A case of xanthogranulomatous epididymitis complicated by florid testicular cystic ectasia

Abstract

Xanthogranulomatous inflammation (XGI) is a rare, benign inflammatory condition of unclear pathogenesis, characterised by infiltration and subsequent destruction of normal tissue by lipid-laden macrophages together with lymphocytes and plasma cells. A 56-year-old gentleman was referred to the urology department of our hospital due to concerns that his right testicle felt firmer than his left over the preceding six months. He was investigated and subsequently underwent a right sided orchidectomy. The overall histopathological findings were consistent with chronic xanthogranulomatous epididymitis with testicular outflow tract obstruction and cystic ectasia of the rete testis and testicular parenchyma.

1 Introduction

Xanthogranulomatous inflammation (XGI) is a rare, benign inflammatory condition of unclear pathogenesis, characterised by infiltration and subsequent destruction of normal tissue by lipid-laden macrophages together with lymphocytes and plasma cells. ^, Xanthogranulomatous inflammation is best known for its ability to produce tumefactive masses, which may mimic malignancy clinically and radiologically. While XGI is well-recognised to affect the genitourinary system (with xantogranulomatous pyelonephritis being the prototypical example), involvement of the genital system is rare. Herein, we present a case of xanthogranulomatous epididymo-orchitis (XGEO) complicated by testicular outflow obstruction.

2 Case-report

A 56-year-old male was referred to the Urology Outpatient Department due to concerns that his right testicle felt firmer than his left over the preceding six months. There were no signs or symptoms to suggest an infective aetiology. He had a past medical history of coeliac disease and gastro-oesophageal reflux disease, for which he was on proton-pump inhibitor treatment . Clinical examination showed diffuse swelling, induration and tenderness of the right testis with no discreet palpable mass. The left testis was clinically normal. All blood tests including testicular tumour markers were within normal range. Urine cultures were negative.

An initial ultrasound of the right testicle was performed, which suggested a right-sided spermatocele ( Fig. 1 ). Persistent right testicular discomfort was noted at three-month follow-up. Repeat ultrasound studies showed a swollen right testicle with a normal echogenic structure and Doppler flow exhibiting extensive ectasia of the rete testis and multicyst formation at the level of the testicular hilum ( Fig. 2 ). Given his persistent discomfort, after discussion with him, it was agreed to proceed to a right orchidectomy. Since malignancy was not suspected, the testicle was removed via a midline scrotal incision however, the testicle and the spermatic cord were removed with the tunica vaginalis layer intact. His post-operative recovery was uneventful and he was followed-up as an outpatient following the surgery.

Pathological examination confirmed an enlarged, firm right testicle measuring 62mm superoinferiorly, 35mm mediolaterally and 32mm anteroposteriorly, with the tunica vaginalis firmly adherent to the tunica albuginea in the region of the head of the epididymis and the testicular hilum. On sectioning, the hilar half of the testicle was completely replaced by numerous microcysts inspissated with mucoid material. The lateral half of the testicle exhibited a grossly normal architecture. The epididymis was diffusely firm and indurated, with the head of the epididymis also exhibiting a microcystic architecture. The spermatic cord was grossly normal. No testicular masses were identified ( Fig. 3 ).