Abstract
BackgroundIn 2010, we observed children with atypical presentations of hand-foot-mouth disease (HFMD), such as rashes on earlobes and faces, or bullae on trunks and bilateral limbs. Hyperpigmentation later developed as the bullous lesions crusted. Thus, we intended to study the etiology of the illness and the phylogeny of the pathogens.MethodPatients were prospectively enrolled in a tertiary medical center in Taipei, Taiwan. The definition of atypical HFMD includes symptoms of acute viral infection with either of the following presentations: (1) maculopapular rashes presenting on the trunks, buttocks or facial areas, or (2) large vesicles or bullae on any sites of the body. Patients were classified into two groups according to vesicle sizes by two pediatricians at different points in time. The large vesicle group was defined as having vesciculobullous lesions ≥ 1 cm in diameter; the small rashes group had maculopapular rashes < 1cm in diameter. Two throat swabs were collected from each patient for virus isolation and reverse transcription polymerase chain reactions.ResultsWe enrolled 101 patients between March and December 2010. The mean age of the participants was 3.3 ± 3.0 years (median age: 2.5 years, range: 21 days-13.5 years). The ratio of males to females was 1.8 to 1. All samples were enterovirus-positive, including coxsackievirus A6 (80%), coxsackievirus A16 (6%), enterovirus 71 (1%), coxsackievirus A5 (1%) and 12 non-typable enterovirus (12%). Bullous fluid aspirated from 2 patients also grew coxsackievirus A6. Among the patients infected with coxsackievirus A6, 54% (45/81) had bullae, compared to 25% (5/20) of those having non-coxsackievirus A6 infections (P=0.02). Fourteen cases had myoclonic jerks and one boy was diagnosed with febrile convulsions. None had complications or sequelae. Phylogenetic analysis showed the strains in Taiwan in 2010 shared more commonality with strains from Finland in 2009 (GenBank: FJ870502-FJ870508), and were close to those circulating in Japan in 2011 (GenBank: AB649286-AB649291).ConclusionsCoxsackievirus A6 infections may cause atypical manifestations of HFMD, including vesicles or papules on faces or bullae on trunks. These features could provide valuable information to distinguish this versatile enterovirus infection from other virus-induced vesiculobullous diseases.
Highlights
In 2010, we observed children with atypical presentations of hand-foot-mouth disease (HFMD), such as rashes on earlobes and faces, or bullae on trunks and bilateral limbs
Bullous fluid aspirated from 2 patients grew coxsackievirus A6
Among the patients infected with coxsackievirus A6, 54% (45/81) had bullae, compared to 25% (5/20) of those having noncoxsackievirus A6 infections (P=0.02)
Summary
In 2010, we observed children with atypical presentations of hand-foot-mouth disease (HFMD), such as rashes on earlobes and faces, or bullae on trunks and bilateral limbs. Handfoot-mouth disease (HFMD), a common and potentially fatal infectious disease in children, largely relies on clinical manifestations for early diagnosis, including maculopapular or vesicular rashes on soles, palms and buttocks, and oral ulcers in the pharynx [3]. Since spring 2010, we have observed an unusual type of skin lesion presenting on children in Taiwan, including large vesicles or bullae over the limbs, trunks or buttocks and papules on faces, accompanied by fevers, stomatitis, and sore throats. A few children developed onychomadesis, desquamation and skin pigmentation over the areas previously presenting with large vesicles or bullae. These presentations shared several of the traits of HFMD, including vesicular regions on limbs and oral ulcers, yet with distinct vesiculobullous lesions from the typical maculopapular rashes. The association between onychomadesis and enterovirus infection has been reported in coxsackievirus A5 (CVA5), coxsackievirus A6 (CVA6) and coxsackievirus A10 [7,8]
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