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Porencephalic cyst: A complication of fetal intravascular transfusion
Porencephalic cyst: A complication of fetal intravascular transfusion Gary A. Dildy III, MD, Leon G. Smith, Jr., MD, Kenneth J. Moise, Jr., MD, Lorraine E. Cano, RN, and Diane E. Hesketh, RN Houston, Texas A case of unilateral porencephalic cyst as a result of intravascular intrauterine transfusion is reported. Although other factors may have contributed, fetal bradycardia and relative hyperviscosity as a result of increased fetal hematocrit value were considered to be related to this event. Because some authors recommend direct intravascular transfusion to supraphysiologic hematocrits to prolong the interval between procedures, we would caution the use of this approach on the basis of theoretic considerations and the occurrence of this complication. (AM J OBSTET GVNECOL 1991 ;165:76-8.)
Key words: Porencephalic cyst, intravascular transfusion, intrauterine transfusion, Rh alloimmunization Direct fetal intravascular transfusion has become an important method of treating the anemic fetus. Reported complications of intravascular transfusion include umbilical cord hematoma and fetal bradycardia. We report a patient treated for Rh alloimmunization whose fetus developed a left cerebral porencephalic cyst after intravascular transfusion complicated by fetal bradycardia and an elevated fetal hematocrit value.
Case report A 28-year-old gravida 2, para 1 woman presented at 12 weeks' gestation for prenatal care with an anti-D titer of 64. In her only previous pregnancy she was delivered by cesarean section at term because of fetal distress of a 3992 gm male infant whose blood type was reportedly Rh negative. The patient gave an otherwise negative medical history with the exception of having received a blood transfusion at age 16 years for undetermined reasons. Her husband's blood type was A positive, heterozygous for the D antigen. At 21.4 weeks' gestation, the indirect Coombs titer rose to 512 and fetal blood sampling was performed i:hrough an anterior fundal placenta after documentation of normal results of an ultra so no graphic examination. The fetal hematocrit was 34.9% and the fetal blood type was 0 positive. An indirect Coombs titer of 8192 obtained at 24.1 weeks' gestation to exFrom the Division of Maternal-Fetal Medicine, Department of Obstetrics and Gynecology, Baylor College of Medicine. Received for publication October 5, 1990; revised December 12, 1990; accepted January 18,1991. Reprint requests: Kenneth J. M oise, Jr., MD, Division of M aternalFetal Medicine, Department of Obstetrics and Gynecology, Baylor College of Medicine, Texas Medical Center, Houston, TX 77030. 611 /28059
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clude an amnestic maternal antibody response necessitated repeat fetal blood sampling. The hematocrit value was 28.8%. Five days later, fetal ascites and a small pericardial effusion were noted on ultrasonographic examination at the time of preparation for intrauterine transfusion. Pretransfusion fetal hematocrit value was 17%. Forty milliliters of filtered maternal packed red blood cells (hematocrit, 84%) were transfused intravascularly into the fetus. Blood was filtered twice, once through a leukocyte-poor filter during blood bank processing and again with a standard blood transfusion filter at the time of transfusion. The interval fetal hematocrit value was 55%. Umbilical venous pressure was noted to rise from 10 mm Hg at the onset of the procedure to 23 mm Hg after completion of the transfusion. Nine milliliters of fetal blood was slowly removed to reduce umbilical venous pressure and avoid hyperviscosity. At the completion of the procedure, a fetal bradycardia to 60 beats/min was noted, resolving spontaneously after 7 to 10 minutes. Ultrasonographic examination at 26.1 weeks' gestation, before the second intrauterine transfusion, demonstrated a new finding of left cerebral ventricular enlargement (Fig. 1). Transfusions were repeated at 26.1, 28.1, 31.1, and 33.9 weeks' gestation without difficulty. There was no progression of the cystic cerebral lesion on serial ultrasonography. A 2200 gm female infant with Apgar scores of 7 at 1 minute and 9 at 5 minutes was delivered by repeat cesarean section at 35.9 weeks' gestation. Phototherapy was required for 3 days. Computed tomography scan without contrast on the day 9 after birth demonstrated atrophic changes of the left cerebral hemisphere with atrophic enlargement of the left lateral ventricle (Fig. 2). The infant was discharged home on the day 10 after birth; neurologic status was noted to be normal.
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Fig. 1. Ultrasonographic examination of tetal head at 26.1 weeks' gestation, 9 days after the initial intrauterine transfusion. A cystic structure was noted in the left cerebral hemisphere.
Fig. 2. Computed tomography of the head without contrast on day 9 after birth showing a leftsided porencephalic cyst with cerebral atrophy.
Comment To our knowledge, congenital malformations as a result of intrauterine transfusion have not been previously described. The potential exists for vascular accidents to occur as a result of several mechanisms. Air
embolism may potentially occur during intravascular transfusion; however, at our institution a closed circuit transfusion system with a three-way stopcock is used, and the apparatus is flushed with blood at each step of the procedure to avoid air leakage. Another potential
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explanation of vascular accidents might be that of microemboli from transfused blood; however, we minimize this risk by filtering the packed red blood cells twice before transfusion. Alterations in regional blood flow during these procedures may theoretically occur. Two mechanisms that are potentially contributory to the development of the porencephalic cyst in our case include the dramatic increase in blood viscosity and concommitant fetal bradycardia. In the adult, blood hyperviscosity is a recognized cause of cerebral ischemia and infarction. I In addition, fetal bradycardia encountered during the intravascular transfusion in our case may have contributed to the cerebrovascular event by producing hypotension, sluggish blood flow, and markedly reduced cerebral perfusion. Controversy exists with regard to the optimal method of transfusion for hemolytic disease of the fetus. Some centers use only the intravascular method, routinely transfusing packed red blood cells to target hematocrit values of at least 55%.2 We routinely use the intravas-
July 1991 Am J Obstet Gynecol
cular technique, achieving hematocrit values in the range of 35% to 40%, followed by a standard intraperitoneal transfusion to lengthen the interval between procedures. Until more data concerning the effects of relative hyperviscosity on fetal organ perfusion become available, we recommend intravascular transfusion of blood to physiologic hematocrits. A combined technique of intrauterine transfusion that includes the intraperitoneal transfusion may be used effectively to allow prolonged intervals between successive transfusions. REFERENCES 1. Barnett HJM. Cerebral ischemia and infarction. In: Wyngaarden JB, Smith LH, eds. Cecil textbook of medicine. Philadelphia: WB Saunders, 1985:2090. 2. Harman CR, Bowman JM, Manning FA, et al. Intrauterine transfusion-intraperitoneal versus intravascular approach: a case-control comparison. AM J OBSTET GYNECOL 1990; 162: 1053-9.
Key words: Porencephalic cyst, intravascular transfusion, intrauterine transfusion, Rh alloimmunization Direct fetal intravascular transfusion has become an important method of treating the anemic fetus. Reported complications of intravascular transfusion include umbilical cord hematoma and fetal bradycardia. We report a patient treated for Rh alloimmunization whose fetus developed a left cerebral porencephalic cyst after intravascular transfusion complicated by fetal bradycardia and an elevated fetal hematocrit value.
Case report A 28-year-old gravida 2, para 1 woman presented at 12 weeks' gestation for prenatal care with an anti-D titer of 64. In her only previous pregnancy she was delivered by cesarean section at term because of fetal distress of a 3992 gm male infant whose blood type was reportedly Rh negative. The patient gave an otherwise negative medical history with the exception of having received a blood transfusion at age 16 years for undetermined reasons. Her husband's blood type was A positive, heterozygous for the D antigen. At 21.4 weeks' gestation, the indirect Coombs titer rose to 512 and fetal blood sampling was performed i:hrough an anterior fundal placenta after documentation of normal results of an ultra so no graphic examination. The fetal hematocrit was 34.9% and the fetal blood type was 0 positive. An indirect Coombs titer of 8192 obtained at 24.1 weeks' gestation to exFrom the Division of Maternal-Fetal Medicine, Department of Obstetrics and Gynecology, Baylor College of Medicine. Received for publication October 5, 1990; revised December 12, 1990; accepted January 18,1991. Reprint requests: Kenneth J. M oise, Jr., MD, Division of M aternalFetal Medicine, Department of Obstetrics and Gynecology, Baylor College of Medicine, Texas Medical Center, Houston, TX 77030. 611 /28059
76
clude an amnestic maternal antibody response necessitated repeat fetal blood sampling. The hematocrit value was 28.8%. Five days later, fetal ascites and a small pericardial effusion were noted on ultrasonographic examination at the time of preparation for intrauterine transfusion. Pretransfusion fetal hematocrit value was 17%. Forty milliliters of filtered maternal packed red blood cells (hematocrit, 84%) were transfused intravascularly into the fetus. Blood was filtered twice, once through a leukocyte-poor filter during blood bank processing and again with a standard blood transfusion filter at the time of transfusion. The interval fetal hematocrit value was 55%. Umbilical venous pressure was noted to rise from 10 mm Hg at the onset of the procedure to 23 mm Hg after completion of the transfusion. Nine milliliters of fetal blood was slowly removed to reduce umbilical venous pressure and avoid hyperviscosity. At the completion of the procedure, a fetal bradycardia to 60 beats/min was noted, resolving spontaneously after 7 to 10 minutes. Ultrasonographic examination at 26.1 weeks' gestation, before the second intrauterine transfusion, demonstrated a new finding of left cerebral ventricular enlargement (Fig. 1). Transfusions were repeated at 26.1, 28.1, 31.1, and 33.9 weeks' gestation without difficulty. There was no progression of the cystic cerebral lesion on serial ultrasonography. A 2200 gm female infant with Apgar scores of 7 at 1 minute and 9 at 5 minutes was delivered by repeat cesarean section at 35.9 weeks' gestation. Phototherapy was required for 3 days. Computed tomography scan without contrast on the day 9 after birth demonstrated atrophic changes of the left cerebral hemisphere with atrophic enlargement of the left lateral ventricle (Fig. 2). The infant was discharged home on the day 10 after birth; neurologic status was noted to be normal.
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Fig. 1. Ultrasonographic examination of tetal head at 26.1 weeks' gestation, 9 days after the initial intrauterine transfusion. A cystic structure was noted in the left cerebral hemisphere.
Fig. 2. Computed tomography of the head without contrast on day 9 after birth showing a leftsided porencephalic cyst with cerebral atrophy.
Comment To our knowledge, congenital malformations as a result of intrauterine transfusion have not been previously described. The potential exists for vascular accidents to occur as a result of several mechanisms. Air
embolism may potentially occur during intravascular transfusion; however, at our institution a closed circuit transfusion system with a three-way stopcock is used, and the apparatus is flushed with blood at each step of the procedure to avoid air leakage. Another potential
78
Dildy et al.
explanation of vascular accidents might be that of microemboli from transfused blood; however, we minimize this risk by filtering the packed red blood cells twice before transfusion. Alterations in regional blood flow during these procedures may theoretically occur. Two mechanisms that are potentially contributory to the development of the porencephalic cyst in our case include the dramatic increase in blood viscosity and concommitant fetal bradycardia. In the adult, blood hyperviscosity is a recognized cause of cerebral ischemia and infarction. I In addition, fetal bradycardia encountered during the intravascular transfusion in our case may have contributed to the cerebrovascular event by producing hypotension, sluggish blood flow, and markedly reduced cerebral perfusion. Controversy exists with regard to the optimal method of transfusion for hemolytic disease of the fetus. Some centers use only the intravascular method, routinely transfusing packed red blood cells to target hematocrit values of at least 55%.2 We routinely use the intravas-
July 1991 Am J Obstet Gynecol
cular technique, achieving hematocrit values in the range of 35% to 40%, followed by a standard intraperitoneal transfusion to lengthen the interval between procedures. Until more data concerning the effects of relative hyperviscosity on fetal organ perfusion become available, we recommend intravascular transfusion of blood to physiologic hematocrits. A combined technique of intrauterine transfusion that includes the intraperitoneal transfusion may be used effectively to allow prolonged intervals between successive transfusions. REFERENCES 1. Barnett HJM. Cerebral ischemia and infarction. In: Wyngaarden JB, Smith LH, eds. Cecil textbook of medicine. Philadelphia: WB Saunders, 1985:2090. 2. Harman CR, Bowman JM, Manning FA, et al. Intrauterine transfusion-intraperitoneal versus intravascular approach: a case-control comparison. AM J OBSTET GYNECOL 1990; 162: 1053-9.