11/83. Sn-Mesoporphyrin interdiction of severe hyperbilirubinemia in Jehovah's Witness newborns as an alternative to exchange transfusion.OBJECTIVE: The religious convictions of parents who are Jehovah's Witness adherents lead them to reject the use of exchange transfusions as therapy for severe hyperbilirubinemia in newborns in whom intensive phototherapy has failed to control this problem. Consequently, physicians caring for such infants may be obliged to initiate legal action to compel use of the procedure when severe hyperbilirubinemia not sufficiently responsive to phototherapy warrants an exchange transfusion. Our goal was to determine if we could use the potent inhibitor of bilirubin production, Sn-Mesoporphyrin (SnMP), to resolve the troubling medical-legal issues in such situations in 2 infants with hemolytic disease of the newborn who required exchange transfusions for severe hyperbilirubinemia but whose Jehovah's Witness parents rejected the procedure. SnMP was administered in a single dose, as in previous studies, at the time when exchange transfusion would have been initiated and plasma bilirubin levels were monitored at close intervals thereafter. methods: SnMP is a potent inhibitor of heme oxygenase, the rate-limiting enzyme in catabolism of heme to bilirubin. We found in earlier studies that in single doses of 6 micromol/kg birth weight, SnMP is extremely effective in moderating the course of hyperbilirubinemia and in eliminating the need for supplemental phototherapy in jaundiced newborns. In the 2 cases described, a single dose of SnMP (6 micromol/kg birth weight) was administered intramuscularly to severely jaundiced infants with immune hemolysis at a time when clinical circumstances dictated the need for exchange transfusion. CASE 1: This patient was a preterm male infant (gestational age: 35 5/7 weeks; birth weight: 2790 g) whose plasma bilirubin concentration (PBC) at 1 hour after birth was 5.0 mg/dL. Despite intensive phototherapy with 3 banks of lights and 1 biliblanket, the PBC increased steadily with no diminution in the rate of increase for 75 hours. In view of the problems of immune hemolysis, and prematurity, and the inability of phototherapy to stop progression of hyperbilirubinemia, a decision to carry out an exchange transfusion was made; the decision was, however, rejected by the Jehovah's Witness parents. Pending legal action to compel use of the procedure, a request to this (Rockefeller) laboratory for SnMP was made; its use was approved by the food and Drug Administration; and the inhibitor was delivered to the physician-in-charge (D.P.M.) in Sioux Falls, south dakota. The single dose of SnMP was administered to the infant at 75 hours after birth; the course of hyperbilirubinemia before and after the use of the inhibitor is shown in Fig 1. [figure: see text]. CASE 2: This female term infant (gestational age: 38-39 weeks; birth weight: 4140 g) with immune hemolysis was delivered by cesarean section and because of problems related to meconium aspiration required helicopter transfer to the Special Care Nursery in Abilene, texas, where 10 hours after birth the first PBC was determined to be 18.0 mg/dL. Double-bank phototherapy plus a biliblanket was initiated; a third bank of lights was later ordered. The PBC fluctuated in the ensuing 2 days between 13.8 to 25.8 mg/dL during which suggestive clinical signs of possible bilirubin encephalopathy became manifest. In view of the clinical circumstances and the continued severe hyperbilirubinemia, permission for a double-exchange transfusion was requested. The parents, who were Jehovah's Witness adherents, refused the procedure. While preparing legal action to compel use of the exchange, a request was made to this (Rockefeller) laboratory for use of SnMP to attempt control of hyperbilirubinemia. With FDA approval, the SnMP was delivered to the attending neonatologist (J. R. M.) in Abilene and administered in a single dose (6 micromol/kg birth weight) at 56 hours after birth when the PBC was 19.5 mg/dL. The course of bilirubinemia before and after SnMP use is shown in Fig 2. [figure: see text]. RESULTS AND CONCLUSIONS: The use of SnMP to moderate or prevent the development of severe hyperbilirubinemia in newborns (preterm, near-term, term with high PBCs [15-18 mg/dL], ABO-incompatibility; glucose-6-phosphate dehydrogenase deficiency) has been extensively studied in carefully conducted clinical trials the results of which have been reported earlier. This inhibitor of bilirubin production has demonstrated marked efficacy in moderating the course of hyperbilirubinemia in all diagnostic groups of unconjugated neonatal jaundice. The 2 cases described in this report confirmed the efficacy of SnMP in terminating progression of hyperbilirubinemia in infants in whom phototherapy had failed to sufficiently control the problem and whose parents, for religious reasons, would not permit exchange transfusions. Interdiction of severe hyperbilirubinemia by inhibiting the production of bilirubin with SnMP can be an effective alternative to the use of exchange transfusion in the management of severe newborn jaundice that has not responded sufficiently to light treatment to ease concern about the development of bilirubin encephalopathy.- - - - - - - - - - ranking = 1keywords = gestation (Clic here for more details about this article) |
12/83. rupture of the spleen in erythroblastosis fetalis.A male infant of 36 weeks' gestation, weighing 3080 g, with erythroblastosis, ruptured spleen, and bilateral suprarenal haemorrhages is described. The infant survived after exchange transfusions and splenectomy.- - - - - - - - - - ranking = 0.5keywords = gestation (Clic here for more details about this article) |
13/83. Successful treatment of extremely severe fetal anemia due to Kell alloimmunization.BACKGROUND: Repeated plasmapheresis was used to prevent fetal death from severe anti-Kell alloimmunization until intrauterine transfusions were feasible. CASE: Repeated maternal plasma exchanges (N = 40) beginning at 7 weeks' gestation were used to treat severe anti-Kell alloimmunization. Ultrasound examination at 19 weeks' gestation revealed diffuse hydrops in this fetus (umbilical venous hemoglobin, 1.2 g/dL), which then required nine intrauterine transfusions through 34 weeks. A healthy 3840-g girl was delivered by cesarean delivery at 36 weeks. Despite aplastic anemia during the first 3 months of life, she is healthy and has no observable abnormalities at age 8. CONCLUSION: A highly aggressive course of plasmapheresis and intrauterine transfusions can successfully treat fetal anemia caused by anti-Kell alloimmunization even when fetal hemoglobin is extremely low.- - - - - - - - - - ranking = 1keywords = gestation (Clic here for more details about this article) |
14/83. Anti-Ce complicating two consecutive pregnancies with increasing severity of haemolytic disease of the newborn.The Ce antigen is expressed on red cells of individuals with Rh haplotypes, R1 or CDe and r' or Cde. However, anti-Ce is rare, and only two cases of severe haemolytic disease of the newborn (HDN) caused by this antibody have been described (Malde et al., 2000; Wagner et al., 2000). We describe a woman who was found to have anti-Ce in her second pregnancy, resulting in a neonate with HDN that required exchange transfusion. She subsequently had a twin pregnancy, where both the twins were affected by severe haemolytic disease (HD) of the fetus because of anti-Ce and required repeated fetal transfusions, followed by exchange transfusions after birth. This is the first reported case of HD caused by anti-Ce requiring fetal transfusions.- - - - - - - - - - ranking = 0.85163237975047keywords = pregnancy (Clic here for more details about this article) |
15/83. Complementary therapy for severe Rh-alloimmunization.PURPOSE OF INVESTIGATION: This report describes successful treatment, using invasive and noninvasive techniques, of a 36-year-old woman (gravida 10, para 0) referred to our center at 13 weeks' gestation for severe Rh alloimmunization. Pre-pregnancy indirect Coombs titers ranged from 1:1024-2048. All nine past pregnancies (conceived with three different partners) had ended in abortion, intrauterine death or neonatal death methods: The patient was treated with a single session of plasmapheresis (week 14) immediately followed by five days of immunoglobulin therapy and immunosuppressive therapy based on azathioprine and prednisone (weeks 15-22). Seven fetal transfusions (one intraperitoneal, six intravascular) were performed beginning at 16 weeks. RESULTS: The pregnancy, which was characterized by insulin-dependent gestational diabetes, spontaneously resolving polyhydramnios and peak indirect Coombs titers of 1:65,536, ended at 27 weeks with cesarean section delivery of a viable female weighing 1,000 g. In spite of numerous neonatal complications, the child is physically well at age 3, with normal intellectual and psychomotor development. CONCLUSION: In light of the negative outcomes of the patient's nine past pregnancies, our experience suggests that the early initiation of an integrated approach based on noninvasive and invasive techniques can play a potentially decisive role in the management of severe Rh-alloimmunization.- - - - - - - - - - ranking = 1.8516323797505keywords = gestation, pregnancy (Clic here for more details about this article) |
16/83. Anti-E alloimmunization in pregnancy: management dilemmas.Compared to anti-D alloimmunization, anti-E alloimmunization is a less common cause of hemolytic diseases of the newborn. Being a less potent immunogen, clinical manifestations of anti-E alloimmunization are more variable and usually of less severity. However, the clinical obstetric management of these cases of anti-E alloimmunization is just as challenging. We report here the management of a patient with anti-E alloimmunization to illustrate the controversies of invasive and non-invasive monitoring in the management of such cases.- - - - - - - - - - ranking = 1.7032647595009keywords = pregnancy (Clic here for more details about this article) |
17/83. Anti-Diego a red blood cell alloantibody as a possible cause of anemia in a 3-week-old infant.Our aim in this case report was to describe anemia caused by anti-Diego(a) red blood cell (RBC) antibody in a 3-week-old infant derived during pregnancy to low-frequency Diego(a) RBC antigen. Pre- and postnatal maternal serum screening for unexpected RBC antibodies and determination of RBC antibody specificity in the sera of the mother and child and in the elute of the child were performed by use of microcards (Diamed, Basel, switzerland; BioVue, Ortho Clinical diagnosis, Raritan, NJ, USA) with commercially prepared test RBCs (Diamed, Ortho Clinical diagnosis, and Gamma Biologicals, Houston, TX, USA) at 37 degrees C and indirect antiglobulin test (IAT) according to manufacturer instructions. Direct antiglobulin test (DAT) was performed by use of microcards (Diamed, Ortho Clinical diagnosis) with both polyspecific and monospecific IgG anti-human globulin. Di(a) antigen was determined on maternal, paternal, and infant's red cells by commercial reagent (Gamma Biologicals). Determination of RBC antibody specificities in maternal and child sera and in the child's RBC eluate showed 2 positive reactions only with two Di(a ) test RBCs. Father and baby were positive and mother was negative for Di(a) antigen. When a newborn has positive DAT and there are no clinical reasons for this, the possibility of positive DAT resulting from alloimmunization to low-frequency RBC antigens should be considered.- - - - - - - - - - ranking = 0.42581618987523keywords = pregnancy (Clic here for more details about this article) |
18/83. Diagnosing hemolytic disease of the newborn.A Rh negative, pregnant female presented to a major medical center for possible Rh alloimmunization. This female had nine previous pregnancies, including three spontaneous abortions, four live births, and two fetal demises. Because of poor prenatal care, the immunization Rh immune globulin was administered to only the first two pregnancies. After much laboratory testing and treatment, this tenth pregnancy also ended in fetal demise.- - - - - - - - - - ranking = 0.42581618987523keywords = pregnancy (Clic here for more details about this article) |
19/83. pancytopenia due to suppressed hematopoiesis in a case of fatal hemolytic disease of the newborn associated with anti-K supported by molecular K1 typing.The authors report on a fatal case of hemolytic disease of the newborn (HDN) due to anti-K antibodies with subsequent trilineage pancytopenia in a preterm infant of 28 weeks gestational age, with pronounced leukopenia and neutropenia. In addition, molecular typing of the Kk polymorphism was necessary to confirm HDN. This case of HDN associated with anti-K provides additional evidence that trilineage pancytopenia due to suppressed hematopoiesis is part of the disease. Therefore, antibodies against antigens of the Kell blood group system should be considered as a potential cause of unexplained inhibition of myelopoiesis.- - - - - - - - - - ranking = 0.5keywords = gestation (Clic here for more details about this article) |
20/83. Fetal hemolytic disease due to anti-Rh17 alloimmunization.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.- - - - - - - - - - ranking = 2.1290809493762keywords = pregnancy (Clic here for more details about this article) |
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