Background: Juvenile xanthogranulomas (JXG) is a histiocytic disorder, in a child below 5 years of age. Xanthogranulomatous orchitis (XGO) is a rare disease, it is an extremely rare inflammatory non-neoplastic destructive lesion of the testis, and a few cases have been reported to date. Aim: We reported a case of JXO presenting as a solitary mass in the left testicle causing testicular torsion in an 11-month-old child without manifestations of JXG elsewhere. Case presentation: the patient presented with an acute history of pain in his left hemi scrotum. An ultrasound study showed a 10 x 13 mm round tumour with a distinct boundary between the tumour and the irregular heterogeneous testicular tissue, Doppler ultrasound images submitted changes suggestive of testicular torsion. Surgery was performed that showed a necrotic left testicle, with and tumour-like lesion attached to the cord and overlapping the body of epididymis. A left orchiectomy was performed. Histopathology reported that the architecture of the testis was lost. Two cut sections in the mass nodular collections of abundant xanthomatous foamy macrophages. Xanthogranulomas were suggested. Conclusion: Because of the aggressive nature of this disease, curative treatment is by either radical or partial orchiectomy considered the goal standard for the final management outcome.
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