P = 0.040) and hematocrit levels (32% vs 33%, P = 0.048). Conclusions Hoarseness was found to be prevalent as a presenting sign of acute KD in younger children. Anemia may indicate the presence of active inflammation.

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Section: Discussioncontrasting

confidence: 61%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Kawasaki disease presenting with hoarseness: A multinational study of the REKAMLATINA network

Gámez-González

Ulloa‐Gutiérrez

Murata

et al. 2021

Pediatrics International

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“…Although studies have established that host genetics influence susceptibility and disease outcome, the etiologic agent that triggers KD remains elusive, and extensive searches for an infectious agent have not yielded a consistent pathogen. Several lines of evidence suggest an upper respiratory tract portal of entry for the environmental trigger of KD including inflammation of the tissues of the oropharynx and upper airway and infiltration of the bronchi by IgA-secreting plasma cells 6 – 8 . An antecedent minor infection associated with immune activation followed by an environmental exposure to a specific trigger have been postulated as a sequence that can induce the vasculitis in genetically susceptible children 9 , 10 .…”

Section: Introductionmentioning

confidence: 99%

Clustering and climate associations of Kawasaki Disease in San Diego County suggest environmental triggers

Rypdal

Burney

et al. 2018

Sci Rep

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Kawasaki Disease (KD) is the most common cause of pediatric acquired heart disease, but its etiology remains unknown. We examined 1164 cases of KD treated at a regional children’s hospital in San Diego over a period of 15 years and uncovered novel structure to disease incidence. KD cases showed a well-defined seasonal variability, but also clustered temporally at much shorter time scales (days to weeks), and spatiotemporally on time scales of up to 10 days and spatial scales of 10–100 km. Temporal clusters of KD cases were associated with strongly significant regional-scale air temperature anomalies and consistent larger-scale atmospheric circulation patterns. Gene expression analysis further revealed a natural partitioning of KD patients into distinct groups based on their gene expression pattern, and that the different groups were associated with certain clinical characteristics that also exhibit temporal autocorrelation. Our data suggest that one or more environmental triggers exist, and that episodic exposures are modulated at least in part by regional weather conditions. We propose that characterization of the environmental factors that trigger KD in genetically susceptible children should focus on aerosols inhaled by patients who share common disease characteristics.

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“… 3 Clinical features of the illness, including mucosal inflammation of the lips, tongue, and upper airway coupled with cervical lymphadenopathy and hoarseness, all point to a trigger that enters through the nasopharynx. 4 , 5 Specific atmospheric patterns have been linked to clusters of cases of Kawasaki disease that may increase exposure to the causative trigger. 3 …”

mentioning

confidence: 99%

Temporal Clusters of Kawasaki Disease Cases Share Distinct Phenotypes That Suggest Response to Diverse Triggers

Burns

DeHaan

Shimizu

et al. 2021

The Journal of Pediatrics

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Objective To test the hypothesis that cases of Kawasaki disease within a temporal cluster have a similar pattern of host response that is distinct from cases of Kawasaki disease in different observed clusters and randomly constructed clusters. Study design We designed a case-control study to analyze 47 clusters derived from 1332 patients with Kawasaki disease over a 17-yr. period (2002-2019) from a single clinical site and compared the cluster characteristics with two control groups of synthetic KD clusters. We defined a “true” KD cluster as at least 5 patients within a 7-day moving window. The observed and synthetic KD clusters were compared with respect to demographic and clinical characteristics and median values for standard laboratory data using univariate analysis and a multivariate, Rotated Empirical Orthogonal Function Analysis (REOFs). Results In a univariate analysis, the median values for age, coronary artery Z score, white blood cell count, erythrocyte sedimentation rate, C-reactive protein, and age-adjusted hemoglobin for several of the true KD clusters exceeded the 95th percentile for the two synthetic clusters. REOFs revealed distinct patterns of demographic and clinical measures within clusters. Conclusions Cases of Kawasaki disease within a cluster were more similar with respect to demographic and clinical features, and levels of inflammation than would be expected by chance. These observations suggest that different triggers and/or different intensity of exposures result in clusters of cases of Kawasaki disease that share a similar response pattern. Analyzing cases within clusters or cases who share demographic and clinical features may lead to new insights into the etiology of KD.

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Hoarseness as a Presenting Sign in Children With Kawasaki Disease

Cited by 8 publications

References 15 publications

Kawasaki disease presenting with hoarseness: A multinational study of the REKAMLATINA network

Kawasaki disease presenting with hoarseness: A multinational study of the REKAMLATINA network

Clustering and climate associations of Kawasaki Disease in San Diego County suggest environmental triggers

Temporal Clusters of Kawasaki Disease Cases Share Distinct Phenotypes That Suggest Response to Diverse Triggers

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