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Year : 2021  |  Volume : 22  |  Issue : 2  |  Page : 148-150

An atypical manifestation of hand-foot and mouth disease involving the genitalia

1 Department of Dermatology, Mahatma Gandhi Medical College and Research Institute, Sri Balaji Vidyapeeth University, Karaikal, India
2 Department of Dermatology, Mahatma Gandhi Medical College and Research Institute, Sri Balaji Vidyapeeth University; Department of Dermatology and STD, Jawaharlal Institute of Postgraduate Medical Education and Research, Karaikal, India

Date of Submission01-Jul-2020
Date of Decision07-Jul-2020
Date of Acceptance10-Dec-2020
Date of Web Publication31-Mar-2021

Correspondence Address:
Manobalan Karunanandhan
Department of Dermatology, Venereology and Leprology, Mahatma Gandhi Medical College and Research Institute, Sri Balaji Vidyapeeth University, Pillayarkuppam, Puducherry - 607 402
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ijpd.IJPD_113_20

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Hand-foot and mouth disease (HFMD), an acute viral illness, is caused by the Coxsackievirus (CV) that commonly affects young children. We report a case of a 15-month-old female child with painful erosions over the genitalia, following which she developed erythematous macules over the palms and soles. Viral polymerase chain reaction was positive for CV. She was diagnosed to have atypical HFMD. HFMD with atypical manifestations involving the genitalia as initial presentation is a quite unusual or under-reported entity. Prompt recognition of atypical features of HFMD aids in early diagnosis and defers detailed evaluation.

Keywords: Atypical hand-foot and mouth disease, Coxsackie A6, genital erosions

How to cite this article:
Ravikumar B, Munusamy R, Karunanandhan M. An atypical manifestation of hand-foot and mouth disease involving the genitalia. Indian J Paediatr Dermatol 2021;22:148-50

How to cite this URL:
Ravikumar B, Munusamy R, Karunanandhan M. An atypical manifestation of hand-foot and mouth disease involving the genitalia. Indian J Paediatr Dermatol [serial online] 2021 [cited 2021 Jun 24];22:148-50. Available from: https://www.ijpd.in/text.asp?2021/22/2/148/312824

  Introduction Top

Hand-foot and mouth disease (HFMD) is an acute viral illness that commonly affects the pediatric population. Enteroviruses (EVs) are the main pathogens of HFMD, of which Coxsackievirus A16 (CV-A16) and EV-A71 or EV-71 are the common causative agents. However, the number of HFMD cases due to CV-A6 and CV-A10 are on the rise in recent years.[1] The clinical features of HFMD include fever and exanthema characterized by the presence of cloudy vesicles with an erythematous halo that favors the palms and soles. Sometimes, papulovesicles and erosions are noted at these predilected sites. Oral mucosal manifestations may range from petechial spots to multiple tiny ulcers involving the palate, buccal or gingival mucosa, and the tongue. Very rarely, the genital mucosa may also be involved in HFMD. Atypical HFMD presents as a widely distributed rash with varying morphology that makes clinical diagnosis and treatment challenging.[2],[3] We report this case due to the unusual and atypical presentation of HFMD lesions involving the genitalia.

  Case Report Top

A 15-month-old girl otherwise healthy, born of nonconsanguineous marriage presented with a history of fever, painful erosions on the genitalia, and ulcers over the hard palate. There was no history of other skin lesions or prodromal symptoms. On examination, the child was irritable. Physical examination revealed few discrete 2–4 mm sized papulovesicles over the thigh and gluteal region; few erythematous macules were seen over the palms and soles [Figure 1]. There were multiple discrete erosions (measuring 2 mm) and petechial spots noted over the hard palate. Examination of genital mucosa revealed few erosions with minimal slough over the labia majora and minora [Figure 2]. Tzanck smear for multinucleated giant cells was negative. Swabs for polymerase chain reaction (PCR) from oral and vaginal erosions were positive for CV. Hence, the diagnosis of atypical HFMD was made. The child was conservatively managed with supportive care and saline compresses. After one week, there was a spontaneous resolution of the lesions [Figure 3].
Figure 1: Erythematous papulovesicles over right palm (green arrow) (a) and over right sole (b)

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Figure 2: Multiple discrete erosions with surrounding erythema over the hard palate (a), few erosions (yellow arrow) on the genitalia (b) and a vesicle (red arrow) on the right thigh

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Figure 3: Resolved genital lesions and crusted vesicles (white arrow) noted after a week

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  Discussion Top

HFMD, a viral illness caused by some of the EVs such as CV-A16 and EV-71, commonly affects children. HFMD typically manifests as a mild, febrile illness and is characterized by mouth sores and a vesicular (cloudy vesicles) rash involving the palms and soles.[1],[2]

Clinical presentation of HFMD varies from the classic erythematous papules, vesicles, erosions (usually oval-shaped and small) to atypical manifestations that include widespread distribution, varicelliform eruptions, and grouped vesicles.[3]

HFMD lesions commonly have a predilection to involve the hand, feet, and the oral mucosa. The lesions can also affect the elbows, knees and buttocks.[4] These clinical features differentiate HFMD from other common viral exanthems such as varicella, eczema herpeticum, and Gianotti–Crosti Syndrome. However, in cases where the diagnosis is doubtful, further laboratory evaluation such as Tzanck smear (for multinucleated giant cells), viral culture, and PCR can be helpful.

Atypical HFMD is defined when there are symptoms of acute viral infection (such as fevers coughs or diarrhea) with either of the following clinical manifestations: (1) maculopapular rash involving the trunk, buttocks, or facial areas, or (2) large vesicles or bullae affecting any site of the body.[4]

There are few reports of HFMD caused by CV-A6 with extensive cutaneous involvement though they mostly follow a benign course.[3],[5] HFMD caused by CV-A6 often presents with atypical cutaneous signs that include vesiculobullous and erosive eruptions, eczema coxsackium, Gianotti–Crosti-like eruption, petechial and purpuric rash. Sometimes, onychomadesis and acral desquamation can be the only manifestation when the patient presents late.[6] The exact mechanism behind the onset of these eruptions is unknown. Moreover, these atypical presentations often do not portend a worse prognosis as they resolve spontaneously.

HFMD is usually a self-limiting viral illness. However, HFMD caused by EV-71 may sometimes lead to neurologic complications such as aseptic meningitis, encephalitis, or polio-like syndromes in children.[7]

In our case, the child predominantly had painful erosions involving the oral and genital mucosa with subtle cutaneous involvement. The lesions were self-limiting in a few days.

  Conclusion Top

HFMD can present with unusual cutaneous and mucosal manifestations. This case illustrates the importance of recognizing possible atypical presentations of HFMD involving the genitalia. Identifying these atypical presentations may aid in prompt diagnosis of HFMD, amid other viral and bacterial infections.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship


Conflicts of interest

There are no conflicts of interest.

  References Top

Kimmis BD, Downing C, Tyring S. Hand-foot-and-mouth disease caused by Coxsackievirus A6 on the rise. Cutis 2018;102:353-6.  Back to cited text no. 1
Buttery VW, Kenyon C, Grunewald S, Oberste MS, Nix WA. Atypical presentations of hand, foot, and mouth disease caused by Coxsackievirus A6--Minnesota, 2014. MMWR Morb Mortal Wkly Rep 2015;64:805.  Back to cited text no. 2
Mathes EF, Oza V, Frieden IJ, Cordoro KM, Yagi S, Howard R, et al. “Eczema coxsackium” and unusual cutaneous findings in an Enterovirus outbreak. Pediatrics 2013;132:e149-57.  Back to cited text no. 3
Hubiche T, Schuffenecker I, Boralevi F, Léauté-Labrèze C, Bornebusch L, Chiaverini C, et al. Dermatological spectrum of hand, foot and mouth disease from classical to generalized exanthema. Pediatr Infect Dis J 2014;33:e92-8.  Back to cited text no. 4
Huang WC, Huang LM, Lu CY, Cheng AL, Chang LY. Atypical hand-foot-mouth disease in children: A hospital-based prospective cohort study. Virol J 2013;10:209.  Back to cited text no. 5
Lott JP, Liu K, Landry ML, Nix WA, Oberste MS, Bolognia J, et al. Atypical hand-foot-and-mouth disease associated with Coxsackievirus A6 infection. J Am Acad Dermatol 2013;69:736-41.  Back to cited text no. 6
Kim SJ, Kim JH, Kang JH, Kim DS, Kim KH, Kim KH, et al. Risk factors for neurologic complications of hand, foot and mouth disease in the Republic of Korea, 2009. J Korean Med Sci 2013;28:120-7.  Back to cited text no. 7


  [Figure 1], [Figure 2], [Figure 3]


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