Two cases of platelet transfusion refractoriness and one case of possible <scp>FNAIT</scp> caused by antibodies against <scp>CD36</scp> in China

Xia, Wenjie; Ye, X.; Xu, Xiuzhang; Ding, Haoqiang; Liu, J.; Deng, Jing; Chen, Y.; Shao, Yuan; Wang, J.; Li, H.; Fu, Yongshui

doi:10.1111/tme.12137

Cited by 16 publications

(13 citation statements)

References 5 publications

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“…18 Based on this fact, routine diagnostics as well as treatment strategy for anti-CD36-mediated bleeding disorders with use of CD36-compatible platelets should be seriously taken into consideration. 19,20 The most frequent mutation associated with CD36 deficiency found in our cohort was the single c.1163A > T mutation located in Exon 12 and leading to the amino acid exchange p.Gln388Leu. This mutation, in the homozygous state, was reported in a case of PTR in a Chinese individual with detectable anti-CD36 antibodies.…”

Section: Discussionmentioning

confidence: 79%

“…Indeed, a case of FNAIT has been reported in Thailand . Based on this fact, routine diagnostics as well as treatment strategy for anti‐CD36–mediated bleeding disorders with use of CD36‐compatible platelets should be seriously taken into consideration …”

Section: Discussionmentioning

confidence: 99%

“…The most frequent mutation associated with CD36 deficiency found in our cohort was the single c.1163A > T mutation located in Exon 12 and leading to the amino acid exchange p.Gln388Leu. This mutation, in the homozygous state, was reported in a case of PTR in a Chinese individual with detectable anti‐CD36 antibodies . To date, the reason for the complete lack of CD36 expression caused by p.Gln388Leu mutation is not clear.…”

Section: Discussionmentioning

confidence: 99%

“…This mutation, in the homozygous state, was reported in a case of PTR in a Chinese individual with detectable anti-CD36 antibodies. 19 To date, the reason for the complete lack of CD36 expression caused by p. Gln388Leu mutation is not clear. Interestingly, the c.1163A > T mutation is not the most common mutation in other Asian populations.…”

Section: Discussionmentioning

confidence: 99%

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Frequency of CD36 deficiency in Thais analyzed by quantification of CD36 on cell surfaces and in plasma

et al. 2020

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BACKGROUND Anti‐CD36s, developing after transfusion or during pregnancy, play an important role in immune‐mediated bleeding disorders among Asian populations. Currently, little is known about the clinical relevance of anti‐CD36. Here, we aimed to determine the frequency of CD36 deficiency in Thais by analyzing CD36 expression on cell surfaces and in plasma. STUDY DESIGN AND METHODS The expression and deficiency of CD36 on platelets and monocytes were determined by flow cytometry. Mutations in the CD36 gene were analyzed by nucleotide sequencing. Soluble CD36 (sCD36) in plasma was quantified with enzyme‐linked immunosorbent assay. RESULTS Fifteen of 700 blood donors (2.14%) were identified as CD36 deficient. The frequencies of Type I and II CD36 deficiency were 0.43% and 1.71%, respectively. Type I individuals exhibited c.1163A > T, c.429 + 4insG, and c.1156C > T. Type II individuals exhibited c.879 T > C, c.329‐330delAC, c.818 + 108delAACT, c.1125 + 13C > A, and c.1163A > T. CD36 on donor platelets (n = 685) showed a wide distribution of expression levels (mean fluorescence intensity, 16.71 ± 8.68). In the normal phenotype (n = 14), sCD36 concentration was 58.84 ± 11.68 ng/mL, which was significantly correlated with platelet CD36 expression (r2 = 0.8551). In Type II–deficient individuals (n = 6), a similar sCD36 concentration was detected (53.67 ± 8.17 ng/mL). However, sCD36 could not be detected in Type I individuals (n = 3). CONCLUSION CD36 Type I deficiency was found, indicating the potential for immune‐mediated platelet disorders in Thais. However, the underlying mutations differed from those reported in Japan and China. Interestingly, sCD36 could not be detected in plasma of Type I–deficient individuals. This finding may lead to the use of plasma to identify individuals at risk and to allow screening of large cohorts.

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Section: Discussionmentioning

confidence: 79%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Frequency of CD36 deficiency in Thais analyzed by quantification of CD36 on cell surfaces and in plasma

et al. 2020

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“…28 Interestingly, variable clinical pictures of FNAIT mediated by anti-CD36 isoantibodies, including mild and severe thrombocytopenia, hydrops fetalis, and recurrent abortions, have been observed. [9][10][11][12][13]29 However, the precise mechanism of this phenomenon is not known.…”

Section: Discussionmentioning

confidence: 99%

Fetal/neonatal alloimmune thrombocytopenia due to anti‐CD36 antibodies: antibody evaluations by CD36‐transfected cell lines

Lin

Lee

et al. 2017

Transfusion

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BACKGROUND: Isoantibodies against CD36 (platelet glycoprotein 4), developed in Type I CD36-deficient mothers are frequently reported as the cause of fetal/ neonatal alloimmune thrombocytopenia in the Asian population. Therefore, further detailed characterization of anti-CD36-mediated fetal/neonatal alloimmune thrombocytopenia is warranted. Here, we report the characterization of a patient with fetal/neonatal alloimmune thrombocytopenia in a Taiwanese family caused by anti-CD36 isoantibodies using a novel antigen-capture method. STUDY DESIGN AND METHODS:Platelets and monocytes were analyzed for CD36 expression by flow cytometry. Sequencing analysis of the CD36 gene was performed to identify the mutation underlying the CD36 deficiency. Stable transfected human embryonic kidney HEK293 cells expressing recombinant CD36 were established. These cells were used for the characterization of anti-CD36 isoantibodies by flow cytometry, immunoprecipitation, and antigen-capture assay. RESULTS:Flow cytometry analysis revealed a total absence of CD36 on both platelets and monocytes of the mother (Type I CD36-deficient) caused by heterozygous deletions of the CD36 gene (332_333delCA and c.1254 1 6_1254 1 11delTATTTG). Analysis of maternal serum with CD36-transfected HEK293 cells by flow cytometry, immunoprecipitation, and antigen-capture assay demonstrated the presence of anti-CD36 isoantibodies in maternal serum. Interestingly, this antibody could not be detected by the monoclonal antibody immobilization of platelet antigens assay when anti-CD36 monoclonal antibody (clone FA6-152) was used as the capture antibody.CONCLUSION: This case reemphasizes the role of anti-CD36 isoantibodies on the pathomechanism of fetal/ neonatal alloimmune thrombocytopenia. The fact that the monoclonal antibody immobilization of platelet antigens assay does not seem to be reliable for the identification of all anti-CD36 antibodies indicates that screening of anti-CD36 isoantibodies by a monoclonal antibody-independent method, as presented here, should be considered. Fetal/neonatal alloimmune thrombocytopenia (FNAIT), which occurs in from 1 of 800 to 1 of 1000 live births, is the most common cause of severe thrombocytopenia and intracranial hemorrhage in the fetus and in term newborns.1,2 FNAIT is caused by maternal antibodies, which recognize paternalderived alloantigens on fetal platelets, leading to platelet destruction. In Caucasians, more than 75% of FNAIT cases are induced by alloantibodies against human platelet antigen-1a (HPA-1a), 3,4 whereas the most common antibodies causing FNAIT in Japanese are anti-HPA-4b alloantibodies. The clinical importance of anti-CD36 isoantibodies in the development of FNAIT has been reported in different populations.9-13 Currently, the antigen-capture assay represents the gold standard for detecting platelet antibodies. 14 With this assay, platelet antigens captured by mouse monoclonal antibody are used as the target for platelet antibodies. Unfortunately, this approach does not seem to be reliable for the identi...

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

Xia

et al. 2017

Int J Hematol

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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 × 10/L). After 2 days the platelet count rose to 121 × 10/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.

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Two cases of platelet transfusion refractoriness and one case of possible FNAIT caused by antibodies against CD36 in China

Cited by 16 publications

References 5 publications

Frequency of CD36 deficiency in Thais analyzed by quantification of CD36 on cell surfaces and in plasma

Frequency of CD36 deficiency in Thais analyzed by quantification of CD36 on cell surfaces and in plasma

Fetal/neonatal alloimmune thrombocytopenia due to anti‐CD36 antibodies: antibody evaluations by CD36‐transfected cell lines

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

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