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Case report

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  2. Case report
  3. Comment
  4. References
  5. Authors

A 70-year-old man underwent endoscopic holmium laser prostatectomy; he received flomoxef sodium intravenously for 3 days after treatment. The urethral balloon was withdrawn on the second day after surgery. On the fifth day he developed a fever of 39.6 °C and complained of right scrotal pain. The right epididymis was indurated, swollen and markedly tender; the testis was also swollen. The serum leukocyte count was 12 300/mL, while the C-reactive protein was 41 mg/L. Urine analysis revealed pyuria and urine culture was positive for Pseudomonas aeruginosa (105 c.f.u./mL). The patient received amikacin for 10 days. The tenderness subsequently subsided and both the serum leukocyte count and C-reactive protein values improved. Thereafter, the patient was treated with oral ciprofloxacin hydrochloride. Induration of the epididymis remained unchanged and there was partial adhesion to the skin. This condition continued until the right epididymal abscess spontaneously ruptured and drained, 1 month after its onset. The microbial culture of the pus was positive for P. aeruginosa. Epididymectomy was selected but as the epididymis could not be separated from the testis, the epididymis and testis were resected together. Macroscopically, most of the epididymis appeared bright yellow and was associated with the abscess. Histopathological findings showed the presence of numerous xanthoma cells and inflammatory granulomas, accompanied by lymphocyte and plasma cell infiltration around the dilated ductus, in the head and through the body to the tail of the epididymis, leading to the diagnosis of xanthogranulomatous inflammation (Fig. 1 ).

image

Figure 1. (a) Diffuse chronic inflammatory cells with granulomatous formation and fibrosis are observed around epididymal tubules. Haematoxylin and eosin × 20. (b) Numerous xanthoma cells and the formation of inflammatory granulomatous tissue accompanied by lymphocyte and plasma cell infiltration. Haematoxylin and eosin × 100.

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Comment

  1. Top of page
  2. Case report
  3. Comment
  4. References
  5. Authors

Although most cases of xanthogranulomatous inflammation within urology have been xanthogranulomatous pyelonephritis, it remains a rare disease with a frequency of 0.6–1.4% [1]. Xanthogranulomatous epididymitis is extremely rare. The present is only the third case reported in the English and Japanese literature [2,3]. The first case of xanthogranulomatous epididymitis was reported by Wiener et al.[2]; who reported suppurative tissue changes arising from the epididymis that progressed into further xanthogranulomatous involvement. Findings of this type have been reported in xanthogranulomas arising in other organs [4–6]. The present case had a severe inflammatory lesion concomitant with intrascrotal tissue destruction. The pathological findings revealed a dominant lymphocyte and plasma cell infiltration, surrounded by accumulating foamy cells, resulting in xanthogranulomatous changes. Specific inflammatory findings, such as epithelioid granuloma, were not seen and there was no spermatogenic granuloma. Also, xanthogranulomatous inflammation must be distinguished from malacoplakia; in the present case, routine haematoxylin and eosin, periodic acid-Schiff and Von Kossa staining did not show Michaelis-Gutmann bodies. Accordingly, the diagnosis of malacoplakia was excluded.

The pathogens that generally cause xanthogranulomatous pyelonephritis are Proteus and Escherichia coli[1]. The causative organism in our case was P. aeruginosa. Nevertheless, the aetiology is not limited to infections by specific bacteria, as xanthogranulomatous inflammation is the result of chronic bacterial infection. The two previously reported cases both had diabetes mellitus, although the onsets were unrelated to surgery. In contrast, the onset of the present case was related to surgery, but free from a diabetic condition. Thus, xanthogranulomatous epididymitis should be included in the differential diagnosis of intrascrotal disorders even in patients who do not have diabetes, and this type of inflammatory destructive lesion should be removed surgically.

References

  1. Top of page
  2. Case report
  3. Comment
  4. References
  5. Authors
  • 1
    Schaeffer AJ. Infections of the urinary tract. In WalshPC, RetikAB, VaughanED, WeinAJ eds, Campbell’s Urology 7th edn, Vol. 1. Chapt 15. Philadelphia; WB Saunders, 1998: 275276
  • 2
    Wiener LB, Riehl PA & Baum N. Xanthogranulomatous epididymitis: A case report. J Urol l987; 138: 6212
  • 3
    Matsubara T, Kasagi Y, Hoshino T, Mimata H & Nomura Y. Xanthogranulomatous epididymitis. A case report. Jpn J Urol 1999; 90: 87881
  • 4
    Minkowitz S, Friedman F & Henniger G. Xanthogranuloma of the ovary. Arch Path 1965; 80: 20913
  • 5
    Fligiel S & Lewin K. Xanthogranulomatous cholecystitis. Case report and review of the literature. Arch Pathol Lab Med 1982; 106: 3024
  • 6
    Chung MK, Seol MY, Cho WY, Seo HK & Kim JS. Xanthogranulomatous cystitis associated with suture material. J Urol 1998; 159: 9812

Authors

  1. Top of page
  2. Case report
  3. Comment
  4. References
  5. Authors

K. Matsuoka, MD.

H. Yano, MD.

M. Inoue, MD.

S. IIda, MD.

Y. Hirabayashi, MD.

S. Noda, MD.