Emergency. 2016; 4 (3): 163-165 CA S E RE P O RT Erythema Multiforme as a Result of Orf Disease; a Case Re- port Tahmine Biazar1, Mehran Shokri1, Hajar Hosseinnia2, Masomeh Bayani1∗ 1. Infectious Diseases and Tropical Medicine Research Center, Babol University of Medical Sciences, Babol, Iran. 2. Clinical Research Development Unit, Rouhani Hospital, Babol University of Medical Science. Babol, Iran. Received: February 2016; Accepted: March 2016 Abstract: Orf is a mucocutaneous disease that occurs when non-intact skin comes into contact with contaminated sheep saliva. The lesions may complicate to lymphangitis or secondary bacterial infection, but systemic complications such as erythema multiforme, maculopapular rash, and generalized lymphadenopathy are rare. In this paper, we present two cases of erythema multiforme following Orf disease. Keywords: Orf virus; erythema multiforme; emergency department; infectious disease medicine © Copyright (2016) Shahid Beheshti University of Medical Sciences Cite this article as: Biazar T, shokri M, Hosseinnia M, Bayani M. Erythema Multiforme as a Result of Orf Disease; a Case Report. Emergency. 2016; 4(3):163-165. 1. Introduction Orf is a mucocutaneous disease caused by double-stranded DNA parapoxviruses that is also known as sheep pox, ec- thyma contagiosum, and contagious pustular dermatitis (1- 3). Human transmission occurs when non-intact skin comes into contact with contaminated sheep and goat saliva and their dead body (4). The incubation period varies from 3 to 10 days and then single or multiple lesions evolve, which arise mostly in the hands and face. At the onset of disease, pri- mary lesions are the papules that gradually progress to nodu- lar patterns. The nodules change into tubercule or crusted form within 4-6 weeks (5). The lesions may complicate to lymphangitis or secondary bacterial infection but systemic complications such as erythema multiforme, maculopapu- lar rash, and generalized lymphadenopathy are rare (6, 7). In this paper, we present two cases of erythema multiforme fol- lowing Orf disease. 2. Case report 2.1. Case 1 A 45-year-old woman was admitted to the emergency de- partment with chief complaint of generalized erythema, low- ∗Corresponding Author: Masomeh Bayani; Infectious Diseases and Tropical Medicine Research Center, Babol University of Medical Sciences, Babol, Iran. Tel:+989113112559; E-Mail:m_baiany@yahoo.com. grade fever, and mild itching. She had a history of hyper- tension and captopril consumption from 3 years ago. On ar- rival, vital signs were stable and only a low fever was detected. On physical examination, disseminated maculopapular rash and target lesions in favor of erythema multiforme were seen. Moreover, there were several purple nodules with a brief fluc- tuation in the proximal phalange of right index finger and distal phalange of third right finger (figure 1). Finger lesions appeared 5 days after contact with a sheep and gradually en- larged during the 25 days before present complaint. Labo- ratory data showed mild leukocytosis. Based on clinical fea- tures, history of contact with sheep, and the high prevalence of disease in Mazandaran province, Iran, diagnosis of Orf disease was made with a high pretest probability for finger lesions, which is complicated to erythema multiforme. Pa- tient was treated with warm compress, and low dose of in- travenous corticosteroid and antihistamine. The generalized eruptions disappeared within five days with complete recov- ery after 6 weeks. 2.2. Case 2 The second case was a 32-year-old woman admitted to the emergency department with generalized maculopapular rash, low fever and sore throat. She had a negative medical and drug history. On arrival, vital signs were stable and physi- cal examination revealed papulopustular lesions on first pha- lange of right thumb and generalized maculopapular rash with target lesions (figure 2). Patient remembered exposure This open-access article distributed under the terms of the Creative Commons Attribution NonCommercial 3.0 License (CC BY-NC 3.0). Downloaded from: www.jemerg.com E-Mail: m_baiany@yahoo.com Biazar et al. 164 Figure 1: Skin lesions of case 1. Figure 2: Skin lesions of case 2. to the head of a sheep several days ago. Same as the first pre- sented case, erythema multiforme following Orf disease was diagnosed and supportive therapy with low doses of intra- venous corticosteroid and antihistamine was administered. The systemic eruptions were cured within 6 days and Orf le- sions disappeared after 5 weeks. 3. Discussion Orf or pustular dermatitis or milker’s nodule is a disease caused by parapaxvirus transmitted to humans via contact with infected goat and sheep (8). The disease occurs most frequently on the fingers, hands and face. After an incuba- tion period of 4-6 days, an erythematous papule appears and possibly goes to nodular and pustular form (9). Milker’s nod- ule can complicate to fever, lymphangitis, lymphadenopa- thy or bacterial super-infection. Rare complications of the disease are vesioculo-pustular eruptions such as erythema multiforme (9, 10). These reactions are considered to be an immune response to the Orf infection (10). Differen- tial diagnoses include pyoderma, herpetic whitlow, cowpox, cat-scratch disease, anthrax, tularemia, tuberculosis, other mycobacteria, syphilis, sportrichosis, keratoacanthoma, and pyogenic granuloma (10). Diagnosis of Orf is usually based on clinical findings and history of exposure of non-intact skin to contaminated sheep and goat saliva and their dead body. Virus isolation, tissue culture and polymerase chain reaction (PCR) in some cases could be helpful but they are expen- sive and difficult (9, 10). Orf is a self-limiting disease and is completely resolved in about 4-6 weeks (2, 10). Conserva- tive therapy and local antiseptic to prevent bacterial supper- infection are recommend, but for large lesions cryotherapy or topical cidofovir cream could be used (2, 11). Low dose systemic steroid and antihistamines are useful in treatment of erythema multiforme (12). Orf disease should be consid- ered as a possible underlying cause of erythema multiforme in endemic area. 4. Appendix 4.1. Acknowledgements We express our thanks to all staff at Rohani Hospital, Babol, Iran for their help and assistance in management of these cases. 4.2. Author’s contributions All authors passed four criteria for authorship contribution based on recommendations of the International Committee of Medical Journal Editors. 4.3. Funding and support None. 4.4. 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ORF FOLLOWED BY ERYTHEMA MUL- TIFORME (CASE REPORT). 2007. 12. Lamoreux MR, Sternbach MR, Hsu WT. Erythema multi- forme. Am Fam Physician. 2006;74(11):1883-8. This open-access article distributed under the terms of the Creative Commons Attribution NonCommercial 3.0 License (CC BY-NC 3.0). Downloaded from: www.jemerg.com Introduction Case report Discussion Appendix References