A Patient with Severe Cerebral Sinus Thrombosis in Whom Mechanical Thrombolysis with a Balloon and Thrombectomy with a Stent Retriever Were Effective

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1 DOI: /jnet.cr Patient with Severe Cerebral Sinus Thrombosis in Whom Mechanical Thrombolysis with a alloon and Thrombectomy with a Stent Retriever Were Effective Issei Takano, Yoshiyuki Matsumoto, Yoshiko Fujii, Yuki Inoue, Yoshiki Sugiura, Koji Hirata, Yousuke Kawamura, Ryotaro Suzuki, Ryuta Nakae, Yoshihiro Tanaka, Masaya Nagaishi, Tomoji Takigawa, kio Hyodo, and Kensuke Suzuki Objective: We report a patient with severe cerebral sinus thrombosis (CST) in whom mechanical thrombolysis with a balloon and thrombectomy with a stent retriever were effective. Case Presentation: The patient was a 32-year-old male. Headache occurred, and magnetic resonance venography (MRV) showed occlusion of the superior sagittal sinus. Transvenous anticoagulant therapy was performed, but consciousness disorder and paralysis progressed in a few days. Head CT revealed marked edema of the bilateral frontal lobes and cerebral hemorrhage. Cerebral angiography showed occlusion of the superior sagittal sinus, and endovascular treatment with a balloon and stent retriever was performed, leading to recanalization. Finally, the course was favorable. Conclusion: Endovascular treatment with a stent retriever may be safe and effective for severe CST. Keywords cerebral venous sinus thrombosis, mechanical thrombectomy, stent retriever Introduction Cerebral sinus thrombosis (CST) is rare among patients with stroke. Routinely, conservative treatment with anticoagulants is performed, but recanalization of the sinus is not achieved when thrombus-related occlusion involves an extensive area, leading to a severe condition in some cases. In this study, we report a patient with rapidly deteriorating CST for whom mechanical thrombolysis with a balloon and thrombectomy with a stent retriever were performed. Department of Neurosurgery, Dokkyo Medical University Saitama Medical Center, Koshigaya, Saitama, Japan Received: November 8, 2017; ccepted: pril 11, 2018 Corresponding author: Issei Takano. Department of Neurosurgery, Dokkyo Medical University Saitama Medical Center, Minamikoshigaya, Koshigaya, Saitama , Japan itakano@dokkyomed.ac.jp This work is licensed under a Creative Commons ttribution-noncommercial- NoDerivatives International License The Japanese Society for Neuroendovascular Therapy Case Presentation Case: 32-year-old male. Complaints: Headache and left incomplete paralysis. Family history: Not contributory. Medical history: He had taken Warfarin to treat lower limb venous thrombosis. Present illness: Mild headache had persisted for 1 week, but he did not consult a hospital. Severe headache suddenly occurred while working at his desk. Subsequently, mild left hemiparesis was noted, and he consulted a local clinic of neurosurgery. MRI revealed CST, and he was referred to our hospital. Neurologic findings on admission: Concerning the consciousness level, the Japan Coma Scale (JCS) score was I-0, and the Glasgow Coma Scale (GCS) score was 15 points (E4V5M6). Left incomplete paralysis (MMT: 4/5) was observed. Hematological data on admission: ST 54 IU/L, LT 106 IU/L, LD 196 IU/L, γ-gt 117 IU/l, UN 7.2 mg/dl, CRE 0.60 mg/dl, RC /μL, Hb 16.5 g/dl, Hct 47.9%, Plt /μL, PT 12.3 sec, PT(%) 89.5%, PT-INR 1

2 Takano I, et al. Fig. 1 () Head CT (axial section) showed high-density areas in the superior sagittal sinus and bridging vein. () On a coronal section, high-density areas were also detected in the superior sagittal sinus and bridging vein INR, PTT 31.3 sec, Fibrinogen 364 mg/dl, T-III 114%, FDP 4.3 μg/ml, D-dimer 2.5 μg/ml Concerning the blood coagulation system, there was a slight increase in the D-dimer level. In addition, mild liver dysfunction was noted. Radiological findings on admission: CT showed highdensity areas in the superior sagittal sinus and bridging vein (Fig. 1). Magnetic resonance venography (MRV) revealed the disappearance of the superior sagittal sinus (Fig. 2). Course after admission: fter admission, conservative treatment by anticoagulant therapy with heparin was started. However, systemic tonic-clonic convulsion occurred 3 days after admission. Head CT showed low-density areas in the bilateral frontal lobes. Paralysis of the bilateral upper/lower limbs progressed 6 days after admission, and the consciousness level reduced (JCS: 100). Head CT revealed increases in the low-density areas in the bilateral frontal lobes and partial hemorrhage-related high-density areas (Fig. 3). ssuming that no improvement may be achieved by further conservative treatment, emergency endovascular treatment was performed. Endovascular treatment: s severe consciousness disorder was present, general anesthesia was performed after intubation. 4 Fr sheath was inserted into the left femoral artery through the left inguinal region. Internal carotid angiography was conducted. In the arterial phase, there were no abnormalities. In the venous phase, the superior sagittal sinus was not visualized, and the bilateral transverse sinuses were slightly visualized. The occipital sinus was advanced; this was considered to be a primary etiological factor for venous return. Under systemic heparin administration, the activated coagulation time (CT) was maintained at 300. To secure the back-up system of Fig. 2 On MRV, the former half (2/3) of the superior sagittal sinus was not visualized. MRV: magnetic resonance venography a guiding catheter, a 6 Fr shuttle sheath (Cook Medical, loomington, IN, US) was inserted into the left internal jugular vein through the right femoral vein. In addition, a 6 Fr FUUKI (sahi Intecc Co., Ltd, ichi, Japan) was coaxially inserted to an area adjacent to the confluence of the sinus. Subsequently, an SL10 (Stryker, Kalamazoo, MI, US) was carefully guided to the forehead region of the superior sagittal sinus using a Cruise 14 (sahi Intecc Co., Ltd). ngiography through a microcatheter was performed. fter confirming the contrast enhancement of a portion of the sinus, urokinase at units was administered as several divided doses. However, recanalization was not achieved, and the SL10 was exchanged for an mphirion PT balloon catheter (Medtronic, Minneapolis, MN, US) 2

3 Thrombectomy of CST by alloon and Stent Retriever Fig. 3 () Head CT (axial section) showed low-density areas in the bilateral frontal lobes. Partially, a high-density area related to hemorrhage was noted. () On a coronal section, extensive low-density areas were also detected in the bilateral frontal lobes. Partially, a high-density area related to hemorrhage was observed. measuring mm. Its position is changed so that all thrombi at the site of occlusion of the superior sagittal sinus may be crushed, percutaneous transluminal angioplasty (PT) was conducted three times. Subsequently, the balloon catheter was again exchanged for an SL10, and angiography was performed. The superior sagittal sinus was slightly visualized. Subsequently, a stent retriever was adopted, considering that sufficient recanalization cannot be achieved by PT alone. The microcatheter was exchanged for an XT27 (Stryker), and a Trevo 6 25 mm (Stryker) was guided to the forehead region of the superior sagittal sinus. Subsequently, the Trevo was deployed, and slowly removed so that thrombi might be arrested (Fig. 4). fter removal, internal carotid angiography confirmed the visualization of the superior sagittal sinus, and the procedure was completed (Fig. 5). Course after treatment: CT immediately after surgery did not show any increase in the site of hemorrhage, which was detected before surgery, or new onset of hemorrhage. To protect the brain, anesthesia with propofol was continued for 3 days after surgery. For anticoagulant therapy, heparin was administered immediately after surgery to gradually prolong the activated partial thromboplastin time (PTT), targeting a value two times higher than the control value. Heparin was switched to Warfarin 2 weeks after surgery. Subsequently, rehabilitation was conducted, leading to an improvement in paralysis. The patient became able to walk. MRI confirmed the recanalization of the superior sagittal sinus. There was no new hemorrhage or infarction, and the patient was referred to the recovery-phase ward for further rehabilitation. Discussion In the present case, conservative treatment with an anticoagulant was started to treat CST. However, consciousness disorder rapidly progressed, and brain edema with hemorrhage deteriorated. Endovascular treatment with a stent retriever was performed, leading to a favorable treatment course. CST accounts for 0.5% 1.0% of all stroke patients. 1) Previously, treatment had not been established, and the mortality rate was high. 2) However, recently, the International Study on Cerebral Vein and Dural Sinus Thrombosis (ISCVT) reported that the early administration of adequate anticoagulants, such as undifferentiated or lowmolecular-weight heparin, improved the prognosis, and that patients with unfavorable outcomes, including death, accounted for approximately 13%. 3) However, endovascular treatment is considered for patients with hemorrhage in whom the systemic administration of anticoagulants is difficult, those in whom the site of occlusion of the venous sinus is extensive, and those in whom there is no improvement despite the systemic administration of anticoagulants. 4) In the present case, the systemic administration of an anticoagulant was performed, but, subsequently, paralysis of the limbs and consciousness disorder rapidly progressed, inducing systemic convulsion. Head CT showed the progression of brain edema with cerebral hemorrhage related to intracranial perfusion disorder, and early revascularization by endovascular treatment was considered to be necessary. 3

4 Takano I, et al. C D Fig. 4 () Lateral view on angiography: n Excelsior XT-27 was guided to the tip of the forehead region of the superior sagittal sinus with a microguidewire. () Magnification. (C) Lateral view on angiography: Trevo 6 25 mm was deployed. (D) Magnification (trevo: arrows). Excelsior XT-27: Stryker, Kalamazoo, MI, US Currently, it is recommended that endovascular treatment should be considered for patients with CST who do not respond to local fibrinolysis therapy in several guidelines. 4,5) However, its usefulness has not been established. Therefore, endovascular treatment is considered for many severe-status patients in whom standard treatment is not effective for clinical deterioration during anticoagulant therapy, venous infarction-/cerebral hemorrhage-related compression effects, or an increase in the intracranial pressure. s endovascular treatment, selective fibrinolysis therapy and mechanical thrombolysis have been reported. The favorable results of selective fibrinolysis therapy with urokinase 6,7) or tissue plasminogen activator (tp) 8,9) were published, but neither the dose nor continuation method has been established. On the other hand, a study indicated the exacerbation of hemorrhage in patients with cerebral hemorrhage before treatment. 9) Mechanical thrombolysis procedures include thrombolysis with a balloon, 10) that using an ngiojet (oston Scientific, Minneapolis, MN, US), 11) and thrombus aspiration with a Penumbra system (Penumbra, Inc., lameda, C, US). 12) In addition, a recent study reported the favorable results of mechanical thrombolysis with a stent retriever. 13) In the present case, initially, urokinase was administered through the tip of the superior sagittal sinus, but the site of occlusion involved the entire superior sagittal sinus; thrombolysis was considered to be difficult. Furthermore, cerebral hemorrhage occurred, and urokinase administration was switched to thrombectomy by mechanical thrombolysis, which may less frequently induce hemorrhagic complications, considering that further urokinase administration may promote hemorrhage. lthough several studies reported thrombus aspiration with a Penumbra system, thrombosis involved 2/3 of the superior sagittal sinus in the present case, and it was considered difficult to guide a large-profile catheter to the tip; a balloon and stent retriever were used. 13) Several sessions of mechanical thrombolysis with a balloon and thrombectomy using a stent retriever may have contributed to early recanalization. ccording to systematic reviews of endovascular treatment for venous sinus thrombosis, 14,15) favorable prognosis patients with a modified Rankin Scale score of 0-2 4

5 Thrombectomy of CST by alloon and Stent Retriever C D Fig. 5 () On left common carotid angiography, the visualization of the former half (2/3) of the superior sagittal sinus was unfavorable in the venous phase. () microcatheter was inserted to the forehead region of the superior sagittal sinus, and angiography was performed. thrombus-related shadow defect was observed. (C) fter treatment, the entire superior sagittal sinus was visualized. (C) On postoperative MRV, the superior sagittal sinus was favorably visualized. MRV: magnetic resonance venography accounted for 76.0% 84.0%, and the compete recanalization rate ranged from 69.0% to 74.0%. In addition, the incidence of hemorrhage was 8.7% 10%; the results were not better than those of standard treatment. However, endovascular treatment is frequently performed for severe-status patients who do not respond to standard treatment or those with hemorrhage according to guidelines. Under such conditions, the results were acceptable. Concerning the selection of devices, an ngiojet had been the most frequently used according to a review published by Siddiqui et al. 14) However, the profile of this device is large and hard; therefore, it is difficult to guide it to the lesion site. They reported the usefulness of a Penumbra system with a more favorable guiding property and a profile that facilitates thrombus aspiration. With respect to a stent retriever, the number of patients in whom it was used is still limited, and favorable results may be achieved using this device in the future. Its merits include a profile smaller than that of the above aspiration-type catheter and a favorable guiding property. On the other hand, mechanical thrombectomy may induce hemorrhage related to vascular injury, but a soft device with a smaller profile may reduce the risk; a stent retriever may be advantageous. Ilyas et al. 15) suggested that the venous sinus may not be damaged due to its large diameter and wall thicker than the vein wall. Furthermore, a study examined selective fibrinolysis therapy combined with mechanical thrombectomy, and reported that the incidence of hemorrhage was slightly higher in the selectivefibrinolysis-therapy-combined group although there was no significant difference; combination therapy should be carefully performed. On the other hand, the perioperative systemic administration of anticoagulants is recommended even for patients with intracranial hemorrhage. 4,5) If there is no further increase in bleeding at the site of hemorrhage after surgery, anticoagulant therapy should be continued. To date, no study has prospectively investigated 5

6 Takano I, et al. endovascular treatment for CST or demonstrated its usefulness. In the future, further reports should be published. Conclusion Mechanical thrombolysis with a balloon and endovascular treatment using a stent retriever may be safe and effective for severe CST. Disclosure Statement There is no conflict of interest. References 1) Stam J: Thrombosis of the cerebral veins and sinuses. N Engl J Med 2005; 352: ) Nagpal RD: Dural sinus and cerebral venous thrombosis. Neurosurg Rev 1983; 6: ) Ferro JM, Canhão P, Stam J, et al: Prognosis of cerebral vein and dural sinus thrombosis: results of the International Study on Cerebral Vein and Dural Sinus Thrombosis (ISCVT). Stroke 2004; 35: ) Saposnik G, arinagarrementeria F, rown RD, et al: Diagnosis and management of cerebral venous thrombosis: a statement for healthcare professionals from the merican heart association/merican stroke association. Stroke 2011; 42: ) Ferro JM, ousser MG, Canhão P, et al: European stroke organization guideline for the diagnosis and treatment of cerebral venous thrombosis - endorsed by the European academy of neurology. Eur J Neurol 2017; 24: ) Smith TP, Higashida RT, arnwell SL, et al: Treatment of dural sinus thrombosis by urokinase infusion. JNR m J Neuroradiol 1994; 15: ) Spearman MP, Jungreis C, Wehner JJ, et al: Endovascular thrombolysis in deep cerebral venous thrombosis. JNR m J Neuroradiol 1997; 18: ) Yamini, Loch Macdonald R, Rosenblum J: Treatment of deep cerebral venous thrombosis by local infusion of tissue plasminogen activator. Surg Neurol 2001; 55: ) Frey JL, Muro GJ, McDougall CG, et al: Cerebral venous thrombosis: combined intrathrombus rtp and intravenous heparin. Stroke 1999; 30: ) 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: ; discussion ) Chow K, Gobin YP, Saver J, et al: Endovascular treatment of dural sinus thrombosis with rheolytic thrombectomy and intra-arterial thrombolysis. Stroke 2000; 31: ) Mammen S, Keshava SN, Moses V, et al: Role of penumbra mechanical thrombectomy device in acute dural sinus thrombosis. Indian J Radiol Imaging 2017; 27: ) Pukenas, Kumar M, Stiefel M, et al: Solitaire FR device for treatment of dural sinus thrombosis. J Neurointerv Surg 2014; 6: e2. 14) Siddiqui FM, Dandapat S, anerjee C, et al: Mechanical thrombectomy in cerebral venous thrombosis: systematic review of 185 cases. Stroke 2015; 46: ) Ilyas, Chen CJ, Raper DM, et al: Endovascular mechanical thrombectomy for cerebral venous sinus thrombosis: a systematic review. J Neurointerv Surg 2017; 9:

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