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Tuberculous epididymitis with extensive retroperitoneal and mediastinal involvement
CASE REPORT
TUBERCULOUS EPIDIDYMITIS WITH EXTENSIVE RETROPERITONEAL AND MEDIASTINAL INVOLVEMENT FELIX T. OBEN, RENEE D. E. WRIGHT,
AND
CHILEDUM A. AHAGHOTU
ABSTRACT The incidence of tuberculosis in the United States is on the rise, in part, because of its association with acquired immunodeficiency syndrome. Genitourinary tuberculosis remains one of the most common forms of secondary or extrapulmonary disease. We present an unusual case of tuberculous epididymitis with extensive retroperitoneal and mediastinal spread. The possible routes of dissemination, as well as the efficacy of antimycobacterial therapy in the management of tuberculous epididymitis, are discussed and the relevant literature is reviewed. UROLOGY 64: 156.e3–156.e5, 2004. © 2004 Elsevier Inc.
A
pproximately 25,000 new cases of tuberculosis are reported annually in the United States.1 The incidence has been increasing, because it is the major communicable complication of acquired immunodeficiency syndrome. Although it is primarily a pulmonary infection, extrapulmonary tuberculosis accounts for 15% of cases.1 Genital tuberculosis is a rare form of the disease, representing approximately 0.43% of cases in the United States.2 The most common genital sites are the epididymis, prostate, and seminal vesicles; the testes are the least commonly infected. Up to 88% of patients with genital tuberculosis have extragenital involvement.2 The following case report demonstrates that extensive disease may have limited external manifestations and excellent results may be obtained with appropriate antimycobacterial therapy. CASE REPORT The patient was a 22-year-old man, a recent immigrant from Sierra Leone, who presented with a 4-week history of a painful right groin swelling. The pain radiated along the distribution of the right spermatic cord. The patient denied nausea, vomiting, urinary symptoms, or urethral discharge. He also denied cough or hemoptysis, but admitted to a 2-month history of fever, chills, night From the Division of Urology, Howard University Hospital, Washington, DC Address for correspondence: Felix T. Oben, M.D., Division of Urology, Howard University Hospital, 2041 Georgia Avenue, Suite 4C-02, Washington, DC 20060 Submitted: June 11, 2003, accepted: March 5, 2004 © 2004 ELSEVIER INC. ALL RIGHTS RESERVED
sweats, and weight loss. He also had pain and paresthesia in the upper back. He denied any previous history of tuberculosis, but admitted contact with an infected family member in Sierra Leone. His physical examination revealed a 12 ⫻ 5-cm fluctuant mass along the right inguinal canal (Fig. 1). The right spermatic cord was indurated and tender. Right inguinal lymphadenopathy was present. The left spermatic cord and testis were palpably normal. Routine studies, including complete blood count, serum electrolytes, prothrombin time and partial thromboplastin time, and urinalysis were within normal limits. Human immunodeficiency virus and Venereal Disease Research Laboratory tests were negative. Ultrasonography of the scrotum revealed normal testes bilaterally, an echogenic right epididymis with overlying subcutaneous edema suggestive of epididymitis, and large cystic collections in the pelvis and right lower quadrant of the retroperitoneum. Abdominal/pelvic computed tomography (CT) scan demonstrated a large, complex, hypoattenuated mass consistent with the ultrasound findings in the right retroperitoneum involving the iliacus muscle (Fig. 2). Fine needle aspiration of the inguinal mass was performed. Zeihl-Neilson staining of the aspirate showed the presence of acid-fast tubercle bacilli. CT scan of the chest revealed a 4 ⫻ 3.5 ⫻ 5-cm superior mediastinal mass displacing the superior vena cava anteriorly and the trachea posteriorly (Fig. 3). The lung fields were unremarkable. The patient was administered a course of rifampicin 600 mg, isoniazid 300 mg, and pyrazinamide 1500 mg daily for 4 months, followed by rifampi0090-4295/04/$30.00 doi:10.1016/j.urology.2004.03.014 156.e3
scan after 7 months demonstrated reduction in the retroperitoneal mass. Additional imaging at 9 months showed complete resolution of the retroperitoneal and mediastinal masses, with no gross evidence of residual epididymal disease. COMMENT
FIGURE 1. Painful mass in right groin.
FIGURE 2. CT of abdomen and pelvis showing large complex multiloculated collection in retroperitoneum, involving iliacus and extending into right hemiscrotum.
FIGURE 3. CT of chest showing superior mediastinal extension of process with anterior displacement of inferior vena cava and posterior displacement of trachea.
cin 600 mg and isoniazid 600 mg three times per week for 5 months. The patient was seen 2 weeks after the onset of treatment, at which time the inguinal mass was no longer visible, with residual induration of the inguinal cord. The patient presented at monthly intervals for 4 months with an uneventful course. A repeat abdominal/pelvic CT 156.e4
Tuberculous (TB) epididymitis occurs most often in young, sexually active men. Historically, the typical patient presented between the ages of 16 and 40 years. Currently, more than 70% of men with TB epididymitis are older than 35 years, with 15% to 20% older than 65 years.3 The typical presentation is painful, unilateral scrotal swelling. With exclusively external genital involvement, urinary symptoms, as well as constitutional symptoms such as fever and chills, are usually absent. In this case, the patient presented with a painful inguinal swelling radiating to the scrotum. He also had constitutional symptoms that had begun 1 month before the appearance of the inguinal swelling. This suggests that he had extragenital involvement of tuberculosis, which is often the case in patients with TB epididymitis. This was confirmed by abdominal and chest CT scans, showing a large retroperitoneal mass with superior mediastinal involvement. The most common sites of extragenital involvement in patients with TB epididymitis are the kidneys and lungs. Pulmonary and renal involvement is seen in 50% and 80% of patients with genital tuberculosis, respectively.4 Renal involvement is often suggested by irritative voiding symptoms, pyuria, and urinary acid-fast bacilli. All of these were absent in this patient, and CT scan showed no evidence of renal disease. However, TB epididymitis may be the first manifestation of genitourinary tuberculosis in cases in which upper urinary tract studies are normal and urine cultures for Mycobacterium tuberculosis are negative.5 In such cases, the renal focus is often microscopic and, therefore, undetectable with intravenous urography. In these cases, tubercle bacilli are believed to spread to the prostate from a lesion in the kidney, by way of the urinary tract, and from the prostate to the epididymis by way of the vas deferens. This route was described by Skoutelis et al.6 in 2 patients who presented with epididymitis. In both cases, a significant delay in diagnosis and treatment occurred, resulting in psoas abscess formation and other systemic manifestations. Similarly, our patient had mild constitutional symptoms that may have been overlooked for several months. Another possible mechanism is direct extension from the retroperitoneum along the iliacus muscle and inguinal canal into the epididymis. Bench studies and clinical reports to support this mechaUROLOGY 64 (1), 2004
nism have been documented. Duchek and Winblad7 introduced tubercle bacilli into the bladder wall of guinea pigs after one half had undergone bilateral ductus deferens resection. Most animals with bilateral resection developed epididymitis and demonstrated extensive lymphatic involvement under histopathologic examination. In another study, tuberculous infection spread to the periureteral and para-aortic lymph nodes after ligation of the ureter and introduction of tubercle bacilli proximal to the ligature.8 A case report by Winblad9 also demonstrated lymphatic involvement exterior to the vas deferens in a patient with TB epididymitis and prostatitis. Another potential route of spread is hematogenous. Primary pulmonary tuberculosis may be complicated by blood-borne dissemination to sites, including the epididymis. Although our patient denied a history of active tuberculosis, previous exposure raises the possibility of subclinical pulmonary involvement. A final, unusual mechanism involves venereal transmission of tuberculosis. Nine cases of suspected male-to-female transmission10 and one case of suspected female-to-male transmission4 have been reported. This mode of transmission is supported by evidence of acid-fast bacilli in the semen of some patients with TB epididymitis.11 Currently, no consensus has been reached concerning how to treat patients with TB epididymitis with extensive retroperitoneal involvement. Blumental et al.12 recommended drainage of large collections followed by antituberculous treatment. Varying success has been achieved with medical management. Ferrie and Rundle13 reported surgery was necessary in 9 of 14 reported cases. Others have recommended that surgery be reserved only for those cases refractory to medical treatment.14,15 The present case demonstrates the efficacy of medical management as a first-line treatment for TB epididymitis, reserving surgical intervention for medical failure. CONCLUSIONS TB epididymitis is an unusual form of extrapulmonary tuberculosis. Limited clinical manifestations may be misleading with regard to the extent
UROLOGY 64 (1), 2004
of the disease. The various potential pathogenic pathways often interfere with a clear understanding of its natural history. Our case report demonstrates the efficacy of multidrug therapy as firstline management of the disease. REFERENCES 1. Lederman MM: Diseases of the lower respiratory tract, in Andreoli TE, Carpenter CCJ, Griggs RC, et al (Eds): Cecil’s Essentials of Medicine. Philadelphia, WB Saunders, 2001, pp 794 –795. 2. Gorse GJ, and Belshe RB: Male genital tuberculosis: a review of the literature with instructive case reports. Rev Infect Dis 7: 511–524, 1985. 3. Wolf JS Jr: Epididymal tuberculosis, in eMedicine (world medical library [on-line]). http://www.emedicine. com/MED/topic703.htm 2002. 4. Wolf JS Jr, and McAninch JW: Tuberculous epididymoorchitis: diagnosis by fine needle aspiration. J Urol 145: 836 – 838, 1991. 5. Johnson WD Jr, Johnson CW, and Lowe FC: Tuberculosis and parasitic diseases of the genitourinary system, in Walsh PC, Retik AB, Vaughan ED Jr, et al (Eds): Campbell’s Urology, 8th ed. Philadelphia, WB Saunders, 2002, pp 750 – 751. 6. Skoutelis A, Marangos M, Petsas T, et al: Serious complications of tuberculous epididymitis. Infection 28: 193–195, 2000. 7. Duchek M, and Winblad B: Spread of tuberculosis from the urinary bladder to the male genital organs: an experimental study. Urol Res 1: 141–144, 1973. 8. Winblad B, and Duchek M: Spread of tuberculosis from obstructed and non-obstructed upper urinary tract: an experimental study in male guinea pigs. Acta Pathol Microbiol Immunol Scand [A] 83: 229 –236, 1975. 9. Winblad B: Male genital tuberculosis—the possibility of lymphatic spread: a case report. Acta Pathol Microbiol Scand [A] 83: 425–428, 1975. 10. Lattimer JK: Transmission of genital tuberculosis from husband to wife via semen. Am Rev Tuberculosis 69: 618 – 624, 1954. 11. Shafik A: Treatment of tuberculous epididymitis by intratunical rifampicin injection. Arch Androl 36: 239 –246, 1996. 12. Blumental E, Gottehrer N, Dollberg M, et al: A giant tuberculous lymphangioma extending from the mediastinum to the inguinal region. Chest 105: 1279 –1280, 1994. 13. Ferrie BG, and Rundle JS: Tuberculous epididymo-orchitis: a review of 20 cases. Br J Urol 55: 437–439, 1983. 14. Kunichika N, Murakami K, Makihata K, et al: Orchiectomy for tuberculous epididymitis: a report of two cases with intractable to antituberculosis treatment. Kekkaku 76: 673– 676, 2001. 15. Gueye SM, Ba M, Sylla C, et al: Epididymal manifestations of urogenital tuberculosis. Prog Urol 8: 240 –243, 1998.
156.e5
TUBERCULOUS EPIDIDYMITIS WITH EXTENSIVE RETROPERITONEAL AND MEDIASTINAL INVOLVEMENT FELIX T. OBEN, RENEE D. E. WRIGHT,
AND
CHILEDUM A. AHAGHOTU
ABSTRACT The incidence of tuberculosis in the United States is on the rise, in part, because of its association with acquired immunodeficiency syndrome. Genitourinary tuberculosis remains one of the most common forms of secondary or extrapulmonary disease. We present an unusual case of tuberculous epididymitis with extensive retroperitoneal and mediastinal spread. The possible routes of dissemination, as well as the efficacy of antimycobacterial therapy in the management of tuberculous epididymitis, are discussed and the relevant literature is reviewed. UROLOGY 64: 156.e3–156.e5, 2004. © 2004 Elsevier Inc.
A
pproximately 25,000 new cases of tuberculosis are reported annually in the United States.1 The incidence has been increasing, because it is the major communicable complication of acquired immunodeficiency syndrome. Although it is primarily a pulmonary infection, extrapulmonary tuberculosis accounts for 15% of cases.1 Genital tuberculosis is a rare form of the disease, representing approximately 0.43% of cases in the United States.2 The most common genital sites are the epididymis, prostate, and seminal vesicles; the testes are the least commonly infected. Up to 88% of patients with genital tuberculosis have extragenital involvement.2 The following case report demonstrates that extensive disease may have limited external manifestations and excellent results may be obtained with appropriate antimycobacterial therapy. CASE REPORT The patient was a 22-year-old man, a recent immigrant from Sierra Leone, who presented with a 4-week history of a painful right groin swelling. The pain radiated along the distribution of the right spermatic cord. The patient denied nausea, vomiting, urinary symptoms, or urethral discharge. He also denied cough or hemoptysis, but admitted to a 2-month history of fever, chills, night From the Division of Urology, Howard University Hospital, Washington, DC Address for correspondence: Felix T. Oben, M.D., Division of Urology, Howard University Hospital, 2041 Georgia Avenue, Suite 4C-02, Washington, DC 20060 Submitted: June 11, 2003, accepted: March 5, 2004 © 2004 ELSEVIER INC. ALL RIGHTS RESERVED
sweats, and weight loss. He also had pain and paresthesia in the upper back. He denied any previous history of tuberculosis, but admitted contact with an infected family member in Sierra Leone. His physical examination revealed a 12 ⫻ 5-cm fluctuant mass along the right inguinal canal (Fig. 1). The right spermatic cord was indurated and tender. Right inguinal lymphadenopathy was present. The left spermatic cord and testis were palpably normal. Routine studies, including complete blood count, serum electrolytes, prothrombin time and partial thromboplastin time, and urinalysis were within normal limits. Human immunodeficiency virus and Venereal Disease Research Laboratory tests were negative. Ultrasonography of the scrotum revealed normal testes bilaterally, an echogenic right epididymis with overlying subcutaneous edema suggestive of epididymitis, and large cystic collections in the pelvis and right lower quadrant of the retroperitoneum. Abdominal/pelvic computed tomography (CT) scan demonstrated a large, complex, hypoattenuated mass consistent with the ultrasound findings in the right retroperitoneum involving the iliacus muscle (Fig. 2). Fine needle aspiration of the inguinal mass was performed. Zeihl-Neilson staining of the aspirate showed the presence of acid-fast tubercle bacilli. CT scan of the chest revealed a 4 ⫻ 3.5 ⫻ 5-cm superior mediastinal mass displacing the superior vena cava anteriorly and the trachea posteriorly (Fig. 3). The lung fields were unremarkable. The patient was administered a course of rifampicin 600 mg, isoniazid 300 mg, and pyrazinamide 1500 mg daily for 4 months, followed by rifampi0090-4295/04/$30.00 doi:10.1016/j.urology.2004.03.014 156.e3
scan after 7 months demonstrated reduction in the retroperitoneal mass. Additional imaging at 9 months showed complete resolution of the retroperitoneal and mediastinal masses, with no gross evidence of residual epididymal disease. COMMENT
FIGURE 1. Painful mass in right groin.
FIGURE 2. CT of abdomen and pelvis showing large complex multiloculated collection in retroperitoneum, involving iliacus and extending into right hemiscrotum.
FIGURE 3. CT of chest showing superior mediastinal extension of process with anterior displacement of inferior vena cava and posterior displacement of trachea.
cin 600 mg and isoniazid 600 mg three times per week for 5 months. The patient was seen 2 weeks after the onset of treatment, at which time the inguinal mass was no longer visible, with residual induration of the inguinal cord. The patient presented at monthly intervals for 4 months with an uneventful course. A repeat abdominal/pelvic CT 156.e4
Tuberculous (TB) epididymitis occurs most often in young, sexually active men. Historically, the typical patient presented between the ages of 16 and 40 years. Currently, more than 70% of men with TB epididymitis are older than 35 years, with 15% to 20% older than 65 years.3 The typical presentation is painful, unilateral scrotal swelling. With exclusively external genital involvement, urinary symptoms, as well as constitutional symptoms such as fever and chills, are usually absent. In this case, the patient presented with a painful inguinal swelling radiating to the scrotum. He also had constitutional symptoms that had begun 1 month before the appearance of the inguinal swelling. This suggests that he had extragenital involvement of tuberculosis, which is often the case in patients with TB epididymitis. This was confirmed by abdominal and chest CT scans, showing a large retroperitoneal mass with superior mediastinal involvement. The most common sites of extragenital involvement in patients with TB epididymitis are the kidneys and lungs. Pulmonary and renal involvement is seen in 50% and 80% of patients with genital tuberculosis, respectively.4 Renal involvement is often suggested by irritative voiding symptoms, pyuria, and urinary acid-fast bacilli. All of these were absent in this patient, and CT scan showed no evidence of renal disease. However, TB epididymitis may be the first manifestation of genitourinary tuberculosis in cases in which upper urinary tract studies are normal and urine cultures for Mycobacterium tuberculosis are negative.5 In such cases, the renal focus is often microscopic and, therefore, undetectable with intravenous urography. In these cases, tubercle bacilli are believed to spread to the prostate from a lesion in the kidney, by way of the urinary tract, and from the prostate to the epididymis by way of the vas deferens. This route was described by Skoutelis et al.6 in 2 patients who presented with epididymitis. In both cases, a significant delay in diagnosis and treatment occurred, resulting in psoas abscess formation and other systemic manifestations. Similarly, our patient had mild constitutional symptoms that may have been overlooked for several months. Another possible mechanism is direct extension from the retroperitoneum along the iliacus muscle and inguinal canal into the epididymis. Bench studies and clinical reports to support this mechaUROLOGY 64 (1), 2004
nism have been documented. Duchek and Winblad7 introduced tubercle bacilli into the bladder wall of guinea pigs after one half had undergone bilateral ductus deferens resection. Most animals with bilateral resection developed epididymitis and demonstrated extensive lymphatic involvement under histopathologic examination. In another study, tuberculous infection spread to the periureteral and para-aortic lymph nodes after ligation of the ureter and introduction of tubercle bacilli proximal to the ligature.8 A case report by Winblad9 also demonstrated lymphatic involvement exterior to the vas deferens in a patient with TB epididymitis and prostatitis. Another potential route of spread is hematogenous. Primary pulmonary tuberculosis may be complicated by blood-borne dissemination to sites, including the epididymis. Although our patient denied a history of active tuberculosis, previous exposure raises the possibility of subclinical pulmonary involvement. A final, unusual mechanism involves venereal transmission of tuberculosis. Nine cases of suspected male-to-female transmission10 and one case of suspected female-to-male transmission4 have been reported. This mode of transmission is supported by evidence of acid-fast bacilli in the semen of some patients with TB epididymitis.11 Currently, no consensus has been reached concerning how to treat patients with TB epididymitis with extensive retroperitoneal involvement. Blumental et al.12 recommended drainage of large collections followed by antituberculous treatment. Varying success has been achieved with medical management. Ferrie and Rundle13 reported surgery was necessary in 9 of 14 reported cases. Others have recommended that surgery be reserved only for those cases refractory to medical treatment.14,15 The present case demonstrates the efficacy of medical management as a first-line treatment for TB epididymitis, reserving surgical intervention for medical failure. CONCLUSIONS TB epididymitis is an unusual form of extrapulmonary tuberculosis. Limited clinical manifestations may be misleading with regard to the extent
UROLOGY 64 (1), 2004
of the disease. The various potential pathogenic pathways often interfere with a clear understanding of its natural history. Our case report demonstrates the efficacy of multidrug therapy as firstline management of the disease. REFERENCES 1. Lederman MM: Diseases of the lower respiratory tract, in Andreoli TE, Carpenter CCJ, Griggs RC, et al (Eds): Cecil’s Essentials of Medicine. Philadelphia, WB Saunders, 2001, pp 794 –795. 2. Gorse GJ, and Belshe RB: Male genital tuberculosis: a review of the literature with instructive case reports. Rev Infect Dis 7: 511–524, 1985. 3. Wolf JS Jr: Epididymal tuberculosis, in eMedicine (world medical library [on-line]). http://www.emedicine. com/MED/topic703.htm 2002. 4. Wolf JS Jr, and McAninch JW: Tuberculous epididymoorchitis: diagnosis by fine needle aspiration. J Urol 145: 836 – 838, 1991. 5. Johnson WD Jr, Johnson CW, and Lowe FC: Tuberculosis and parasitic diseases of the genitourinary system, in Walsh PC, Retik AB, Vaughan ED Jr, et al (Eds): Campbell’s Urology, 8th ed. Philadelphia, WB Saunders, 2002, pp 750 – 751. 6. Skoutelis A, Marangos M, Petsas T, et al: Serious complications of tuberculous epididymitis. Infection 28: 193–195, 2000. 7. Duchek M, and Winblad B: Spread of tuberculosis from the urinary bladder to the male genital organs: an experimental study. Urol Res 1: 141–144, 1973. 8. Winblad B, and Duchek M: Spread of tuberculosis from obstructed and non-obstructed upper urinary tract: an experimental study in male guinea pigs. Acta Pathol Microbiol Immunol Scand [A] 83: 229 –236, 1975. 9. Winblad B: Male genital tuberculosis—the possibility of lymphatic spread: a case report. Acta Pathol Microbiol Scand [A] 83: 425–428, 1975. 10. Lattimer JK: Transmission of genital tuberculosis from husband to wife via semen. Am Rev Tuberculosis 69: 618 – 624, 1954. 11. Shafik A: Treatment of tuberculous epididymitis by intratunical rifampicin injection. Arch Androl 36: 239 –246, 1996. 12. Blumental E, Gottehrer N, Dollberg M, et al: A giant tuberculous lymphangioma extending from the mediastinum to the inguinal region. Chest 105: 1279 –1280, 1994. 13. Ferrie BG, and Rundle JS: Tuberculous epididymo-orchitis: a review of 20 cases. Br J Urol 55: 437–439, 1983. 14. Kunichika N, Murakami K, Makihata K, et al: Orchiectomy for tuberculous epididymitis: a report of two cases with intractable to antituberculosis treatment. Kekkaku 76: 673– 676, 2001. 15. Gueye SM, Ba M, Sylla C, et al: Epididymal manifestations of urogenital tuberculosis. Prog Urol 8: 240 –243, 1998.
156.e5