Unusual clinical manifestation of linear IgA dermatosis: A report of two cases

Cauza, Karla; Hinterhuber, Gabriele; Sterniczky, Barbara; Brugger, Karin; Pieczkowski, Friederike; Karlhofer, Franz; Wolff, Klaus; Foedinger, Dagmar

doi:10.1016/j.jaad.2004.01.059

Cited by 29 publications

(16 citation statements)

References 38 publications

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“…Although 8 cases of localized LABD have been reported (4)(5)(6)(7)(12)(13)(14)(15), no cases of localized LAGBD have been described. We believe this to be the first case of localized LAGBD whose skin lesions were restricted to the perianal area.…”

Section: Discussionmentioning

confidence: 99%

Localized Linear IgA/IgG Bullous Dermatosis

Shimizu

Natsuga²,

Shinkuma³

et al. 2010

Acta Derm Venerol

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Linear IgA/IgG bullous dermatosis (LAGBD) is an auto-immune blistering disease characterized by the local accumulation of IgA- and IgG-class anti-basement membrane autoantibodies. It typically presents as a generalized pruritic vesiculobullous eruption. No cases of localized LAGBD have yet been reported. We report a case of a 78-year-old man with LAGBD localized to the perianal area. The patient complained of suffering from persistent ulcers around the anus for more than 3 years. Physical examination revealed several blisters and ulcers up to 2-cm in diameter around the anus. No lesions were found elsewhere on the body. Histological analysis of a skin biopsy revealed subepidermal blistering, while direct immunofluorescence showed the linear deposition of IgA and IgG antibodies at the dermoepidermal junction. Indirect immunofluorescence of normal human skin whose layers had been separated using 1M NaCl showed the binding of both IgA and IgG to the epidermal side. Immunoblotting demonstrated the presence of circulating IgA and IgG autoantibodies that bound to a 120-kDa protein. This is the first case of localized LAGBD whose skin lesions were restricted to the perianal region.

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Section: Discussionmentioning

confidence: 99%

Localized Linear IgA/IgG Bullous Dermatosis

Shimizu

Natsuga²,

Shinkuma³

et al. 2010

Acta Derm Venerol

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“…Five previous cases have been reported in the literature, all of them presenting a favorable response to IVIg always given in addition to corticosteroids and or dapsone. [30][31][32][33][34] The different clinical results among the studies could be related to several causes. One important factor could be the IVIg product used in each study 38 ; data were not available in most of the articles.…”

Section: Discussionmentioning

confidence: 99%

High-dose intravenous immunoglobulins for the treatment of autoimmune mucocutaneous blistering diseases: Evaluation of its use in 19 cases

Segura¹,

Iranzo²,

Pablo³

et al. 2007

Journal of the American Academy of Dermatology

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“…Dapsone and sulfonamides are usually given in conjunction with topical glucocorticosteroids, and in patients whose disease cannot be adequately controlled, oral prednisolone (0.5 mg/kg/day) is recommended [2]. Various other drugs including mycophenolate mofetil, mycophenolate sodium, colchicine, cyclosporine, methotrexate, intravenous immunoglobulin, co-trimoxazole, erythromycin, oxacillin, flucloxacillin, dicloxacillin, tetracycline and nicotinamide were successfully applied in single patients and small case studies (table 1) [27,28,29,30,31,32,33,34,35,36,37,38,39,40,41,42,43,44,45,46,47,48,49,50,51,52,53,54,55,56,57,58,59]. No prospective controlled clinical trial has yet been performed in LAD.…”

Section: Discussionmentioning

confidence: 99%

Linear IgA Disease: Successful Application of Immunoadsorption and Review of the Literature

et al. 2010

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Linear IgA disease (LAD) is an autoimmune subepidermal blistering disorder characterized by IgA autoantibodies at the dermal-epidermal junction. Conventional first-line treatments include dapsone with or without oral glucocorticosteroids. Various other therapeutic approaches have been used in refractory patients. Immunoadsorption (IA) has been previously successfully applied in severe and/or otherwise treatment-resistant IgG-mediated immunobullous disorders. Here, we report a patient with a severe generalized LAD in whom adjuvant tryptophan IA was associated with rapid healing of skin lesions. Our observation suggests that IA may also be a helpful adjuvant treatment option for severe LAD.

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Unusual clinical manifestation of linear IgA dermatosis: A report of two cases

Cited by 29 publications

References 38 publications

Localized Linear IgA/IgG Bullous Dermatosis

Localized Linear IgA/IgG Bullous Dermatosis

High-dose intravenous immunoglobulins for the treatment of autoimmune mucocutaneous blistering diseases: Evaluation of its use in 19 cases

Linear IgA Disease: Successful Application of Immunoadsorption and Review of the Literature

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