A 71-year-old woman was admitted complaining of left flank pain, and presented with oliguria and hypotension. Pyuria and severe sepsis led to the diagnosis of acute pyelonephritis. An abdominal computed tomography intravenous pyelogram revealed normal-sized kidneys, however, the differentiation between cortex and medulla was poorly determined. The left kidney showed an amorphic, low attenuation lesion that was consistent with acute pyelonephritis (Fig. 1A). Both kidneys were incompletely malrotated and anteriorly facing hila were observed. The renal vessels and ureter were located laterally. The right kidney was 5 cm lower than the left kidney, indicating renal ectopia. Mild dilation was found at the proximal ureter of the left kidney. Additionally, a three-dimensional computed tomography reconstruction image showed anteriorly rotated kidneys (Fig. 1B). The patient initially received piperacillin-tazobactam empirically, then switched to cefotaxime after detecting an E. coli infection in the bloodstream. Three days later, her hemodynamic state had improved, and she was released from the intensive care unit.