Severe and prolonged thrombocytopenia is not uncommon among ill preterm infants. Pseudothrombocytopenia, which has the appearance of severe and prolonged thrombocytopenia, has not been described in this population. We observed a preterm neonate who had EDTA-independent pseudothrombocytopenia and conclude that this condition should be considered when severe and prolonged thrombocytopenia occurs in a neonate in the absence of clinical signs of platelet-type hemorrhage.