Stesura Seveso 151Archivio Italiano di Urologia e Andrologia 2015; 87, 2 SHORT COMMUNICATION Different presentation types of primary Brucella epididimo-orchitis Huseyin Aydemir 1, Gokcen Budak 2, Salih Budak 3, Orcun Celik 3, Okan Yalbuzdag 3, !brahim Keles 4 1 Department of Urology, Ministry of Health Sakarya Teaching and Research Hospital, Sakarya, Turkey; 2 Department of Infectious Disease Clinic, Tire State Hospital, Sakarya, Turkey; 3 Tepecik Educational and Research Hospital, Department of Urology, Izmir, Turkey; 4 Department of Urology, School of Medicine, Afyon Kocatepe University, Afyonkarahisar, Turkey. Brucellosis is a zoonotic disease that involved genitourinary system in 2-20% and most commonly cause single sided epididymo-orchitis. In our country Brucella is an endemic disease and causes serious and different diagnosis of acute scrotum and epididy- moorchitis. In this paper six cases of epididymo-orchitis cases which were resistant to classical treatment were dis- cussed according to clinical and laboratory findings. We describe different types of presentation of Brucella epi- didymoorchitis with diagnosis and treatment modalities. KEY WORDS: Brucellosis; Testis; Epididymo-orchitis; Acute scrotum; Infection. Submittec 5 November 2014; Accepted 28 January 2015 Summary No conflict of interest declared. Patient developed joint and muscle pain lately. At physi- cal examination, body temperature was 37.2°C, swelling on right testicle with extensive tenderness was present, epididymis was hard, scrotum was erythematous and local temperature increased. Examination of other sys- tems was normal. Scrotal color Doppler ultrasonography reported findings consistent with right epididymo-orchi- tis. Laboratory findings included leucocytes 8700/mm3, sedimentation rate 27 mm/h, CRP 30 mg/L and tube agglutination test positive at 1/160 titers. There was no growth in blood cultures. Patient was treated with rifampicin 600 mg/day, doxycycline 200 mg/day and anti-inflammatory treatment for 6 weeks. Symptoms were regressed after first week of treatment. There was no recurrence on the follow up. Case 2 (septicemia) A 63 year old male patient working with farm animals, presented with fever, night sweating and joint pain for 15 days For the last 2 days he had dysuria, swelling in the right testicle and pain. Physical examination showed 37.8°C body temperature, right epididymis very tender and swelling of the testicle, local erythema of the scro- tum with temperature increase. Physical examination of other systems was normal. Laboratory results showed leucocyte count 13500/mm3, sedimentation rate 67 mm/h, CRP 70 mg/L, Brucella tube agglutination test positive for 1/640 titer. Brucella spp. growth was docu- mented in blood cultures. Whereas there was no growth in urine culture. Patient’s treatment was planned for 6 weeks with rifampicin 600 mg/day, doxycycline 200 mg/day and anti-inflammatory treatment but in the sec- ond week of his treatment testicular pain was not regressed and patient continued to have frequent fever (38°C). One g/day streptomycin IM was added to treat- ment for two weeks and treatment was finished up to 6 weeks. After addition of streptomycin on the 3rd day of treatment patient complaints were relieved dramatically. There was no relapse in 18 months follow up. Case 3 (acute scrotum, septicemia) A 27 year old male farmer presented with acute left tes- ticular pain, fever, shivering, nausea and vomiting start- DOI: 10.4081/aiua.2015.2.151 INTRODUCTION Brucellosis is spread to humans by infected or contami- nated animals and less than sufficiently pasteurized milk and milk products (1, 2). Brucellosis can infect many organs and present with different clinical symptoms (3). Brucellosis infects genitourinary system in 2-20% and most commonly causes single sided epididymo-orchitis (3). Brucella epididymoorchitis (BEO) generally causes acute clinical symptoms (78%) (3-5). Differential diag- nosis of the disease with other emergency urological con- ditions causing acute scrotum (testicular torsion, testicu- lar tumors, etc.) must be conducted precisely to prevent consequences of surgery. This paper is aimed to describe differential diagnosis of epididymo-orchitis cases that are resistant to classical treatment or recurrent considering diagnosis and treat- ment of Brucellosis that still maintains its importance. It also calls attention to different clinical pictures of Brucella epididymoorchitis. CASE REPORTS Case 1 (unsuccessful treatment) A 42 years old male patient with right testicular pain, edema and fever started 2 months before was treated withoute success for recurrent orchitis in another center. Aydemir _Stesura Seveso 02/07/15 11:26 Pagina 151 Archivio Italiano di Urologia e Andrologia 2015; 87, 2 Aydemir Huseyin, Budak Gokcen, Budak Salih, Celik Orcun, Yalbuzdag Okan, Keles !brahim 152 ed one day before. At physical examination his body temperature was 38.7°C and blood pressure was 90/60 mmHg, he had minimal swelling of left testis, severe ten- derness whereas examination of other systems was nor- mal. Laboratory findings showed leucocyte count 21000/mm3, sedimentation rate 50 mm/h, CRP 77 mg/L. Scrotal color Doppler ultrasonography revealed finding of left epididymo-orchitis. Patient was hospitalized, monitored and treated with 2 g/day ceftriaxone. On the 3rd day of treatment, symptoms and vital findings were not recovered enough, left testicular edema increased dramatically and erythema and edema involved the left side of scrotum. Brucella was suspected and Coombs test for Brucella was positive, tube agglutination test was pos- itive for 1/320 titer. In blood culture Brucella spp. growth was observed. Patient was treated with rifampicin 600 mg/day, doxicycline 200 mg/day and streptomycin 1 gr/day IM. Vital findings were normalized after the 2nd day of treatment. Testicular findings recessed after 2 weeks. Patient’s treatment was completed in 6 weeks (streptomycin at 15th day). Patient did not have any recurrence orchitis but due to recurrent arteritis patient was transferred to specialized clinic. Case 4 (nonspecific epididmoorchitis) A 50 years old male patient presented with dysuria and right testicular pain for 6 weeks. At physical examination tenderness of right testicular and inguinal canal was present, vital findings and other systems examination was normal. In laboratory findings leucocyte count was 4500/mm3, sedimentation rate was 12 mm/h and CRP was 9 mg/L. Scrotal color Doppler ultrasonography showed findings consistent with right epididymo-orchi- tis. Patient was treated with ciprofloxacin 100 mg/day. At the 4th day of treatment patient presented again with high fever and swelling of right testicle. Physical exami- nation revealed swelling of right testicle, erythema and temperature increase at the right side of the scrotum. His body temperature was 38.2°C. Patient detailed history revealed that he was treated for Brucellosis 6 months prior. Brucellosis Coombs test was positive and tube agglutination test was positive for 1/160 titer. There was no growth in blood and urine cultures. Patient was treat- ed with rifampicin 600 mg/day, doxycycline 200 mg/day and anti-inflammatory drugs for 6 weeks. His symptoms were diminished after the first week of treatment. There was no recurrence in 6 months follow up of the patient. Case 5 (bilateral epididymoorchitis) A 22 years old male patient presenting with long lasting bilateral testicular pain. Physical examination showed minimal tenderness of both testicles, vital findings and other system examination were normal. Laboratory find- ings showed no specific findings in urinary analysis, leu- cocyte count 4100/mm3, sedimentation rate 8 mm/h, and CRP 6 mg /L. Scrotal color Doppler ultrasonography revealed bilateral testicular increased blood flow. Patient’s family was treated for Brucellosis, so Brucellosis panel was studied. Rose Bengal test was positive and tube agglutina- tion test was positive for 1/160 titer. There was no growth in blood or urine culture. Patient was treated with rifampicin 600 mg/day, doxycycline 200 mg/day and anti- inflammatory drugs for 6 weeks. Patient’s symptoms were decreased after the 2nd week of the treatment. There was no recurrence in the 3 month follow up of the patient. Case 6 (testicular tumor suspicion) A 30 years old male patient presented to the urology clinic with a left testicular painful mass. Physical exami- nation revealed left testicular stiffness at palpation. Examination of other system was normal. There was no significant clinical or family history. Laboratory findings included leucocyte count 10200/mm3, CRP 72 mg/L, α- feto protein (AFP) 1.8 (< 13.4), BHCG < 1.2. Emergency scrotal ultrasonographyshowed an hypodense 31.8 mm solid mass of left testicle. After a preliminary diagnosis of seminoma, magnetic resonance imaging (RMI) was scheduled that demonstrated unclear demarcation of the mass with heterogeneous pattern that was reported as consistent with epididymo-orchitis (Figure 1). Brucella tube agglutination test was positive for 1/250 titer. There was no growth in blood culture. Patient was treated with rifampicin 600 mg/day and doxycycline 200 mg/day. Pain and swelling regressed and scrotal ultra- sonography 2 week after treatment showed dramatic regression. Ultrasonography conducted 3 months later showed further decrease of mass size and at 1 year follow up ultrasonography findings were normal. There was no recurrence at 2 years follow up. DISCUSSION Brucellosis is very rare in developed countries. But it is an endemic zoonotic disease in the Mediterranean basin and Middle East (2). Turkey is in the Brucellosis endemic region and its incidence is 2-6% (6). In systemic brucel- losis, epididymo-orchitis is most commonly seen as uni- lateral (3, 6, 7). In a study of 12 cases with genitourinary Figure 1. Left testicular heterogeneous mass, consistent with epididymoorchitis. Aydemir _Stesura Seveso 02/07/15 11:26 Pagina 152 153Archivio Italiano di Urologia e Andrologia 2015; 87, 2 Different presentation types of primary Brucella epididimo-orchitis complications due to Brucellosis, 10 cases presented with epididymoorchitis (5). In the same study the ten BEO patients were compared with 15 non-specific epididy- moorchitis (EO) cases and BEO was characterized by long-lasting clinical presentation, history of unpasteur- ized milk consumption, lower urinary system symptoms, normal urinary analysis and frequent leukocytosis (5). Some tests are required for the diagnosis of brucellosis, in addition to patient history and physical examination (rose bengal, tube agglutination, Coombs test, blood cul- ture). Values of tube test higher of 1/160 titer and blood culture positivity are important for diagnosis (8). Delayed diagnosis and treatment, can lead to various complications (e.g., testicular abscess, atrophy, necrosis and infertility) (9). It is very difficult to distinguish BEO from non-specific EO (10). Detailed history from the patient is important for diagnosis in case of EO unre- sponsive to conventional therapy, that should bring to mind brucella EO. Acute epididymo-orchitis is a frequent disease in urolo- gy clinic and one of the causes of acute scrotum. Differential diagnosis of acute scrotum includes testicular torsion, testicular tumors, appendicular testicular torsion and testicular trauma. Diagnosis of torsion and tumor are crucial because they require fast treatment, so futher radiological tests must be considered. In our 6th case, even though ultrasonography diagnosis was consistent with a testicular tumor MRI results demonstrated epi- didymoorchitis. Tumor markers in the normal range and tube agglutination test consistent with Brucellosis helped the clinical diagnosis. In consideration of the association of tumor and epididymo-orchitis, frequent and close fol- low up is suggested. World Health Organization recommends a 45 day course of oral doxycycline 200 mg/day and streptomycin 1 g/day IM for Brucellosis treatment. An alternative is a 45 day course of oral rifampicin 15 mg/kg/day (600-800 mg) and doxycycline 200 mg/day (11). In cases unre- sponsive to medical treatment orchiectomy is practiced. Afsar et al. reported 2 cases requiring orchiectomy in a 13 cases study with doxycycline and rifampicin treat- ment (12). 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Correspondence Aydemir Huseyin, MD Department of Urology, Ministry of Health Sakarya Teaching and Research Hospital, Sakarya, Turkey Gokcen Budak, MD Department of Infectious Disease Clinic, Tire State Hospital Sakarya, Turkey Salih Budak, MD (Corresponding Author) salihbudak1977@gmail.com Orcun Celik, MD Okan Yalbuzdag, MD Tepecik Educational and Research Hospital, Department of Urology 35140 Izmir, Turkey !brahim Keles, MD Department of Urology, School of Medicine, Afyon Kocatepe University Afyonkarahisar, Turkey Aydemir _Stesura Seveso 02/07/15 11:26 Pagina 153