The occurrence of unconjugated hyperbilirubinemia in infants with pyloric stenosis is common. We report an infant who presented with projectile vomiting secondary to pyloric stenosis. In addition, he had conjugated hyperbilirubine mia which proved to be due to alpha-1-antitrypsin deficiency of PiZZ pheno type. This is the first case report of such an association. Infants with pyloric stenosis and conjugated hyperbilirubinemia should be investigated for under lying infectious, metabolic causes or anatomical defects of the biliary tree.
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