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Clinical and Serological Characterization of Orf-Induced Immunobullous Disease Publisher Pubmed



Yilmaz K1 ; Goletz S2 ; Pas HH3 ; Van Den Bos RR4 ; Blauvelt A5 ; White WL6 ; Bouaziz JD7 ; Zuelgaray E7 ; Daneshpazhooh M8 ; Yancey KB9 ; Goebeler M10 ; Schmidt E1, 2
Authors
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Authors Affiliations
  1. 1. Department of Dermatology, University of Lubeck, Lubeck, Germany
  2. 2. Lubeck Institute of Experimental Dermatology (LIED), University of Lubeck, Ratzeburger Allee 160, Lubeck, 23538, Germany
  3. 3. Department of Dermatology, University of Groningen, Groningen, Netherlands
  4. 4. Department of Dermatology, Erasmus MC, Rotterdam, Netherlands
  5. 5. Department of Dermatology, Oregon Health & Science University, Portland, United States
  6. 6. Aurora Diagnostics, GPA Laboratories, Greensboro, NC, United States
  7. 7. Dermatology Department, AP-HP, St-Louis Hospital, University of Paris, Paris, France
  8. 8. Autoimmune Bullous Diseases Research Center, Razi Hospital, Tehran University of Medical Sciences, Tehran, Iran
  9. 9. Department of Dermatology, UT Southwestern Medical Center in Dallas, Dallas, TX, United States
  10. 10. Department of Dermatology, University of Wurzburg, Wurzburg, Germany

Source: JAMA Dermatology Published:2022


Abstract

Importance: Ecthyma contagiosum, or orf, is a viral zoonotic infection caused by Poxviridae. Although human orf infection is considered to follow a self-limited course, various immunological reactions may be triggered, including immunobullous diseases. In the majority of the latter cases, the antigenic target remained enigmatic. Objective: To characterize the predominant autoantigen in orf-induced immunobullous disease and further describe this clinical entity. Design, Setting, and Participants: This multicenter case series sought to provide detailed clinical, histopathological and immunological characteristics of a patient with orf-induced pemphigoid. Based on this index patient, serological analyses were conducted of 4 additional patients with previously reported orf-induced immunobullous disease. Immunoblotting with extracellular matrix and a recently established indirect immunofluorescence assay for detection of serum anti-laminin 332 IgG were performed. Exposures: The disease course and clinical characteristics of orf-induced immunobullous disease were observed. Main Outcomes and Measures: Orf-induced immunobullous disease is primarily characterized by anti-laminin 332 autoantibodies, predominant skin involvement, and a self-limiting course. The study provides further details on epidemiological, clinical, immunopathological, diagnostic, and therapeutic aspects of orf-induced immunobullous disease. Results: In all 5 patients, IgG1 and/or IgG3 autoantibodies against laminin 332 were identified. The α3, β3, and γ2 chains were recognized in 2, 4, and 1 patient(s), respectively. Conclusions and Relevance: In this case series, laminin 332, a well-known target antigen in mucous membrane pemphigoid, was a major autoantigen in orf-induced immunobullous disease, even though predominant mucosal lesions were lacking in this autoimmune blistering disease. Orf-induced anti-laminin 332 pemphigoid is proposed as distinct clinical entity.. © 2022 American Medical Association. All rights reserved.