International Journal of Urology (2004) 11, 686–688

Blackwell Science, LtdOxford, UKIJUInternational Journal of Urology0919-81722004 Blackwell Publishing Asia Pty LtdApril 2004114686688Original ArticleXanthogranulomatous orchitisD Demırcı

et al.

Correspondence: Deniz Demırcı MD, Erciyes UniversitySchool of Medicine, Department of Urology, Kayserı 38039,Turkey. Email: demirci@erciyes.edu.tr.

Received 25 November 2003; accepted 9 January 2004.

Case Report

Xanthogranulomatous orchitis with scrotal fistulas

DENIZ DEM RC , O UZ EKMEKÇ O LU, I IN SOYUER AND MUSTAFA EM RDO AN

Department of Urology, Erciyes University School of Medicine, Kayserı, Turkey

Abstract Xanthogranulomatous orchitis is an extremely rare inflammatory change of testis which is difficultto distinguish from testicular tumor. We report on a 21-year-old man who presented with lefttesticular swelling and pyogenic discharge from the scrotum. Testicular tumor markers were normal.Scrotal ultrasonography showed a testicular tumor in the left testis. Because of severe adhesionbetween the scrotum and intrascrotal structures, radical orchiectomy combined with hemiscrotec-tomy was performed to exclude possible malignancy. Histopathological findings showed xanthogran-ulomatous orchitis.

Key words testis, xanthogranulomatous infection.

I.

I.

G I.

G SI.

G

Introduction

Xanthogranulomatous inflammation (XGI) is anuncommon and non-neoplastic process characterized bythe destruction of tissue. Diabetes mellitus, urolithiasis,obstructive uropathy and urinary infections with Proteusmirabilis or Escherichia coli are possible predisposalfactors.1

We report an unusual case of XGI in one testis withscrotal fistulas.

Case report

A 21-year-old man presented with left testicular swell-ing and scrotal discomfort for 15 days. The patient com-plained of pyogenic discharge from the scrotum. Therewas no positive history of urinary tract infection, uroli-thiasis, trauma or diabetes. His body temperature was37.2∞C. On physical examination, three fistulas at thelower part of the hyperemic and edematous scrotumwere detected. Whereas the right testis was normal, lefttestis showed severe tenderness with enlargement.Direct plain film and urinalysis were normal. Urine andpus cultures were negative. Laboratory results included

a white blood cell count of 11 ¥ 109/L, a hemoglobinlevel of 13.8 g/dL and a platelet count of 407.000/mm3.Tumor markers aFP, bHCG and LDH were normal.Ultrasonography of the testis showed a testicular tumorin the left testis which was infiltrative and hyper-echogenic (Fig. 1).

Surgical exploration revealed severe adhesion of thescrotal contents. The testis could not be separated fromthe scrotum by dissection. Because of the severe adhe-sion between scrotum and intrascrotal structures, radicalorciectomy combined with hemiscrotectomy was per-formed to exclude possible malignancy. Microscopicexamination revealed the replacement of the tubules andinterstitial tissue by numerous foamy macrophages,dense lymphocytes and plasma cell infiltration(Figs 2,3), and an examination of the epididymis wasnormal.

Discussion

Xanthogranulomatous infection is characterized byfoamy macrophages, dense lymphocytes and plasmacell infiltration in affected tissue. Although many theo-ries have been proposed to explain the development ofthis pathology, the etiology of xanthogranulomatousinfection remains obscure. Possible etiological factorsinclude urinary tract infections with ineffective antibi-otic therapy, calculous or non-calculous obstructions,malnutrition, abnormal lipid metabolism, altered immu-

Xanthogranulomatous orchitis 687

nologic response, lymphatic blockage and congenitalurinary anomalies.2 We could not find any possible eti-ological factors in our case.

Although the common clinical form of the pathologyis diffuse or local pyelonephritis, it remains a rare dis-ease with frequency of 0.6% to 1.4%. However, it canoccur in other organs such as the gall bladder, ovary,appendix or urinary bladder.3 Genital system involve-ment, such as epidymitis or funiculitis with orchitis, arealso very rare.3,4 Reflux of urine to the seminal vesiclesor vas deferens and urethral manipulation (catheteriza-tion or surgery) were reported as possible risks for thedevelopment of the inflammation.3,4 The present casewas unrelated to the above-mentioned conditions. Fur-ther investigation of the pathogenesis of xanthogranulo-

matous infection in the urological field may benecessary.

Although computed tomography appears to be themost valuable tool in the diagnosis of xanthogranuloma-tous pyelonephritis (XGP), a differential diagnosis ofXGP from a renal tumor is very difficult.5 Xanthogran-ulomatous pyelonephritis is treated with surgery andusually diagnosed after total or partial nephrectomy.Likewise, the accurate diagnosis of xanthogranuloma-tous orchitis is also difficult.6 Tumor markers can alsobe used, but tumor markers may not be increased insome testicular tumors. In the present case, surgicalexploration of the left testis was an adequate procedureto establish the precise diagnosis.

Because serious tissue destruction is a feature ofxanthogranulomatous inflammation; surgical removalis the curative treatment in most cases because of inef-fective medical treatment.5 However, successful medi-cal management of the focal XGP has been reported.7

Medical therapy was not a viable option in the presentcase, so we performed radical orchiectomy and hemis-crotectomy to exclude the possibility of the testiculartumor.

In conclusion, surgical excision seems to be the stan-dard treatment for xanthogranulomatous orchitis.

References

1 Samuel M, Duffy P, Capps S, Mouriquand P, WilliamsD, Ransley P. Xanthogranulomatous pyelonephritis inchildhood. J. Pediatr. Surg. 2001; 36: 598–601.

2 Hammadeh MY, Nicholls G, Calder CJ, Buick RG, Gor-nall P, Corkery JJ. Xanthogranulomatous pyelonephritis

Fig. 1 Ultrasonographic appearance of the infiltrativeand hyperechoic testicular tumor in the left testis.

Fig. 2 The tubules and interstitial tissue of the left testiswere replaced by foamy macrophages, dense lymphocytesand plasma cells. HE ¥ 100.

Fig. 3 Numerous xanthoma cells accompanied by lym-phocyte and plasma cell infiltration. HE ¥ 400.

688 D Demırcı et al.

in childhood: pre-operative diagnosis is possible. Br. J.Urol. 1994; 73: 83–6.

3 Vaidyanathan S, Mansour P, Parsons KF et al. Xan-thogranulomatous funiculitis and epididymo-orchitis ina tetraplegic patient. Spinal Cord 2000; 38: 769–72.

4 Matsuoka K, Yano H, Inoue M, Iida S, Hirabayasi Y,Noda S. Xanthogranulomatous epididymitis. BJU Int.2001; 87: 275–6.

5 Osca JM, Peiro MJ, Rodrigo M, Martinez-Jabaloyas JM,Jimenez-Cruz JF. Focal xanthogranulomatous pyelone-

phritis: partial nephrectomy as definitive treatment. Eur.Urol. 1997; 32: 375–9.

6 Scott RF, Bayliss AP. Ultrasound in the diagnosis ofgranulomatous orchitis. Br. J. Radiol. 1985; 58: 907–9.

7 Mollier S, Descotes JL, Pasquier D et al. Pseudoneo-plastic xanthogranulomatous pyelonephritis. A typicalclinical presentation, but unusual diagnosis and treat-ment. Eur. Urol. 1995; 27: 170–3.