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Refractory High Intracranial Pressure following Intraventricular Hemorrhage due to Moyamoya Disease in a Pregnant Caucasian Woman.

Montiel V, Grandin C, Goffette P, Fomekong E, Hantson P - Case Rep Neurol (2009)

Bottom Line: Refractory high intracranial pressure developed and required bilateral ventricular drainage and intensive care treatment with barbiturates and hypothermia.Magnetic resonance imaging and cerebral angiography revealed a moyamoya syndrome with rupture of the abnormal collateral vascular network as the cause of the hemorrhage.Despite an initially low GCS, this patient made a good functional recovery at one year follow-up.

View Article: PubMed Central - PubMed

Affiliation: Departments of Intensive Care, Brussels, Belgium.

ABSTRACT
Intraventricular hemorrhage during pregnancy is usually followed by a poor recovery. When caused by moyamoya disease, ischemic or hemorrhagic episodes may complicate the management of high intracranial pressure. A 26-year-old Caucasian woman presented with generalized seizures and a Glasgow Coma Score (GCS) of 3 during the 36th week of pregnancy. The fetus was delivered by caesarean section. The brain CT in the mother revealed bilateral intraventricular hemorrhage, a callosal hematoma, hydrocephalus and right frontal ischemia. Refractory high intracranial pressure developed and required bilateral ventricular drainage and intensive care treatment with barbiturates and hypothermia. Magnetic resonance imaging and cerebral angiography revealed a moyamoya syndrome with rupture of the abnormal collateral vascular network as the cause of the hemorrhage. Intracranial pressure could only be controlled after the surgical removal of the clots after a large opening of the right ventricle. Despite an initially low GCS, this patient made a good functional recovery at one year follow-up. Management of refractory high intracranial pressure following moyamoya related intraventricular bleeding should require optimal removal of ventricular clots and appropriate control of cerebral hemodynamics to avoid ischemic or hemorrhagic complications.

No MeSH data available.


Related in: MedlinePlus

Brain MR-angiography. Occlusion of the right internal carotid artery (ICA), severe stenosis of the left ICA and of the M1 segment of the left middle cerebral arteries (MCA), occlusion of the left posterior cerebral artery, collateral vessels (arrows) arising from the external carotid arteries, and an extensive abnormal vascular network appearing as multiple small collateral vessels developed mainly in basal cisterna and in the thalami.
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Figure 2: Brain MR-angiography. Occlusion of the right internal carotid artery (ICA), severe stenosis of the left ICA and of the M1 segment of the left middle cerebral arteries (MCA), occlusion of the left posterior cerebral artery, collateral vessels (arrows) arising from the external carotid arteries, and an extensive abnormal vascular network appearing as multiple small collateral vessels developed mainly in basal cisterna and in the thalami.

Mentions: A 26-year-old Caucasian pregnant woman at 36 weeks of gestation presented to the Emergency Department of a first hospital for a severe headache. Her past medical history was not relevant. There was no sign of toxemia during pregnancy and no history of hypertension. She had delivered her first child 3 years ago without any complication. Soon after admission, she developed generalized tonic-clonic seizures with a GCS at 3 immediately after seizure. A caesarian section was performed without any delay under general anesthesia and mechanical ventilation. The brain computerized tomography (CT) performed immediately after revealed extensive bilateral intraventricular hemorrhage and a large hematoma in the splenium of the corpus callosum. Routine laboratory tests including coagulation tests had normal results. An external ventricular drain was inserted bilaterally. Despite bilateral ventricular drainage and intensive care management, intracranial pressure (ICP) remained above 40 mm Hg. Diabetes insipidus was noted on day 2. Treatment with intravenous barbiturates and hypothermia was started. Hemodynamic conditions worsened and catecholamines administration (dobutamine and norepinephrine) was required. The patient was transferred to the university hospital on day 8. On admission, GCS was 3/15 with abolition of brainstem reflexes. Central body temperature was 32°C. As the EEG showed burst suppression, barbiturate overdose was suspected and continuous infusion was stopped. Blood thiopental concentration was excessive (68.8 μg/ml). Cardiocirculatory status improved after barbiturate withdrawal. Echocardiography disclosed normal left ventricle contractility. However, high intracranial pressure persisted above 40 mm Hg. On brain CT (fig. 1), the size of the intraventricular hemorrhage had decreased on the left but not on the right side and the sulci were diffusely obliterated, confirming the intracranial hypertension. A right subcortical frontal ischemic lesion was also visible. The right drain was impinged in the ventricular clot and was ineffective, even after the in loco infusion of a fibrinolytic agent (5 mg rt-PA), leading to refractory high intracranial pressure. Endoscopic removal of the right ventricular clots was first tried, but it was necessary to perform a larger right frontal craniotomy. Intracranial pressure decreased postoperatively but the GCS remained at 3/15 with absence of brainstem reflexes. A brain magnetic resonance imaging (MRI) performed on day 20 showed the partial removal of the intraventricular clot with decreased ventricular size and reopening of the sulci. MR-angiography disclosed extensive arterial anomalies with occlusion of the right internal carotid artery (ICA), severe stenosis of the left ICA and of the M1 segment of the left middle cerebral arteries (MCA), occlusion of the left posterior cerebral artery, collaterals arising from the external carotid arteries, and an extensive abnormal vascular network appearing as multiple small collateral vessels developed mainly in basal cisterna and in the thalami (fig. 2). This pattern evoked a moyamoya syndrome that occurred very early in age as demonstrated by the hypoplastic right bony carotid canal (fig. 3). Perfusion-weighted MRI showed moderate hemodynamic disturbances in both hemispheres, predominant on the left side, with delayed time-to-peak, prolonged mean-transit time and a compensatory increased cerebral blood volume. Conventional cerebral angiography (day 39) confirmed these observations.


Refractory High Intracranial Pressure following Intraventricular Hemorrhage due to Moyamoya Disease in a Pregnant Caucasian Woman.

Montiel V, Grandin C, Goffette P, Fomekong E, Hantson P - Case Rep Neurol (2009)

Brain MR-angiography. Occlusion of the right internal carotid artery (ICA), severe stenosis of the left ICA and of the M1 segment of the left middle cerebral arteries (MCA), occlusion of the left posterior cerebral artery, collateral vessels (arrows) arising from the external carotid arteries, and an extensive abnormal vascular network appearing as multiple small collateral vessels developed mainly in basal cisterna and in the thalami.
© Copyright Policy
Related In: Results  -  Collection

Show All Figures
getmorefigures.php?uid=PMC2875849&req=5

Figure 2: Brain MR-angiography. Occlusion of the right internal carotid artery (ICA), severe stenosis of the left ICA and of the M1 segment of the left middle cerebral arteries (MCA), occlusion of the left posterior cerebral artery, collateral vessels (arrows) arising from the external carotid arteries, and an extensive abnormal vascular network appearing as multiple small collateral vessels developed mainly in basal cisterna and in the thalami.
Mentions: A 26-year-old Caucasian pregnant woman at 36 weeks of gestation presented to the Emergency Department of a first hospital for a severe headache. Her past medical history was not relevant. There was no sign of toxemia during pregnancy and no history of hypertension. She had delivered her first child 3 years ago without any complication. Soon after admission, she developed generalized tonic-clonic seizures with a GCS at 3 immediately after seizure. A caesarian section was performed without any delay under general anesthesia and mechanical ventilation. The brain computerized tomography (CT) performed immediately after revealed extensive bilateral intraventricular hemorrhage and a large hematoma in the splenium of the corpus callosum. Routine laboratory tests including coagulation tests had normal results. An external ventricular drain was inserted bilaterally. Despite bilateral ventricular drainage and intensive care management, intracranial pressure (ICP) remained above 40 mm Hg. Diabetes insipidus was noted on day 2. Treatment with intravenous barbiturates and hypothermia was started. Hemodynamic conditions worsened and catecholamines administration (dobutamine and norepinephrine) was required. The patient was transferred to the university hospital on day 8. On admission, GCS was 3/15 with abolition of brainstem reflexes. Central body temperature was 32°C. As the EEG showed burst suppression, barbiturate overdose was suspected and continuous infusion was stopped. Blood thiopental concentration was excessive (68.8 μg/ml). Cardiocirculatory status improved after barbiturate withdrawal. Echocardiography disclosed normal left ventricle contractility. However, high intracranial pressure persisted above 40 mm Hg. On brain CT (fig. 1), the size of the intraventricular hemorrhage had decreased on the left but not on the right side and the sulci were diffusely obliterated, confirming the intracranial hypertension. A right subcortical frontal ischemic lesion was also visible. The right drain was impinged in the ventricular clot and was ineffective, even after the in loco infusion of a fibrinolytic agent (5 mg rt-PA), leading to refractory high intracranial pressure. Endoscopic removal of the right ventricular clots was first tried, but it was necessary to perform a larger right frontal craniotomy. Intracranial pressure decreased postoperatively but the GCS remained at 3/15 with absence of brainstem reflexes. A brain magnetic resonance imaging (MRI) performed on day 20 showed the partial removal of the intraventricular clot with decreased ventricular size and reopening of the sulci. MR-angiography disclosed extensive arterial anomalies with occlusion of the right internal carotid artery (ICA), severe stenosis of the left ICA and of the M1 segment of the left middle cerebral arteries (MCA), occlusion of the left posterior cerebral artery, collaterals arising from the external carotid arteries, and an extensive abnormal vascular network appearing as multiple small collateral vessels developed mainly in basal cisterna and in the thalami (fig. 2). This pattern evoked a moyamoya syndrome that occurred very early in age as demonstrated by the hypoplastic right bony carotid canal (fig. 3). Perfusion-weighted MRI showed moderate hemodynamic disturbances in both hemispheres, predominant on the left side, with delayed time-to-peak, prolonged mean-transit time and a compensatory increased cerebral blood volume. Conventional cerebral angiography (day 39) confirmed these observations.

Bottom Line: Refractory high intracranial pressure developed and required bilateral ventricular drainage and intensive care treatment with barbiturates and hypothermia.Magnetic resonance imaging and cerebral angiography revealed a moyamoya syndrome with rupture of the abnormal collateral vascular network as the cause of the hemorrhage.Despite an initially low GCS, this patient made a good functional recovery at one year follow-up.

View Article: PubMed Central - PubMed

Affiliation: Departments of Intensive Care, Brussels, Belgium.

ABSTRACT
Intraventricular hemorrhage during pregnancy is usually followed by a poor recovery. When caused by moyamoya disease, ischemic or hemorrhagic episodes may complicate the management of high intracranial pressure. A 26-year-old Caucasian woman presented with generalized seizures and a Glasgow Coma Score (GCS) of 3 during the 36th week of pregnancy. The fetus was delivered by caesarean section. The brain CT in the mother revealed bilateral intraventricular hemorrhage, a callosal hematoma, hydrocephalus and right frontal ischemia. Refractory high intracranial pressure developed and required bilateral ventricular drainage and intensive care treatment with barbiturates and hypothermia. Magnetic resonance imaging and cerebral angiography revealed a moyamoya syndrome with rupture of the abnormal collateral vascular network as the cause of the hemorrhage. Intracranial pressure could only be controlled after the surgical removal of the clots after a large opening of the right ventricle. Despite an initially low GCS, this patient made a good functional recovery at one year follow-up. Management of refractory high intracranial pressure following moyamoya related intraventricular bleeding should require optimal removal of ventricular clots and appropriate control of cerebral hemodynamics to avoid ischemic or hemorrhagic complications.

No MeSH data available.


Related in: MedlinePlus