A Case of Fatal Hemolytic Disease of the Newborn Associated With-D-/-D-Phenotype

Han, Kyou Sup; Kim, H C; Shim, Woo Sup

doi:10.1055/s-2007-994188

Cited by 14 publications

(22 citation statements)

References 5 publications

(6 reference statements)

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“…In Western populations, -D-/-D-is an extremely rare Rh phenotype, with only 3 cases of hemolytic disease of the fetus and newborn in sensitized -D-/-D-women reported in the English language literature [2][3][4]. On the other hand, the frequency of the -D-/-D-phenotype among Japanese is much higher (approximately 1 in 100,000) and 50 cases of hemolytic disease of the fetus and newborn in -D-/-D-women have been reported in Japan.…”

Section: Discussionmentioning

confidence: 99%

Fetal Hemolytic Disease due to Anti-Rh17 Alloimmunization

Hirose¹,

Nakanishi²,

Kaku³

et al. 2004

Fetal Diagn Ther

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Objective: To delineate clinical features of a case of fetal hemolytic disease due to anti-Rh17, along with a review of relevant studies published in English and Japanese. Methods: We present clinical features of a –D-/-D- phenotype woman with anti-Rh17 alloimmunization during pregnancy. Relevant English literature in the MEDLINE database was reviewed, while Japanese studies were searched in the Japana Centra Revuo Medicina database. Results: A Japanese –D-/-D- woman with anti-Rh17 (Hro) was treated during pregnancy. Serial ultrasonography, antibody titers, amniocenteses, and cordocenteses were conducted for perinatal management. Amniocentesis results demonstrated a high delta optical density level of 450 in the amniotic fluid, while cordocentesis revealed alloimmunization between the mother and the fetus as well as fetal hemolytic anemia. Blood flow velocity in the middle cerebral artery indicated a rapid development of fetal anemia. The newborn demonstrated severe anemia and hyperbilirubinemia, which were successfully treated with exchange transfusions. Two cases of prenatally diagnosed fetal hemolytic disease due to anti-Rh17 were found published in English and 5 in Japanese. Conclusion: A –D-/-D- phenotype patient with anti-Rh17 was successfully managed during pregnancy and a good outcome for the neonate was achieved. Our results and a review of related literature led to the following suggestions. The first pregnancy in a –D-/-D- woman may be affected, an anamnestic immune response can easily occur during pregnancy, the level of anti-Rh17 titer is indicative of the degree of fetal hemolysis, and appropriate intrauterine intervention is warranted for achievement of a good outcome.

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Section: Discussionmentioning

confidence: 99%

Fetal Hemolytic Disease due to Anti-Rh17 Alloimmunization

Hirose¹,

Nakanishi²,

Kaku³

et al. 2004

Fetal Diagn Ther

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“…Okubo et al found 7 -D- individuals (0.001%) among 692,000 Japanese blood donors and estimated the frequency of the -D- haplotype as 0.0032 among Japanese [1]. With the exception of 3 cases reported since the initial case report in 1998, there is no data about the prevalence of -D- phenotype in Korea [2-6]. The significance of the -D- phenotype is that individuals who have it can make multiple Rh antibodies against C, c, E, or e antigens if they are sensitized to Rh antigens, and this puts them at risk of massive hemolytic transfusion reactions.…”

mentioning

confidence: 99%

“…The significance of the -D- phenotype is that individuals who have it can make multiple Rh antibodies against C, c, E, or e antigens if they are sensitized to Rh antigens, and this puts them at risk of massive hemolytic transfusion reactions. Thus far, the clinical relevance of the -D- phenotype has been predominantly reported in pregnant women, causing a mild to fatal hemolytic disease of the fetus and newborn [2-7]. Nevertheless, it is generally not indicated to transfuse C, c, E, or e antigen-positive red blood cells (RBCs) to a -D- phenotype patient.…”

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confidence: 99%

Sensitization to Multiple Rh Antigens by Transfusion of Random Donor Platelet Concentrates in a -D- Phenotype Patient

Yun

Kang

et al. 2012

Ann Lab Med

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The -D- phenotype is a rare Rh phenotype that strongly expresses D antigen without C, c, E, or e antigens. In -D- phenotype individuals, anti-Rh17 (Hro) is commonly found if there is a history of pregnancy or transfusion with red blood cells (RBCs) that express C, c, E, or e antigens. We report the first case of a -D- phenotype patient with multiple Rh antibodies including anti-Rh17 who had a history of two occasions of transfusion with eight random donor platelet concentrates two and six years ago. We found that a trivial amount of RBCs in the platelet components was able to trigger sensitization to RBC antigens, especially the highly immunogenic and clinically significant Rh antigens, including C, c, E, e or CcEe polypeptides. To avoid unnecessary sensitization and to minimize the risk of hemolytic transfusion reactions in patients with this rare Rh phenotype, a modified strategy for pretransfusion screenings needs to be discussed in the field of transfusion medicine.

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“…Anti-Rh17 [4] and anti-Jk a antibodies, however, can lead to massive hemolysis. Therefore, only erythrocytes of donors with the -D- phenotype, negative for Jk a , could be selected for transfusion.…”

mentioning

confidence: 99%

“…To date the clinical relevance of the -D- phenotype has been predominantly described in pregnant women, causing mild to fatal hemolytic disease of the newborn [4, 7–10]. We describe the successful treatment of a patient with recurrent need of transfusions despite the presence of antibodies against the high-prevalence antigens of the RhCcEe polypeptide combined with anti-Jk a .…”

mentioning

confidence: 99%