Cases reported "Pemphigoid, Bullous"

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1/79. Immunoadsorption for the treatment of bullous pemphigoid.

    We treated 2 intractable patients with pemphigoid by absorbent plasmapheresis using dextran sulfate conjugated cellulose columns with an automated regenerating unit. During a 2 week period, this plasmapheresis was performed 4 to 6 times for Cases 1 and 2, respectively. Clinical findings including skin eruptions were remarkably improved, and the titers of antibasement membrane zone antibodies were decreased after the treatment of 1.8 to 4.2 L of plasma. These results suggest that the absorbent column is effective for intractable pemphigoid.
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2/79. Severe subepidermal blistering disorder with features of bullous pemphigoid and herpes gestationis.

    Herpes gestationis (HG) and bullous pemphigoid (BP) are blistering disorders with similar features, including urticarial lesions that progress to blisters and immunodeposition of C3 in a linear pattern at the basement membrane zone. Among their differences, HG is distinguished by its association with pregnancy, the puerperium, or hormonal perturbation. We describe the immunopathologic findings and clinical course in a multiparous woman with a severe blistering eruption. The patient was not pregnant. Malignancy evaluation was negative, and hormonal testing was normal. Histologic examination demonstrated a subepidermal bulla with eosinophils. Direct immunofluorescence showed C3 in a strong linear band at the dermal-epidermal junction. Indirect immunofluorescence demonstrated circulating IgG and IgG3 antibodies to basement membrane zone (epidermal component on salt-split skin), and complement-fixing IgG. immunoprecipitation demonstrated antibodies to a 180-kd keratinocyte protein antigen. By clinical definition, this patient has BP. However, her disease presentation demonstrated features of both BP and HG.
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3/79. Bullous systemic lupus erythematosus with autoantibodies recognizing multiple skin basement membrane components, bullous pemphigoid antigen 1, laminin-5, laminin-6, and type VII collagen.

    BACKGROUND: Bullous systemic lupus erythematosus is a generalized subepidermal blistering skin eruption in patients suffering from systemic lupus erythematosus. Type VII collagen was initially identified as the target antigen. observation: We studied an unusual patient who had bullous systemic lupus crythematosus. The patient fulfilled the criteria of systemic lupus with an antinuclear antibody titer of 1:5120. Immunopathological testing revealed in vivo deposition of all IgG subclasses, secretory IgA1, and both light chains at the patient's skin basement membrane. The in vivo-bound IgG and IgA were localized at the hemidesmosomes and lamina densa. The patient's IgG and IgA circulating autoantibodies labeled both the epidermal roof and the dermal floor of salt-split skin and recognized the hemidesmosomal protein BP230 as well as the full-length native form and the recombinant noncollagenous domain 1 of type VII collagen (anchoring fibril). In addition, the patient's IgG autoantibodies recognized the anchoring filament proteins laminin-5 and laminin-6 (alpha3 chain and gamma2 chain). CONCLUSIONS: We conclude that patients with bullous systemic lupus erythematosus may have autoantibodies to multiple basement membrane components critical for epidermal-dermal junctional adhesion. Possible pathogenic mechanisms in this patient's clinical diseases include provocation of organ-specific disease (bullous disease) by systemic autoimmunity (lupus) and the "epitope spreading" immune phenomenon.
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4/79. Bullous pemphigoid developing during systemic therapy with chloroquine.

    Bullous pemphigoid has been reported to be induced or precipitated by systemic therapy with several drugs, including penicillamine, captopril, frusemide and ampicillin. We report an African male patient with sarcoidosis who was prescribed chloroquine for progressive dyspnoea. After 3 months he developed generalized pruritus which evolved into a widespread bullous eruption with acral targetoid lesions resembling erythema multiforme. The histological and immunofluorescence findings were diagnostic of bullous pemphigoid. The atypical clinical features of this case resemble the phenotype that has been noted in previous reports of drug-induced bullous pemphigoid.
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5/79. A subepidermal bullous eruption associated with IgG autoantibodies to a 200 kd dermal antigen: the first case report from the united states.

    We describe an 81-year-old white man in whom a subepidermal bullous eruption developed that clinically resembled bullous pemphigoid. The eruption promptly responded to oral tetracycline and niacinamide and topical clobetasol. Histologic examination of perilesional skin revealed neutrophilic infiltration with formation of papillary microabscesses and subepidermal cleavage. Direct immunofluorescence showed linear deposition of IgG and C3 along the basement membrane zone. By indirect immunofluorescence, circulating IgG autoantibodies bound exclusively to the dermal side of salt-split normal human skin. Immunoblot analysis demonstrated that the patient's autoantibodies reacted with a 200 kd dermal protein that was different from type VII collagen, the epidermolysis bullosa acquisita autoantigen. This patient represents the first confirmed case from the united states with a recently reported novel autoimmune subepidermal bullous disease associated with IgG autoantibodies to a 200 kd dermal antigen.
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6/79. Generalized pruritic eruption with suprabasal acantholysis preceeding the development of bullous pemphigoid.

    We report a patient who presented with a papular pruritic eruption of a 3-month duration that histologically showed suprabasal acantholysis accompanied of an eosinophilic inflammatory infiltrate that was consistent with the diagnosis of Grover's disease. Later, erythematous plaques and vesicles appeared which showed a histopathological pattern of eosinophilic spongiosis. The direct immunofluorescence (DIF) study showed lineal IgG and C'3 at the dermal epidermal junction which was consistent with the diagnosis of bullous pemphigoid. No anti-intercellular deposits of immunoglobulin g (IgG) or C'3 were observed. We consider that suprabasal acantholysis may represent the early phase of bullous pemphigoid.
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7/79. Drug-induced linear immunoglobulin a bullous disease that clinically mimics toxic epidermal necrolysis.

    Drug-induced linear immunoglobulin a bullous disease is a subepidermal blistering disorder that most commonly occurs after exposure to vancomycin. It can clinically mimic toxic epidermolytic necrolysis. We describe an 87-year-old white woman in whom linear immunoglobulin a bullous disease developed while she was taking vancomycin and phenytoin. A few days after the linear immunoglobulin a bullous disease developed, both medications were discontinued. No new bullae developed, and the eruption completely resolved within 2 weeks. The patient was treated with only topical therapy.
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8/79. Eosinophilic spongiosis in association with bullous pemphigoid and chronic lymphocytic leukaemia.

    We report the onset of an unusual blistering eruption following a diagnosis of B-cell chronic lymphocytic leukaemia (CLL). The histology consistently showed eosinophilic spongiosis, but the diagnosis of bullous pemphigoid was only confirmed after 13 years by repeated immunofluorescence studies. The occurrence of subepidermal blistering diseases in association with B-cell lymphoproliferative disorders is rare; a recent study showed that the majority of these cases are epidermolysis bullosa acquisita, confirmed by immunological studies. Only two cases of immunofluorescence-proven bullous pemphigoid in association with CLL have been previously reported.
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9/79. pemphigus with pemphigoid-like presentation, associated with squamous cell carcinoma of the tongue.

    A 53-year-old woman presented with an inoperable squamous cell carcinoma of the tongue associated with tense large bullae consistent with bullous pemphigoid, preceded by a prodrome of urticarial plaques. The histological findings showed a regenerating subepidermal blister with eosinophils and no acantholysis. Direct immunofluorescence study, however, showed positive staining for IgG and C3 throughout the epidermis consistent with pemphigus. The blistering eruption had no mucosal involvement and responded to low dose corticosteroids. Our patient may represent another presentation of a 'paraneoplastic pemphigus spectrum'.
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10/79. Bullous eruption associated with scabies: evidence for scabetic induction of true bullous pemphigoid.

    Some patients with scabies develop bullae concomitantly with, or subsequently after, the occurrence of scabetic lesions. Although several immunofluorescence studies have demonstrated immunoglobulin deposition in the basement membrane zone of bullous lesions, it remained unclear whether these antibodies are directed to bullous pemphigoid antigens. We clearly show that two scabetic patients with bullous eruptions had circulating antibodies against BP180 and/or BP230 as determined by Western blotting analysis. This is the first report to demonstrate that at least some of the bullous eruptions occurring in scabetics are true bullous pemphigoid.
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ranking = 6
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