Infantile pyknocytosis is a rare form of neonatal haemolytic anaemia with unusual red cell morphology. Anaemia is mostly severe and red blood cells transfusion is often needed. In this report, we have described a male child aged 10 days, born at 37 weeks + 3 days, who presented neonatal jaundice and severe anaemia. After a careful peripheral blood smear examination, infantile pyknocytosis was diagnosed. A treatment with recombinant subcutaneous erythropoietin (1,000 UI/prokg/week) in conjunction with iron supplementation (6 mg/kg/day) was started. The therapy was reduced 6 weeks after the beginning and discontinued 4 weeks after the reaching of a steady state of the haemoglobin values. After 12 months of follow up, the patient showed no anaemia and pyknocytosis.