Human orf complicated by epidermolysis bullosa acquisita

Abstract

Orf is a DNA parapoxvirus transmitted to humans by contact with infected goats and sheep. Many complications have been reported after orf infection, including erythema multiforme. A few cases of autoimmune bullous dermatosis complicating orf disease have been reported to date. They are usually characterized by tense blister eruptions with or without mucosal involvement; linear deposition of C3, IgG and/or IgA along the basement membrane; and negativity of indirect immunofluorescence analysis and enzyme-linked immunosorbent assay (ELISA) (performed in four of 11 reported cases). These analyses have targeted antigens of bullous pemphigoid, mucous membrane pemphigoid or epidermolysis bullosa acquisita, except one case of mucosal pemphigoid with antilaminin-332 antibodies. We describe the case of a patient who presented with an ulceration on his finger 10 days after direct contact with a lamb during Eid al-Adha. Four weeks later he developed a severe tense blistering eruption associated with mucous membrane erosions. Indirect immunofluorescence analysis using the patient's serum revealed circulating antibasement membrane IgG that bound the dermal side of salt-split skin. ELISA was positive for recombinant immunodominant NC1 domain of type VII collagen. We finally diagnosed epidermolysis bullosa acquisita complicating probable human orf infection.

A viral infection usually seen after feast of sacrifice: Human orf, in this case accompanied with erythema multiforme

“…Pathophysiology of this immune response is unclear but virus mimicry of host proteins (molecular mimicry) or alteration of the basement membrane proteins by virus can be the responsible mechanism [9,10]. But in our case treatment with antibiotic coexisted so EM might be because of a drug associated reaction or a synergistic effect of concurrent drug and virus exposure.…”

“…Cases of EM and more rarely cases with bullous pemphigoid like eruptions, papulovesicular eruptions, epidermolysis bullosa acquisita have been described after or concurrently with orf infection. [1][2][3]5,9,10] In the literature, patients with orfassociated EM were treated with topical and oral corticosteroids, antihistamines and antibiotics. [1][2][3]5] In a report by Erbağcı et al [4].…”

Synthesis of case reports for meta-assessment of causality

“…For discrimination, they provide several observational paradigms as criteria for causal inference, including morphological (eg, exposure distribution) and spatiotemporal pattern correspondences (eg, multiple exposures), as well as correspondence of test results or predictions with outcomes (eg, microbial susceptibility-guided or specific antidote-guided therapy) 2. In addition to dose–effect correspondence, such criteria are valuable for assessing causality in instances of therapeutic effect (eg, ref 3) or disease manifestations (ie, vs coincidence of pathology; eg, ref  4), and it is within this dimension that qualitative synthesis has novel potential.…”

Orf - a potential trigger for self-limiting epidermolysis bullosa acquisita-like blistering

“…In this issue of the BJD Zuelgaray et al . report a further case of widespread pruritic subepidermal blisters with mucous membrane erosions occurring 4 weeks after a presumptive diagnosis of orf.…”