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Journal of the Chinese Medical Association 75 (2012) 292e295
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Case Report
Isolated tuberculous epididymitis presenting as a painless scrotal tumor
Victor Ka-Siong Kho, Pei-Hui Chan*
Division of Urology, Department of Surgery, Far Eastern Memorial Hospital and Medical Center, Banciao, New Taipei City, Taiwan, ROC
Received December 16, 2010; accepted May 21, 2011
Abstract
Genitourinary tuberculosis, the second most common extrapulmonary tuberculosis (TB), is very difficult to diagnose unless one maintains
a high index of suspicion. Isolated tuberculous epididymitis (ITE), defined as tuberculous epididymitis without clinical evidence of either renal
or prostate involvement, is a rare entity among genitourinary tuberculosis. When diagnosed correctly, ITE can be cured with anti-TB medications. However, patients with poor response to medical treatment may require surgery. Here, we report a 20-year-old man who presented with
a slow-growing painless scrotal tumor for 2 months, with the initial workup suspicious for a right paratesticular tumor. Surgical resection of the
tumor was therefore scheduled. However, severe pain and redness over the patient’s right hemi-scrotum were noted on the day of surgery. A
repeat scrotal ultrasound was performed that revealed findings suggesting a chronic inflammatory process rather than a malignancy. Frozen
section of the lesion confirmed the ultrasonographic findings, and the pathology established the diagnosis of ITE. The patient remained on antiTB therapy postoperatively for 6 months and had an excellent outcome.
Copyright Ó 2012 Elsevier Taiwan LLC and the Chinese Medical Association. All rights reserved.
Keywords: genitourinary tuberculosis; isolated tuberculous epididymitis; orchiectomy; scrotal tumor; scrotal ultrasound
1. Introduction
The present incidence and prevalence of tuberculosis (TB)
has not changed significantly for decades due to the emigration of people born in endemic areas of TB, the growing
population positive for human immunodeficiency virus (HIV)
infection, and the emergence of multidrug resistant strains of
Mycobacterium. Despite strict implementation of control
measures, TB remains one of the leading causes of death
among notifiable diseases in Taiwan, with an estimated 15,000
new cases and approximately 8000 sputum-smear positive
cases reported annually.1 Chang et al1 reported that the incidence and mortality rates of TB for aborigines in eastern
Taiwan were 3.1 and 3.2 times higher, respectively, than the
rates for the general population in Taiwan.
Extrapulmonary TB can develop in a variety of locations,
with the skeletal, genitourinary tract, and central nervous
system as the three most common extrapulmonary sites.2
Evidences from the literature suggest that the infecting
Mycobacterium tuberculosis bacilli reach the kidney through
hematogenous spread from the lungs, then spread down the
ureter, bladder. and/or prostate.3 Isolated tuberculous epididymitis (ITE), defined as TB epididymitis without clinical and
laboratory evidence of renal involvement, is usually rare and
difficult to diagnose.4 Although the typical symptoms and
imaging signs of epididymal TB have been described by
Madeb et al,5 the definitive diagnosis of ITE can only be
confirmed by positive cultures, ZiehleNeelson staining, and/
or histopathologic examination. In this report, we present
a case of ITE initially presenting as a scrotal tumor that was
diagnosed by histopathologic examination of the surgical
specimen and managed later with anti-TB medications for 6
months.
2. Case report
* Correspondence author. Dr. Pei-Hui Chan, Division of Urology, Department of Surgery, Far Eastern Memorial Hospital and Medical Center, 21,
Section 2, Nan-Ya South Road, Banciao, New Taipei City 220, Taiwan, ROC.
E-mail address: [email protected] (P.-H. Chan).
A 20-year-old man, recently discharged from military
service in eastern Taiwan, came to our clinic due to a slowgrowing painless scrotal tumor for the preceding 2 months.
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doi:10.1016/j.jcma.2012.04.014
V.K.-S. Kho, P.-H. Chan / Journal of the Chinese Medical Association 75 (2012) 292e295
He was healthy previously, without a history of pulmonary TB,
and claimed he had received a bacilli CalmetteeGuerin
vaccination during infancy. Physical examination revealed an
afebrile male with a 5 2-cm non-tender, irregular, nodule in
his right hemi-scrotum. Digital rectal examination revealed
a non-tender, firm, and rubbery prostate. Urinalysis was
normal, without evidence of pyuria, and his complete blood
count, biochemistry, C-reactive protein, alpha-feto protein,
and beta-human chorionic gonadotropin levels were all within
normal limits, with only the lactate-dehydrogenase slightly
elevated. Chest X-ray was clear, and the abdominal computed
tomography scan showed a 5.2-cm heterogenous lesion over
the right paratesticular region involving the epididymis
(Fig. 1), with both kidneys normal in appearance (Fig. 2). A
scrotal ultrasound also revealed a 5.2-cm solid heterogenous
paratesticular tumor.
With tentative diagnosis of a paratesticular tumor, the
patient was scheduled for a high right inguinal orchiectomy.
However, on the scheduled operation day, severe pain with
erythema was noted over the lesion site (Fig. 3).
A scrotal ultrasound was repeated, showing that the previous
solid heterogenous lesion had become a focal anechoic lesion
with calcifications and internal echoes, which was compatible
with a chronic inflammatory process. An exploration of the right
testis was performed through an inguinal incision. Approximately
20 mL of purulent pus with caseous necrotic epididymal tissues
was noted intraoperatively (Fig. 4). Frozen sections of the
necrotic tissues were sent for examination and revealed chronic
granulomatous inflammation. The testis was then preserved, and
the pus was sent for cultures and ZiehleNeelson staining. The
remaining necrotic tissues were sent for pathologic examination.
The patient’s urine was negative for TB culture and
ZiehleNeelson staining. Nevertheless, pathologic examination
of the necrotic tissues showed caseating granulomatous reaction
293
Fig. 2. Coronal contrast CT scan of the abdomen showing a normal appearance
of bilateral kidneys.
with Langhan’s giant cells, (Fig. 5A), and with positive
ZiehleNeelson-stained bacilli (Fig. 5B). The pus culture later
was also positive for M. tuberculosis. The patient was then treated
with a four-drug combination (isoniazid þ rifampicin þ
ethambutol þ pyrazinamide) of anti-TB for 2 months, followed
by triple-drug (isoniazid þ rifampicin þ ethambutol) therapy for
4 months. He remained stable at follow-up, with both testes intact
inside the scrotum (Fig. 6). He tested negative for HIV infection
during follow-up at the clinic.
3. Discussion
Despite advances in anti-mycobacterial therapy and strict
implementation of well-known TB control measures, the
prevalence and incidence of TB remains high worldwide.
Epididymal TB, which accounts for about 20% of genitourinary tuberculosis,6 is believed to result from a retrograde
Fig. 1. Contrast computed tomography (CT) scan of the pelvis showing
(arrow) a heterogenous 5.2-cm right paratesticular tumor, with slightly
decreased enhancement of the right testis compared to the left testis.
Fig. 3. Perioperative picture of the swollen and erythematous right scrotum
(arrow).
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V.K.-S. Kho, P.-H. Chan / Journal of the Chinese Medical Association 75 (2012) 292e295
Fig. 4. Caseous-like necrotic tissues noted intraoperatively.
spread of prostate TB, which is usually secondary to a renal
TB.7 ITE, which is defined as TB epididymitis without clinical
and laboratory evidence of renal involvement,4 is a rare and
hard-to-diagnose disease entity. However, some authors have
disputed the existence of true ITE since initial imaging studies
or microscopic examination of the urine may fail to reveal
a renal lesion.4 In addition, urine culture can be falsely
negative due to its low sensitivity (as low as 50%).8 Ross et al
reported that renal TB or positive urine culture could develop
during the later course of the disease (ITE).9
ITE is usually seen in young adults. In a review of 40 ITE
patients, Viswaroop et al4 reported the median age was 32
years (range 21e37 years); our patient falls into this age
category. ITE can be either the clinical onset of HIV infection
or caused by intravesical bacilli CalmetteeGuerin instillation
of superficial bladder cancer.2 Clinically, ITE usually presents
with a painful scrotal swelling; however, it can also present as
a painless scrotal mass, acting as a first clue to the presence of
TB infection of the prostate and seminal vesicles.2 Irritative
voiding symptoms commonly seen in acute inflammation of
the epididymis and testis are not common in ITE,10,11 as was
Fig. 6. Intact bilateral testis inside the scrotum noted during follow-up at
clinic.
noted in our patient. Typically, ITE occurs unilaterally, but
bilateral involvement has also been reported.4
Up until now, ITE has shared the same imaging findings as
those of other chronic inflammatory processes or testicular
tumor. Commonly used imaging modalities such as scrotal
ultrasonography, computed tomography scan, or magnetic
resonance imaging may show diffuse or focal heterogenous
lesions in the enlarged epididymis, with or without hydrocele,
septation, extratesticular calcification, scrotal abscess, or
scrotal sinus tract.4,10 Therefore, correct preoperative diagnosis of ITE relies on having a high index of suspicion. A
definitive diagnosis of ITE is based on pathological material
obtained from fine-needle aspiration cytology or surgical
resection of the epididymis.4,11,12 However, as we know, fineneedle aspiration is contraindicated in a patient presenting
with a painless scrotal tumor, because if a malignancy is
Fig. 5. (A) Microscopic findings of the necrotic soft tissues with positive ZiehleNeelson stained bacilli (arrowhead) (acid-fast stain 400). (B) Pathologic
specimen from necrotic scrotal tissues showed caseating granulomatous inflammation with Langhans giant cell formation (arrow) and epithelioid histiocytes
(hematoxylin and eosin stain, 100).
V.K.-S. Kho, P.-H. Chan / Journal of the Chinese Medical Association 75 (2012) 292e295
proven later, possible lymphatic spread of malignant cells may
occur during aspiration cytology.
ITE can be cure by anti-TB medications, with a combined oral
regimen of isoniazid, rifampicin, ethambutol, and pyrazinamide
given daily. Suggested duration of therapy varies from 2 months
to 2 years, although a 9- to 12- month regimen is generally
accepted.4 Currently, the standard treatment for genitourinary
tuberculosis in Taiwan consists of 2 months of quadruple-drug
therapy, followed by a triple regimen for an additional 4
months, which our patient received immediately after pathologic
confirmation of ITE.13 The present consensus recommends
surgical intervention if there are no signs of resolution within 2
months of medical treatment or if intrascrotal abscess is identified.14 Surgical resection (epididymo-orchiectomy) is usually
reserved for patients who do not respond to medical therapy.
In conclusion, we have presented a case of ITE, the initial
presentation of which was a painless scrotal tumor. ITE is
a very rare disease entity which usually presents as a painful
scrotal tumor. Nevertheless, ITE can also present as a painless
scrotal tumor. The differential diagnosis includes malignant
testicular and paratesticular tumors and inflammatory conditions such as epididymitis and epididymo-orchitis. It is difficult
to achieve an early and correct diagnosis, which sometimes
leads to unnecessary orchiectomy. A high index of suspicion
cannot be overemphasized. ITE is usually curable with anti-TB
medication, while surgical resection (epididymo-orchiectomy)
is reserved for patients who do not respond to medical therapy.
We also recommend a scrotal ultrasound for patients when the
initial diagnosis of painless scrotal tumor has become questionable, to prevent unwarranted orchiectomy.
295
References
1. Chang YM, Tsai BY, Wu YC, Yang SY, Chen CH. Risk of Mycobacterium
tuberculosis transmission in an aboriginal village, Taiwan. Southeast
Asian J Trop Med Public Health 2006;37:161e4.
2. Lai AYU, Lu SH, Yu HJ, Kuo YC, Huang CY. Tuberculous epididymitis
presenting as huge scrotal tumor. Urology 2009;73:1163. e5ee7.
3. Cinman AC. Genitourinary tuberculosis. Urology 1982;20:353e8.
4. Viswaroop BS, Kekre N, Gopalakrishnan G. Isolated tuberculous
epididymitis: a review of forty cases. J Postgrad Med 2005;51:109e11.
discussion 111.
5. Madeb R, Marshall J, Natif O, Ertuk E. Epididymal tuberculosis: case
report and review of the literature. Urology 2005;65:798.
6. Chattopadhyay A, Bhatnagar V, Agarwala S, Mitra DK. Genitourinary
tuberculosis in a pediatric surgical patient. J Pediatr Surg 1997;32:
1283e6.
7. Cabral DA, Johnson HW, Coleman GU, Nigro M, Speert DP. Tuberculous
epididymitis as a cause of testicular pseudomalignancy in two young
children. Pediatr Infect Dis 1985;4:59e62.
8. Cousins DV, Wilton SD, Francis BR, Gow BL. Use of polymerase chain
reaction for rapid diagnosis of tuberculosis. J Clin Microbiol 1992;30:
255e8.
9. Ross JC, Gow JG, St Hill CA. Tuberculous epididymitis: a review of 170
patients. Br J Sur 1962;48:663e6.
10. Liu HY, Fu YT, Wu CJ, Sun GH. Tuberculous epididymitis: a case report
and literature review. Asian J Androl 2005;7:329e32.
11. Wolf JS Jr, McAninch JW. Tuberculous epididymo-orchitis: diagnosis by
fine-needle aspiration. J Urol 1991;145:836e8.
12. Shafik A. Treatment of tuberculous epididymitis by intratunical rifampicin
injection. Arch Androl 1996;36:239e46.
13. Chang FY, Chang SC, Chen YS, Chen YC, Chiang CY, Chiang IH, et al.
Guidelines for chemotherapy of tuberculosis in Taiwan. J Microbiol
Immunol Infect 2004;37:382e4.
14. Gow JG, Barbosa S. Genitourinary tuberculosis: a study of 1117 cases
over a period of 34 years. Br J Urol 1984;56:449e55.