Case reports
mGNS 9 Springer-Verlag 1991
Child's Nerv Syst (1992) 8:462-464
Chronic subdural hematoma in utero R e p o r t o f a case with survival after t r e a t m e n t Guillermo Javier Nogueira* Department of Neurosurgery, Hamad General Hospital, Doha, Qatar Received March 15, 1991
Abstract. A case o f c h r o n i c s u b d u r a l h e m a t o m a a n d hyd r o c e p h a l u s d i a g n o s e d in u t e r o is presented. N o h i s t o r y o f t r a u m a c o u l d be elicited. L a b o r a t o r y i n v e s t i g a t i o n s failed to show a c o a g u l a t i o n disorder. Both the hem a t o m a a n d h y d r o c e p h a l u s were surgically treated 8 days after delivery. A t 14 m o n t h s o f age the child is showing m o d e r a t e l y delayed d e v e l o p m e n t . T h e literature is reviewed a n d discussed. T h e i m p o r t a n c e o f correct anten a t a l diagnosis is stressed. Key words: C h r o n i c s u b d u r a l h e m a t o m a - I n u t e r o
Since the a d v e n t o f u l t r a s o u n d e x a m i n a t i o n d u r i n g pregn a n c y , fetal i n t r a c r a n i a l h e m o r r h a g e has b e e n a d d e d to the list of c o n d i t i o n s which it is n o w possible to d i a g n o s e [2, 6, 11]. However, m o s t o f the r e p o r t e d cases are of either i n t r a v e n t r i c u l a r or i n t r a p a r e n c h y m a t o u s h e m o r rhages [2]. C h r o n i c s u b d u r a l h e m a t o m a s are a rarity, a n d t h a t is w h a t p r o m p t e d this c o m m u n i c a t i o n , together with c o n s i d e r a t i o n s a b o u t correct diagnosis a n d the implications o f the r e l a t i o n s h i p o f this lesion with h y d r o cephalus.
Case report The patient was the child of a 38-year-old Qatari woman, gravida 13, para 11, 1 spontaneous abortion, diabetic receiving insulin treatment. There was no history of consanguinity and all siblings were reported to be normal. On April 27, 1989, the first ultrasound study was performed and reported as showing a gestational age of 10 weeks, in high implantation. Follow-up ultrasonography on October 7, 1989 (Fig. 1), showed an enlarged, asymmetric ventricular system, the left side larger than right. Biparietal diameter was 9.7 cm, head circumference 35.6 cm, and lateral ventricular ratio 0.80. These values correspond to a gestational age of 40 weeks, while the abdominal circumference and femur length were represen* Present address and address f o r correspondence: Calle Rivas 4029,
7600 Mar del Plata, Argentina
tative of 33 weeks' gestational age. The weight was estimated at 2459_+ 200 g. Since the pregnancy was considered uneventful otherwise, and there was no evidence of fetal distress, no action was taken. On October 17, 1989, a boy weighing 2570 g was born. A spontaneous, normal vaginal delivery was accomplished. Gestational age was 37 weeks by date and 34 weeks by estimation. The Apgar score was 9 and 10 at 1 and 5 min respectively. Head circumference was 36.5 cm (90th percentile). Blood group of the mother and baby were 0 R h + . Clinical findings at that time were within normal limits except for the head size. Ophthalmological examination reported normal results, as did the laboratory work-up, including coagulation tests on both the baby and the mother. TORCH test and chromosomal studies were later reported as giving normal findings also. The baby developed a mild jaundice that resolved with phototherapy. A new ultrasound examination was performed on October 18, 1989 (Fig. 2), showing again asymmetric ventricular enlargement with obliteration of the right occipital horn. A single ventricle was apparent in the anterior part, and the suspicion of a complex malformation was raised. CT scanning was performed the next day (Fig. 3) and showed massive dilatation of the left ventricle with occlusion and displacement of the posterior portion of the right ventricle. A mixed-density lesion was visible on the right parietaloccipital area, surrounded by an enhancing membrane. The anterior portion of the lateral ventricles seemed to form a single cavity. In retrospect the ultrasound of October 7 showed similar findings. With the diagnosis of hydrocephalus and chronic subdural hematoma, the patient was taken to surgery on October 25. A left-sided ventriculo peritoneal shunt, medium pressure, was inserted, and through a right parietal burr-hole a partly solid, old subdural hematoma was evacuated. The membrane was clearly visible and was biopsied. Cerebrospinal fluid (CSF) obtained from the left ventricle was xanthochromic with 20000 RBC and 3 WBC, the sugar level was 34 mg/dl and albumin 150 mg/dl. Cultures were negative. The post-operative course was uneventful and the baby was discharged home at 20 days of age. At 6 months of age examination showed him to be developing within normal limits and CT showed marked reduction of the hydrocephalus and disappearance of the right parietal hematoma (Fig. 4). Follow-up at 14 months revealed a moderately delayed development with spastic diplegia. Motor performance was at about the 6-month-old level but performance in other areas was at the 10- to 12-month-old level. Head circumference is at the 25th percentile. The shunt appeared clinically to be functioningwelt. The parents were repeatedly questioned but could not recall any trauma during the pregnancy. The biopsy showed fibrous tissue with hemosiderin deposits, compatible with hematoma capsule.
463
Fig. 1. Ultrasonogram 10 days before birth, showing an enlarged, asymmetric ventricular system in which the left side is larger than the right Fig. 2. Ultrasonogram the day after birth, still showing asymmetric ventricular enlargement, with obliteration of the right occipital horn Fig. 3 a, b. CT scans taken the day after the ultrasonogram in Fig. 2, showing massive dilatation of the left ventricle with occlusion and displacement of the posterior portion of the right ventricle. Mixed density lesion and enhancing capsule in the right parieto-occipital area
Fig. 4a, b. CT scans taken 6 months after birth, showing marked reduction of the hydrocephalus and disappearance of the right parietal hematoma
Discussion Subdural h e m a t o m a s in utero are quite rare. The first reported case in the absence of trauma [8] dates from 1977, and the first one actually diagnosed in utero and treated was reported in 1985 [5]. The fact that antenatal ultrasonography is routinely used in m a n y places without a significant increase in the n u m b e r of reported cases points in the same direction. N o n e of the known causes apply to our case as there was no history of trauma or clotting disorders [9, 10]. A high incidence in babies born of Samoan mothers was reported by G u n n e t al. [4] and was presumed to relate to " a b d o m i n a l massage" by traditional healers. N o such practice is used in Qatar and the ethnic background does not apply either. A review of the literature available to us showed that out of 7 reported cases [1, 3 - 5, 8, 9], only one was unilateral [4] like our case. Association with hydrocephalus is frequent (5 out of 8, including our case). Prognosis is p o o r (3 out of 8 died and 5 out of 8 had abnormal development). Once the obvious causes such as trauma, sepsis, coagulation disorders, and vascular anomalies are excluded, there are no clues to the etiology. The fact is that obvious causes are seldom present in the cases diagnosed. The association with hydrocephalus, especially in the presence of hemorrhagic CSF, poses several interesting questions. The existence of intracranial hemorrhages, not related to the delivery and therefore antenatal, is now well documented [2, 6, 7, 10]. Consequently, one possibility for subdural h e m a t o m a s is that they represent just one variety of the same entity with a particular location and magnitude. Hydrocephalus m a y be a direct consequence of the subdural collection or m a y be due to the presence of blood in the CSF pathways. More complex possibili-
ties can be guessed at, if we consider intracranial pressure changes and their relationship with maternal abdominal and uterine pressures, as well as spatial shifting of the fetus or the intracranial contents. F r o m the clinician's point of view, and on the basis of our experience with this case, it is important to be aware of the diagnostic possibility of chronic subdural hem a t o m a at the time of ultrasound examination. The study should be repeated, or another examination technique (MRI, CT) used, until the intracranial structures are well visualized and can be clearly interpreted. In our case the description of the ultrasonogram was correct but the interpretation was wrong, probably because the frequency of malformations biased the interpreter against considering subdural hematoma. Correct diagnosis should lead to a p r o m p t and aggressive m a n a g e m e n t policy, in order to minimize brain damage, which in these patients is due to multiple factors: hydrocephalus, local pressure by the hematomas, increased intracranial pressure, anemia, presence of blood in the CSF, and hemodynamic changes, either independent or related to all the above.
References 1. Atluru VL, Kumar IR (1987) Intrauterine chronic subdural hematomas with postoperative tension pneumocephalus. Pediatr Neurol 3:306-309 2. Bondurant S, Boehm FH, Fleischer AC, Machin JE (1984) Antepartum diagnosis of fetal intracranial hemorrhage by ultrasound. Obstet Gynecol 64 [Suppl]: 25-27 3. Demir RH, Gleicher N, Myers SA (1989) Atraumatic anteparturn subdural hematoma causing fetal death. Am J Obstet Gynecol 160:619-620 4. Gunn TR, Mok PM, Becroft DMO (1985) Subdural hemorrhage in utero. Pediatrics 76:605 610
464 5. Hanigan WC, Maqbool BA, Cusack TJ, Miller TC, Shah JJ (1985) Diagnosis of subdural hemorrhage in utero. Case report. J Neurosurg 63:977-979 6. Jackson JC, Blumhagen JD (1983) Congenital hydrocephalus due to prenatal intracranial hemorrhage. Pediatrics 72: 344346 7. Lebed MR, Platt LD, Schifrin BS, Sims M, Afriat C (1980) Fetal subdural hemorrhage presenting as hydrocephalus. Br Med J 281:679-680 8. MacDonald JT, Weitz R, Sher PK (1977) Intrauterine chronic subdural hematoma. Arch Neurol 34:777-778
9. Robinson MJ, Cameron MD, Smith MF, Ayers AB (1980) Fetal subdural hemorrhages presenting as hydrocephalus. Br Med J 281:35 10. Sokal MM, Katz M, Lell ME, Fox A (1980) Neonatal survival after traumatic fetal subdural hematoma. J Reprod Med 24:131-133 11. Stifling HF, Hendry M, Brown JK (1989) Prenatal intracranial hemorrhage. Dev Med Child Neurol 31:798-815
mGNS 9 Springer-Verlag 1991
Child's Nerv Syst (1992) 8:462-464
Chronic subdural hematoma in utero R e p o r t o f a case with survival after t r e a t m e n t Guillermo Javier Nogueira* Department of Neurosurgery, Hamad General Hospital, Doha, Qatar Received March 15, 1991
Abstract. A case o f c h r o n i c s u b d u r a l h e m a t o m a a n d hyd r o c e p h a l u s d i a g n o s e d in u t e r o is presented. N o h i s t o r y o f t r a u m a c o u l d be elicited. L a b o r a t o r y i n v e s t i g a t i o n s failed to show a c o a g u l a t i o n disorder. Both the hem a t o m a a n d h y d r o c e p h a l u s were surgically treated 8 days after delivery. A t 14 m o n t h s o f age the child is showing m o d e r a t e l y delayed d e v e l o p m e n t . T h e literature is reviewed a n d discussed. T h e i m p o r t a n c e o f correct anten a t a l diagnosis is stressed. Key words: C h r o n i c s u b d u r a l h e m a t o m a - I n u t e r o
Since the a d v e n t o f u l t r a s o u n d e x a m i n a t i o n d u r i n g pregn a n c y , fetal i n t r a c r a n i a l h e m o r r h a g e has b e e n a d d e d to the list of c o n d i t i o n s which it is n o w possible to d i a g n o s e [2, 6, 11]. However, m o s t o f the r e p o r t e d cases are of either i n t r a v e n t r i c u l a r or i n t r a p a r e n c h y m a t o u s h e m o r rhages [2]. C h r o n i c s u b d u r a l h e m a t o m a s are a rarity, a n d t h a t is w h a t p r o m p t e d this c o m m u n i c a t i o n , together with c o n s i d e r a t i o n s a b o u t correct diagnosis a n d the implications o f the r e l a t i o n s h i p o f this lesion with h y d r o cephalus.
Case report The patient was the child of a 38-year-old Qatari woman, gravida 13, para 11, 1 spontaneous abortion, diabetic receiving insulin treatment. There was no history of consanguinity and all siblings were reported to be normal. On April 27, 1989, the first ultrasound study was performed and reported as showing a gestational age of 10 weeks, in high implantation. Follow-up ultrasonography on October 7, 1989 (Fig. 1), showed an enlarged, asymmetric ventricular system, the left side larger than right. Biparietal diameter was 9.7 cm, head circumference 35.6 cm, and lateral ventricular ratio 0.80. These values correspond to a gestational age of 40 weeks, while the abdominal circumference and femur length were represen* Present address and address f o r correspondence: Calle Rivas 4029,
7600 Mar del Plata, Argentina
tative of 33 weeks' gestational age. The weight was estimated at 2459_+ 200 g. Since the pregnancy was considered uneventful otherwise, and there was no evidence of fetal distress, no action was taken. On October 17, 1989, a boy weighing 2570 g was born. A spontaneous, normal vaginal delivery was accomplished. Gestational age was 37 weeks by date and 34 weeks by estimation. The Apgar score was 9 and 10 at 1 and 5 min respectively. Head circumference was 36.5 cm (90th percentile). Blood group of the mother and baby were 0 R h + . Clinical findings at that time were within normal limits except for the head size. Ophthalmological examination reported normal results, as did the laboratory work-up, including coagulation tests on both the baby and the mother. TORCH test and chromosomal studies were later reported as giving normal findings also. The baby developed a mild jaundice that resolved with phototherapy. A new ultrasound examination was performed on October 18, 1989 (Fig. 2), showing again asymmetric ventricular enlargement with obliteration of the right occipital horn. A single ventricle was apparent in the anterior part, and the suspicion of a complex malformation was raised. CT scanning was performed the next day (Fig. 3) and showed massive dilatation of the left ventricle with occlusion and displacement of the posterior portion of the right ventricle. A mixed-density lesion was visible on the right parietaloccipital area, surrounded by an enhancing membrane. The anterior portion of the lateral ventricles seemed to form a single cavity. In retrospect the ultrasound of October 7 showed similar findings. With the diagnosis of hydrocephalus and chronic subdural hematoma, the patient was taken to surgery on October 25. A left-sided ventriculo peritoneal shunt, medium pressure, was inserted, and through a right parietal burr-hole a partly solid, old subdural hematoma was evacuated. The membrane was clearly visible and was biopsied. Cerebrospinal fluid (CSF) obtained from the left ventricle was xanthochromic with 20000 RBC and 3 WBC, the sugar level was 34 mg/dl and albumin 150 mg/dl. Cultures were negative. The post-operative course was uneventful and the baby was discharged home at 20 days of age. At 6 months of age examination showed him to be developing within normal limits and CT showed marked reduction of the hydrocephalus and disappearance of the right parietal hematoma (Fig. 4). Follow-up at 14 months revealed a moderately delayed development with spastic diplegia. Motor performance was at about the 6-month-old level but performance in other areas was at the 10- to 12-month-old level. Head circumference is at the 25th percentile. The shunt appeared clinically to be functioningwelt. The parents were repeatedly questioned but could not recall any trauma during the pregnancy. The biopsy showed fibrous tissue with hemosiderin deposits, compatible with hematoma capsule.
463
Fig. 1. Ultrasonogram 10 days before birth, showing an enlarged, asymmetric ventricular system in which the left side is larger than the right Fig. 2. Ultrasonogram the day after birth, still showing asymmetric ventricular enlargement, with obliteration of the right occipital horn Fig. 3 a, b. CT scans taken the day after the ultrasonogram in Fig. 2, showing massive dilatation of the left ventricle with occlusion and displacement of the posterior portion of the right ventricle. Mixed density lesion and enhancing capsule in the right parieto-occipital area
Fig. 4a, b. CT scans taken 6 months after birth, showing marked reduction of the hydrocephalus and disappearance of the right parietal hematoma
Discussion Subdural h e m a t o m a s in utero are quite rare. The first reported case in the absence of trauma [8] dates from 1977, and the first one actually diagnosed in utero and treated was reported in 1985 [5]. The fact that antenatal ultrasonography is routinely used in m a n y places without a significant increase in the n u m b e r of reported cases points in the same direction. N o n e of the known causes apply to our case as there was no history of trauma or clotting disorders [9, 10]. A high incidence in babies born of Samoan mothers was reported by G u n n e t al. [4] and was presumed to relate to " a b d o m i n a l massage" by traditional healers. N o such practice is used in Qatar and the ethnic background does not apply either. A review of the literature available to us showed that out of 7 reported cases [1, 3 - 5, 8, 9], only one was unilateral [4] like our case. Association with hydrocephalus is frequent (5 out of 8, including our case). Prognosis is p o o r (3 out of 8 died and 5 out of 8 had abnormal development). Once the obvious causes such as trauma, sepsis, coagulation disorders, and vascular anomalies are excluded, there are no clues to the etiology. The fact is that obvious causes are seldom present in the cases diagnosed. The association with hydrocephalus, especially in the presence of hemorrhagic CSF, poses several interesting questions. The existence of intracranial hemorrhages, not related to the delivery and therefore antenatal, is now well documented [2, 6, 7, 10]. Consequently, one possibility for subdural h e m a t o m a s is that they represent just one variety of the same entity with a particular location and magnitude. Hydrocephalus m a y be a direct consequence of the subdural collection or m a y be due to the presence of blood in the CSF pathways. More complex possibili-
ties can be guessed at, if we consider intracranial pressure changes and their relationship with maternal abdominal and uterine pressures, as well as spatial shifting of the fetus or the intracranial contents. F r o m the clinician's point of view, and on the basis of our experience with this case, it is important to be aware of the diagnostic possibility of chronic subdural hem a t o m a at the time of ultrasound examination. The study should be repeated, or another examination technique (MRI, CT) used, until the intracranial structures are well visualized and can be clearly interpreted. In our case the description of the ultrasonogram was correct but the interpretation was wrong, probably because the frequency of malformations biased the interpreter against considering subdural hematoma. Correct diagnosis should lead to a p r o m p t and aggressive m a n a g e m e n t policy, in order to minimize brain damage, which in these patients is due to multiple factors: hydrocephalus, local pressure by the hematomas, increased intracranial pressure, anemia, presence of blood in the CSF, and hemodynamic changes, either independent or related to all the above.
References 1. Atluru VL, Kumar IR (1987) Intrauterine chronic subdural hematomas with postoperative tension pneumocephalus. Pediatr Neurol 3:306-309 2. Bondurant S, Boehm FH, Fleischer AC, Machin JE (1984) Antepartum diagnosis of fetal intracranial hemorrhage by ultrasound. Obstet Gynecol 64 [Suppl]: 25-27 3. Demir RH, Gleicher N, Myers SA (1989) Atraumatic anteparturn subdural hematoma causing fetal death. Am J Obstet Gynecol 160:619-620 4. Gunn TR, Mok PM, Becroft DMO (1985) Subdural hemorrhage in utero. Pediatrics 76:605 610
464 5. Hanigan WC, Maqbool BA, Cusack TJ, Miller TC, Shah JJ (1985) Diagnosis of subdural hemorrhage in utero. Case report. J Neurosurg 63:977-979 6. Jackson JC, Blumhagen JD (1983) Congenital hydrocephalus due to prenatal intracranial hemorrhage. Pediatrics 72: 344346 7. Lebed MR, Platt LD, Schifrin BS, Sims M, Afriat C (1980) Fetal subdural hemorrhage presenting as hydrocephalus. Br Med J 281:679-680 8. MacDonald JT, Weitz R, Sher PK (1977) Intrauterine chronic subdural hematoma. Arch Neurol 34:777-778
9. Robinson MJ, Cameron MD, Smith MF, Ayers AB (1980) Fetal subdural hemorrhages presenting as hydrocephalus. Br Med J 281:35 10. Sokal MM, Katz M, Lell ME, Fox A (1980) Neonatal survival after traumatic fetal subdural hematoma. J Reprod Med 24:131-133 11. Stifling HF, Hendry M, Brown JK (1989) Prenatal intracranial hemorrhage. Dev Med Child Neurol 31:798-815
