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International Journal of Hematology

, Volume 107, Issue 2, pp 251–256 | Cite as

Successful management of a hydropic fetus with severe anemia and thrombocytopenia caused by anti-CD36 antibody

  • Xiuzhang Xu
  • Lin Li
  • Wenjie Xia
  • Haoqiang Ding
  • Dawei Chen
  • Jing Liu
  • Jing Deng
  • Yangkai Chen
  • Zhiming He
  • Jiali Wang
  • Yuan Shao
  • Sentot Santoso
  • Xin Ye
  • Qun Fang
Case Report

Abstract

Cases of CD36 deficiency are not rare in Asian populations, foetal and neonatal alloimmune thrombocytopenia (FNAIT) caused by anti-CD36 isoantibodies appears more frequent than other HPA alloantibodies. However, little is known about the treatment of anti-CD36 mediated FNAIT in this region. A Chinese male foetus, whose mother had a history of multiple intrauterine foetal demise and/or hydrops, was diagnosed with severe FNAIT at 27 weeks of gestational age. Immunological analysis revealed total absence of CD36 on platelets and monocytes from mother, caused by a 329–330delAC mutation of the CD36 gene. Anti-CD36 and anti-HLA class I antibodies were detected in the maternal serum, whereas only anti-CD36 isoantibodies were detectable in the foetal blood sample. Serial intrauterine transfusions with red blood cells (RBC) and platelets from a CD36null donor were performed to improve the severe anaemia and thrombocytopenia. The baby (2250 g; Apgar scores 10) was delivered vaginally at 32 weeks of gestation with normal haemoglobin (186 g/L) but low platelet count (48 × 109/L). After 2 days the platelet count rose to 121 × 109/L. This report suggests that intrauterine transfusions with compatible RBC and CD36null platelets are useful in preventing the deleterious clinical effects of anti-CD36-mediated severe FNAIT.

Keywords

Foetal and neonatal alloimmune thrombocytopenia Anti-CD36 antibody Foetal anaemia and hydrops Intrauterine transfusion 

Notes

Acknowledgements

This work was supported by the grant from National Natural Science Foundation of China (Ref. number: 81601451), by the Natural Science Foundation of Guangdong Province of China (no. 2016A030313124/no. 2016A030313123), by the German Research Foundation, Excellence Cluster Cardiopulmonary System (to S. S.), and by Science and Technology and Innovative Commission of Guangzhou City (no. 201607010007/no. 201509010009/no. 201707010021).

Compliance with ethical standards

Conflict of interest

All authors declare that there is no conflict of interest.

References

  1. 1.
    Kamphuis MM, Paridaans NP, Porcelijn L, Lopriore E, Oepkes D. Incidence and consequences of neonatal alloimmune thrombocytopenia: a systematic review. Pediatrics. 2014;133:715–21.CrossRefPubMedGoogle Scholar
  2. 2.
    Turner ML, Bessos H, Fagge T, Harkness M, Rentoul F, Seymour J, et al. Prospective epidemiologic study of the outcome and cost-effectiveness of antenatal screening to detect neonatal alloimmune thrombocytopenia due to anti-HPA-1a. Transfusion. 2005;45:1945–56.CrossRefPubMedGoogle Scholar
  3. 3.
    Mueller-Eckhardt C, Kiefel V, Grubert A, Kroll H, Weisheit M, Schmidt S, et al. 348 cases of suspected neonatal alloimmune thrombocytopenia. Lancet. 1989;1:363–6.CrossRefPubMedGoogle Scholar
  4. 4.
    Peterson JA, McFarland JG, Curtis BR, Aster RH. Neonatal alloimmune thrombocytopenia: pathogenesis, diagnosis and management. Br J Haematol. 2013;161:3–14.CrossRefPubMedPubMedCentralGoogle Scholar
  5. 5.
    Ohto H, Miura S, Ariga H, Ishii T, Fujimori K, Morita S, et al. The natural history of maternal immunization against foetal platelet alloantigens. Transfus Med. 2004;14:399–408.CrossRefPubMedGoogle Scholar
  6. 6.
    Xia W, Ye X, Xu X, Ding H, Liu J, Deng J, et al. Two cases of platelet transfusion refractoriness and one case of possible FNAIT caused by antibodies against CD36 in China. Transfus Med. 2014;24:254–6.CrossRefPubMedGoogle Scholar
  7. 7.
    Wu G, Zhou Y, Li L, Zhong Z, Li H, Li H, et al. Platelet immunology in China: research and clinical applications. Transfus Med Rev. 2017;31:118–25.CrossRefPubMedGoogle Scholar
  8. 8.
    Silverstein RL, Febbraio M. CD36, a scavenger receptor involved in immunity, metabolism, angiogenesis, and behavior. Sci Signal. 2009;2:re3.CrossRefPubMedPubMedCentralGoogle Scholar
  9. 9.
    Yamamoto N, Akamatsu N, Sakuraba H, Yamazaki H, Tanoue K. Platelet glycoprotein IV (CD36) deficiency is associated with the absence (type I) or the presence (type II) of glycoprotein IV on monocytes. Blood. 1994;83:392–7.PubMedGoogle Scholar
  10. 10.
    Ikeda H, Mitani T, Ohnuma M, Haga H, Ohtzuka S, Kato T, et al. A new platelet-specific antigen, Naka, involved in the refractoriness of HLA-matched platelet transfusion. Vox Sang. 1989;57:213–7.CrossRefPubMedGoogle Scholar
  11. 11.
    Tomiyama Y, Take H, Ikeda H, Mitani T, Furubayashi T, Mizutani H, et al. Identification of the platelet-specific alloantigen, Naka, on platelet membrane glycoprotein IV. Blood. 1990;75:684–7.PubMedGoogle Scholar
  12. 12.
    Porta C, Caporali R, Montecucco C. Thrombotic thrombocytopenic purpura and autoimmunity: a tale of shadows and suspects. Haematologica. 1999;84:260–9.PubMedGoogle Scholar
  13. 13.
    Morishita K, Wakamoto S, Miyazaki T, Sato S, Fujihara M, Kaneko S, et al. Life-threatening adverse reaction followed by thrombocytopenia after passive transfusion of fresh frozen plasma containing anti-CD36 (Nak) isoantibody. Transfusion. 2005;45:803–6.CrossRefPubMedGoogle Scholar
  14. 14.
    Kankirawatana S, Kupatawintu P, Juji T, Veerakul G, Ngerncham S, Chongkolwatana V, et al. Neonatal alloimmune thrombocytopenia due to anti-Nak(a). Transfusion. 2001;41:375–7.CrossRefPubMedGoogle Scholar
  15. 15.
    Curtis BR, Ali S, Glazier AM, Ebert DD, Aitman TJ, Aster RH. Isoimmunization against CD36 (glycoprotein IV): description of four cases of neonatal isoimmune thrombocytopenia and brief review of the literature. Transfusion. 2002;42:1173–9.CrossRefPubMedGoogle Scholar
  16. 16.
    Taketani T, Ito K, Mishima S, Kanai R, Uchiyama A, Hirata Y. Neonatal Isoimmune thrombocytopenia caused by type I CD36 deficiency having novel splicing isoforms of the CD36 gene. Eur J Haematol. 2008;81:70–4.CrossRefPubMedGoogle Scholar
  17. 17.
    Okajima S, Cho K, Chiba H, Azuma H, Mochizuki T, Yamaguchi M, et al. Two sibling cases of hydrops fetalis due to alloimmune anti-CD36 (Naka) antibody. Thromb Haemost. 2006;95:267–71.PubMedGoogle Scholar
  18. 18.
    Xu X, Ye X, Xia W, Liu J, Ding J, Chen Y, et al. Studies on CD36 deficiency in South China: two cases demonstrating the clinical impact of anti-CD36 antibodies. Thromb Haemost. 2013;110:1199–206.CrossRefPubMedGoogle Scholar
  19. 19.
    Brojer E, Husebekk A, Debska M, Uhrynowska M, Guz K, Orzińska A, et al. Fetal/neonatal alloimmune thrombocytopenia: pathogenesis, diagnostics and prevention. Arch Immunol Ther Exp. 2016;64:279–90.CrossRefGoogle Scholar
  20. 20.
    Segata M, Chaoui R, Khalek N, Bahado-Singh R, Paidas MJ, Mari G. Fetal thrombocytopenia secondary to parvovirus infection. Am J Obstet Gynecol. 2007;196:61.e1–4.CrossRefPubMedGoogle Scholar
  21. 21.
    Burrows RF, Kelton JG. Fetal thrombocytopenia and its relation to maternal thrombocytopenia. N Engl J Med. 1993;329:1463–6.CrossRefPubMedGoogle Scholar
  22. 22.
    Yougbaré I, Lang S, Yang H, Chen P, Zhao X, Tai WS, et al. Maternal anti-platelet β3 integrins impair angiogenesis and cause intracranial hemorrhage. J Clin Investig. 2015;125:1545–56.CrossRefPubMedPubMedCentralGoogle Scholar
  23. 23.
    Santoso S, Wihadmadyatami H, Bakchoul T, Werth S, Al-Fakhri N, Bein G, et al. Antiendothelial αvβ3 antibodies are a major cause of intracranial bleeding in fetal/neonatal alloimmune thrombocytopenia. Arterioscler Thromb Vasc Biol. 2016;36:1517–24.CrossRefPubMedPubMedCentralGoogle Scholar
  24. 24.
    Bussel JB, Primiani A. Fetal and neonatal alloimmune thrombocytopenia: progress and ongoing debates. Blood Rev. 2008;22:33–52.CrossRefPubMedGoogle Scholar

Copyright information

© The Japanese Society of Hematology 2017

Authors and Affiliations

  • Xiuzhang Xu
    • 1
    • 2
  • Lin Li
    • 3
  • Wenjie Xia
    • 1
  • Haoqiang Ding
    • 1
  • Dawei Chen
    • 1
  • Jing Liu
    • 1
  • Jing Deng
    • 1
  • Yangkai Chen
    • 1
  • Zhiming He
    • 3
  • Jiali Wang
    • 1
  • Yuan Shao
    • 1
  • Sentot Santoso
    • 1
    • 2
  • Xin Ye
    • 1
  • Qun Fang
    • 3
  1. 1.Institute of Blood TransfusionGuangzhou Blood CentreGuangzhouPeople’s Republic of China
  2. 2.Institute for Clinical Immunology and Transfusion MedicineJustus-Liebig UniversityGiessenGermany
  3. 3.Fetal Medicine Center, Department of Obstetrics and GynecologyThe First Affiliated Hospital of Sun Yat-sen UniversityGuangzhouPeople’s Republic of China

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