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Patients with autosomal recessive polycystic kidney disease (ARPKD) often present with renal insufficiency and hypertension. We present two children with ARPKD and end-stage renal disease who developed anterior ischemic optic neuropathy and vision loss. Anterior ischemic optic neuropathy occurs rarely in children and has never been reported in children with ARPKD or end-stage renal disease. Both of...