Brucellar epididymo-orchitis: a retrospective study of 25 cases

Abstract

Objective: Brucellosis is a multisystemic disease which may affect all organs. Epididymo-orchitis is the most common form of genitourinary involvement. The aim of this study was to present our eight-year experience regarding the management of patients with brucellar epididymo-orchitis (BEO).

Materials and method: The medical records of male brucellosis patients treated in two centers, between 2010 and 2018 were analyzed retrospectively. The diagnosis of epididymo-orchitis was made when the patients had scrotal pain, swelling, and enlarged tender testicles and/or epididymis on clinical examination. Brucellosis was diagnosed with a positive standard tube agglutination test or a positive blood culture.

Results: Brucellosis was diagnosed in 996 male patients. Of these patients, 25 had a diagnosis of BEO (2.5%). All BEO patients suffered from enlarged painful testicles, however, testicular complaints were the only presentation symptoms in three of them. All patients received medical treatment alone except one patient with testicular abscess who underwent surgical drainage besides medical treatment. All patients recovered completely and no relapses have been detected during six-month follow-up.

Conclusion: Patients with epididymo-orchitis should be investigated for brucellosis especially in endemic regions. To our knowledge, BEO patients may present with isolated testicular symptoms that make a diagnostic challenge.

Keywords:

• Brucellosis
• genitourinary involvement
• epididymo-orchitis

Introduction

Brucellosis is an endemic zoonosis in many parts of the world such as the Southern Europe, the Middle East, and the South American countries. It is transmitted to humans from contaminated raw milk products or direct contact with infected tissues. Also known as the Mediterranean or Malta fever, brucellosis is a multisystemic disease with many different organ or body system involvement during its clinical course. The most common complication is osteoarticular involvement mainly sacroileitis, spondylitis, and spondylodiscitis followed by genitourinary involvement such as epididymo-orchitis, prostatitis, cystitis, pyelonephritis, renal and testicular abscess. Brucellar epididymo-orchitis (BEO) is the most widespread genitourinary complication [1–4]. In this retrospective study, we aimed to present epidemiological, clinical, and laboratory findings of 25 BEO cases along with their treatment options and outcomes.

Materials and methods

This study was approved by the Local Ethics Committee of Bolu Abant Izzet Baysal University, Medical Faculty, Turkey (Decision number: 2018/194). The medical records of male brucellosis patients with epididymo-orchitis treated in two centers between 2010 and 2018 were retrospectively analyzed. Collected data from the patient files included medical history, physical examination findings, laboratory analysis including hemogram, biochemical tests, C-reactive protein (CRP), erythrocyte sedimentation rate (ESR), serological tests for brucellosis, and radiological findings. The diagnosis of brucellosis was based on clinical findings consistent with brucellosis and a positive blood culture or a positive standard tube agglutination test. Wright tube agglutination test was positive when the titer was ≥1/160 with immuncapture method. Blood cultures were performed in the BACTEC 9240 blood culture system (Becton Dickinson, USA). Brucella spp. isolates were identified using fully-automated system (Phoenix, Becton Dickinson). The diagnosis of epididymo-orchitis was made when the patients had scrotal pain, swelling, and enlarged tender testicles and/or epididymis on clinical examination. The diagnosis of epididymo-orchitis was confirmed using scrotal Doppler ultrasonography in male brucellosis patients who were diagnosed either with Wright agglutination test and/or positive blood culture. Testicular enlargement, non-homogenous echotexture, hypoechoic or heterogeneous echogenecity, and testicular hypervascularity on Doppler ultrasonography are the supportive findings of epididymo-orchitis. Scrotal Doppler ultrasonography was also used to determine whether there was any other complication such as necrosis, atrophy or abscess formation.

Results

Among 996 male patients diagnosed with brucellosis during the analyzed period, 25 of them (2.5%) had BEO. Out of 25 BEO patients, 19 also had other coexisting focal forms of brucellosis: 12 hepatitis, 7 sacroiliitis, 5 spondylitis, and 4 peripheral arthritis. The median age of the patients was 36 years (range: 18–76). Most of the BEO patients (80%) had an epidemiological risk factor for brucellosis. Out of 25 BEO patients, 20 had been living in rural area, 15 had been consuming raw mik products, and 14 had a history of animal contact. Brucellosis was also detected in the family of 14 patients. The median duration of symptoms was 20 days (range: 2–90). All BEO patients had enlarged, painful testicles, however, three of them had no other symptoms of brucellosis other than these complaints. Clinical and laboratory findings of the patients were summarized in Table 1.

Table 1. Clinical and laboratory findings of the brucellar epididymo-orchitis patients (n = 25).

Median age, years	36 years (range 18–76)
Clinical findings
Testicular pain and enlargement	25 (100%)
Fever	16 (64%)
Testicular erythema	15 (60%)
Malaise	14 (56%)
Arthralgia	14 (56%)
Anorexia	12 (48%)
Night sweats	12 (48%)
Splenomegaly	4 (16%)
Dysuria	4 (16%)
Weight loss	4 (16%)
Hepatomegaly	2 (8%)
Laboratory findings
Positive Wright agglutination (≥1/160)	25 (100%)
Positive Rose Bengal test	24 (96%)
CRP > 5 mg/dL	22 (88%)
ESR > 20 mm/h	12/16 (75%)
ALT > 40 U/L	12/24 (50%)
Anemia	8 (32%)
AST > 40 U/L	6/23 (26%)
Positive blood culture	1/4 (25%)
Leukocytosis	4 (16%)

On physical examination, all patients had enlarged, tender testicles and 16 patients had fever. Two patients had bilateral testicular involvement. The Rose Bengal test was positive in all but one patient and Wright tube agglutination test was positive in all patients (1/160 in nine patients, 1/320 in four, 1/640 in five, 1/1280 in five, 1/2560 in one, and 1/5120 in one patient). Leukocytosis was found in four patients. Of the blood cultures drawn in four patients, one was positive for Brucella species. The most common laboratory abnormality was CRP elevation followed by ESR and ALT elevations. Scrotal Doppler ultrasonography was performed on 21 patients. Of these patients, eight had unilateral epididymo-orchitis, seven had unilateral orchitis, one had bilateral orchitis, and one had bilateral epididymo-orchitis. Bilateral varicocele, reactive hydrocele, and testicular abscess were other sonographic findings each of which were found in one patient.

All but one patient received medical treatment alone. However, one patient with testicular abscess underwent surgical drainage in addition to medical treatment. Doxycycline 100 mg twice orally plus streptomycin 1 g once intramuscularly, and doxycycline 100 mg twice orally plus rifampin 600 mg once orally were the most preferred combinations. The duration of therapy was 6 weeks in 16 patients. However, the remaining 9 patients were treated for 12 weeks because of multiple involvement besides epididymo-orchitis. All of the patients recovered completely and no relapses have been detected during six-month follow-up.

Discussion

Epididymoorchitis is an important genitorinary complication of brucellosis that has serious impacts on male reproductive health. In previous studies, the incidence of BEO ranged between 1.4% and 25% [4]. We found epididymo-orchitis in 2.5% of male patients with brucellosis. BEO is usually seen in young adults [5,6]. We obtained a similar finding that the median age of our patients was 36 years (range: 18–76). We think that this finding is important, because it means that brucellosis may negatively impact reproductive health of sexually active young adults.

Interestingly, BEO has an insidious course rather than an acute onset [7]. In the present study, the median duration of symptoms was found to be 20 days. We think that this anamnestic clue may be helpful to differentiate BEO from other causes of epididymo-orchitis.

Previous studies reported that patients with BEO mostly present with testicular pain and enlargement, fever, scrotal erythema, and sweating. Besides these symptoms, BEO patients may also exhibit other brucellosis symptoms such as myalgia, arthralgia, malaise, anorexia, and weight loss [4–11]. In the present study, testicular pain and enlargement were the most common symptoms as seen in all patients. Other less common symptoms were fever, scrotal erythema, malaise, arthralgia, anorexia, and night sweats, respectively. Dysuria and weight loss were not frequent, each found in four patients (16%).

Patients with BEO may sometimes present with testicular symptoms only. Even urinary symptoms may be absent. In such cases, brucellosis can be difficult to diagnose due to isolated testicular symptoms. If these cases are missed, delay in the treatment can result in negative consequences such as testicular abscess, necrosis, atrophy, orchiectomy, and infertility [5,12]. In previous studies, the frequency of dysuria in BEO patients was generally reported to be low (22%, 29%, 57%) [5,10,11]. In our study, three patients presented with testicular symptoms only. Neither dysuria nor pollacuria was present in these cases.

Many previous studies showed that leukocytosis is not a typical feature of BEO [13,14]. Our study also supported this finding, as we detected leukocytosis only in four patients (16%). The range of leukocytosis was between 10.9 and 13.2 K/μL in our study. In addition, blood culture positivity was reported between 14% and 69% previously [9,15], however, we detected positive blood culture in one of the four patients (25%).

As an acute phase reactant, elevated CRP and ESR levels may be seen in brucellosis. Increased CRP levels were also associated with brucellosis complications [7]. Consistently, elevated CRP and ESR levels were the most common laboratory findings in our study found in 88% and 75% of the patients, respectively.

Scrotal Doppler ultrasonography has an important function in the management of BEO. It helps to exclude other possible causes of testicular involvement such as malignancy or testicular abscess. The most common ultrasonographic findings are enlargement and hypervascularity of the epididymis and testicles [5,16]. BEO is often unilateral [4,7,8,10]. Similarly, ultrasonography confirmed unilateral testicular involvement in 23 out of 25 patients in our study. Only two patients had bilateral testicular involvement due to brucellosis.

Different antibiotic combinations can be used for the treatment of BEO [4,5,15]. The most common combination used in the current study was doxycycline plus streptomycine. Other combinations with gentamycine, rifampicine, trimethoprim-sulfamethoxazole, and ciprofloxacin were also used. Irrespective of the different antibiotic combinations, all patients were treated successfully. Based on our experience, it is difficult to conclude that one specific combination is superior in the treatment of BEO.

There are some limitations in the present study. First, this is a retrospective study with two center data and relatively small number of patients. Second, follow-up period of the patients is relatively short. Lastly, there was missing data for some patients.

In conclusion, BEO still remains an important health concern in the aging male in several parts of the world. Thus, patients with epididymo-orchitis should be investigated for brucellosis especially in endemic regions. To our knowledge, BEO patients may present with isolated testicular symptoms that make a diagnostic challenge.

Disclosure statement

No potential conflict of interest was reported by the authors.