1356 | Sudden Decline in Semen Volume Due To Seminal Vesicle Fistula in a Patient with Crohn’s Disease: A Case Report Yasushi Yumura,1,2 Kazumi Noguchi,1 Masatoshi Moriyama,2 Akira Iwasaki3 Keywords: seminal vesicles; fistula; etiology; Crohn's disease; complications. INTRODUCTION Seminal vesicle fistula (SVF) is an uncommon condition. Many cases of seminal vesicle fistula are related to bowel lesions. We report a very rare case of SVF associated with a perianal abscess that developed in a Crohn’s disease patient complaining of decline in semen volume. CASE REPORT A 34-year-old man with a history of Crohn’s disease was referred to our department in July 2007 complaining of a decrease in ejaculated semen volume and pneumaturia for the past 2 months. He had previously undergone two perineal abscess drainage procedures (Seton’s meth- od) in 2000 and 2007 due to perianal and ischiorectal abscesses arising from Crohn’s disease. His erectile function was normal. Physical examination revealed induration of the tail of the left epididymis. He was afebrile. Testicular size, measured with an orchidometer, was normal bilaterally (14 mL). The prostate could not be examined because of anal stricture and a Penrose drain that was indwelling from the perineum to a perianal abscess. On ejaculation, watery fluid was discharged from the perineal drain. We did not examine whether spermatozoa existed in the fluid discharged from the perineal drain. Semen analysis revealed very low semen volume (0.3 mL and 0.7 mL) and azoospermia. Serum sex hormones levels including follicle-stimulating hormone, luteinizing hormone and testosterone were within normal limits. Cystogram and cystoscopy revealed no abnormalities. Magnetic resonance imaging (MRI) be- fore the second perineal drainage showed bilateral contracted seminal vesicles and a high-in- Corresponding Author: Yasushi Yumura, MD Department of Urology, Yokohama City University, Medical Center, Yokohama, Japan. Tel: +81 45 261 5656 Fax: +81 45 253 1962 E-mail: yumura@yokohama-cu.ac.jp Received August 2012 Accepted March 2013 1 Department of Urology, Yokohama City University, Medical Center, Yokohama, Japan. 2 Department of Urology, Yoko- hama Municipal Citizen’s Hospital, Yokohama, Japan. 3 Department of Urology, Yoko- hama Minato Red Cross Hospital, Yokohama, Japan. CASE REPORT Case Report 1357Vol. 11 | No. 01 | Jan-Feb 2014 |U R O LO G Y J O U R N A L Decline in Semen Volume Due To Seminal Vesicle Fistula | Yumura et al tensity cystic lesion that was located posterior to the seminal vesicle, and we suspected this was an abscess (Figure 1A). MRI after the second drainage failed to show seminal vesicle (Figure 1B). Vasography showed bilateral obstruction at the level of the ejaculatory duct. Left seminal vesicle was not visualized. Leakage of contrast from the right seminal vesicle to the per- ineal drain via the posterior lesion of the urinary bladder was demonstrated (Figure 2). Subsequently, he underwent colos- tomy because the perianal and ischiorectal abscess had not improved. Seminal tract reconstruction was not performed because of the operation difficulties expected with severe perivesical adhesion. After the surgery, the abscess gradually healed and semen volume increased to 1.0 mL. We found sperm (64.0×106/mL) in his ejaculate 16 months after the operation. DISCUSSION In this case, the causes of azoospermia and sudden decline in semen volume were obstruction of the ejaculatory duct and leakage of seminal vesicle fluid to an abscess. We speculated that the abscess that had been drained unsuccessfully extend- ed to the peri- and rear- prostatic and vesical lesions and led to seminal vesicle destruction and seminal tract obstruction. Because of seminal vesicle destruction, we speculated that the right seminal vesicle was connected to the abscess and seminal vesicle fluid was discharged through the Seton drain. We considered that the colostomy led to reduction of the ab- scess and repair of the seminal tract. SVF is a very rare disease. To our knowledge, approximate- ly 20 cases have been reported to date. The most common causes of fistulae are diverticulitis,(1) colorectal cancer (2-4) and Crohn’s disease.(2,5) Other causes include trauma,(6) ra- diotherapy, infectious cyst (7) and unknown causes.(8) Almost all reported cases had fistula that connected the semi- nal vesicle with the colon or rectum.(1,3-8) To our knowledge, cases of a fistula connected to a perianal abscess are very un- common. Many seminal vesicle fistulas present with symp- toms relating to febrile genitourinary tract infection or lower urinary tract symptom, such as pollakiuria or dysuria. This SVF patient had a unique clinical course. There has been no report concerning SVF with a chief complaint of sudden decline in semen volume and obstruction of the ejaculatory duct, as in our patient. Crohn’s disease may cause fistula in the extra-intestinal tract, including refractory perianal ab- scesses. Carlin and colleagues also reported a case of SVF related Crohn’s disease.(2) In their case, seminal vesicle com- municated with perineum area and the patient had recurrent white discharge from the perineal fistula. It was not reported that the patient had a complaint of decline in semen volume. Computed tomography is usually used for the diagnosis of SVF. Vesiculography,(5, 8) contrast enema (1,2,4) and fistulog- Figure 1. A) Magnetic resonance imaging (MRI) before perineal drainage showed bilateral contracted seminal vesicles and a high- intensity cystic lesion that was located posterior to the seminal vesicle, and we suspected this was an abscess (white arrow). B) MRI after drainage did not show seminal vesicle. 1358 | Case Report raphy have also been used. In general, vesiculography MRI of the seminal vesicle are alternative diagnostic tests for low ejaculate volume. In our case, these examinations were useful for diagnosis of SVF and scrutiny around the semi- nal vesicle. The reported treatment modalities for SVF are conservative treatment,(2,5,8) surgical drainage (transurethral unroofing,(9) transperinael approach,(7) percutaneous ap- proach (4,6) and laparotomy and excision of the culprit lesion. (1) When excision of the culprit lesion is difficult, colostomy is a valuable tool for treatment. In our patient, the abscess gradually healed, the semen volume increased and spermato- zoa appeared in ejaculate after the surgery. CONFLICT OF INTEREST None declared. Figure 2. Vesiculography showed bilateral obstruction at the level of the ejaculatory duct. Left seminal vesicle was not shown and leakage of contrast from the right seminal vesicle to the perineal drain via the posterior lesion of the urinary bladder was demonstrated. REFERENCES 1. LaSpina M, Facklis K, Posalski I, Fleshner P. Coloseminal Vesicle Fis- tula: Report of a Case and Review Of the Literature. Dis Colon Rec- tum. 2006;49:1791-3. 2. Carlin J, Nicholson DA, Scott NA. Two Cases of Seminal Vesicle Fis- tula. Clin Radiol. 1999;54:309-11. 3. Goldman SM, Fishman EK, Gatewood OM, Jones B, Siegelman SS. CT in the diagnosis of enterovesical fistulae. AJR Am J Roentgenol. 1985;144:1229-33. 4. Kollmorgen TA, Kollmorgen CF, Lieber MM, Wolff BG. Seminal vesicle fistula following abdominoperineal resection for recurrent adenocarcinoma of the rectum. A case report. Dis Colon Rectum. 1994;37:1325-7. 5. Hamidinia A. Recto-ejaculatory duct fistula: an unusual complica- tion of Crohn's disease. J Urol. 1984;131:123-4. 6. Maeda H, Arai Y, Aoki Y, Okubo K, Okada T. Successful treatment of a persistent cutaneous fistula to the seminal tract: imaging with three-dimensional computed tomography. Br J Urol. 1998;82:595-6. 7. Hammad FT. Seminal vesicle cyst. Scand J Urol Nephrol. 2006;40:426-8. 8. Izumi K, Takase Y, Kobayashi T, Tokunaga S, Namiki M. Seminal Vesi- cle-Rectal Fistula with Preceding Right Acute Epididymitis. Urol Int. 2007;78:367-9. 9. Frye K, Loughlin K. Successful transurethral drainage of bilateral seminal vesicle abscesses. J Urol. 1988;139:1323-4.