Neurol Sci DOI 10.1007/s10072-014-1676-x
LETTER TO THE EDITOR
Two cases of cerebral venous sinus thrombosis due to intracranial hypotension after epidural anesthesia as a diagnostic challenge Fulya Basoglu • Ayhan Koksal • Batuhan Kara • Neslihan Hatice Sutpideler Koksal • Sevim Baybas
Received: 11 January 2014 / Accepted: 5 February 2014 Ó Springer-Verlag Italia 2014
Dear Editor, Intracranial hypotension after spinal interventions is known to occur due to iatrogenic severance of the dura resulting in cerebrospinal fluid leakage. It is characterized by postural headache and it is mostly undertreated or underdiagnosed. We present two cases of cerebral venous sinus thrombosis (CVST) due to intracranial hypotension after epidural anesthesia.
Case 1 A 27-year-old female patient was admitted to our hospital with a generalized tonic–clonic seizure. She had a caesarian section delivery under epidural anesthesia with an atraumatic needle a week ago. She had been suffering from a moderate headache which started 3 days after surgery. Her headache got worse in upright position and subsided when she lied down. Her family and birth histories were unremarkable. She only had an iron-deficiency anemia. This was her third uneventful pregnancy and delivery. When she was admitted to the emergency room, she was F. Basoglu (&) A. Koksal S. Baybas Department of Neurology, Bakirkoy Research and Training Hospital for Neurology, Neurosurgery and Psychiatry, Istanbul, Turkey e-mail: [email protected] B. Kara Department of Radiology, Bakirko¨y Dr. Sadi Konuk Teaching Hospital, Istanbul, Turkey N. H. S. Koksal Department of Neurosurgery, Samatya Research and Training Hospital, Istanbul, Turkey
postictal and mildly confused and had no other neurological signs or symptoms. Contrast-enhanced cranial magnetic resonance imaging (MRI) revealed a slightly hemorrhagic venous infarction area on the left frontal lobe and bilateral dural thickening, and contrast enhancement more prominent on the left cerebral hemisphere suggesting the presence of intracranial hypotension (Fig. 1a). Cranial MRI venography confirmed partial thrombosis of the anterior part of superior sagittal sinus (Fig. 1b). There were hypochromic microcytic erythrocytes in the peripheral blood smear consistent with iron-deficiency anemia. No abnormal findings were present in the laboratory values including prothrombin and partial thromboplastin times, antithrombin III, fibrinogen level, serum glucose level, electrolytes, liver enzymes, urea, creatinine, protein, vitamin B12, folate, and homocysteine. Extensive laboratory investigations for infections, vasculitic disorders and hereditary and acquired hypercoagulable states revealed no pathological findings. The patient was diagnosed with CVST due to intracranial hypotension and was started on standard heparin infusion along with hydration and bed rest for a week followed with warfarin treatment for 6 months.
Case 2 A 28-year-old female patient, who had a term delivery under epidural anesthesia performed with an atraumatic needle 10 days ago, gradually developed headache, pain and hearing loss in the left ear a week after the operation and had been admitted to a hospital where she was diagnosed with otitis media. The headache increased when she stood up and decreased when she lied down. 2 days later
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Neurol Sci
Fig. 1 a Contrast-enhanced cranial MRI reveals bilateral dural thickening and contrast enhancement which is more prominent on the left cerebral hemisphere suggesting the presence of intracranial hypotension. b Cranial MRI venography confirms partial thrombosis in the anterior part of superior sagittal sinus
the headache lost its postural character and became more intense. On the third day, she had a generalized tonic– clonic seizure and was admitted to our emergency room. She was mildly confused without any focal neurologic deficits. She had no previous history of disease and her family history was also unremarkable. Contrast-enhanced cranial MRI confirmed cerebral venous infarction on the left temporal lobe and revealed CVST of the left transverse sinus and dural contrast enhancement (Fig. 2a). Cranial MRI venography also confirmed the diagnosis (Fig. 2b). Laboratory investigations disclosed no abnormality. EEG showed focal epileptiform activity in the temporal region of the left hemisphere. The patient received standard heparin infusion for 1 week followed by warfarin treatment for 6 months.
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Fig. 2 a Contrast-enhanced cranial MRI depicts dural contrast enhancement. b Cranial MRI venography confirms left transverse sinus thrombosis
Oxcarbazepine 600 mg/day was added to her treatment and seizures were controlled.
Discussion Lumbar puncture and epidural or peridural anesthesia with accidental dural puncture may rarely cause CVST [1]. Symptoms of CVST include focal or generalized seizures
Neurol Sci
followed by hemiparesis, aphasia, hemianopia or other focal deficits, often without signs of elevated intracranial pressure [2]. In both of our cases, change in the characteristics of headache and generalized seizures was prominent findings without any other focal neurological deficit. Postpartum state is a well-established risk factor for CVST. In our patients, pregnancy was a hypercoagulable state and in the second case otitis media and intracranial hypotension possibly worked together to cause a strong hypercoagulative state that resulted in venous thrombosis. CVST is difficult to diagnose because of the lack of a gold standard; thus, it might be overlooked [3]. Recent reports have emphasized MRI findings in intracranial hypotension [4]. In conclusion, CVST may be a diagnostic challenge after intracranial hypotension due to accidental dural damage from spinal interventions. Any change in the characteristics of a headache following lumbar puncture should be evaluated to exclude CVST. Seizures should also
warn the physician about CVST even in the absence of any focal neurological signs and symptoms suggesting raised intracranial pressure.
References 1. Aidi S, Chaunu MP, Biousse V, Bousser MG (1999) Changing pattern of headache pointing to cerebral venous thrombosis after lumbar puncture and intravenous high-dose steroids. Headache 39:559–564 2. Linn J, Bru¨ckmann H (2009) Differential diagnosis of nontraumatic intracerebral hemorrhage. Klin Neuroradiol 19:45–61 3. Boukobza M, Crassard I, Bousser MG, Chabriat H (2009) MR imaging features of isolated cortical vein thrombosis: diagnosis and follow-up. Am J Neuroradiol 30:344–348 4. Linn J, Michl S, Katja B, Pfefferkorn T, Wiesmann M, Hartz S, Dichgans M, Bru¨ckmann H (2010) Cortical vein thrombosis: the diagnostic value of different imaging modalities. Neuroradiology 52:899–911
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LETTER TO THE EDITOR
Two cases of cerebral venous sinus thrombosis due to intracranial hypotension after epidural anesthesia as a diagnostic challenge Fulya Basoglu • Ayhan Koksal • Batuhan Kara • Neslihan Hatice Sutpideler Koksal • Sevim Baybas
Received: 11 January 2014 / Accepted: 5 February 2014 Ó Springer-Verlag Italia 2014
Dear Editor, Intracranial hypotension after spinal interventions is known to occur due to iatrogenic severance of the dura resulting in cerebrospinal fluid leakage. It is characterized by postural headache and it is mostly undertreated or underdiagnosed. We present two cases of cerebral venous sinus thrombosis (CVST) due to intracranial hypotension after epidural anesthesia.
Case 1 A 27-year-old female patient was admitted to our hospital with a generalized tonic–clonic seizure. She had a caesarian section delivery under epidural anesthesia with an atraumatic needle a week ago. She had been suffering from a moderate headache which started 3 days after surgery. Her headache got worse in upright position and subsided when she lied down. Her family and birth histories were unremarkable. She only had an iron-deficiency anemia. This was her third uneventful pregnancy and delivery. When she was admitted to the emergency room, she was F. Basoglu (&) A. Koksal S. Baybas Department of Neurology, Bakirkoy Research and Training Hospital for Neurology, Neurosurgery and Psychiatry, Istanbul, Turkey e-mail: [email protected] B. Kara Department of Radiology, Bakirko¨y Dr. Sadi Konuk Teaching Hospital, Istanbul, Turkey N. H. S. Koksal Department of Neurosurgery, Samatya Research and Training Hospital, Istanbul, Turkey
postictal and mildly confused and had no other neurological signs or symptoms. Contrast-enhanced cranial magnetic resonance imaging (MRI) revealed a slightly hemorrhagic venous infarction area on the left frontal lobe and bilateral dural thickening, and contrast enhancement more prominent on the left cerebral hemisphere suggesting the presence of intracranial hypotension (Fig. 1a). Cranial MRI venography confirmed partial thrombosis of the anterior part of superior sagittal sinus (Fig. 1b). There were hypochromic microcytic erythrocytes in the peripheral blood smear consistent with iron-deficiency anemia. No abnormal findings were present in the laboratory values including prothrombin and partial thromboplastin times, antithrombin III, fibrinogen level, serum glucose level, electrolytes, liver enzymes, urea, creatinine, protein, vitamin B12, folate, and homocysteine. Extensive laboratory investigations for infections, vasculitic disorders and hereditary and acquired hypercoagulable states revealed no pathological findings. The patient was diagnosed with CVST due to intracranial hypotension and was started on standard heparin infusion along with hydration and bed rest for a week followed with warfarin treatment for 6 months.
Case 2 A 28-year-old female patient, who had a term delivery under epidural anesthesia performed with an atraumatic needle 10 days ago, gradually developed headache, pain and hearing loss in the left ear a week after the operation and had been admitted to a hospital where she was diagnosed with otitis media. The headache increased when she stood up and decreased when she lied down. 2 days later
123
Neurol Sci
Fig. 1 a Contrast-enhanced cranial MRI reveals bilateral dural thickening and contrast enhancement which is more prominent on the left cerebral hemisphere suggesting the presence of intracranial hypotension. b Cranial MRI venography confirms partial thrombosis in the anterior part of superior sagittal sinus
the headache lost its postural character and became more intense. On the third day, she had a generalized tonic– clonic seizure and was admitted to our emergency room. She was mildly confused without any focal neurologic deficits. She had no previous history of disease and her family history was also unremarkable. Contrast-enhanced cranial MRI confirmed cerebral venous infarction on the left temporal lobe and revealed CVST of the left transverse sinus and dural contrast enhancement (Fig. 2a). Cranial MRI venography also confirmed the diagnosis (Fig. 2b). Laboratory investigations disclosed no abnormality. EEG showed focal epileptiform activity in the temporal region of the left hemisphere. The patient received standard heparin infusion for 1 week followed by warfarin treatment for 6 months.
123
Fig. 2 a Contrast-enhanced cranial MRI depicts dural contrast enhancement. b Cranial MRI venography confirms left transverse sinus thrombosis
Oxcarbazepine 600 mg/day was added to her treatment and seizures were controlled.
Discussion Lumbar puncture and epidural or peridural anesthesia with accidental dural puncture may rarely cause CVST [1]. Symptoms of CVST include focal or generalized seizures
Neurol Sci
followed by hemiparesis, aphasia, hemianopia or other focal deficits, often without signs of elevated intracranial pressure [2]. In both of our cases, change in the characteristics of headache and generalized seizures was prominent findings without any other focal neurological deficit. Postpartum state is a well-established risk factor for CVST. In our patients, pregnancy was a hypercoagulable state and in the second case otitis media and intracranial hypotension possibly worked together to cause a strong hypercoagulative state that resulted in venous thrombosis. CVST is difficult to diagnose because of the lack of a gold standard; thus, it might be overlooked [3]. Recent reports have emphasized MRI findings in intracranial hypotension [4]. In conclusion, CVST may be a diagnostic challenge after intracranial hypotension due to accidental dural damage from spinal interventions. Any change in the characteristics of a headache following lumbar puncture should be evaluated to exclude CVST. Seizures should also
warn the physician about CVST even in the absence of any focal neurological signs and symptoms suggesting raised intracranial pressure.
References 1. Aidi S, Chaunu MP, Biousse V, Bousser MG (1999) Changing pattern of headache pointing to cerebral venous thrombosis after lumbar puncture and intravenous high-dose steroids. Headache 39:559–564 2. Linn J, Bru¨ckmann H (2009) Differential diagnosis of nontraumatic intracerebral hemorrhage. Klin Neuroradiol 19:45–61 3. Boukobza M, Crassard I, Bousser MG, Chabriat H (2009) MR imaging features of isolated cortical vein thrombosis: diagnosis and follow-up. Am J Neuroradiol 30:344–348 4. Linn J, Michl S, Katja B, Pfefferkorn T, Wiesmann M, Hartz S, Dichgans M, Bru¨ckmann H (2010) Cortical vein thrombosis: the diagnostic value of different imaging modalities. Neuroradiology 52:899–911
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