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1/7. tetracycline and niacinamide: treatment alternatives in ocular cicatricial pemphigoid.

    Cicatricial pemphigoid (CP) is one of the subepidermal autoimmune bullous dermatoses in which pathologic separation occurs between the epidermis and dermis. Ocular findings characteristic of CP include conjunctival cicatrization, subepithelial fibrosis, and symblepharon formation, which may progress to blindness. Ocular CP (OCP) is usually treated with steroids or immunosuppressive agents, which are problematic in and of themselves within the elderly population, which is most often afflicted with OCP. We describe the utility and effectiveness of therapy with tetracycline and niacinamide in elderly patients with OCP.
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2/7. Benign mucous membrane pemphigoid of the upper aero-digestive tract: rare paraneoplastic syndrome presentation in renal cell carcinoma.

    Benign mucous membrane pemphigoid is a rare autoimmune disorder affecting the upper aero-digestive tract and conjunctivae. This is a case presentation of benign mucous membrane pemphigoid affecting the oral mucosa, pharynx, oesophagus and larynx, leading to cicatricial lesions in the pharynx and larynx, causing dysphagia, hoarseness and stridor. The alternative forms of management for laryngeal scarring due to this disease are explained. The patient was later diagnosed with advanced renal cell carcinoma, raising the possibility of cicatricial pemphigoid manifesting as a paraneoplastic syndrome of underlying renal cell carcinoma.
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3/7. Successful treatment of mucous membrane pemphigoid with etanercept in 3 patients.

    BACKGROUND: mucous membrane pemphigoid (MMP), also known as cicatricial pemphigoid, is a serious, autoimmune, blistering disorder that can result in blindness and other complications as a result of scarring of the mucous membranes. Effective treatment modalities are often toxic. Herein, we describe a novel therapeutic approach that is based on 2 reports in the literature of the successful use of etanercept to treat MMP. OBSERVATIONS: Three patients with MMP were treated with subcutaneous injections of 25 mg of etanercept twice weekly. All 3 patients had oral mucosal involvement, and 1 had severe, recalcitrant, ocular disease. Oral mucosal disease improved in all 3 patients. The patient with ocular involvement experienced stabilization of progression. CONCLUSIONS: Effective treatment modalities for MMP are often toxic. Etanercept may be an effective treatment option for MMP of the oral and ocular mucous membranes. This therapy should be considered as an alternative treatment option for patients who would require other aggressive systemic treatments, such as cyclophosphamide, corticosteroids, azathioprine sodium, and intravenous immunoglobulin.
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4/7. Cicatricial pemphigoid. Identification of two distinct sets of epidermal antigens by IgA and IgG class circulating autoantibodies.

    A patient with severe cicatricial pemphigoid demonstrated both in vivo bound and circulating anti-basement membrane zone antibodies of the IgA and IgG classes. Complement component 3 (C3) was also deposited in the basement membrane zone of lesional skin as well as in normal-appearing buccal mucosa of the patient. However, C1q was absent, while granular deposits of two factors of the alternative complement activating pathway, properdin and properdin factor B, were present only in the basement membrane zone of lesional skin, but not in normal buccal mucosa. Deposition of alternative complement pathway reactants in the lesion suggests that complement activation by IgA was associated with lesion development. Western blot analysis of the patient's serum on electrophoresed cultured keratinocyte antigens identified two distinct sets of epidermal antigens. While IgG bound antigens of 230, 205, 140, and 90 kd, the patient's IgA antibodies bound a distinct set of antigens, 180 and 130 kd. The potential pathogenic role of IgA in cicatricial pemphigoid is discussed.
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5/7. Ocular cicatricial pemphigoid with granular IgA and complement deposition.

    A 65-year-old woman developed chronic redness of both eyes, and, over the ensuing 2 1/2 years, she had progressive conjunctival scarring with symblepharon formation. Other mucosal surfaces were not involved. A conjunctival biopsy specimen 12 months following onset of her disease showed areas of epithelial separation from the basement membrane zone as well as subepithelial chronic inflammation and scarring. Two years later, another conjunctival biopsy specimen showed granular deposition of IgA and C3 along the epithelial basement membrane zone using direct immunofluorescent staining. Electron microscopy confirmed the presence of deposits that were morphologically consistent with antigen-antibody complexes. These findings suggest that antigen-antibody (IgA) immune-complex deposition may provide an alternative pathogenetic mechanism to basement membrane zone autoantibody formation for development of progressive conjunctival scarring.
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6/7. Treatment of cicatricial (benign mucous membrane) pemphigoid with dapsone.

    Treatment of cicatricial pemphigoid is a problem because patients afflicted are elderly, the disease is chronic, systemic agents required for control are potentially toxic (particularly to older patients), and the disease is often in an advanced stage when the diagnosis is established and requires aggressive therapy for control. We selected dapsone as an alternative anti-inflammatory agent because it has provided control in a subset of patients with bullous pemphigoid, five of six who had oral lesions. Twenty-four patients with cicatricial pemphigoid were treated with dapsone. Twenty (83.3%) had partial or complete control with mild to no inflammatory activity. Control was obtained within 4 weeks in eleven of the twenty patients. The use of dapsone was discontinued in two patients because it failed to control their disease and in four patients (two with control) because of drug-related side effects. Cicatricial pemphigoid may be added to the list of dapsone-responsive dermatoses.
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7/7. Cicatricial pemphigoid.

    This case of CP is of interest because of its "not-so-benign" course in this patient, its unusual immunofluorescence patterns, and the need for a complex therapeutic regimen to achieve control. This patient had severe ocular; laryngeal, and oropharyngeal involvement leading to visual problems, hoarseness, and marked weight loss and dehydration. He also had anemia thought to be partially related to dapsone use. We believe the side effects of dapsone, combined with fluid retention due to prednisone therapy, contributed to cardiac failure. The diagnosis of CP is usually established by correlation of clinical findings with immunofluorescence studies. However, indirect immunofluorescence may show strong intercellular antibody binding in the epidermis (ie, pemphigus-like antibodies). Treatment alternatives for patients with CP who have adverse reactions to, or no significant benefit from, conventional agents such as dapsone or prednisone may include immunosuppressive agents such as azathioprine or cyclophosphamide. As this case demonstrates, care of patients having CP involves a cooperative effort from a number of different specialties, including dermatology, primary care, ophthalmology, and otolaryngology.
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