Skip to main content
Top
Published in: International Journal of Hematology 2/2018

01-02-2018 | Case Report

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

Authors: 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

Published in: International Journal of Hematology | Issue 2/2018

Login to get access

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.
Literature
1.
go back to reference 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.CrossRefPubMed 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.CrossRefPubMed
2.
go back to reference 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.CrossRefPubMed 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.CrossRefPubMed
3.
go back to reference 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.CrossRefPubMed 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.CrossRefPubMed
4.
go back to reference Peterson JA, McFarland JG, Curtis BR, Aster RH. Neonatal alloimmune thrombocytopenia: pathogenesis, diagnosis and management. Br J Haematol. 2013;161:3–14.CrossRefPubMedPubMedCentral Peterson JA, McFarland JG, Curtis BR, Aster RH. Neonatal alloimmune thrombocytopenia: pathogenesis, diagnosis and management. Br J Haematol. 2013;161:3–14.CrossRefPubMedPubMedCentral
5.
go back to reference 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.CrossRefPubMed 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.CrossRefPubMed
6.
go back to reference 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.CrossRefPubMed 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.CrossRefPubMed
7.
go back to reference 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.CrossRefPubMed 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.CrossRefPubMed
9.
go back to reference 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.PubMed 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.PubMed
10.
go back to reference 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.CrossRefPubMed 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.CrossRefPubMed
11.
go back to reference 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.PubMed 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.PubMed
12.
go back to reference Porta C, Caporali R, Montecucco C. Thrombotic thrombocytopenic purpura and autoimmunity: a tale of shadows and suspects. Haematologica. 1999;84:260–9.PubMed Porta C, Caporali R, Montecucco C. Thrombotic thrombocytopenic purpura and autoimmunity: a tale of shadows and suspects. Haematologica. 1999;84:260–9.PubMed
13.
go back to reference 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.CrossRefPubMed 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.CrossRefPubMed
14.
go back to reference 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.CrossRefPubMed 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.CrossRefPubMed
15.
go back to reference 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.CrossRefPubMed 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.CrossRefPubMed
16.
go back to reference 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.CrossRefPubMed 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.CrossRefPubMed
17.
go back to reference 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.PubMed 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.PubMed
18.
go back to reference 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.CrossRefPubMed 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.CrossRefPubMed
19.
go back to reference 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.CrossRef 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.CrossRef
20.
go back to reference 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.CrossRefPubMed 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.CrossRefPubMed
21.
go back to reference Burrows RF, Kelton JG. Fetal thrombocytopenia and its relation to maternal thrombocytopenia. N Engl J Med. 1993;329:1463–6.CrossRefPubMed Burrows RF, Kelton JG. Fetal thrombocytopenia and its relation to maternal thrombocytopenia. N Engl J Med. 1993;329:1463–6.CrossRefPubMed
22.
go back to reference 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.CrossRefPubMedPubMedCentral 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.CrossRefPubMedPubMedCentral
23.
go back to reference 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.CrossRefPubMedPubMedCentral 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.CrossRefPubMedPubMedCentral
24.
go back to reference Bussel JB, Primiani A. Fetal and neonatal alloimmune thrombocytopenia: progress and ongoing debates. Blood Rev. 2008;22:33–52.CrossRefPubMed Bussel JB, Primiani A. Fetal and neonatal alloimmune thrombocytopenia: progress and ongoing debates. Blood Rev. 2008;22:33–52.CrossRefPubMed
Metadata
Title
Successful management of a hydropic fetus with severe anemia and thrombocytopenia caused by anti-CD36 antibody
Authors
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
Publication date
01-02-2018
Publisher
Springer Japan
Published in
International Journal of Hematology / Issue 2/2018
Print ISSN: 0925-5710
Electronic ISSN: 1865-3774
DOI
https://doi.org/10.1007/s12185-017-2310-5

Other articles of this Issue 2/2018

International Journal of Hematology 2/2018 Go to the issue
Webinar | 19-02-2024 | 17:30 (CET)

Keynote webinar | Spotlight on antibody–drug conjugates in cancer

Antibody–drug conjugates (ADCs) are novel agents that have shown promise across multiple tumor types. Explore the current landscape of ADCs in breast and lung cancer with our experts, and gain insights into the mechanism of action, key clinical trials data, existing challenges, and future directions.

Dr. Véronique Diéras
Prof. Fabrice Barlesi
Developed by: Springer Medicine