Cases reported "Erythroblastosis, Fetal"

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1/103. The association of Hb Khartoum [beta124(H2)Pro-->Arg] with gamma -thalassemia is responsible for hemolytic disease in the newborn of a Sudanese family.

    The unstable Hb Khartoum with a Pro-->Arg replacement at position beta124 was identified by isoelectrofocusing, high performance liquid chromatography, and peptide mapping in a mother and two male children of a Sudanese family. All three were heterozygous for the abnormal hemoglobin; the father and a third male child did not carry the mutation. The mother was also homozygous for two putative gamma -thalassemia point mutations, one affecting both Agamma and Ggamma genes at IVS-II-115 (A-->G), and one affecting the Ggamma gene at the 3' untranslated region (-A) at position -6 from the polyadenylation site. The father had normal gamma genes. All three children were heterozygous for both the gamma -thalassemia mutations. The two older children, who were compound heterozygotes for Hb Khartoum/gamma -thalassemia, presented at birth with severe neonatal jaundice which necessitated exchange blood transfusions. Other causes of neonatal jaundice were excluded. The third male child, who did not carry the Hb Khartoum anomaly but was heterozygous for gamma -thalassemia, did not develop neonatal jaundice. It is concluded that the instability of Hb Khartoum in combination with gamma -thalassemia is responsible for neonatal hemolytic anemia in this family.
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2/103. Severe hemolytic disease from rhesus anti-C antibodies in a surrogate pregnancy after oocyte donation. A case report.

    BACKGROUND: Maternal sensitization with rhesus anti-C antibodies is comparatively rare and usually benign. In pregnancies conceived using donor oocytes, the mother's blood group may differ from that of both the father and the oocyte donor, making blood group incompatibility more likely. CASE: twins, the result of a surrogate pregnancy using donor oocytes, were born with severe hemolytic disease due to rhesus anti-C antibodies. Both infants required exchange transfusion for profound anemia at birth. Isoimmunization in the surrogate mother was not detected antenatally. The twins were delivered by emergency cesarean section due to fetal compromise, detected fortuitously when the mother attended for routine fetal assessment at 35 weeks' gestation. CONCLUSION: Isoimmunization with anti-C antibodies is not always benign and may cause significant hemolytic disease. With the success of in vitro fertilization and oocyte donation, more infertile couples may use these methods to conceive, with or without surrogacy arrangements. In such cases, the provision of antenatal care may become a complex matter, involving several parties, and good communication between everyone involved is vital. In pregnancies conceived with donor oocytes, there may be a higher risk of blood group incompatibility, and special vigilance is warranted.
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3/103. Anti-G in a pregnant patient.

    BACKGROUND: Anti-G is a red cell (RBC) antibody of the Rh system. It has been described in pregnant women only in association with anti-D or anti-C; therefore, the ability of this antibody alone to cause hemolytic disease of the newborn is uncertain. One case in which this antibody caused no clinical sequelae is reported. CASE REPORT: The patient was a 35-year-old primigravida with type O, D-, C-, E-, c RBCs who was given 4 units of type O, D- allogeneic RBCs and 2 units of autologous RBCs 2 years antepartum. She was found to have anti-D and anti-C by an outside laboratory as part of a routine prenatal work-up. Further evaluation by our laboratory revealed the presence of anti-G and possible anti-C without anti-D. Titers at 22 weeks' gestation were 64 against r'r RBCs and 16 against R2R2 RBCs; these remained unchanged throughout the pregnancy. amniocentesis performed at Weeks 28 and 32 showed no evidence of hemolytic disease of the newborn. A healthy 3.3-kg infant was delivered at 36 weeks' gestation. Prophylactic Rh immune globulin was administered antepartum and postpartum. The infant's RBCs were type O, D , c C-, E-, and the direct antiglobulin test was positive. An acid eluate prepared from the baby's RBCs revealed anti-G. The total bilirubin was 5.5 mg per dL at birth, and the hematocrit was 66 percent. Total bilirubin peaked on Day 5 at 11.9 mg per dL, and no therapeutic intervention was required. CONCLUSIONS: Anti-G alone caused little if any fetal or neonatal hemolysis in this case. Although further study is needed, invasive fetal monitoring may be unnecessary if anti-G is the sole cause of fetomaternal RBC incompatibility.
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keywords = fetomaternal
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4/103. Use of recombinant human erythropoietin (EPO-alfa) in a mother alloimmunized to the Js(b) antigen.

    erythropoietin (EPO) is a glycoprotein hormone and the principal regulator of erythropoiesis in the fetus, newborn, and adult. EPO-alfa is erythropoietin manufactured by recombinant human dna technology (rhEPO). After counseling, a pregnant woman with anti-Js(b) in her serum was started on rhEPO (600 U/Kg, biweekly) to prevent anemia secondary to serial donations of her blood for fetal transfusions. After a total of 25 rhEPO infusions and autologous donation of 8 units of whole blood, maternal hemoglobin prior to the elective cesarean section at 37 weeks was 11.3 gm/dL. serum EPO concentration was determined in paired maternal and fetal blood samples, before ultrasound guided intravascular transfusions, in this alloimmunized Js(b)-negative and another Rh(D) alloimmunized pregnancy to determine possible correlations between maternal and fetal serum EPO. rhEPO prevented anemia in a patient who donated 8 units of blood from 18-37 weeks of pregnancy without inducing adverse biological effects such as hypertension or thrombotic complications in the placenta. Data presented in this study suggest that EPO does not cross the human placenta.
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5/103. Haemolytic disease of the newborn caused by anti-c, anti-E and anti-Fya antibodies: report of five cases.

    Fetal haemolytic disease caused by irregular antibodies is discussed on the basis of three cases with maternal anti-c alone, one with anti-E along with anti-c, and one with anti-Fya sensitization. All fetuses suffering from maternal c-allo-immunization alone were treated with intra-uterine transfusions and the newborns received exchange transfusions. These interventions were also required in the case of simultaneous E and c-allo-immunization, and this was the most severe of the five cases. Delta OD450 results were consistent with the severity of the fetal condition in the c and/or E allo-immunization cases. Maternal anti-Fya sensitization caused only mild jaundice of the neonate, but the results of amniotic fluid analysis were quite misleading in that case. Antibody titres did not prove to have good prognostic values (though they were all above the critical level), and the direct antiglobulin test from cord blood was negative in three cases. Regular sonographic evaluations were performed and fetal blood samplings were a cornerstone of management.
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6/103. Perinatal management of fetal hemolytic disease due to Rh incompatibility combined with fetal alloimmune thrombocytopenia due to HPA-5b incompatibility.

    We report out experience in the perinatal management of a complex case of fetal hemolytic disease primarily due to Rhesus incompatibility combined with fetal alloimmune thrombocytopenia. The lowest fetal hemoglobin and platelet levels were 2.6 g/dl and 13,000/microliter, respectively. Intrauterine treatment consisted of six transfusions of packed red cells into the umbilical vein and one transfusion of platelets. The neonate required four transfusions of packed red cells to correct her hyporegenerative erythropoiesis. Postnatal management also included one platelet transfusion, intravenous immunoglobulins and erythropoietin. Although some degree of fetal thrombocytopenia may invariably be found in fetal red cell incompatibility, other rare causes need to be excluded.
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ranking = 4
keywords = transfusion
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7/103. Hemolytic transfusion reaction due to anti-Tc(a).

    BACKGROUND: Anti-Tc(a) detects a high-incidence antigen in the Cromer blood group system. Cromer system antibodies have not usually been associated with hemolytic transfusion reactions or hemolytic disease of the newborn. CASE REPORT: Anti-Tc(a) (initially identified in the patient's serum in 1982) was not detected when she was admitted to the hospital with upper gastrointestinal. bleeding. Three units of red cells were administered. The patient was discharged, but was readmitted to the hospital after her hemoglobin fell to 7.1 g per dL. Antibody detection tests remained negative and three additional units were transfused. Over the next 7 days, her hemoglobin steadily fell to 5.5 g per dL. The level of lactate dehydrogenase rose to 1257, the plasma hemoglobin rose to >16 mg per dL, and the haptoglobin decreased to <6 mg per dL. Five days after transfusion, her direct antiglobulin test was weakly reactive with complement-specific antiglobulin reagents. Eluates were nonreactive. Anti-Tc(a) was detected in her serum; no other antibodies were detected. Differential typing failed to detect any circulating Tc(a ) red cells. The antibody was strongly reactive in a monocyte monolayer assay. CONCLUSION: Although Cromer system antibodies have generally not been proven to be clinically significant in transfusion therapy, the destruction of red cells from six units of transfused Tc(a ) red cells in this patient indicates that anti-Tc(a) may have destructive potential in some patients.
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ranking = 7
keywords = transfusion
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8/103. Use of a PCR-based assay for fetal Cw antigen genotyping in a patient with a history of moderately severe hemolytic disease of the newborn due to anti-Cw.

    Anti-Cw is an uncommon cause of clinically significant hemolytic disease of the newborn (HDN). We report an unusually severe case of HDN due to anti-Cw that required phototherapy and exchange transfusion. We also describe a novel PCR-RFLP method for Cw typing of fetal genomic dna that was used for prenatal diagnosis in a subsequent pregnancy. Following PCR amplification of a 163 bp segment of the RHCE gene containing the nucleotide 122 G to A substitution that corresponds to the Cw allele, Cw types were distinguished by TaqI digestion. PCR-RFLP analysis confirmed that the father and previously affected child were Cw-positive. The fetus was Cw-negative, thus excluding HDN in the current pregnancy and obviating the need for further invasive or noninvasive diagnostic procedures for the remainder of the pregnancy. This case illustrates the utility of PCR-based fetal genotype determination in pregnancies at risk of HDN due to uncommon red cell antibodies such as anti-Cw.
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ranking = 1
keywords = transfusion
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9/103. Intra-uterine transfusion in the management of pregnant women with severe rhesus isoimmunization. II. Results and discussion.

    The results of 51 intra-uterine transfusions in 34 pregnant women with severe rhesus isoimmunization are presented. The survival rate is 56%. Factors influencing the fetal and neonatal mortality are considered, e.g. the gestational age at the initial intra-uterine transfusion, the presence of fetal hydrops, and the interval from the initial intra-uterine transfusion to delivery. The difficulties of the selection procedure are emphasized and it is stated that in some cases intra-uterine transfusion should perhaps not be performed because of a predictable poor fetal prognosis. The significance of accumulating experience is discussed.
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10/103. "Bloodless" treatment of a Jehovah's Witness infant with ABO hemolytic disease.

    An ABO-incompatible term infant girl born to parents who are jehovah's witnesses was admitted to our neonatal unit with a high serum bilirubin level necessitating exchange transfusion. The parents signed a request that blood should not be administered under any circumstances. However, they authorized us to apply the possible alternative treatments of orally administered D-penicillamine (300 mg/kg per day divided in three doses for 3 days), phototherapy, intravenous fluids, and recombinant human erythropoietin (200 U/kg subcutaneously on every second day for 2 weeks). Herein, we report the outcome of this baby, who was discharged from the our unit in good condition after treatment. Her physical growth and motor milestones at 14 months of age revealed no red flags for neurodevelopmental maturation. To our knowledge, this is the first case of an infant who received such a combined alternative (and "bloodless") treatment of serious ABO hemolytic disease of the newborn.
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