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Cerebral venous sinus thrombosis complicated with acute development of dural arteriovenous fistula: A case report
Journal of Clinical Neuroscience xxx (2017) xxx–xxx
Contents lists available at ScienceDirect
Journal of Clinical Neuroscience journal homepage: www.elsevier.com/locate/jocn
Case report
Cerebral venous sinus thrombosis complicated with acute development of dural arteriovenous fistula: A case report Ji-gang Chen 1, Zhen-xing Li 1, Dan-feng Zhang 1, Jun-yu Wang ⇑, Li-jun Hou ⇑ Department of Neurosurgery, Shanghai Neurosurgical Institute, Changzheng Hospital, Shanghai, China
a r t i c l e
i n f o
Article history: Received 9 January 2017 Accepted 8 June 2017 Available online xxxx Keywords: Cerebral venous sinus thrombosis Dural arteriovenous fistula Todd’s paralysis
a b s t r a c t Development of dural arteriovenous fistula (dAVF) after cerebral venous sinus thrombosis (CVST) was very uncommon and for all these reported cases, the dAVF was a chronic complication. We present a case of acute development of dAVF after CVST. A 40-year-old female was admitted into our department with 2 day’s headache and vomiting for 9 h. Head computed tomography (CT) scan showed only scattered minor hematomas over the right frontal lobe. Blood test indicated an elevated D-dimer. The patient experienced transient paralysis (Todd’s paralysis) after intermittent focal epilepsy from day 3, which progressed into sustained epilepsy on day 6. Magnetic resonance imaging (MRI) on day 7 confirmed the thrombosis of the superior sagittal sinus and a large area of infarction and edema in the left frontal and parietal lobe. She was then treated with heparin and warfarin. Cerebral angiography on day 9 demonstrated a dAVF which was classified as Borden Type II and fed by the left occipital artery. Subsequently, endovascular occlusion of the fistula was conducted and the patient recovered well with only slight right limbs weakness at 1 year follow up. Ó 2017 Elsevier Ltd. All rights reserved.
1. Introduction Cerebral venous sinus thrombosis (CVST) is a rare form of venous thromboembolism with an estimated annual incidence of 3–7/1,000,000 among populations [1]. The development of dural arteriovenous fistula (dAVF) after CVST is rare and have been reported for only several cases [2,3]. Moreover, in all these cases, dAVF was presented as the chronic complication of CVST, which developed several months or years later. In this study, a case of acute development of dAVF after CVST was reported. 2. Case presentation A 40-year-old female was admitted with 2 day’s of headache and vomiting for 9 h. She also complained intermittent convulsion over right cheek. Physical examination was unremarkable. A noncontrast head computed tomography (CT) scan on admission showed only scattered hemorrhages over right frontal lobe (Fig. 1A). Findings of other tests, including haematology and coagulation profile, were normal and D-dimer level was significantly ⇑ Corresponding authors. E-mail addresses: [email protected] (J.-y. Wang), [email protected] (L.-j. Hou). 1 These authors contributed equally to this work and should be considered co-first authors.
elevated (>20,000 mg/L). She was previously healthy other than gallstones treated with laparoscopic cholecystectomy 15 days ago. Regular treatment including anti-epilepsy therapy and hemostatic was performed. The patient experienced transient paralysis (Todd’s paralysis) over right limbs after she was struck by focal epilepsy from day 3. The intermittent focal epilepsy evolved into sustained epilepsy and this patient fell into sedative state gradually with the decreased myodynamia in right limbs on day 6 after the using of large dose of diazepam and sodium valproate. Enhanced brain magnetic resonance imaging (MRI) on day 7 confirmed thrombosis of the superior sagittal sinus (Fig. 1B and C), as well as a large area of infarction and edema in the left frontal and parietal lobe (Fig. 1D). She was then treated with low-molecular-weight heparin injection and oral warfarin tablets. The cerebral angiography performed on day 9 revealed an inadequate drainage over the superior sagittal sinus (Fig. 2A). What’s more, a dAVF classified as Borden Type II next to left transverse sinus was found. It’s fed by the left occipital artery and drained into the sphenoparietal sinus with cortical reflux (Fig. 2B). We performed an endovascular occlusion of the fistula with liquid embolic agent and complete occlusion was achieved. The post-operative course was uneventful. She recovered gradually with improved consciousness and myodynamia. Repeated cerebral angiography suggested a complete occlusion of the dAVF and relief of thrombosis (Fig. 2C and D). She was
http://dx.doi.org/10.1016/j.jocn.2017.06.011 0967-5868/Ó 2017 Elsevier Ltd. All rights reserved.
Please cite this article in press as: Chen J-g et al. Cerebral venous sinus thrombosis complicated with acute development of dural arteriovenous fistula: A case report. J Clin Neurosci (2017), http://dx.doi.org/10.1016/j.jocn.2017.06.011
2
Case report / Journal of Clinical Neuroscience xxx (2017) xxx–xxx
discharged to rehabilitation center 24 days later and follow up at 1 year indicated good recovery with only slight right limbs weakness. 3. Discussion
Fig. 1. Head CT scan shows scattered minor hemorrhages on the right frontal lobe (A). Enhanced brain MRI demonstrates the thrombosis of the superior sagittal sinus and a large area of infarction and edema in the left frontal and parietal lobe is confirmed (D).
Despite constant investigation about relationships between dAVF and CVST, whether CVST is the cause or result of dAVF [4] remains elusive. In our report, it’s difficult to decide dAVF was primary or not without baseline cerebral angiography. However, according to patient’s initial head CT scan, only scattered hemorrhages on right frontal lobe was detected, which could be hardly associated with dAVF on left side of brain. Thus, CVST was considered as the trigger or at least the aggravating factor for the development of dAVF. Different mechanisms about the pathogenesis of dAVF have been proposed. Some studies reported that it’s due to the opening of pre-existing physiological shunts between meningeal arterial networks and dural venous sinus because of CVST induced venous hypertension [5,6]. Other studies considered the essential role of myogenic vasoregulation of dural vascular and sinus thrombus organization in the development of dAVF [7,8]. However, these mechanisms need further confirmation. Nearly 1/3 of patients with CVST would have seizures as the presenting feature and among patients with seizures [9], Todd’s paralysis occured in half of all [10]. Yarnell [11] postulated that Todd’s paralysis was owing to cortical ischemia and arteriovenous shunts. In this way, cerebrovascular diseases could lead to a higher rate of Todd’s paralysis. In our report, seizures couldn’t be controlled well under anti-epilepsy drug until occlusion of dAVF and relief of CVST. Thus, cerebrovascular diseases should be suspected for patients of unclear diagnosis with intractable epilepsy and Todd paralysis. References
Fig. 2. Cerebral angiography presents an inadequate drainage over the superior sagittal sinus (A) and a dAVF fed by the left occipital artery is discovered. This dAVF drains into the sphenoparietal sinus (B). Repeated cerebral angiography indicates a complete occlusion of the dAVF and resolving of thrombosis (C and D).
[1] Stam J. Thrombosis of the cerebral veins and sinus. N Engl J Med 2005;352:1791–8. [2] Morales H, Jones BV, Leach JL, et al. Documented development of a dural arteriovenous fistula in an infant subsequent to sinus thrombosis: case report and review of the literature. Neuroradiology 2010;52:225–9. [3] Micieli JA, Derkatch S, Pereira VM, et al. Development of dural arteriovenous fistulas after cerebral venous sinus thrombosis. J Neuroophthalmol 2016;36:53–7. [4] Nishijima M, Takaku A, Endo S, et al. Etiological evaluation of dural arteriovenous malformations of the lateral and sigmoid sinuses based on histopathological examinations. J Neurosurg 1992;76:600–6. [5] Uranishi R, Nakase H, Sakaki T. Expression of angiogenic growth factors in dural arteriovenous fistula. J Neurosurg 1999;91:781–6. [6] Herman JM, Spetzler RF, Bederson JB, et al. Genesis of a dural arteriovenous malformation in a rat model. J Neurosurg 1995;83:539–45. [7] Mironov A. Pathogenetical consideration of spontaneous dural arterio-venous fistulas (DAVFS). Acta Neurochir 1994;131:45–8. [8] Terada T, Higashida RT. Development of aquired arteriovenous fistula in rats due to venous hypertension. J Neurosurg 1994;80:8884–9. [9] Kalita J, Chandra S, Misra UK. Significance of seizure in cerebral venous sinus thrombosis. Seizure 2012;21:639–42. [10] Masuhr F, Busch M, Amberger N, et al. Risk and predictors of early epileptic seizures in acute cerebral venous and sinus thrombosis. Eur J Neurol 2006;13:852–6. [11] Yarnell PR. Todd’s paralysis: a cerebrovascular phenomenon? Stroke 1975;6:301–3.
Please cite this article in press as: Chen J-g et al. Cerebral venous sinus thrombosis complicated with acute development of dural arteriovenous fistula: A case report. J Clin Neurosci (2017), http://dx.doi.org/10.1016/j.jocn.2017.06.011
Contents lists available at ScienceDirect
Journal of Clinical Neuroscience journal homepage: www.elsevier.com/locate/jocn
Case report
Cerebral venous sinus thrombosis complicated with acute development of dural arteriovenous fistula: A case report Ji-gang Chen 1, Zhen-xing Li 1, Dan-feng Zhang 1, Jun-yu Wang ⇑, Li-jun Hou ⇑ Department of Neurosurgery, Shanghai Neurosurgical Institute, Changzheng Hospital, Shanghai, China
a r t i c l e
i n f o
Article history: Received 9 January 2017 Accepted 8 June 2017 Available online xxxx Keywords: Cerebral venous sinus thrombosis Dural arteriovenous fistula Todd’s paralysis
a b s t r a c t Development of dural arteriovenous fistula (dAVF) after cerebral venous sinus thrombosis (CVST) was very uncommon and for all these reported cases, the dAVF was a chronic complication. We present a case of acute development of dAVF after CVST. A 40-year-old female was admitted into our department with 2 day’s headache and vomiting for 9 h. Head computed tomography (CT) scan showed only scattered minor hematomas over the right frontal lobe. Blood test indicated an elevated D-dimer. The patient experienced transient paralysis (Todd’s paralysis) after intermittent focal epilepsy from day 3, which progressed into sustained epilepsy on day 6. Magnetic resonance imaging (MRI) on day 7 confirmed the thrombosis of the superior sagittal sinus and a large area of infarction and edema in the left frontal and parietal lobe. She was then treated with heparin and warfarin. Cerebral angiography on day 9 demonstrated a dAVF which was classified as Borden Type II and fed by the left occipital artery. Subsequently, endovascular occlusion of the fistula was conducted and the patient recovered well with only slight right limbs weakness at 1 year follow up. Ó 2017 Elsevier Ltd. All rights reserved.
1. Introduction Cerebral venous sinus thrombosis (CVST) is a rare form of venous thromboembolism with an estimated annual incidence of 3–7/1,000,000 among populations [1]. The development of dural arteriovenous fistula (dAVF) after CVST is rare and have been reported for only several cases [2,3]. Moreover, in all these cases, dAVF was presented as the chronic complication of CVST, which developed several months or years later. In this study, a case of acute development of dAVF after CVST was reported. 2. Case presentation A 40-year-old female was admitted with 2 day’s of headache and vomiting for 9 h. She also complained intermittent convulsion over right cheek. Physical examination was unremarkable. A noncontrast head computed tomography (CT) scan on admission showed only scattered hemorrhages over right frontal lobe (Fig. 1A). Findings of other tests, including haematology and coagulation profile, were normal and D-dimer level was significantly ⇑ Corresponding authors. E-mail addresses: [email protected] (J.-y. Wang), [email protected] (L.-j. Hou). 1 These authors contributed equally to this work and should be considered co-first authors.
elevated (>20,000 mg/L). She was previously healthy other than gallstones treated with laparoscopic cholecystectomy 15 days ago. Regular treatment including anti-epilepsy therapy and hemostatic was performed. The patient experienced transient paralysis (Todd’s paralysis) over right limbs after she was struck by focal epilepsy from day 3. The intermittent focal epilepsy evolved into sustained epilepsy and this patient fell into sedative state gradually with the decreased myodynamia in right limbs on day 6 after the using of large dose of diazepam and sodium valproate. Enhanced brain magnetic resonance imaging (MRI) on day 7 confirmed thrombosis of the superior sagittal sinus (Fig. 1B and C), as well as a large area of infarction and edema in the left frontal and parietal lobe (Fig. 1D). She was then treated with low-molecular-weight heparin injection and oral warfarin tablets. The cerebral angiography performed on day 9 revealed an inadequate drainage over the superior sagittal sinus (Fig. 2A). What’s more, a dAVF classified as Borden Type II next to left transverse sinus was found. It’s fed by the left occipital artery and drained into the sphenoparietal sinus with cortical reflux (Fig. 2B). We performed an endovascular occlusion of the fistula with liquid embolic agent and complete occlusion was achieved. The post-operative course was uneventful. She recovered gradually with improved consciousness and myodynamia. Repeated cerebral angiography suggested a complete occlusion of the dAVF and relief of thrombosis (Fig. 2C and D). She was
http://dx.doi.org/10.1016/j.jocn.2017.06.011 0967-5868/Ó 2017 Elsevier Ltd. All rights reserved.
Please cite this article in press as: Chen J-g et al. Cerebral venous sinus thrombosis complicated with acute development of dural arteriovenous fistula: A case report. J Clin Neurosci (2017), http://dx.doi.org/10.1016/j.jocn.2017.06.011
2
Case report / Journal of Clinical Neuroscience xxx (2017) xxx–xxx
discharged to rehabilitation center 24 days later and follow up at 1 year indicated good recovery with only slight right limbs weakness. 3. Discussion
Fig. 1. Head CT scan shows scattered minor hemorrhages on the right frontal lobe (A). Enhanced brain MRI demonstrates the thrombosis of the superior sagittal sinus and a large area of infarction and edema in the left frontal and parietal lobe is confirmed (D).
Despite constant investigation about relationships between dAVF and CVST, whether CVST is the cause or result of dAVF [4] remains elusive. In our report, it’s difficult to decide dAVF was primary or not without baseline cerebral angiography. However, according to patient’s initial head CT scan, only scattered hemorrhages on right frontal lobe was detected, which could be hardly associated with dAVF on left side of brain. Thus, CVST was considered as the trigger or at least the aggravating factor for the development of dAVF. Different mechanisms about the pathogenesis of dAVF have been proposed. Some studies reported that it’s due to the opening of pre-existing physiological shunts between meningeal arterial networks and dural venous sinus because of CVST induced venous hypertension [5,6]. Other studies considered the essential role of myogenic vasoregulation of dural vascular and sinus thrombus organization in the development of dAVF [7,8]. However, these mechanisms need further confirmation. Nearly 1/3 of patients with CVST would have seizures as the presenting feature and among patients with seizures [9], Todd’s paralysis occured in half of all [10]. Yarnell [11] postulated that Todd’s paralysis was owing to cortical ischemia and arteriovenous shunts. In this way, cerebrovascular diseases could lead to a higher rate of Todd’s paralysis. In our report, seizures couldn’t be controlled well under anti-epilepsy drug until occlusion of dAVF and relief of CVST. Thus, cerebrovascular diseases should be suspected for patients of unclear diagnosis with intractable epilepsy and Todd paralysis. References
Fig. 2. Cerebral angiography presents an inadequate drainage over the superior sagittal sinus (A) and a dAVF fed by the left occipital artery is discovered. This dAVF drains into the sphenoparietal sinus (B). Repeated cerebral angiography indicates a complete occlusion of the dAVF and resolving of thrombosis (C and D).
[1] Stam J. Thrombosis of the cerebral veins and sinus. N Engl J Med 2005;352:1791–8. [2] Morales H, Jones BV, Leach JL, et al. Documented development of a dural arteriovenous fistula in an infant subsequent to sinus thrombosis: case report and review of the literature. Neuroradiology 2010;52:225–9. [3] Micieli JA, Derkatch S, Pereira VM, et al. Development of dural arteriovenous fistulas after cerebral venous sinus thrombosis. J Neuroophthalmol 2016;36:53–7. [4] Nishijima M, Takaku A, Endo S, et al. Etiological evaluation of dural arteriovenous malformations of the lateral and sigmoid sinuses based on histopathological examinations. J Neurosurg 1992;76:600–6. [5] Uranishi R, Nakase H, Sakaki T. Expression of angiogenic growth factors in dural arteriovenous fistula. J Neurosurg 1999;91:781–6. [6] Herman JM, Spetzler RF, Bederson JB, et al. Genesis of a dural arteriovenous malformation in a rat model. J Neurosurg 1995;83:539–45. [7] Mironov A. Pathogenetical consideration of spontaneous dural arterio-venous fistulas (DAVFS). Acta Neurochir 1994;131:45–8. [8] Terada T, Higashida RT. Development of aquired arteriovenous fistula in rats due to venous hypertension. J Neurosurg 1994;80:8884–9. [9] Kalita J, Chandra S, Misra UK. Significance of seizure in cerebral venous sinus thrombosis. Seizure 2012;21:639–42. [10] Masuhr F, Busch M, Amberger N, et al. Risk and predictors of early epileptic seizures in acute cerebral venous and sinus thrombosis. Eur J Neurol 2006;13:852–6. [11] Yarnell PR. Todd’s paralysis: a cerebrovascular phenomenon? Stroke 1975;6:301–3.
Please cite this article in press as: Chen J-g et al. Cerebral venous sinus thrombosis complicated with acute development of dural arteriovenous fistula: A case report. J Clin Neurosci (2017), http://dx.doi.org/10.1016/j.jocn.2017.06.011