| Summary: | Abstract Background Although paediatric patients with acute lymphoblastic leukaemia (ALL) have improved survival following modern chemotherapy treatment, disease relapses still occur in 20–25% of them. Our case had relapsed ALL with testicular leukaemic infiltration, which is rare, with less than 2% incidence. Case presentation We describe a case of an 11-year-old boy with a background history of ALL presenting with painless unilateral scrotal swelling. An initial ultrasound of the testes showed heterogeneity throughout with hypervascularity on colour-flow imaging on the affected testis. Some areas are hypoechoic with dilated tubules. The histopathology of the follow-up testicular biopsy revealed malignant cells and leukaemic infiltration. Patient subsequently received chemotherapy treatment at another centre. Conclusions Testicular relapse of ALL is relatively rare with the use of improved contemporary frontline treatment with better outcomes. With its non-specific symptoms, radiological imaging can significantly facilitate timely diagnosis and contribute to appropriate further management and improved prognosis.
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