CASE REPORT Behçet's Disease Detecting by Attacks of Recurrent Epididymo-Orchitis: Case Reports Emre Ulukaradag*, Omur Memik, Bekir Voyvoda, Levent Ozcan Keywords: Behcet syndrome; comorbidity; male; epididymitis; orchitis; etiology. INTRODUCTION Behçet's disease (BD) is a systemic vasculitis characterized by the involvement of joints, gastrointestinal sys-tem, central nervous system, as well as recurrent genital and oral ulcerations, and ocular involvement. BD also may affect any sizes of arteries and veins. Central nervous system and major vessel involvement account for most of the deaths from this disease.(1) Epididymitis, occasionally recurrent, occur in 4% to 11% of patients. We report two BD patients presenting with attacks of recurrent epididymo-orchitis. CASE REPORT Thirty two and twenty seven years old two male patients were admitted to our clinic with sensitivity in left testis in both. In their medical history, recurrent attacks of epididymo-orchitis have occurred since 2-3 weeks ago, but never treated with different types of antibiotic and anti-inflammatory therapies. They also had genital ulcers and recurrent aphthous stomatitis history occurred 5-6 times per year. But they never admitted to any dermatology clinic with these complaints. When they came to our clinic with those symptoms, we made a physical examination and performed blood tests. They had genital ulcers and aphthous stomatitis. Because of epididymo-orchitis is a rare symptom of BD, we performed a pathergy test and dermatology consultation. The pathergy tests were positive in both. Then we diagnosed BD and began colchicine and prednisone (1 mg/kg/day). After the initiation of the therapy epididymo-orchitis resolved spontaneously. DISCUSSION Acute epididymo-orchitis is an inflammatory disease of testis and epididymis. It generally presents unilaterally and occurs because of a specific or nonspecific urinary tract infection like cystitis, prostatitis or urethritis that seeds to the epididymis and testis through the lymphatic vessels or ductus deferens.(2) It can also occur as a result of viral infections, trauma, autoimmune disorders, or amiodarone use. Surgeries on the lower urinary tract, different uro- genital malformations, bladder outlet obstruction may also play a role in the etiology of acute epididymo-orchitis. Our patients have no history like amiodarone use, trauma, urogenital malformation, or surgery. BD was first described by Dr. Hulusi Behçet in 1937 which is characterized by the triad of recurrent aphthous stomatitis, genital ulcerations, and recurrent uveitis.(3) The disease may start with one or more symptoms but others symptoms may gradually appear over the years.(1) Epididymo-orchitis is not the first symptom of BD but appears during follow up. The etiology of BD is unknown, but presumed to be multifactorial, including genetic predispo- sition, infectious triggers, and dysregulation of the immune system. Its prevalence is the highest in Middle East, Asia and Mediterranean region. The prevalence is 80 to 370 cases per 100,000 population in Turkey.(4) The usual age of onset is around 30 years, and male to female ratio shows variation as 1:1 to 1:3.(1) Generally its main clinical manifestation is oral ulcers, and disease occurs mainly between 18 to 40 years.(4) There is no specific test for the definite diagnosis of BD, it depends on the clinical criteria. The criteria of the International Study Group have been widely accepted since 1990.(3) According to them, the presence of oral ulcerations in addition to the presence of two criteria from among recurrent genital ulcerations, skin lesions, ocular lesions or positive pathergy test is sufficient for diagnosis of BD.(3) In our cases, the presence of oral ulcers, positive pathergy test, and genital ulcer confirmed the diagnosis of BD. These two patients also had epididymo-orchitis as a rare symptom of BD. The interesting part of our cases is that, untreated epididymo-orchitis with antibiotic therapy might be a symptom of BD, and in our cases the way to deter- mine this disease was began from this point. CONCLUSIONS In any patient with epididymo-orchitis, BD should be remembered and investigations for other symptoms of the disease especially in regions with a high prevalence of it should be made. Department of Urology, Derince Training and Research Hospital, Kocaeli, Turkey. *Correspondence: Department of Urology, 41900 Derince Training and Research Hospital, Kocaeli, Turkey. Tel: +90 262 3178000. Fax: +90 262 2334641. E- mail: eulukaradag@gmail.com. Received April 2015 & Accepted July 2015 Vol 12 No 05 September-October 2015 2379 CONFLICT OF INTEREST None declared. REFERENCES 1. Saleh Z, Arayssi T. Update on the therapy of Behçet disease. Ther Adv Chronic Dis. 2014;5:112-34. 2. Banyra O, Shulyak A. Acute epididymo- orchitis: staging and treatment. Cent European J Urol. 2012;65:139-43. 3. Pektaş A, Devrim I, Beşbaş N, Bilginer Y, Cengiz AB, Ozen S. A child with Behçet's disease presenting with a spectrum of inflammatory manifestations including epididymoorchitis. Turk J Pediatr. 2008;50:78- 80. 4. Saadoun D, Wechsler B. Behçet's disease. Orphanet J Rare Dis. 2012;7:20. Behçet's Disease Epididymo-Orchitis-Ulukaradag et al. Case Report 2380