The Ce antigen is expressed on red cells of individuals with Rh haplotypes, R1 or CDe and r' or Cde. However, anti-Ce is rare, and only two cases of severe haemolytic disease of the newborn (HDN) caused by this antibody have been described (Malde et al., 2000; Wagner et al., 2000). We describe a woman who was found to have anti-Ce in her second pregnancy, resulting in a neonate with HDN that required exchange transfusion. She subsequently had a twin pregnancy, where both the twins were affected by severe haemolytic disease (HD) of the fetus because of anti-Ce and required repeated fetal transfusions, followed by exchange transfusions after birth. This is the first reported case of HD caused by anti-Ce requiring fetal transfusions.