Infantile hypertrophic pyloric stenosis (IHPS) is a common surgical cause of vomiting, which requires surgery in infant. There is the complete or partial obstruction of pylorus due to hypertrophy of circular muscle of the pylorus leading gastric outlet obstruction. The occurrence of IHPS in dizygotic twins is rarer and the theory of genetic origin alone does not explain it. Recent literature points to the association of bottle feeding in singletons to be a major aetiological factor for this condition. Here, we present a rare case of dizygotic twins who were bottle-fed and were affected with IHPS. We review the literature and focus on the question of whether this condition in twins supports a genetic or environmental origin of IHPS.