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DOI 10.3389/fimmu.2026.1907863

Медицинская библиотека · Frontiers in immunology · 2026

Case report: Atypical SJS/TEN-like fulminant pemphigus vulgaris: diagnostic challenges and the potential impact of anti-Dsg1 titers and CMV reactivation.

Verdelli Alice, Daher Rachel, Sanna Luca, Corti Virginia, Magnatta Alessandro, Landini Simone, Donati Marta, Bonanni Irene, Corrà Alberto, Ruffo di Calabria Valentina, Biancamaria Mariotti Elena, Della Bella Chiara, Giovannoni Lorenzo, D'Elios Mario Milco, Moggi Pignone Alberto, Caproni Marzia

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Аннотация

Pemphigus vulgaris (PV) is a chronic autoimmune blistering disease characterized by intraepidermal flaccid blisters of the skin and mucous membranes. Although typically chronic and relapsing, rare fulminant variants have been described. We report a life-threatening case of PV in a previously healthy 55-year-old woman who developed a rapidly progressive, Stevens-Johnson syndrome/toxic epidermal necrolysis (SJS/TEN)-like phenotype with extensive epidermal detachment and mucocutaneous involvement. Histopathological analysis demonstrated suprabasal acantholysis, and direct immunofluorescence showed intercellular IgG and C3 deposition. Serology revealed markedly elevated anti-Desmoglein-1 (107.5 IU/mL) and modest anti-Desmoglein-3 titers (9.6 IU/mL). The clinical course was complicated by Cytomegalovirus reactivation, while malignancy screening remained negative. Treatment with rituximab and intravenous immunoglobulin led to rapid re-epithelialization and clinical stabilization. This case expands the clinical spectrum of PV and underscores diagnostic and therapeutic challenges posed by fulminant autoimmune blistering disease exhibiting overlapping features. As rat-bladder indirect immunofluorescence and extended plakin serology were not available, a paraneoplastic pemphigus (PNP)-spectrum disorder could not be formally excluded, although the clinical, histological, and serological picture strongly favored PV. We propose that markedly elevated anti-Dsg1 antibodies, complement activation, and CMV reactivation may each have contributed to this severe phenotype.

Для цитирования: Verdelli Alice, Daher Rachel, Sanna Luca, Corti Virginia, Magnatta Alessandro, Landini Simone, Donati Marta, Bonanni Irene, Corrà Alberto, Ruffo di Calabria Valentina, Biancamaria Mariotti Elena, Della Bella Chiara, Giovannoni Lorenzo, D'Elios Mario Milco, Moggi Pignone Alberto, Caproni Marzia. Case report: Atypical SJS/TEN-like fulminant pemphigus vulgaris: diagnostic challenges and the potential impact of anti-Dsg1 titers and CMV reactivation.. Frontiers in immunology. 2026. DOI: 10.3389/fimmu.2026.1907863.

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