We describe a case of a 24-year-old female renal transplant recipient who, 10 years after receiving a deceased-donor kidney, presented with acute and massive increases in serum creatinine and proteinuria levels of 13 g over 24 hours. At a previous outpatient clinic visit, her baseline serum creatinine was noted to be 87 μmol/L; on admission, serum creatinine was 1377 μmol/L. Renal biopsy results were consistent with acute cellular rejection with severe interstitial lymphoplasmacytic infiltrates and edema with no evidence of glomerular pathology, including transplant glomerulopathy. The immunofluorescence test results were negative, and the ultrastructural features were consistent with podocytopathy with no immune deposits present. We believe thatthis is the first case of acute cellular rejection typified by severe interstitial lymphoplasmacytic infiltrates and edema with severe proteinuria secondary to minimal change disease (or podocytopathy).