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Pemphigus is an autoimmune bullous dermatosis in which intercellular antibodies are associated with loss of intercellular cohesion. Pemphigus vegetans, a rare variant of pemphigus, is manifested by vegetating plaques, usually in intertriginous areas of patients who probably have an increased resistance to the disease. We report a case of chronic balanitis owing to pemphigus vegetans, and discuss proposed pathogenic mechanisms and treatment.
Pemphigus vegetans is a variant of pemphigus vulgaris with different clinical manifestations. Diagnosis may be difficult because of the varied presentations. A 46-year-old woman with this condition had been treated for Behcet's disease because of oral lesions. On admission, prior to dermatological consultation, she was thought to have an infectious disease. The diagnosis of pemphigus vegetans was confirmed by histological and immunofluorescence studies.
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Pemphigus foliaceus (PF) sera react with a 150 kDa antigen, desmoglein (DG), while pemphigus vulgaris (PV) sera react with a 130 kDa PV antigen. Recent molecular cloning studies have revealed that both DG and PV antigen are members of the cadherin family of cell adhesion molecules and that PV antigen shows very high homology to DG. We have recently noticed that other desmosoal cadherin molecules, desmocollins I and II (DC I/II), are also recognized by some pemphigus antibodies. In the present study we report two cases of Hallopeau type pemphigus vegetans. Immunoblot study revealed that the sera of these cases reacted not only with the PV antigen but also with both DG (PF antigen) and DC I/II. Although the significance of these findings is not known at present, further studies for the antigen profile for more Hallopeau type pemphigus vegetans patients may unravel the pathogenesis of this rare type of pemphigus.
Pemphigus vegetans was diagnosed in a 12-year-old boy based on clinical, histopathological and immunohistological findings. To our knowledge, this is the first case of juvenile pemphigus vegetans to be published in the literature. Suggested treatment with prednisone and azathioprine was refused, and the patient was treated with a decoction of herbs prescribed by a practitioner of traditional Chinese herbal medicine. This treatment gave excellent results. Possible active components of the treatment are discussed.
BACKGROUND: Pemphigus vegetans, a variant of pemphigus vulgaris, constitutes a rare form of all pemphigus cases, and oral involvement is common. Two clinical subtypes of pemphigus vegetans exist, characterized initially by flaccid bullae and erosions (Neumann) or pustules (Hallopeau). Both subtypes subsequently develop into hyperpigmented vegetative plaques with pustules and hypertrophic granulation tissue at the periphery. METHODS: We report three cases of pemphigus vegetans with oral manifestations exclusively. Two patients were male aged 30 and 45 years old, respectively, while one was a 51-year-old female. CONCLUSION: Oral lesions in all cases consisted of erosions and whitish, vegetating plaques. The histopathological characteristics were in all cases identical. The spinous cell layer was characterized by intense acanthosis and by the presence of vesicles between the spinous and basal cell layers. Inside the vesicles exudative elements were observed consisting mainly of eosinophils. In the upper lamina propria severe inflammatory reaction was observed. Streptavidin-biotin immunoperoxidase technique showed in all cases intercellular epithelial deposition of IgG and C3.
BACKGROUND: Pemphigus vegetans, a rare form of pemphigus vulgaris, consists of vegetating plaques localized to flexural areas. Two types, the Neumann and the Hallopeau type, are recognized with their own characteristics. METHODS: Three patients with pemphigus vegetans were examined, two with Hallopeau type and one with Neumann type. The microscopic and immunofluorescence findings were recorded. RESULTS: Two remarkable features were present. In one case pemphigus vegetans was possibly induced by the use of enalapril. Only in three previous cases has enalapril been described in relation to pemphigus. A second case was associated with a malignant lung tumor, a phenomenon which could not be traced in the literature. CONCLUSIONS: Two types of pemphigus vegetans must be distinguished. Induction of pemphigus (also vegetans) is an accepted side effect of captopril. The effect of enalapril on pemphigus is still in debate. To the best of our knowledge, this is the first time that a patient with pemphigus vegetans and a simultaneously occurring internal malignancy is described.
Pemphigus vegetans is a rare variant of pemphigus which is mainly localized in the intertriginous areas (Neumann type, Hallopeau type) or primarily involves the mucous membranes (pyostomatitis vegetans). A 18-year-old patient with erosive stomatitis developed a vegetating plaque with papillomatous and verruciform features in her left axilla. Histopathological examination of the axilla revealed papillomatosis and acanthosis as well as suprabasal clefting with acantholytic cells. By direct immunofluorescent examination, deposits of immunoglobulin IgG and complement (C3) were found in the intercellular space of the epidermis. Serological examination by indirect immunofluorescent techniques was indicative of pemphigus autoantibodies at a titer of 1:40. This case report demonstrates that the classical differentiation of pemphigus vegetans in two types, Neumann type and Hallopeau type, is mainly of historical importance, because both entities may represent variants of the same disease. In addition, other disorders such as IgA pemphigus may also present with vegetating plaques in intertriginous sites.
OBJECTIVE: To investigate the clinicopathological features of oral pemphigus vegetans. METHODS: Seven cases of pemphigus vegetans involving oral mucosa were included in this study. The paraffin sections were analyzed by routine light microscope. RESULTS: There were 5 females and 2 males in this group, with an average age of 52.7 years. The course of diseases was from 3 months to 10 years. Oral manifestations included verrucous or papilliform hyperplasia of oral mucosa, with extensive edema and exudation. Similar skin lesions were found in 5 patients. Under the microscope, pemphigus vegetans showed epithelial thickening with prolonged rete pegs, intraepithelial clefts in suprabasal cells. Eosinophilic microabscesses was often seen within the epithelium. Other microscopic changes were intraepithelial edema and eosinophils infiltration, chronic inflammatory cells infiltration in the lamina propria. CONCLUSION: The microscopic features pemphigus vegetans described in this study are of valuable for pathological diagnosis of oral pemphigus vegetans.
Pemphigus vegetans is a rare variant of pemphigus vulgaris and is characterized by flaccid bullae, which become eroded and form vegetations or papillomatous proliferations, especially in the intertriginous areas. Oral administration of corticosteroids alone does not always induce disease remission in pemphigus vegetans. We report a 44-year-old Japanese man with pemphigus vegetans. Although corticosteroid therapy resulted in healing of the oral ulcers and skin bullae, verrucous vegetations continued to develop. In contrast, by combining corticosteroid with etretinate, verrucous vegetations improved. Thus we propose that the combination therapy of steroid and etretinate might be an effective adjunct in the therapy of pemphigus vegetans.
BACKGROUND: Pemphigus vegetans, a variant of pemphigus vulgaris, most commonly occurs in the flexural area. OBJECTIVE: To describe an unusual case of pemphigus vegetans occurring in a skin graft recipient site and to discuss the possible etiology. METHODS: We present a 41-year-old man who developed vegetating plaques from the graft recipient site of his left leg for 8 months. RESULTS: Based on the histopathologic findings of a skin biopsy, this case was diagnosed as pemphigus vegetans. The patient's condition was successfully treated with systemic corticosteroids and acitretin. CONCLUSION: Our case is unique in its presentation of pemphigus vegetans shortly after a split-thickness skin graft. Physicians should be aware of this entity while differentiating cutaneous lesions arising from a skin graft.
We report the case of a 25-year-old woman with bullous lesions diagnosed as the rare pustular type of pemphigus described by Hallopeau. Direct immunofluorescence studies revealed deposits of IgG and C3 in the intercellular spaces of the epidermis; the indirect immunofluorescence technique revealed circulating antiepithelial IgG antibodies. The skin lesions cleared completely following treatment with systemic corticosteroids and azathioprine. These findings indicate the disease first described by Hallopeau as "pyodermite végétante" does indeed belong to the pemphigus group, being a rare type of pemphigus vegetans.
Pemphigus vegetans of the Hallopeau type was diagnosed in aged dog. Diagnosis was based on (a) history, (b) physical examination, (c) histopathologic examination of affected skin, and (d) response to therapy. Clinical response to immunosuppressive doses of prednisolone was excellent.
Desmosomes of a patient suffering from pemphigus vegetans were studied by electron microscopy and analysed by morphometry, The desmosomes were fewer in the patient's skin than in healthy individuals. The desmosomes of the patient were thin and short, showing pathological ultrastructures, viz. lack of median lines, single-sided attachment plaques and direct cell contact with some thickened cell membranes. These desmosomal changes were more manifest in the involved than in the uninvolved skin. The changes suggest deterioration or defective formation of desmosomes in pemphigus vegetans.
A 49-year old woman had an eruption resembling 'dermatite pustuleuse chronique en foyers a progression excentrique' as initially described by Hallopeau. Numberous vegetating lesions occurred over the genitocrural, pubic, umbilical and submammary regions. The finger-nails showed onycholysis and sterile pustules. No bullae were seen at any time. Histopathological study revealed the presence of acantholysis. Direct immunofluorescent microscopy showed IgG deposits in the intercellular epidermal space. The titer of pemphigus antibody was 1/1,600: the results of these immunofluorescence studies on early pustular lesions as well as vegetating lesions support the view that Hallopeau disease belongs to the 'pemphigus group'. The patient was treated first with oral prednisone (80 mg/day). In twelve months of follow-up care, the vegetating lesions disappeared completely, leaving hyperpigmentation. Prompt recurrence of lesions was observed when corticosteroids were discontinued; reinstitution of this therapy was followed by their regression. A difference exits between pemphigus vegetans of Hallopeau and pemphigus vegetans of Neumann: (1) the Hallopeau type begins with pustules as the primary lesions, followed by vegetations, with a lack of bullae and a preference for flexural areas. Immunofluorescence studies will be the deciding factor in separating pemphigus vegetans of Hallopeau from pyoderma vegetans. (2) The Neumann type is a variant of pemphigus vulgaris in which the denuded areas have a tendency to heal with papillomatous formations. It begins and ends with bullae.
We report an unusual case of vulvar acantholytic dermatosis with features of pemphigus vegetans in a 22-year-old Indian girl who presented with a "warty" lesion in her left labium majus. Following excision of this lesion, she presented with 2 localized recurrent lesions on the left and right labia majora about 2 1/2 years later which were also excised. All 3 biopsies showed histological features typical of pemphigus which included extensive suprabasal acantholysis with bullae formation, prominent villus-like processes at the base of the bullae, focal hyperkeratosis and papillomatosis, and the occasional mixed neutrophil and eosinophilic intraepidermal abscess. IgG and C3 immunofluorescence was positive in the intercellular spaces of the epidermis. These lesions, which probably represent a form of pemphigus vegetans, have not been previously reported as a cause of localized vulvar acantholytic dermatosis.