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pemphigoid vegetans is a very rare variant of bullous pemphigoid characterized by vegetative and purulent lesions located in groin , face , axillae , thigh , hands , eyelids and perioral areas .
winkelman and su first described this entity in 1979 and till now 11 cases of this entity have been described in the world literature .
all cases have been described in adults with age ranging between 23 and 89 years .
we report a case of pemphigoid vegetans in a 9-year - old girl and to our knowledge this is the first case of childhood pemphigoid vegetans .
a 9-year - old girl presented with complaints of recurrent multiple pruritic vesiculopustular and hypertrophic raised lesions involving forehead , eyelids , periorbital region , perioral areas , lips , vulva and groin since 1 year .
she was treated with topical steroids and antibiotics which led to partial resolution of the lesions .
after 2 months , similar lesions appeared along with vesicles and pustules involving periorbital areas , eyelids , perioral region , periauricular area and vulva .
she was treated with antibiotics , antiviral agents and topical steroids and showed partial resolution of lesions .
six months later , she presented with another recurrence of lesions to our outpatient department . on examination , multiple nontender , vesicopustules coalescing over a brown to black hypertrophic plaque with adherent yellow crust and erosions were seen involving both eyebrows , eyelids , lips and perioral areas [ figure 2a ] .
antidesmoglein antibody 1 ( anti - dsg-1 ) was 4.6 u / ml ( normal value < 14 ) and antidesmoglein antibody 3 ( anti - dsg-3 ) was 6.62 u / ml ( normal value < 7 ) detected by enzyme - linked immunosorbent assay ( elisa ) . a skin biopsy showed the presence of a subepidermal bulla with the presence of eosinophils and neutrophils . surrounding
direct immunofluorescence of perilesional skin showed linear deposition of igg at the basement membrane zone ( bmz ) .
specimen was further treated with 1 m nacl and section of salt split skin were treated with igg .
the bmz band was seen on the epidermal side [ roof pattern ] of the split confirming the diagnosis of bullous pemphigoid [ figure 3b ] .
( b ) cerebriform tongue showing fissuring ( a ) crusted bullous and purulent lesions on eyelids , lips and perioral regions .
( b ) vegetating lesions on vulva ( a ) subepidermal bulla formation with epidermal hyperplasia of the adjacent epidermis .
( b ) direct immunofluorescence showing linear deposition of igg on the epidermal side salt split skin hence , a diagnosis of pemphigoid vegetans was made based on the clinical , histological and immunofluorescent features .
the patient was put on dapsone 100 mg once daily along with tablet prednisolone 30 mg for 2 weeks followed by tapering of dose by 5 mg every fortnight .
there was complete resolution of lesions with hyperpigmentation , scarring and milia formation [ figure 4 ] .
( a ) complete resolution of the lesions on the face with hyperpigmentation and milia formation ( b ) complete resolution of the lesions on vulva showing hyperpigmentation
bullous pemphigoid is a blistering autoimmune disease of the elderly with very few cases reported in childhood .
pemphigoid vegetans has not been reported in children till now , ours being the first case .
the common causes of these type of lesions are pemphigus vegetans , pyoderma vegetans , pyodermatitis , pyostomatitis vegetans and rarely cicatricial and pemphigoid vegetans .
though these lesions can have overlapping clinical features , histology and immunofluorescence studies will distinguish these conditions from each other . on review of the 11 cases of pemphigoid vegetans reported earlier , the age ranged from 23 to 89 years [ table 1 ] .
all patients presented with intertriginous vegetating plaques but vesicles and pustules were seen in 7 out of 11 cases .
mucosal involvement has been seen in 6 out of 11 cases and serum eosinophilia has been documented in four cases .
site of involvement has been localized in some cases and has been more extensive in others , as in the present case .
they include groin , vulva , axillae , face , eyelids , periorbital areas , perioral areas , lips and umbilicus .
target antigens have been reported in only four cases , one patient reacted with bp230 and the 160 kda , 130 kda and 95 kda proteins , while two other patients have shown the presence of both bp230 and bp180 antigens by immunoblot and elisa technique .
one case has shown features of both pemphigus and pemphigoid vegetans with dual reactivity to both bp230 and dsg1 protein .
our case was similar to other cases regarding site , type of lesions , histological features and direct immunofluorescence ( dif ) pattern .
the presence of cerebriform tongue in our case has not been reported in earlier cases .
topical antibiotic has been used in one case , topical corticosteroids in four cases , topical steroid with antibiotic in three cases , dapsone in one case and oral corticosteroids in one case .
four cases have shown complete resolution , two cases have shown relapse after treatment and three cases have not completely resolved . to conclude , pemphigoid vegetans is a very rare entity marked by vegetative and purulent lesions clinically resembling other vegetative lesions .
summary of previously reported cases of pemphigoid vegetans only 11 cases of pemphigoid vegetans have been reported in the world literature .
pemphigoid vegetans is a very rare type of bullous pemphigoid which usually affects the elderly and has not been reported in children . 
 it shows a clinical resemblance to pemphigus vegetans but has distinct histological and immunopathological features of bullous pemphigoid . a 9-year - old girl presented with recurrent purulent and verrucous vegetating lesions on her forehead , groin and vulva along with scaling , crusted , bullous and purulent lesions on the eyelids , periorbital , periauricular , perioral region and lips . 
 she had oral lesions and a cerebriform tongue . 
 though she showed clinical features of pemphigus vegetans , histology revealed a subepidermal blister with the absence of acantholysis . 
 direct immunofluorescence studies were suggestive of bullous pemphigoid . on clinicopathological correlation , a diagnosis of pemphigoid vegetans was made . 
 she responded well to oral corticosteroids and dapsone therapy with complete resolution of the lesions .