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Chronic, massive fetomaternal hemorrhage treated with repeated fetal intravascular transfusions
Chronic, massive fetomatemal hemorrhage treated with repeated fetal intravascular transfusions Richard L. Fischer, MD, Kathleen Kuhlman, MD, Joseph Grover, MD, Owen Montgomery, MD, and RonaldJ. Wapner, MD Philadelphia, Pennsylvania We report the first known case of chronic, massive fetomaternal hemorrhage managed by serial fetal intravascular transfusions. Timing of transfusions was guided by fetal heart rate patterns and fetal movement evaluation. Despite severe anemia and a sinusoidal heart rate pattern, the fetus demonstrated normal blood gases and no sign of hydrops. (AM J OBSTET GVNECOL 1990;162:203-4.)
Key words: Fetomaternal hemorrhage, fetal anemia, fetal intravascular transfusion
Antepartum fetomaternal hemorrhage occurs in approximately 45% of pregnancies, usually in quantities <0.1 ml. Rarely, a massive fetomaternal hemorrhage, defined as> 150 ml, can result in significant fetal and neonatal anemia. In utero intravascular transfusion is now used for treatment of fetal anemia that is secondary to isoimmunization. This report describes a case in which a massive, chronic fetomaternal hemorrhage was identified and treated with serial fetal intravascular transfusions.
Case report A 34-year-old, primigravid, diethylstilbesterolexposed woman was first seen at 31 weeks' gestation with decreased fetal movement for 2 weeks and no movement for 3 days. Prenatal history had been remarkable for an episode of heavy first-trimester vaginal bleeding and a genetic amniocentesis at 16 weeks through an anterior placenta. On admission, a nonstress test showed a sinusoidal heart rate pattern. Ultrasonography revealed normal amniotic fluid volume, no hydropic changes except for an enlarged fetal liver, and no evidence of abruptio placenta or placenta previa. Although the biophysical profile was normal, a contraction stress test result was interpreted as suspicious. A review of prenatal laboratory test results showed a blood type of a positive, negative antibody screen, and nonreactive rapid plasma reagin. A Kleihauer-Betke smear on admission suggested a fetomaternal hemorrhage of 180 ml. A maternal serum u-fetoprotein level of 13,473 ng/ml (normal <430 ng/ml) was supportive of a large fetomaternal hemFrom the DIvision of Maternal-Fetal Medlcme, Department of Obstetrics and Gynecology, jefferson Medical College, Thomas jefferson University. ReceIVed for publicatIOn july 13, 1989; accePtedju(~ 19,1989. Reprint requests: RIchard L. Fischer, MD, Department of Obstetncs and Gynecology, Cooper Hospltall Universzty MedIcal Center, 3 Cooper Plaza, SUIte 211, Camden. Nj 08103. 611115404
orrhage. A cordocentesis was performed, which revealed a fetal hemoglobin of 2.2 gm/dl and hematocrit of 7.6%. An intravascular fetal transfusion of 96 ml of packed, washed, irradiated, cytomegalovirus-negative, a-negative red blood cells was performed. The posttransfusion fetal hemoglobin was 12.4 gm/dl and hematocrit was 39.2%. Fetal venous blood gas values were normal (pH 7.40, P0 2 50 mm Hg, Peo 2 36 mm Hg, base excess - 0.9, oxygen saturation 85%). Fetal viral studies, including cytomegalovirus and parvovirus, were negative. Within 4 hours after the procedure, the patient reported a marked increase in fetal activity, and the fetal heart rate tracing showed accelerations. The fetal heart rate tracing remained reassuring until day 5, when fetal movements decreased and a sinusoidal pattern recurred. A second cordocentesis was performed, with fetal hemoglobin of 4.2 gmt dl and hematocrit of 13.5%. An intravascular transfusion of 100 ml was performed, with a final hemoglobin of 12.2 gm/dl and hematocrit of37.9%. Amniotic fluid analysis demonstrated fetal lung maturity. Color-flow Doppler and magnetic resonance imaging of the placenta showed no abnormalities. On day 6, the sinusoidal pattern returned. In view of the chronicity and rapidity of the massive fetomaternal hemorrhage and with documented mature lung indices, a primary cesarean section was performed because of breech presentation. The appropriate-forgestational-age infant weighed 1750 gm, with Apgar scores of 9 at 1 minute and 10 at 5 minutes. Neonatal hemoglobin was 6.6 gm/dl and hematocrit was 20.4%. Blood type was a positive, and total bilirubin was 1.1 mg/dl. The infant received two blood transfusions and phototherapy for a maximum bilirubin level of 13.4 mg/dl. No ventilatory support was needed. and the infant is currently well. The placenta appeared normal except for a subchorionic hematoma, 2 cm from the cord insertion, which probably resulted from needle displacement during a transfusion. Barium i~ection into the umbilical vein showed an unremarkable arborization pattern and no 203
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Fischer et al.
evidence of vascular malformation. Pathologic evaluation revealed intraluminal thrombi of fetal stem vessels, peripheral endovasculitis, one area suggestive of abruptio placentae, and no evidence of a vascular tumor. Comment
This is the first reported case of a chronic, massive fetomaternal hemorrhage treated with serial fetal intravascular transfusions. As described previously, 1 chronic fetomaternal hemorrhage is associated with hemodynamic compensation and normal acid-base status. Cardwel12 reported a case of fetal hydrops from an acute fetomaternal hemorrhage at 21 weeks; it was treated successfully with a single intraperitoneal transfusion and there was resolution of the hydrops. In our case of chronic, massive fetomaternal hemorrhage, there was hepatomegaly but no evidence of hydrops fetalis, despite a hematocrit of 7.6%. In Rh isoimmunization, hydrops has been observed at hematocrits ranging from 10% to 15%, implying that factors other than fetal anemia may contribute to hydrops fetalis.
January 1990 Am J Obstet Gynecol
Interestingly, the nonstress test and maternal perception of diminished fetal movements were our most sensitive noninvasive indicators of severe fetal anemia. In contrast, the biophysical profile was normal despite severe anemia, suggesting that the biophysical profile may not be useful for this condition. This case demonstrates the importance of the Kleihauer-Betke stain and cordocentesis in the diagnosis of suspected massive fetomaternal hemorrhage, as well as the usefulness of intravascular fetal transfusion for treatment of severe anemia in the preterm fetus. Intensive antepartum fetal monitoring is crucial to detect persistent fetomaternal hemorrhage with recurrent anemia, so that intravascular transfusion or delivery can be effected.
REFERENCES 1. Willis C, Foreman CS. Chronic massive fetomaternal hemorrhage: a case report. Obstet Gynecol 1988;71:459-60. 2. Cardwell MS. Successful treatment of hydrops fetalis caused by fetomaternal hemorrhage: a case report. AM J OBSTET GYNECOL 1988;158:131-2.
Bound volumes available to subscribers Bound volumes of the AMERICAN JOURNAL OF OBSTETRICS AND GYNECOLOGY are available to subscribers (only) for the 1990 issues from the Publisher, at a cost of $56.00 ($79.00 international) for Vol. 162 (January-June) and Vol. 163 (July-December). Shipping charges are included. Each bound volume contains a subject and author index and all advertising is removed. Copies are shipped within 60 days after publication of the last issue in the volume. The binding is durable buckram with the JOURNAL name, volume number, and year stamped in gold on the spine. Payment must accompany all orders. Contact The C. V. Mosby Company, Circulation Department, 11830 Westline Industrial Drive, St. Louis, Missouri 63146-3318, USA; phone (800) 325-4177, ext. 351. Subscriptions must be in force to qualify. Bound volumes are not available in place of a regular JOURNAL subscription.
Key words: Fetomaternal hemorrhage, fetal anemia, fetal intravascular transfusion
Antepartum fetomaternal hemorrhage occurs in approximately 45% of pregnancies, usually in quantities <0.1 ml. Rarely, a massive fetomaternal hemorrhage, defined as> 150 ml, can result in significant fetal and neonatal anemia. In utero intravascular transfusion is now used for treatment of fetal anemia that is secondary to isoimmunization. This report describes a case in which a massive, chronic fetomaternal hemorrhage was identified and treated with serial fetal intravascular transfusions.
Case report A 34-year-old, primigravid, diethylstilbesterolexposed woman was first seen at 31 weeks' gestation with decreased fetal movement for 2 weeks and no movement for 3 days. Prenatal history had been remarkable for an episode of heavy first-trimester vaginal bleeding and a genetic amniocentesis at 16 weeks through an anterior placenta. On admission, a nonstress test showed a sinusoidal heart rate pattern. Ultrasonography revealed normal amniotic fluid volume, no hydropic changes except for an enlarged fetal liver, and no evidence of abruptio placenta or placenta previa. Although the biophysical profile was normal, a contraction stress test result was interpreted as suspicious. A review of prenatal laboratory test results showed a blood type of a positive, negative antibody screen, and nonreactive rapid plasma reagin. A Kleihauer-Betke smear on admission suggested a fetomaternal hemorrhage of 180 ml. A maternal serum u-fetoprotein level of 13,473 ng/ml (normal <430 ng/ml) was supportive of a large fetomaternal hemFrom the DIvision of Maternal-Fetal Medlcme, Department of Obstetrics and Gynecology, jefferson Medical College, Thomas jefferson University. ReceIVed for publicatIOn july 13, 1989; accePtedju(~ 19,1989. Reprint requests: RIchard L. Fischer, MD, Department of Obstetncs and Gynecology, Cooper Hospltall Universzty MedIcal Center, 3 Cooper Plaza, SUIte 211, Camden. Nj 08103. 611115404
orrhage. A cordocentesis was performed, which revealed a fetal hemoglobin of 2.2 gm/dl and hematocrit of 7.6%. An intravascular fetal transfusion of 96 ml of packed, washed, irradiated, cytomegalovirus-negative, a-negative red blood cells was performed. The posttransfusion fetal hemoglobin was 12.4 gm/dl and hematocrit was 39.2%. Fetal venous blood gas values were normal (pH 7.40, P0 2 50 mm Hg, Peo 2 36 mm Hg, base excess - 0.9, oxygen saturation 85%). Fetal viral studies, including cytomegalovirus and parvovirus, were negative. Within 4 hours after the procedure, the patient reported a marked increase in fetal activity, and the fetal heart rate tracing showed accelerations. The fetal heart rate tracing remained reassuring until day 5, when fetal movements decreased and a sinusoidal pattern recurred. A second cordocentesis was performed, with fetal hemoglobin of 4.2 gmt dl and hematocrit of 13.5%. An intravascular transfusion of 100 ml was performed, with a final hemoglobin of 12.2 gm/dl and hematocrit of37.9%. Amniotic fluid analysis demonstrated fetal lung maturity. Color-flow Doppler and magnetic resonance imaging of the placenta showed no abnormalities. On day 6, the sinusoidal pattern returned. In view of the chronicity and rapidity of the massive fetomaternal hemorrhage and with documented mature lung indices, a primary cesarean section was performed because of breech presentation. The appropriate-forgestational-age infant weighed 1750 gm, with Apgar scores of 9 at 1 minute and 10 at 5 minutes. Neonatal hemoglobin was 6.6 gm/dl and hematocrit was 20.4%. Blood type was a positive, and total bilirubin was 1.1 mg/dl. The infant received two blood transfusions and phototherapy for a maximum bilirubin level of 13.4 mg/dl. No ventilatory support was needed. and the infant is currently well. The placenta appeared normal except for a subchorionic hematoma, 2 cm from the cord insertion, which probably resulted from needle displacement during a transfusion. Barium i~ection into the umbilical vein showed an unremarkable arborization pattern and no 203
204
Fischer et al.
evidence of vascular malformation. Pathologic evaluation revealed intraluminal thrombi of fetal stem vessels, peripheral endovasculitis, one area suggestive of abruptio placentae, and no evidence of a vascular tumor. Comment
This is the first reported case of a chronic, massive fetomaternal hemorrhage treated with serial fetal intravascular transfusions. As described previously, 1 chronic fetomaternal hemorrhage is associated with hemodynamic compensation and normal acid-base status. Cardwel12 reported a case of fetal hydrops from an acute fetomaternal hemorrhage at 21 weeks; it was treated successfully with a single intraperitoneal transfusion and there was resolution of the hydrops. In our case of chronic, massive fetomaternal hemorrhage, there was hepatomegaly but no evidence of hydrops fetalis, despite a hematocrit of 7.6%. In Rh isoimmunization, hydrops has been observed at hematocrits ranging from 10% to 15%, implying that factors other than fetal anemia may contribute to hydrops fetalis.
January 1990 Am J Obstet Gynecol
Interestingly, the nonstress test and maternal perception of diminished fetal movements were our most sensitive noninvasive indicators of severe fetal anemia. In contrast, the biophysical profile was normal despite severe anemia, suggesting that the biophysical profile may not be useful for this condition. This case demonstrates the importance of the Kleihauer-Betke stain and cordocentesis in the diagnosis of suspected massive fetomaternal hemorrhage, as well as the usefulness of intravascular fetal transfusion for treatment of severe anemia in the preterm fetus. Intensive antepartum fetal monitoring is crucial to detect persistent fetomaternal hemorrhage with recurrent anemia, so that intravascular transfusion or delivery can be effected.
REFERENCES 1. Willis C, Foreman CS. Chronic massive fetomaternal hemorrhage: a case report. Obstet Gynecol 1988;71:459-60. 2. Cardwell MS. Successful treatment of hydrops fetalis caused by fetomaternal hemorrhage: a case report. AM J OBSTET GYNECOL 1988;158:131-2.
Bound volumes available to subscribers Bound volumes of the AMERICAN JOURNAL OF OBSTETRICS AND GYNECOLOGY are available to subscribers (only) for the 1990 issues from the Publisher, at a cost of $56.00 ($79.00 international) for Vol. 162 (January-June) and Vol. 163 (July-December). Shipping charges are included. Each bound volume contains a subject and author index and all advertising is removed. Copies are shipped within 60 days after publication of the last issue in the volume. The binding is durable buckram with the JOURNAL name, volume number, and year stamped in gold on the spine. Payment must accompany all orders. Contact The C. V. Mosby Company, Circulation Department, 11830 Westline Industrial Drive, St. Louis, Missouri 63146-3318, USA; phone (800) 325-4177, ext. 351. Subscriptions must be in force to qualify. Bound volumes are not available in place of a regular JOURNAL subscription.