Glomerulocystic kidney disease (GCKD) is a rare congenital condition that i
s usually reported in infants and young children. Only five cases of acquir
ed GCKD after an acquired renal disease have been reported. Among these, tw
o patients have developed cystic glomerular lesions following hemolytic ure
mic syndrome (HUS). We report a third case in a 2-year-old patient with thi
s association. Common features between these three cases include atypical H
US, development of GCKD after prolonged peritoneal dialysis treatment, seve
re hypertension, and normal-sized kidneys without development of macroscopi
c cysts. Pathology findings in our patient include heavy expression of epid
ermal growth factor receptor in proximal tubules and evidence of obstructio
n of the glomerular outflow. We speculate that cystic dilatation of the Bow
man's capsule may be secondary to ischemic lesions leading to proximal tubu
lar obstruction.