We are presenting a 2-year male child with large abdominal mass extending from right inguino-scrotal region to suprapubic region of size 4.0 × 5.0 cm. Mass causing pressure effect over bilateral kidneys and ureters with bilateral dilated renal pelvis and extending into the right inguinal region and pelvis. Histopathologically and immunocytochemically patient was confirmed as rhabdomyosarcoma of the retroperitoneum. Patient treated with six cycles of infusion chemotherapy with 3- weekly VAC regimen and was having progressive disease because of the aggressive behaviour of the disease and further treated with second line chemotherapy. The present case is a very unusual and rare site of metastatic presentation of the Rhabdomyosarcoma.
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