BACKGROUND: Hookworm infections (Necator americanus, Ancylostoma duodenale) are common in rural areas of tropical and subtropical countries. Human acquisition results from direct percutaneous invasion of infective larvae from contaminated soil. Overall, almost 472 million people in developing rural countries are infected. According to simulation models, hookworm disease has a global financial impact of over US$100 billion a year. Hookworm infection in newborn or infancy is rare, and most of the cases reported in literature are from endemic countries. Here, we describe the case of an infant with an Ancylostoma duodenale infection and review the literature currently available on this topic. CASE PRESENTATION: An Italian 2-month-old infant presented with vomit and weight loss. Her blood exams showed anemia and eosinophilia and stool analysis resulted positive for hookworms' eggs, identified as Ancylostoma duodenale with real time-PCR. Parasite research on parents' stools resulted negative, and since the mother travelled to Vietnam and Thailand during pregnancy, we assumed a transplacental transmission of the infection. The patient was treated successfully with oral Mebendazole and discharged in good conditions. DISCUSSION: Hookworm helminthiasis is a major cause of morbidity in children in the tropics and subtropics, but rare in developed countries. Despite most of the patients is usually asymptomatic, children are highly exposed to negative sequelae such as malnutrition, retarded growth and impaired cognitive development. In infants and newborns, the mechanism of infection remains unclear. Although infrequent, vertical transmission of larvae can occur through breastfeeding and transplacentally. Hookworm infection should be taken into account in children with abdominal symptoms and unexplained persistent eosinophilia. The treatment of infants infected by hookworm has potential benefit, but further studies are needed to define the best clinical management of these cases