An adult case of hand-foot-mouth disease showing severe mucous involvement
Published Web Locationhttps://doi.org/10.5070/D39wz2c40w
Letter: An adult case of hand-foot-mouth disease showing severe mucous involvementDepartment of Dermatology, Kyoto University, Kyoto, Japan
Emi Shikuma, Akihiro Fujisawa, Miki Tanioka, Yumi Matsumura, Yoshiki Miyachi
Dermatology Online Journal 17 (12): 15
Hand-foot-mouth disease is a common childhood viral infection. Generally, small vesicles and erosions occur on the hands, feet and oral mucosa. We report an unusual case of this disease that occurred in an immunocompent adult and resulted in severe erosion on the lips and mucous membranes.
Hand-foot-mouth disease (HFMD) is an acute virus infectious disease that becomes more common among children in summer. It is a disease primarily located on the hands, feet, and oral mucosa and is characterized by the formation of white and oval vesicles, sized 2-3 mm. It usually resolves in several days.
A 22-year-old male was referred to our department for the evaluation of severe erosion on the lips and oral mucosa. He had suffered from a high fever, headache, and cough for 10 days. Five days before his visit at our clinic, he noticed vesicles on the hands and feet. Painful lesions on the oral mucosa developed 4 days before his consultation. The mucosal lesions rapidly eroded, which made eating difficult. His past medical history was unremarkable.
Physical examination revealed edematous lips and oral mucosa with erosion, bleeding, and pus. The lesions were too painful for him to eat and open his mouth (Figure 1a). A few vesicles sized 5-10 mm were scattered on the hands and feet (Figure 1b). He was admitted to our department. Tzanck test of a vesicle on the hand detected no herpetic giant cells. Direct immunofluorescent examination of a mucosal biopsy, specific antibodies against desmoglein 1 or 3, and HIV antibody testing were all negative. Cerebrospinal fluid examination and a CT scan of the whole body found no abnormality.
Histopathological examination of a vesicle on the hand showed an intraepidermal bulla including degenerated keratinocytes (Figure 2). No multinuclear cells or inclusion bodies were found. Neutralization test for coxsackie virus type 16 using his paired serum samples showed a sixty-four fold increase in serum titer; serology for enterovirus type 71 was negative. These results suggested that the cause of the eruptions were coxsackie virus type 16.
His skin lesions were diagnosed as HFMD and was treated with intravenous fluid replacement. The skin lesions resolved without complications in 10 days.
In 2011, an outbreak of HFMD is occurring in Japan for the first time in the past 10 years.
The characteristic features of HFMD in 2011 in Japan are adult cases with severe systemic symptoms, larger skin eruptions, and a wider distribution on the face and buttocks besides the hands and feet. We have also observed similar adult cases of HFMD looking like varicella zoster this year.
In adult cases of HFMD, more severe skin symptoms than children are common. However, hospital admission is usually not necessary . Severe erosion on the upper and lower lips caused us to suspect pemphigus vulgaris or paraneoplastic pemphigus. However, subsequent testing excluded the possibility.
We have no idea why HFMD is exhibiting unusual clinical manifestations in 2011 in Japan. However, a possible explanation is a gene alternation of coxsackie virus type 16. In the previous outbreak in Singapore in 2008, mutations of coxsackie A strains and enterovirus type 71 were suspected as the cause . Moreover, genetic variation of coxsackie virus B5 was reported to be associated with aseptic meningitis in Greece . Another possibility is that coxsackie virus type 6 may have caused the severe symptoms. This virus was detected in half of the HFMD cases in Japan in 2011. The neutralization test for coxsackie virus type 16 is known to show cross reactivity to type 6. We suggest that dermatologists should be aware of the new manifestations of HFMD.
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2. Wu Y, Yeo A, Phoon MC et al. The largest outbreak of hand; foot and mouth disease in Singapore in 2008: the role of enterovirus 71 and coxsackievirus A strains. Int J Infect Dis 2010; 14: e1076-81. [PubMed]
3. Papa A, Dumaidi K, Franzidou F, Antoniadis A. Genetic variation of coxsackie virus B5 strains associated with aseptic meningitis in Greece. Clin Microbiol Infect 2006; 12: 688-91. [PubMed]
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