In utero diagnosis and management of fetal subdural hematoma Siegfried Rotmensch, MD, Peter A. Grannum, MD, Jose A. Nores, MD, Cynthia Hall, MD, Marc S. Keller, MD, Shirley McCarthy, MD, and John C. Hobbins, MD New Haven, Connecticut The in utero ultrasonographic diagnosis of a fetal subdural hematoma is presented. The value of percutaneous umbilical blood sampling, transvaginal ultrasonography, and magnetic resonance imaging for diagnosis and management is discussed. (AM J OaSTET GVNECOL 1991;164:1246-8.)
Key words: Subdural hematoma, percutaneous umbilical blood sampling, transvaginal ultrasonography, magnetic resonance imaging The in utero diagnosis of subdural hematoma has been reported in two cases. I. 2 Percutaneous umbilical blood sampling, transvaginal ultrasonography, and magnetic resonance imaging have added new dimensions to the identification and management of this entity. We report a case and suggest guidelines for management.
Case history A 37-year-old Haitian woman, gravida 4, para 3, was first seen at 32 weeks' gestation. Physical examination revealed a uterine fundal height of 37 em. A transabdominal ultrasonographic scan showed a single live fetus in vertex presentation with moderate polyhydramnios. Biometry of the long bones was consistent with 32 weeks' gestation. Gross fetal anatomy appeared normal except for the intracranium, which could not be well visualized because of engagement of the fetal head in the pelvis. The biparietal diameter was consistent with a gestation of 37 weeks 5 days. A transvaginal scan showed a grossly abnormal intracranial anatomy (Fig. 1). The brain was displaced from the inner table of the cranium by a hypoechoic extraaxial fluid collection. Within this extraaxial fluid, two hyperechoic irregular masses were seen in the right frontoparietal area. The right hemisphere and falx were displaced toward the left. The ventricular system was not dilated, and there was no evidence of intraventricular hemorrhage. The frontal portion of the lateral ventricles and the hippocampal gyri appeared to be symmetrically compressed by the extraaxial fluid. Interestingly, the sulci of the brain tissue were markedly accentuated and appeared as bright echogenic lines. From the Section of Maternal-Fetal Medicine, Department of Obstetrics and Gynecology, and the Department of Diagnostic Imaging, Yale University School of Medicine. Received for publication October 25, 1990; accepted December 28, 1990. Reprint requests: Siegfried Rotmensch, MD, Section of MaternalFetal Medicine, Department of Obstetrics and Gynecology, Yale University School of Medicine, 333 Cedar St., New Haven, CT 06510.
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The central areas of the brain tissue, which correlated with the anatomic location of white matter, appeared significantly more echogenic in comparison with the peripheral brain tissue, raising suspicion of evolving infarction or encephalomalacia. The cerebellar vermis and hemispheres could not be clearly outlined and appeared as an irregularly shaped mass in the posterior fossa. The above findings were suggestive of a fetal subdural hematoma with multiple blood clots in the subdural space and secondary compression of brain tissue. Percutaneous umbilical blood sampling was performed for chromosome analysis, fetal acid-base status, and coagulation profile. The following results were obtained: pH, 7.35; base deficit, -8.2 mmol/L; hematocrit, 14.1 %, prothrombin time, 64.6 seconds (control, 11.1 seconds); partial thromboplastin time, >2 minutes (control, 28.6 seconds), platelet count, 236,OOO/m1; and a weakly positive direct Coomb's test. Fetal liver function tests and plasma protein level were within normal limits. The mother's blood type was 0 negative with an anti-D titer of 1: 64. Screening tests for cytomegalovirus and toxoplasmosis were negative. An intrauterine transfusion of 130 ml of O-negative packed red blood cells was performed, which raised fetal hematocrit to 30%. Because of the unexplained fetal coagulopathy, 30 ml of fresh frozen plasma was transfused empirically. After the transfusion the nonstress test became reactive for the first time, and the biophysical profile scored 10 points on a scale of 10. Amniocentesis revealed mature lung indices. The fetal karyotype was normal 46,XY. To further characterize the intracranial findings, a magnetic resonance imaging scan of the fetal brain was obtained, which confirmed the diagnosis (Fig. 2). Fetal paralysis during the scan was achieved with pancuronium bromide, 0.1 mg/kg, via cordocentesis. In consultation with the neonatologists, delivery was delayed despite mature fetal lung indices, to reduce the potential for other complications of prematurity. Nine days after the intrauterine transfusion, the daily nonstress test became nonreactive again, and the biophys-
Volume 164 Number 5. Part I
ical profile failed to reveal breathing movements and tone. Marked polyhydramnios had acutely formed. An emergency low-transverse cesarean section was performed, and a male fetus weighing 2890 gm with Apgar scores of 3 and 5 at 1 and 5 minutes, respectively, was delivered. The umbilical cord arterial pH was 7.19 with a base deficit of - 8.0 mmol/ L. Marked craniomegaly with widespread sutures and bulging fontanelles were noted. The immediate neonatal course was complicated by multiple episodes of bradycardia. Respiratory efforts were essentially absent, leading to intubation. Neonatal brain scans by both ultrasonography and computed tomography revealed a large subdural hematoma, diffuse cerebral edema, and massive bilateral intraventricular and periventricular hemorrhages. The infant died 6 hours after delivery. Autopsy confirmed the diagnosis of bilateral subdural hematomas with multiple organized clots in the subdural space and massive intraventricular hemorrhage. Comment
The antenatal diagnosis of a subdural hematoma can be based on the findings described. Transvaginal ultrasonography was of great value in complete visualization of the cranium in this fetus with vertex presentation, and magnetic resonance imaging was a useful adjunct for confirmation. Fetal paralysis with pancuronium via cordocentesis allowed for diagnostic magnetic resonance imaging without interference by fetal motion. We believe that the enhanced echogenicity of sulci and the apparent differential echogenicity of white and gray matter in our case reflect brain edema. Furthermore, the rim of fluid surrounding the brain in the subdural space might have provided an additional acoustic window, resulting in accentuation of the brain texture. The prognosis for a fetus affected by subdural hematoma is conceivably dependent on the severity of the hemorrhage and the underlying cause. In one of the previously reported cases hypotonic quadriparesis and bilateral optic atrophy subsequently developed in the surviving infant." In another case the fetus survived but experienced neonatal seizures. I The prognosis thus far appears to be dismal. Our experience with fetal hydrocephalus has taught us that brain function cannot be predicted accurately by ultrasonographic findings except in the most severe cases. In the absence of extensive experience with fetal subdural hematoma, it appears prudent to optimize antenatal treatment and delivery management in the expectation of fetal survival. In contrast to the management of our own case, we suggest delivery as soon as fetal lung maturity is determined. In this case fetal intraventricular hemorrhage must have occurred in the 24-hour period before delivery or immediately post partum, since daily ultrasonographic scans showed no evidence of this complication. Earlier delivery might
Fetal subdural hematoma 1247
Fig. I. Transvaginal coronal scan of fetal cranium. Large clot (c) and fluid (fl) in subdural space are present. Arrows point
toward area of central echodensity. which is consistent with anatomic location of white matter surrounded by more echolucent gray matter.
Fig. 2. Tl-weighted (repetition time 500 msec; echo time 20 msec) magnetic resonance imaging in the coronal plane of mother but in sagittal plane of fetal head. In this sequence brightest signals are obtained from blood and fat. Fetal subdural hematoma surrounds the brain, whereas subcutaneous fat outlines external surface of fetus.
have prevented the occurrence of intraventricular hemorrhage. If indicators of lung maturity are negative, we suggest percutaneous umbilical blood sampling for diagnosis and possible intrauterine transfusion. Percutaneous umbilical blood sampling revealed a severe co-
Rotmensch et al. Am
agulopathy and anemia in this case. Both were partially corrected by transfusion of donor blood cells and freshfrozen plasma. Even though postransfusion clotting function could not be obtained, the coagulation function and hematocrit were improved 9 days later, at the time of delivery. Fetal blood specimens were not sufficient for a complete hematologic evaluation. The route of delivery was by cesarean section in all three reported cases, because of remarkable enlargement of the head size. Undoubtedly, the overall efforts and maternal risks might appear excessive, considering the dismal outcome in at least two of the three cases which have so far been reported. However, physicians
Mav 1991
J Obstet Cynecol
are ethically committed to optimize treatment in cases of a central nervous system lesion with yet undetermined outcome." REFERENCES 1. Hanigan WC, Maqbool BA, Cusack TJ, et al. Diagnosis of subdural hematoma in utero. J Neurosurg 1985;63:977-9. 2. Gunn TR, Mok PM, Becraft DMO. Subdural hemorrhage in utero. Pediatrics 1985;76:605-9. 3. Chervenak FA, McCullogh LB. An ethically justified, clinically comprehensive management strategy for thirdtrimester pregnancies complicated by fetal anomalies. Obstet Gynecol 1990;75:311-6.
Calcium-regulating hormones and osteocalcin levels during pregnancy: A longitudinal study Katsuyoshi Seki, MD, Noriko Makimura, DMSc, Chieko Mitsui, BS, Junko Hirata, BS, and Ichiro Nagata, MD Tokorozauia City, Saitama, Japan We measured serum concentrations of calcium-regulating hormones and osteocalcin in 20 women longitudinally throughout pregnancy. 1,25-0ihydroxyvitamin D levels were high early in pregnancy and increased with advancing gestation. Parathyroid hormone and osteocalcin levels were low in early pregnancy. They declined toward the middle of pregnancy, but increased in late pregnancy. The serum osteocalcin level correlated with the parathyroid hormone level. The synthesis of osteocalcin by osteoblasts is stimulated by the action of 1,25-dihydroxyvitamin D, and serum osteocalcin levels are also related to the levels of parathyroid hormone. During early and mid pregnancy, the stimulatory effect of 1,25-dihydroxyvitamin D on the synthesis of osteocalcin may be overridden by the inhibitory effect of declining parathyroid hormone levels. The increase in osteocalcin level in late pregnancy may be a consequence of increasing levels of both parathyroid hormone and 1,25-dihydroxyvitamin D. (AM J OBSTET GVNECOL 1991 ;164:1248-52.)
Key words: Calcium-regulating hormone, osteocalcin, pregnancy The effect of pregnancy on the maternal calcium content is unclear. I The issue has been investigated by means of photon absorptiometry or x-ray spectrophotometry, but the results appear contradictory. Osteocalcin is a bone-specific protein released into the
From the Department of Obstetrics and Gynecology, National Defense Medical College. Received for publication March 6, 1990; revised November 5, 1990; accepted November 27, 1990. Reprint requests: Katsuyoshi Seki, MD, Department of Obstetrics and Gynecology, National Defense Medical College, Namiki 3-2, Tokorozawa, Saitama 359, Japan.
6/1/27004 1248
circulation proportional to the rate of new bone formarion." The usefulness of serum osteocalcin measurements as a clinical marker of bone turnover is becoming more widely accepted." 4 Serum osteocalcin levels are lower in pregnant women compared with nonpregnant women, indicating reduced bone turnover during pregnancy." However, the cause of the decrease in osteocalcin levels during pregnancy is unclear. The synthesis of osteocalcin by osteoblasts is stimulated by the action of 1,25-dihydroxyvitamin D [1,25(OHhD],'> and serum osteocalcin levels also are influenced by parathyroid hormone." Serum 1,25(OH)2D is increased early in pregnancy and continues to rise until delivery.'
Key words: Subdural hematoma, percutaneous umbilical blood sampling, transvaginal ultrasonography, magnetic resonance imaging The in utero diagnosis of subdural hematoma has been reported in two cases. I. 2 Percutaneous umbilical blood sampling, transvaginal ultrasonography, and magnetic resonance imaging have added new dimensions to the identification and management of this entity. We report a case and suggest guidelines for management.
Case history A 37-year-old Haitian woman, gravida 4, para 3, was first seen at 32 weeks' gestation. Physical examination revealed a uterine fundal height of 37 em. A transabdominal ultrasonographic scan showed a single live fetus in vertex presentation with moderate polyhydramnios. Biometry of the long bones was consistent with 32 weeks' gestation. Gross fetal anatomy appeared normal except for the intracranium, which could not be well visualized because of engagement of the fetal head in the pelvis. The biparietal diameter was consistent with a gestation of 37 weeks 5 days. A transvaginal scan showed a grossly abnormal intracranial anatomy (Fig. 1). The brain was displaced from the inner table of the cranium by a hypoechoic extraaxial fluid collection. Within this extraaxial fluid, two hyperechoic irregular masses were seen in the right frontoparietal area. The right hemisphere and falx were displaced toward the left. The ventricular system was not dilated, and there was no evidence of intraventricular hemorrhage. The frontal portion of the lateral ventricles and the hippocampal gyri appeared to be symmetrically compressed by the extraaxial fluid. Interestingly, the sulci of the brain tissue were markedly accentuated and appeared as bright echogenic lines. From the Section of Maternal-Fetal Medicine, Department of Obstetrics and Gynecology, and the Department of Diagnostic Imaging, Yale University School of Medicine. Received for publication October 25, 1990; accepted December 28, 1990. Reprint requests: Siegfried Rotmensch, MD, Section of MaternalFetal Medicine, Department of Obstetrics and Gynecology, Yale University School of Medicine, 333 Cedar St., New Haven, CT 06510.
6/1/27678
1246
The central areas of the brain tissue, which correlated with the anatomic location of white matter, appeared significantly more echogenic in comparison with the peripheral brain tissue, raising suspicion of evolving infarction or encephalomalacia. The cerebellar vermis and hemispheres could not be clearly outlined and appeared as an irregularly shaped mass in the posterior fossa. The above findings were suggestive of a fetal subdural hematoma with multiple blood clots in the subdural space and secondary compression of brain tissue. Percutaneous umbilical blood sampling was performed for chromosome analysis, fetal acid-base status, and coagulation profile. The following results were obtained: pH, 7.35; base deficit, -8.2 mmol/L; hematocrit, 14.1 %, prothrombin time, 64.6 seconds (control, 11.1 seconds); partial thromboplastin time, >2 minutes (control, 28.6 seconds), platelet count, 236,OOO/m1; and a weakly positive direct Coomb's test. Fetal liver function tests and plasma protein level were within normal limits. The mother's blood type was 0 negative with an anti-D titer of 1: 64. Screening tests for cytomegalovirus and toxoplasmosis were negative. An intrauterine transfusion of 130 ml of O-negative packed red blood cells was performed, which raised fetal hematocrit to 30%. Because of the unexplained fetal coagulopathy, 30 ml of fresh frozen plasma was transfused empirically. After the transfusion the nonstress test became reactive for the first time, and the biophysical profile scored 10 points on a scale of 10. Amniocentesis revealed mature lung indices. The fetal karyotype was normal 46,XY. To further characterize the intracranial findings, a magnetic resonance imaging scan of the fetal brain was obtained, which confirmed the diagnosis (Fig. 2). Fetal paralysis during the scan was achieved with pancuronium bromide, 0.1 mg/kg, via cordocentesis. In consultation with the neonatologists, delivery was delayed despite mature fetal lung indices, to reduce the potential for other complications of prematurity. Nine days after the intrauterine transfusion, the daily nonstress test became nonreactive again, and the biophys-
Volume 164 Number 5. Part I
ical profile failed to reveal breathing movements and tone. Marked polyhydramnios had acutely formed. An emergency low-transverse cesarean section was performed, and a male fetus weighing 2890 gm with Apgar scores of 3 and 5 at 1 and 5 minutes, respectively, was delivered. The umbilical cord arterial pH was 7.19 with a base deficit of - 8.0 mmol/ L. Marked craniomegaly with widespread sutures and bulging fontanelles were noted. The immediate neonatal course was complicated by multiple episodes of bradycardia. Respiratory efforts were essentially absent, leading to intubation. Neonatal brain scans by both ultrasonography and computed tomography revealed a large subdural hematoma, diffuse cerebral edema, and massive bilateral intraventricular and periventricular hemorrhages. The infant died 6 hours after delivery. Autopsy confirmed the diagnosis of bilateral subdural hematomas with multiple organized clots in the subdural space and massive intraventricular hemorrhage. Comment
The antenatal diagnosis of a subdural hematoma can be based on the findings described. Transvaginal ultrasonography was of great value in complete visualization of the cranium in this fetus with vertex presentation, and magnetic resonance imaging was a useful adjunct for confirmation. Fetal paralysis with pancuronium via cordocentesis allowed for diagnostic magnetic resonance imaging without interference by fetal motion. We believe that the enhanced echogenicity of sulci and the apparent differential echogenicity of white and gray matter in our case reflect brain edema. Furthermore, the rim of fluid surrounding the brain in the subdural space might have provided an additional acoustic window, resulting in accentuation of the brain texture. The prognosis for a fetus affected by subdural hematoma is conceivably dependent on the severity of the hemorrhage and the underlying cause. In one of the previously reported cases hypotonic quadriparesis and bilateral optic atrophy subsequently developed in the surviving infant." In another case the fetus survived but experienced neonatal seizures. I The prognosis thus far appears to be dismal. Our experience with fetal hydrocephalus has taught us that brain function cannot be predicted accurately by ultrasonographic findings except in the most severe cases. In the absence of extensive experience with fetal subdural hematoma, it appears prudent to optimize antenatal treatment and delivery management in the expectation of fetal survival. In contrast to the management of our own case, we suggest delivery as soon as fetal lung maturity is determined. In this case fetal intraventricular hemorrhage must have occurred in the 24-hour period before delivery or immediately post partum, since daily ultrasonographic scans showed no evidence of this complication. Earlier delivery might
Fetal subdural hematoma 1247
Fig. I. Transvaginal coronal scan of fetal cranium. Large clot (c) and fluid (fl) in subdural space are present. Arrows point
toward area of central echodensity. which is consistent with anatomic location of white matter surrounded by more echolucent gray matter.
Fig. 2. Tl-weighted (repetition time 500 msec; echo time 20 msec) magnetic resonance imaging in the coronal plane of mother but in sagittal plane of fetal head. In this sequence brightest signals are obtained from blood and fat. Fetal subdural hematoma surrounds the brain, whereas subcutaneous fat outlines external surface of fetus.
have prevented the occurrence of intraventricular hemorrhage. If indicators of lung maturity are negative, we suggest percutaneous umbilical blood sampling for diagnosis and possible intrauterine transfusion. Percutaneous umbilical blood sampling revealed a severe co-
Rotmensch et al. Am
agulopathy and anemia in this case. Both were partially corrected by transfusion of donor blood cells and freshfrozen plasma. Even though postransfusion clotting function could not be obtained, the coagulation function and hematocrit were improved 9 days later, at the time of delivery. Fetal blood specimens were not sufficient for a complete hematologic evaluation. The route of delivery was by cesarean section in all three reported cases, because of remarkable enlargement of the head size. Undoubtedly, the overall efforts and maternal risks might appear excessive, considering the dismal outcome in at least two of the three cases which have so far been reported. However, physicians
Mav 1991
J Obstet Cynecol
are ethically committed to optimize treatment in cases of a central nervous system lesion with yet undetermined outcome." REFERENCES 1. Hanigan WC, Maqbool BA, Cusack TJ, et al. Diagnosis of subdural hematoma in utero. J Neurosurg 1985;63:977-9. 2. Gunn TR, Mok PM, Becraft DMO. Subdural hemorrhage in utero. Pediatrics 1985;76:605-9. 3. Chervenak FA, McCullogh LB. An ethically justified, clinically comprehensive management strategy for thirdtrimester pregnancies complicated by fetal anomalies. Obstet Gynecol 1990;75:311-6.
Calcium-regulating hormones and osteocalcin levels during pregnancy: A longitudinal study Katsuyoshi Seki, MD, Noriko Makimura, DMSc, Chieko Mitsui, BS, Junko Hirata, BS, and Ichiro Nagata, MD Tokorozauia City, Saitama, Japan We measured serum concentrations of calcium-regulating hormones and osteocalcin in 20 women longitudinally throughout pregnancy. 1,25-0ihydroxyvitamin D levels were high early in pregnancy and increased with advancing gestation. Parathyroid hormone and osteocalcin levels were low in early pregnancy. They declined toward the middle of pregnancy, but increased in late pregnancy. The serum osteocalcin level correlated with the parathyroid hormone level. The synthesis of osteocalcin by osteoblasts is stimulated by the action of 1,25-dihydroxyvitamin D, and serum osteocalcin levels are also related to the levels of parathyroid hormone. During early and mid pregnancy, the stimulatory effect of 1,25-dihydroxyvitamin D on the synthesis of osteocalcin may be overridden by the inhibitory effect of declining parathyroid hormone levels. The increase in osteocalcin level in late pregnancy may be a consequence of increasing levels of both parathyroid hormone and 1,25-dihydroxyvitamin D. (AM J OBSTET GVNECOL 1991 ;164:1248-52.)
Key words: Calcium-regulating hormone, osteocalcin, pregnancy The effect of pregnancy on the maternal calcium content is unclear. I The issue has been investigated by means of photon absorptiometry or x-ray spectrophotometry, but the results appear contradictory. Osteocalcin is a bone-specific protein released into the
From the Department of Obstetrics and Gynecology, National Defense Medical College. Received for publication March 6, 1990; revised November 5, 1990; accepted November 27, 1990. Reprint requests: Katsuyoshi Seki, MD, Department of Obstetrics and Gynecology, National Defense Medical College, Namiki 3-2, Tokorozawa, Saitama 359, Japan.
6/1/27004 1248
circulation proportional to the rate of new bone formarion." The usefulness of serum osteocalcin measurements as a clinical marker of bone turnover is becoming more widely accepted." 4 Serum osteocalcin levels are lower in pregnant women compared with nonpregnant women, indicating reduced bone turnover during pregnancy." However, the cause of the decrease in osteocalcin levels during pregnancy is unclear. The synthesis of osteocalcin by osteoblasts is stimulated by the action of 1,25-dihydroxyvitamin D [1,25(OHhD],'> and serum osteocalcin levels also are influenced by parathyroid hormone." Serum 1,25(OH)2D is increased early in pregnancy and continues to rise until delivery.'
