Journal of Clinical Neuroscience xxx (2017) xxx–xxx

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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: jerome2008@163.com (J.-y. Wang), shczsjwk11@126.com
(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).

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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