We report a case of microphthalmia, inferior adherent leukoma, and optic nerve hypoplasia in an infant whose mother underwent biliopancreatic diversion surgery for obesity 7 years before his birth. The pregnancy was complicated by severe, maternal hypovitaminosis A despite oral supplementation. The infant was found to have undetectable serum vitamin A levels in the perinatal period. At 8 weeks of age, the infant underwent sector iridectomies. At 9 months of age, electroretinography suggested rod dysfunction. His visual performance is poor.