Skip to main content
Log in

Successful management of fetal hemolytic disease due to strong anti-Rh17 with plasma exchange and intrauterine transfusion in a woman with the D−− phenotype

International Journal of Hematology Aims and scope Submit manuscript

Abstract

The rare blood phenotype D−− is characterized by the absence of RhCcEe antigens. Women with this blood type who have experienced previous pregnancies may produce anti-Rh17 antibodies, which may cause severe fetal hemolytic anemia or fetal death in subsequent pregnancies. We report successful management of a pregnancy associated with fetal hemolytic disease owing to high titers of anti-Rh17 (1:4096) in a woman with a history of a pregnancy with fetal hydrops and intrauterine fetal death. During her second pregnancy, she received two sets of plasma exchange (PE) per week from weeks 12 till 20. Intrauterine transfusions (IUTs) were performed at 26, 27, 29, and 31 weeks. A male infant was born at 32 weeks and 4 days by normal vaginal delivery, with a birth weight of 1916 g (+ 0.16 SD). He received an exchange transfusion on day 0, immunoglobulin (intravenous immunoglobulin: 1 g/kg) on days 0 and 1, and photo therapy from days 0 to 6. He showed normal development without neurological abnormality and was discharged from the hospital on day 36. We successfully prevented complications caused by the presence of anti-Rh17 antibodies in the mother during pregnancy. The IUT and maternal PE may have promoted this favorable outcome.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1
Fig. 2
Fig. 3

Similar content being viewed by others

References

  1. Race RR, Sanger R. Blood groups in man. Oxford: Blackwell Science Publisher; 1975.

    Google Scholar 

  2. Diao Y, Song N, Guan Z, Zhang L, Wang Y, Wan M. A case of hemolytic disease of the newborn caused by anti-Hro and anti-e. Proc Chin Acad Med Sci Peking Union Med Coll. 1990;5:58–60.

    CAS  PubMed  Google Scholar 

  3. Deitenbeck R, Tutschek B, Crombach G, Stannigel H. Successful management of pregnancy and hemolytic disease of the newborn due to anti-HrO in a woman of the D−− phenotype. Transfusion. 1999;39:1151–2.

    Article  CAS  Google Scholar 

  4. Whang DH, Kim HC, Hur M, Choi JH, Park JS, Han KS. A successful delivery of a baby from a D−−/D−− mother with strong anti-Hr0. Immunohematology. 2000;16:112–4.

    CAS  PubMed  Google Scholar 

  5. Aref K, Boctor FN, Pande S, Uehlinger J, Manning F, Eglowstein M, et al. Successful perinatal management of hydrops fetalis due to hemolytic disease associated with D−− maternal phenotype. J Perinatol. 2002;22:667–8.

    Article  CAS  Google Scholar 

  6. Hirose M, Nakanishi K, Kaku S, Moro H, Hodohara K, Aotani H, et al. Fetal hemolytic disease due to anti-Rh17 alloimmunization. Fetal Diagn Ther. 2004;19:182–6.

    Article  Google Scholar 

  7. Denomme GA, Ryan G, Seaward PG, Kelly EN, Fernandes BJ. Maternal ABO-mismatched blood for intrauterine transfusion of severe hemolytic disease of the newborn due to anti-Rh17. Transfusion. 2004;44:1357–60.

    Article  CAS  Google Scholar 

  8. Salamat N, Bhatti FA, Hussain A. Anti-Rh17 (anti-Hr0): a rare diagnostic and management problem. J Pak Med Assoc. 2004;54:215–8.

    CAS  PubMed  Google Scholar 

  9. Shah SI, Caprio M, Strauss R, Moskowitz N. Management of a full-term infant with hemolytic disease of the newborn due to an anti-Rh17 antibody in a mother with D−− phenotype. Am J Hematol. 2005;80:88–9.

    Article  Google Scholar 

  10. Moise KJ Jr. Hemolytic disease of the fetus and newborn. In: Creasy RK, Resnick R, editors. Maternal fetal medicine principles and practice. Philadelphia: WB Saunders; 2004. p. 551–553.

    Google Scholar 

  11. Schumacher B, Moise KJ Jr. Fetal transfusion for red blood cell alloimmunization in pregnancy. Obstet Gynecol. 1996;88:137–50.

    Article  CAS  Google Scholar 

  12. Schwartz J, Padmanabhan A, Aqui N, Balogun RA, Connelly-Smith L, Delaney M, et al. Guidelines on the use of therapeutic apheresis in clinical practice-evidence-based approach from the Writing Committee of the American Society for Apheresis: the seventh special issue. J Clin Apher. 2016;31:149–62.

    Google Scholar 

  13. Dodd JM, Andersen C, Dickinson JE, Louise J, Deussen A, Grivell RM, et al. Fetal middle cerebral artery Doppler to time intrauterine transfusion in red-cell alloimmunization: a randomized trial. Ultrasound Obstet Gynecol. 2018;51:306–12.

    Article  CAS  Google Scholar 

  14. Dajak S, Culić S, Stefanović V, Lukačević J. Relationship between previous maternal transfusions and haemolytic disease of the foetus and newborn mediated by non-RhD antibodies. Blood Transfus. 2013;11:528–32.

    PubMed  PubMed Central  Google Scholar 

  15. Radunovic N, Lockwood CJ, Alvarez M, Plecas D, Chitkara U, Berkowitz RL. The severely anemic and hydropic isoimmune fetus: changes in fetal hematocrit associated with intrauterine death. Obstet Gynecol. 1992;79:390–3.

    Article  CAS  Google Scholar 

  16. Xu X, Li L, Xia W, Ding H, Chen D, Liu J, et al. Successful management of a hydropic fetus with severe anemia and thrombocytopenia caused by anti-CD36 antibody. Int J Hematol. 2018;107:251–6.

    Article  CAS  Google Scholar 

  17. Van Kamp IL, Klumper FJ, Oepkes D, Meerman RH, Scherjon SA, Vandenbussche FP, et al. Complications of intrauterine intravascular transfusion for fetal anemia due to maternal red-cell alloimmunization. Am J Obstet Gynecol. 2005;192:171–7.

    Article  Google Scholar 

  18. Angela E, Robinson E, Tovey LA. Intensive plasma exchange in the management of severe Rh disease. Br J Haematol. 1980;45:621–31.

    Article  CAS  Google Scholar 

  19. Ruma MS, Moise KJ Jr, Kim E, Murtha AP, Prutsman WJ, Hassan SS, et al. Combined plasmapheresis and intravenous immune globulin for the treatment of severe maternal red cell alloimmunization. Am J Obstet Gynecol. 2007;196(138):e1–6.

    Google Scholar 

  20. Isojima S, Hisano M, Suzuki T, Sago H, Murashima A, Yamaguchi K. Early plasmapheresis followed by high-dose γ-globulin treatment saved a severely Rho-incompatible pregnancy. J Clin Apher. 2011;26:216–8.

    Article  Google Scholar 

  21. Kamei K, Yamaguchi K, Sato M, Ogura M, Ito S, Okada T, et al. Successful treatment of severe rhesus D-incompatible pregnancy with repeated double-filtration plasmapheresis. J Clin Apher. 2015;30:305–7.

    Article  Google Scholar 

Download references

Acknowledgements

The authors would like to thank Enago (www.enago.jp) for the English language review.

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Kazuya Mimura.

Ethics declarations

Conflict of interest

The authors declare that they have no conflict of interest.

Human rights and animal participant statement

This article does not contain any studies with human or animal subjects performed by any of the authors.

Informed consent

Written informed consent was obtained from the patient for the publication of the case report.

Additional information

Publisher's Note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Mimura, K., Endo, M., Takahashi, A. et al. Successful management of fetal hemolytic disease due to strong anti-Rh17 with plasma exchange and intrauterine transfusion in a woman with the D−− phenotype. Int J Hematol 111, 149–154 (2020). https://doi.org/10.1007/s12185-019-02735-6

Download citation

  • Received:

  • Revised:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s12185-019-02735-6

Keywords

Navigation