Clin Neuroradiol DOI 10.1007/s00062-014-0338-y

Correspondence

An acute cerebral venous sinus thrombosis: successful treatment by combining mechanical thrombolysis with continuous urokinase infusion S.-Y. Pan · T.-H. Tsai · W.-H. Chen · C.-C. Shen · Y.-S. Tsuei

Received: 10 February 2014 / Accepted: 13 August 2014 © Springer-Verlag Berlin Heidelberg 2014

Introduction Cerebral venous sinus thrombosis (CVST) is a rare disease with an estimated incidence of five per million, accounting for 0.5 % of all strokes [1]. The International Study on Cerebral Vein and Dural Sinus Thrombosis meta-analysis of several prospective studies describes a 15 % overall death or dependency rate with a 4 % death rate in the acute phase [2]. For severe and long-segment thrombos cases, recanalization with good persistent venous drainage is still challenging. We present a case successfully treated by combined intravenous mechanical thrombolysis with post-op continuous local urokinase infusion. Case presentation A female with inflammatory bowel disease had initial chief complaint of abdominal cramping pain and recurrent bloody diarrhea for 3 days. During hospitalization, she had progressive headache with acute onset of epilepsia partialis conti-

Y.-S. Tsuei, MD () · S.-Y. Pan, MD · C.-C. Shen, MD Department of Neurosurgery, Taichung Veterans General Hospital, No. 160, Sec. 3, Chung-Kang Rd., Taichung, Taiwan e-mail: [email protected] W.-H. Chen, MD Department of Neuroradiology, Taichung Veterans General Hospital, Taichung, Taiwan T.-H. Tsai, MD · C.-C. Shen, MD · Y.-S. Tsuei, MD Department of Neurosurgery, Triservice General Hospital, National Defense Medical Center, Taichung, Taiwan

nua and weakness of left limbs. Other symptoms included nausea, vomiting, photophobia, and phonophobia. Brain computed tomography (CT) with CT angiography revealed high attenuation change with filling defect at the right internal jugular vein, right transverse sinus, and superior sagittal sinus. Magnetic resonance angiography of brain disclosed engorged cortical veins at right temporo-parietooccipital lobe and left frontoparietal lobe. Thrombosis of superior sagittal sinus, right transverse sinus, right sigmoid sinus, and right internal jugular vein were noted (Fig. 1a and b). Anticoagulant treatment was initiated but failed with rapid progression symptoms after 1 day. Glasgow Coma Scale (GCS) decreased from 15 to 11–12. The patient was immediately transferred to the angiography suite where cerebral angiography and venography studies disclosed sinus thrombosis with venous hypertension (Fig. 1c and d). Multi-modality endovascular thrombolysis was applied. Intravenous local mechanical thrombolysis was performed with a large-bore guiding catheter and balloon to break the clot. Using the NEURON Intracranial Access System (Penumbra, San Leandro, CA) as a large-bore guide catheter, direct aspiration for clot disruption was performed as far as the distal sagittal sinus by Sterling monorail balloon catheter (Boston Scientific, Boston, MT) (Fig. 2a and b). At the same time, urokinase (600,000 IU) was infused to dissolve the venous thrombi. After partial recanalization by mechanical thrombolysis, the guiding catheter was left inside the superior sagittal sinus for continuous local urokinase infusion (15,000 IU/h) for 48 h. We removed the catheter and repeated cerebral angiography and venography later. The final follow-up angiogram (Fig. 2c and d) showed patency of the sinuses. She was discharged without residual neurologic deficit and was prescribed warfarin orally for 6 months. Neurologic outcomes, GCS, and neuroimaging results were improved.

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Fig. 1  Brain computed tomography showed high attenuation change at right transverse sinus with filling defect (a). 4D magnetic resonance angiography coronal images revealed thrombosis of superior sagittal sinus, right transverse sinus, right sigmoid sinus, and right internal jugular vein (b). Digital subtraction angiography (DSA) posterior-anterior (PA) view showed long-segment sinus thrombosis from right internal jugular vein to superior sagittal sinus (c). DSA posterioranterior (PA) view with contrast injection from the catheter inside the jugular vein revealed the cutpoint of the filling defect (d)

Discussion There are several predisposing conditions that may lead to the development of CVST. These include medical or surgical causes of deep vein thrombosis, prothrombic disorders, systemic inflammatory disorders, oral contraceptives, pregnancy, puerperium, steroids, cancer, trauma and procedures, such as surgery, lumbar puncture, and jugular catheter placements [2]. A clear correlation between inflammatory bowel disease and increased risk of thrombosis was found in the previous study [3]. The goals of CVST therapy are to re-establish circulation distal to the occlusion, prevent further development of thrombus, treat the underlying cause, and prevent recurrence. Although anticoagulation therapy is the most widely accepted therapeutic option for CVST, more aggressive intervention with endovascular administration of thrombolytics and/or thrombectomy may be indicated if there is clinical deterioration despite use of anticoagulation therapy [4]. The endovascular treatment of CVST includes pharmacological thrombolysis and mechanical thrombectomy. Scott et al. [5] were the first to describe the use of local

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thrombolysis treatment for sagittal sinus thrombosis refractory to systemic anticoagulation by direct administration of urokinase. Horowitz et al. [6] published their protocol for the treatment of refractory sinus thrombosis using mechanical thrombolysis along with intrasinus administration of thrombolytic medication. There is accumulating evidence that local thrombolytic treatment of CVST, often combined with mechanical techniques, yields positive results. In combined pharmacological and mechanical thrombectomy, the dose of the thrombolytic drug and duration of infusion can be decreased because of the increased surface area of the thrombus exposed to the drug [7]. For extensive and long-segment CVST, the optimal treatment is still unclear and no large-scale trial comparing therapeutic options exists. Nimjee et al. [8] presented a similar case that underwent both Angiojet thrombolysis and mechanical thrombectomy with a balloon catheter. Pukenas et al. [9] described a novel technique using the Solitaire FR device which was more easily delivered and deployed within the thrombosed sigmoid and transverse sinus segments for stent retrieval with rapid flow restoration. Here, we present another multimodality endovascular approach.

An acute cerebral venous sinus thrombosis

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Fig. 2  Percutaneous transluminal angioplasty (PTA) was performed up to distal superior sagittal sinus (a). A large-bore guide catheter was placed into distal superior sagittal sinus after PTA with partial recanalization (b). After mechanical thrombolysis with post-op continuous local urokinase infusion, DSA lateral and posterior-anterior (PA) view (c and d) revealed the superior sagittal sinus, right transverse sinus, right sigmoid sinus, and right internal jugular vein were recanalized with good venous flow

In our case, a combination of a large-bore guide catheter, balloon angioplasty, and direct plus post-op continuous local urokinase infusion with thrombolysis led to effective removal of thrombus and rapid recanalization of the superior sagittal, transverse, and sigmoid sinuses. NEURON Intracranial Access System is a new guide catheter with a soft, flexible tip of variable length, a largebore, and a strong platform, allowing placement of the guide catheter further into the intracranial vessels. Previously, approach via tortuous cervical or intracranial vessels was too difficult. This new guide catheter has been employed in several cases of tortuous intracranial vascular anatomy, providing a steady platform [10]. Although the benefit of NEURON catheters in endovascular navigation of tortuous vessels has been published, the data are currently limited to intracranial arteries. We present the first report of successful thrombosis treatment via the intracranial venous approach using a NEURON large-bore guide catheter. We advanced the large-bore guiding catheter to the distal sagittal sinus, almost to the anterior third for the greater clot disruption. Although intravenous therapy with mechanical and medical thrombolysis may rapidly recanalize the main sinuses,

it is still a challenge to maintain the venous flow especially for extensive CVST. A longer time, local infusion of thrombolytics at a lower concentration theoretically increases the probability of drug contact with the clot. In our case, the NEURON guiding catheter was left inside the superior sagittal sinus for continuous local urokinase infusion (15,000 IU/h) for 48 h after the endovascular thrombolysis procedure. Follow-up angiography revealed neither development of venous thrombus nor recurrence. At the moment, mechanical recanalization in combination with the above procedure may be a good alternative to the selected patients with clinical deterioration despite standard treatment with anticoagulation. However, the primary limitation of the report is a single-case report. Therefore, further research is needed to confirm the adequate urokinase dose and the continuous local infusion time in the future. References   1. Bousser MG, Ferro JM. Cerebral venous thrombosis: an update. Lancet Neurol. 2007;6(2):162–70.

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4   2. Ferro JM, Canhão P, Stam J, Bousser MG, Barinagarrementeria F; ISCVT Investigators. Prognosis of cerebral vein and dural sinus thrombosis: results of the International Study on Cerebral Vein and Dural Sinus Thrombosis (ISCVT). Stroke. 2004;35(3):664–70.   3. Kao A, Dlugos D, Hunter JV, Mamula P, Thorarensen O. Anticoagulation therapy in cerebral sinovenous thrombosis and ulcerative colitis in children. J Child Neurol. 2002;17(7):479–82.  4. Chow K, Gobin YP, Saver J, Kidwell C, Dong P, Viñuela F. Endovascular treatment of dural sinus thrombosis with rheolytic thrombectomy and intra-arterial thrombolysis. Stroke. 2000;31(6):1420–5.   5. Scott JA, Pascuzzi RM, Hall PV, Becker GJ. Treatment of dural sinus thrombosis with local urokinase infusion. Case report. J Neurosurg. 1988;68(2):284–7.   6. Horowitz M, Purdy P, Unwin H, Carstens G 3rd, Greenlee R, Hise J, Kopitnik T, Batjer H, Rollins N, Samson D. Treatment of dural sinus thrombosis using selective catheterization and urokinase. Ann Neurol. 1995;38(1):58–67.

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S.-Y. Pan et al.   7. Bagley LJ, Hurst RW, Galetta S, Teener J, Sinson GP. Use of a microsnare to aid direct thrombolytic therapy of dural sinus thrombosis. AJR Am J Roentgenol. 1998;170(3):784–6.   8. Nimjee SM, Powers CJ, Kolls BJ, Smith T, Britz GW, Zomorodi AR. Endovascular treatment of venous sinus thrombosis: a case report and review of the literature. J Neurointerv Surg. 2011;3(1):30–3.   9. Pukenas BA, Kumar M, Stiefel M, Smith M, Hurst R. Solitaire FR device for treatment of dural sinus thrombosis. J Neurointerv Surg. 2014;6(1):e2. 10. Simon SD, Ulm AJ, Russo A, Albanese E, Mericle RA. Distal intracranial catheterization of patients with tortuous vascular anatomy using a new hybrid guide catheter. Surg Neurol. 2009;72(6):737–40.

An acute cerebral venous sinus thrombosis: successful treatment by combining mechanical thrombolysis with continuous urokinase infusion.

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