Novel treatment (new drug/intervention; established drug/procedure in new situation)

CASE REPORT

Intact neurological outcome after
neurointerventional treatment for intracranial venous
thrombosis with straight sinus involvement
M Sidhu,1 D Footitt,2 I Donaldson,3 S Hughes,4 S Wuppalapati,5 H C A Emsley6,7
1

Department of Clinical and
Experimental Epilepsy, UCL/
Institute of Neurology, London,
UK
2
Cumbria Partnership NHS
Foundation Trust, Cumbria, UK
3
Anaesthetics Directorate,
Royal Preston Hospital,
Preston, UK
4
Department of Obstetrics and
Gynaecology, Royal Preston
Hospital, Preston, UK
5
Department of
Neurointervention, Royal
Preston Hospital, Preston, UK
6
Department of Neurology,
Royal Preston Hospital,
Preston, UK
7
University of Manchester,
Manchester, UK
Correspondence to
Dr H C A Emsley, hedley.
emsley@manchester.ac.uk

SUMMARY
The role of neurointerventional treatment, including local
thrombolysis, for intracranial venous thrombosis (ICVT)
currently remains uncertain. We describe a case of
postpartum ICVT involving the straight sinus, with rapid
neurological deterioration and progressive thrombosis,
despite anticoagulation. She underwent multimodality
neurointerventional treatment involving attempted
mechanical clot disruption, microcatheter suction,
intrathrombus thrombolysis and microballoon disruption
and clot retrieval. The patient achieved a full clinical
recovery, with radiological evidence of recanalisation at
follow-up.

INVESTIGATIONS
BACKGROUND
The role of neurointerventional treatment, including local thrombolysis, for intracranial venous
thrombosis (ICVT) currently remains uncertain.
However, we describe a case with excellent clinical
outcome where neurointerventional treatment was
given for straight sinus thrombosis because prognosis was likely to be poor despite adequate anticoagulation (AC).

CASE PRESENTATION
A 23-year-old Asian woman with sickle cell trait, and
2 weeks postpartum, developed headache of 5-day
duration followed by rapidly evolving vomiting, dysphagia, behavioural change, reduced conscious level,
ophthalmoplegia and absent gag reflex.

To cite: Sidhu M, Footitt D,
Donaldson I, et al. BMJ
Case Rep Published online:
[please include Day Month
Year] doi:10.1136/bcr-2013010242

Her family described a preceding 5-day history
of an escalating bifrontal headache. On the
morning of her admission, she displayed bizarre
behaviour and was described to be ‘muddled’. Her
right leg was noted to be jerking intermittently.
Her initial Glasgow Coma Scale was 10 (E3, V1,
M6). She was notably confused and agitated. Pupils
were equal and reactive to light. There was a left,
6th nerve palsy. No papilloedema was seen. There
was marked sialorrhoea, symmetrical poverty of
pharyngeal elevation and an absent gag reflex.
Peripherally, she was neurologically intact with
flexor plantar responses.

The clinical suspicion of ICVT was confirmed on
her initial CT scan which showed a cord sign
(figure 1). Neurological advice was sought and she
was started on intravenous heparin and phenytoin.
Given her rapidly deteriorating neurological condition, she was sedated, intubated and transferred the
same day to our neurosciences centre, direct to the
neurointerventional suite. Given 5 h had elapsed
from initial CT, intracerebral haemorrhage (ICH)
was excluded on a CT scanogram (figure 2), but
this scan did reveal bithalamic low attenuation consistent with venous congestion. Cerebral angiography confirmed thrombus in the cerebral venous
system including the straight sinus (figure 3). The
straight sinus was not filling on the vertebral and
bilateral carotid angiograms. Thrombus was seen in
the left transverse sigmoid junction and left medial

Figure 1 Axial unenhanced CT scans acquired at presentation showing acute thrombus with internal cerebral veins
and veins of Rosenthal (A), vein of Galen and straight sinus (B) and straight sinus with cord sign (C, arrowed).

Sidhu M, et al. BMJ Case Rep 2013. doi:10.1136/bcr-2013-010242

1

Novel treatment (new drug/intervention; established drug/procedure in new situation)

Figure 4 Angiogram showing position of guidewire (tip within
straight sinus).

Figure 2 Axial scanogram acquired on a biplane neurovascular X-ray
system (Philips Allura XperCT) showing bithalamic low attenuation and
cord sign (image degraded by artefact).
transverse sinus. All the veins from thalamostriate to the internal
cerebral vein were ‘hanging in space’. The left sylvian vein
showed stasis and was not draining adequately. She remained
sedated and any further neurological assessment off sedation
would have introduced considerable delay. Coupled with her
likely poor prognosis without effective recanalisation and radiological features of progressive thrombosis, the decision was
taken to proceed with neurointerventional treatment to facilitate
recanalisation.

TREATMENT
Following guidewire insertion (Terumo Medical, Somerset, N
ew Jersey, USA; figure 4), mechanical clot disruption and withdrawal was attempted first using a Penumbra System (PS)
(Penumbra, Alameda, California, USA) followed by the use of a
Solitaire AB stent (ev3 Inc, Irvine, California, USA) with

attempted suction through microcatheter (Prowler select plus
catheter, Cordis Corporation, Bridgewater, New Jersey, USA).
Because these attempts failed to achieve recanalisation (figure
5), intrathrombus recombinant tissue plasminogen activator
(r-tPA) was administered through microcatheter. In total 5 mg
was administered over 40 mins. This was then followed by
microballoon (Eclipse 7, BALT, Montmorency, France) clot disruption/retrieval. There appeared to be partial recanalisation of
the straight sinus albeit with evidence of clot migration to the
left transverse sinus (figure 6).

OUTCOME AND FOLLOW-UP
A CT venogram 18 h postprocedure confirmed partial recanalisation (figure 7). She was successfully extubated 48 h postneurointervention, neurologically intact and was discharged home a
week later on oral AC with warfarin. CT venogram appearances
6 months later were much improved (figure 8).

DISCUSSION
ICVT is a rare but potentially lethal condition that can occur in
any age group including neonates. Estimated annual incidence is

Figure 3 (A) Pretreatment angiogram (left vertebral artery Towne view) showing filling defect within left transverse sinus, arrowed.
(B) Pretreatment angiogram (left vertebral artery lateral view) showing absence of straight sinus (arrowed, yellow) and disconnection of internal
cerebral veins, seen ‘hanging in space’ (arrowed, red). (C) Pretreatment angiogram (left internal carotid artery lateral view) showing absence of
straight sinus (dotted yellow line depicts expected position of straight sinus), disconnection of internal cerebral veins, seen ‘hanging in space’
(arrowed, red) and stagnation in Sylvian vein (arrowed, yellow).
2

Sidhu M, et al. BMJ Case Rep 2013. doi:10.1136/bcr-2013-010242

Novel treatment (new drug/intervention; established drug/procedure in new situation)

Figure 5 Angiogram showing microcatheter within straight sinus
postdeployment of solitaire stent and demonstrating extent of
remaining thrombus within sinus (yellow arrows indicate inferior and
superior margins); contrast seen more prominently towards inferior
margin corresponding to area of greatest mechanical clot disruption.
Septation of thrombus can also be seen (black arrows).

3 –4 cases per 1 million population and up to 7 cases per
million among children. About 75% of the adult patients are
women.1 ICVT accounts for <0.5% of cases of all strokes.
Causes of ICVT are extremely variable. In the majority of
patients, a factor predisposing to thrombosis can be identified.
The greater incidence in women appears to be due to risk
factors such as pregnancy, puerperium, the oral contraceptive
pill and hormone replacement therapy. Other predisposing
factors include infections (eg mastoiditis/sinusitis), hypercoagulable states, inherited prothrombotic tendencies, blood dyscrasias
and malignancy. Thromboembolic complications are exceedingly
rare in patients with sickle cell trait and nevertheless ICVT has
previously been described in this setting.2
Clinical features are varied, including headache (95%), other
features of raised intracranial pressure such as vomiting, seizures
(47%), altered behaviour, decline in conscious level (39%),
papilloedema (43%) and cranial nerve palsies.3 Seizures are generally focal with secondary generalisation and are more commonly seen than in arterial stroke. If the thalami are affected,
there may be behavioural symptoms (eg delirium, amnesia and
mutism). Venous congestion can lead to venous infarction with
or without haemorrhage, cerebral oedema, herniation and
death. In a study of 54 patients, poor outcome at 12-week was
associated with patients presenting with papilloedema, coma or
altered consciousness, age >33 years, diagnostic delay
>10 days, ICH and involvement of the straight sinus.3
Sidhu M, et al. BMJ Case Rep 2013. doi:10.1136/bcr-2013-010242

Figure 6 (A) Vertebral angiogram (venous phase) post-thrombolysis
showing outline of straight sinus (arrowed). (B) Vertebral angiogram
(venous phase) (Towne view) showing partial recanalisation with
migration of thrombus within left transverse sinus (arrowed).

Figure 7 CT venogram 18 h post-treatment showing partial
recanalisation of straight sinus (box indicates area of detail shown with
inverted colours).
3

Novel treatment (new drug/intervention; established drug/procedure in new situation)

Figure 8 CT venogram 6 months post-treatment showing complete recanalisation of the straight sinus with antegrade flow from the deep veins,
and partial recanalisation of the left transverse sinus.

The rationale for treatment is to limit thrombus extension
and promote recanalisation. Treatment with AC in the form of
subcutaneous low molecular weight heparin or intravenous
heparin is widely used.4–6 Thrombolysis with urokinase was first
used in 1971 with encouraging results.7 More recently, r-tPA in
combination with heparin has been used due its many theoretical advantages over urokinase: clot selectivity, a short plasma
half-life of approximately 5 min and low levels of fibrinogen
degradation products. Although there have been promising case
reports, there are no randomised controlled trials supporting
the use of either local or systemic thrombolysis. Frey et al
reported a series of 12 patients who received r-tPA at a loading
dose of 1 mg/cm as a bolus through a microcatheter along the
clot followed by 1–2 mg/h for a mean duration of 29 h delivering a mean dose of 46 mg. Partial or complete flow restoration
was seen in nine patients. Recanalisation rates were superior as
compared to treatment with heparin alone. Importantly,
however, two patients with obvious haemorrhagic infarctions
developed haemorrhage extension on the initial doses of treatment.8 In a separate study, Kim et al used a more aggressive
dosing regime that included repeated boluses plus infusion
(average dose of 134 mg). Although flow was restored in all (9
patients), major intrapelvic bleeding and minor bleeding from
femoral puncture sites were noted.9 The European Federation
of Neurological Societies guidelines conclude that thrombolysis
may be beneficial in patients without ICH or impending herniation from large haemorrhagic infarcts.6
We used a similar bolus to that described by Frey et al of
1 mg/cm along the length of the clot at intervals of approximately 8 min. Various combinations of endovascular techniques
have previously been combined with chemical thrombolysis.10 11
Of these reported cases only two patients had straight sinus
involvement, both requiring more than one procedure (1 case
having 5 procedures had very poor outcome).10 Recently, the PS
was successfully used to treat four cases of ICVT without need
for local thrombolysis, although it appears that the straight sinus
was not involved in any of these patients.12 Owing to logistical
considerations, it would not have been possible to safely administer a continuous infusion of r-tPA as used by previous groups.

4

As the angiogram showed recanalisation of the straight sinus
post-treatment, we decided to follow-up with intravenous
heparin treatment only with an excellent clinical outcome.

Learning points
▸ Intracranial venous thrombosis (ICVT) is rare but potentially
lethal and rapid, accurate clinicoradiological diagnosis is
crucial.
▸ Anticoagulation remains the first-line treatment but
interventional treatment should be considered for continued
neurological deterioration despite adequate
anticoagulation.13
▸ Interventional treatment can facilitate an excellent clinical
outcome, including ICVT involving the straight sinus.
▸ Multiple interventional treatment modalities may be required
during the treatment of a single case.

Contributors MS and HCAE drafted the manuscript; all authors contributed to the
content and approved the final version.
Competing interests None.
Patient consent Obtained.
Provenance and peer review Not commissioned; externally peer reviewed.

REFERENCES
1
2
3

4
5

Stam J. Thrombosis of the cerebral veins and sinuses. N Engl J Med
2005;352:1791–8.
Feldenzer JA, Bueche MJ, Venes JL, et al. Superior sagittal sinus thrombosis with
infarction in sickle cell trait. Stroke 1987;18:656–60.
de Bruin S, de Haan R, Stam J. Clinical features and prognostic factors of cerebral
venous sinus thrombosis in a prospective series of 59 patients. J Neurol Neurosurg
Psychiatry 2001;70:105–8.
Einhäupl KM, Villringer A, Meister W, et al. Heparin treatment in sinus venous
thrombosis. Lancet 1991;338:597–600.
Stam J, de Bruijn S, deVeber G. Anticoagulation for cerebral sinus thrombosis.
Stroke 2003;34:1054–5.

Sidhu M, et al. BMJ Case Rep 2013. doi:10.1136/bcr-2013-010242

Novel treatment (new drug/intervention; established drug/procedure in new situation)
6

7
8
9

10

Einhäupl K, Stam J, Bousser MG, et al. EFNS guideline on the treatment of cerebral
venous and sinus thrombosis in adult patients. Eur J Neurol 2010. [Epub ahead of
print].
Vines FS, Davis DO. Clinical and radiological correlation in cerebral venous occlusive
disease. Radiology 1971;98:9–22.
Frey JL, Muro GJ, McDougall CG, et al. Cerebral venous thrombosis: combined
intra-thrombus rtPA and intravenous heparin. Stroke 1999;30:489–94.
Kim SY, Suh JH. Direct endovascular thrombolytic therapy for dural sinus
thrombosis: infusion of alteplase. AJNR Am J Neuroradiol 1997;18:
639–45.
Baker MD, Opatowsky MJ, Wilson JA, et al. Rheolytic catheter and thrombolysis of
dural venous sinus thrombosis: a case series. Neurosurgery 2001;48:487–93.

11

12

13

Chaloupka JC, Mangla S, Huddle DC. Use of mechanical thrombolysis via
microballoon percutaneous transluminal angioplasty for the treatment of acute dural
sinus thrombosis: case presentation and technical report. Neurosurgery
1999;45:650–6.
Choulakian A, Alexander MJ. Mechanical thrombectomy with the
penumbra system for treatment of venous sinus thrombosis. J Neurointerv Surg
2010;2:153–6.
Saposnik G, Barinagarrementeria F, Brown RD Jr, et al. American Heart Association
Stroke Council and the Council on Epidemiology and Prevention. Diagnosis and
management of cerebral venous thrombosis: a statement for healthcare
professionals from the American Heart Association/American Stroke Association.
Stroke 2011;42:1158–92.

Copyright 2013 BMJ Publishing Group. All rights reserved. For permission to reuse any of this content visit
http://group.bmj.com/group/rights-licensing/permissions.
BMJ Case Report Fellows may re-use this article for personal use and teaching without any further permission.
Become a Fellow of BMJ Case Reports today and you can:
▸ Submit as many cases as you like
▸ Enjoy fast sympathetic peer review and rapid publication of accepted articles
▸ Access all the published articles
▸ Re-use any of the published material for personal use and teaching without further permission
For information on Institutional Fellowships contact consortiasales@bmjgroup.com
Visit casereports.bmj.com for more articles like this and to become a Fellow

Sidhu M, et al. BMJ Case Rep 2013. doi:10.1136/bcr-2013-010242

5