Hemolytic disease of the fetus and newborn (HDFN) due to anti-D was severe and fatal before the development of RhD immune prophylaxis. Proper screening and universal administration of Rh immune globulin has decreased the incidence of HDFN to a great extent. Pregnancy, transfusion, and transplantation still increase the chances of other alloantibody formation and the potential for HDFN. Advanced methods for immunohematology investigation allow for the identification of alloantibodies causative for HDFN other than anti-D. Many antibodies have been reported to cause HDFN, but there is scant literature where isolated anti-C is responsible for HDFN. We present here a case of severe HDFN caused by anti-C leading to severe hydrops and death of the neonate despite three intrauterine transfusions and other measures.
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