title: A case of severe Rh (D) alloimmunization treated by intensive plasma exchange and high-dose intravenous immunoglobulin.
abstract: BACKGROUND
  In extremely severe Rh (D) alloimmunization, during pregnancy, early diagnosis and treatment is essential to avoid hydrops fetalis. Intrauterine transfusion (IUT) is of utmost importance in the prevention of fetal anemia but it is usually feasible only after 20 weeks of pregnancy. Therefore, additional treatment options in early pregnancy are needed.
  
  
  METHODS
  A 27-year-old severely D+C immunized woman was admitted at 8 weeks of gestation in her fifth pregnancy with an extremely high concentration of anti-D. Her first pregnancy was uneventful but resulted in D+C alloimmunization. The next two pregnancies were unsuccessful, because of hydrops fetalis resulting in fetal death in pregnancy week 20 and 24, respectively, despite treatment with high-dose intravenous immunoglobulin (IVIG) and IUT treatment. A fourth pregnancy was terminated with legal abortion. The patient was eager and persistent to accomplish a successful pregnancy. Therefore, a combination of treatments consisting of plasma exchange (PE) three times/week and IVIG 100g/week was started in pregnancy week 12. PE was performed 53 times and totally 159L of plasma was exchanged.
  
  
  RESULTS
  The anti-D concentration was 12mug/mL (IAT titer 2000) before start of treatment by PE and IVIG in pregnancy week 12. The concentration of anti-D was gradually reduced to approximately 3mug/mL after only two weeks of treatment and was maintained at that level until pregnancy week 22. In pregnancy week 26 and 27, signs of hydrops were detected by ultrasonography and IUT were performed at each occasion. Sectio was inevitable at pregnancy week 28+1 and a male baby was born: Hb 58g/L (cord sample) and 68g/L (venous sample); weight 1385g; Apgar score=4-5-7; Bilirubin 56-150mmol/L (4h). Exchange transfusion was performed on day two and day five. Phototherapy was also implemented for eight days. The newborn's recovery thereafter was uneventful and complete.
  
  
  CONCLUSIONS
  A combination of PE and IVIG may be an efficient treatment possible to start in early pregnancy in patients with extremely severe Rh (D) alloimmunization, with a history of hydrops fetalis in previous pregnancies.
authors.affiliation: Department of Transfusion Medicine and Clinical Immunology, Faculty of Health Sciences, Linköping University, 581 85 Linköping, Sweden. miodrag.palfi@lio.se
authors.forename: Miodrag
authors.initials: M
authors.lastname: Palfi
authors.forename: Jan-Olof
authors.initials: JO
authors.lastname: Hildén
authors.forename: Leif
authors.initials: L
authors.lastname: Matthiesen
authors.forename: Anders
authors.initials: A
authors.lastname: Selbing
authors.forename: Gösta
authors.initials: G
authors.lastname: Berlin
mesh_terms: D002585:Cesarean Section; D005260:Female; D006801:Humans; D015160:Hydrops Fetalis / Q000175:diagnosis / Q000628:therapy*; D016756:Immunoglobulins, Intravenous / Q000008:administration & dosage*; D007231:Infant, Newborn; D050498:Live Birth; D008297:Male; D010951:Plasma Exchange; D011247:Pregnancy; D011250:Pregnancy Complications, Hematologic / Q000097:blood / Q000628:therapy*; D012203:Rh Isoimmunization / Q000097:blood / Q000628:therapy*; D012204:Rh-Hr Blood-Group System; D018029:Rho(D) Immune Globulin / Q000097:blood; D012720:Severity of Illness Index
chemical_list: D016756:Immunoglobulins, Intravenous; D012204:Rh-Hr Blood-Group System; D018029:Rho(D) Immune Globulin