Dear Editor,We report a rare case of a 2-year-old male child with Wilms’ tumor with unusual left testicular metastasis. The patient presented with fever, pain abdomen, and left flank mass of approximate size 8.0 cm × 8.0 cm, of 7 months duration. Computed tomography (CT) scan revealed a 7.8 cm × 7.0 cm × 7.8 cm mass in the left kidney. He underwent left radical nephrectomy in August 2015 and final diagnosis was stage I with tumor size of 8.0 cm × 6.5 cm, favorable histology, and with blastemal component on histopathology report. He received five cycles vincristine and cyclophosphamide-based adjuvant chemotherapy at another hospital. In June 2016, he presented with 7.6 cm × 8.6 cm × 9.6 cm retroperitoneal mass with multiple lung metastasis. The boy was given salvage chemotherapy with alternating cycles of cyclophosphamide, etoposide and doxorubicin, and vincristine for two cycles. CT scan at the end of salvage chemotherapy shows complete resolution of lung metastasis and retroperitoneal mass remained unchanged. He received 21.6 Gy/12 fractions to local disease and 12 Gy/8 fractions as lung bath. One month later, a left testicular swelling was detected. Ultrasonography (USG) done in August 2016 showed left testicular mass of size 2.7 cm × 2.6 cm with heterogeneous enhancement. Fine-needle aspiration cytology from testicular mass confirmed metastatic Wilms’ tumor. Patient was then given radiotherapy (10.8 Gy/6 fractions) to scrotum in August 2016. USG of scrotum showed decrease in the mass up to 2 cm × 1.6 cm (partial response). Additional radiotherapy of 19.8 Gy was given followed by chemotherapy (ifosfamide, carboplatin, and etoposide).This is an interesting case since testicular metastasis in Wilms’ tumor is rare, ipsilateral spread can be caused through spermatic vein, and use or radiation therapy is feasible (with partial response) even after multiple recurrences.[123] It also teaches us that retrograde venous and transcoelomic spread may potentially be prevented by spermatic vein ligation immediately before nephrectomy.