Treatment of cerebral venous sinus thrombosis by combined endovascular technique

Dear Editor,

Cerebral sinus thrombosis is a rare cause of stroke (0.5-3%) [1]. It mainly affects younger patients (< 55) and women (2/3) [1]. Symptoms tend to include cephalea (90%), motor disturbances (20-50%) and seizures (20-40%) [1,2]. The most important risk factors are female sex, hormonal contraception, pregnancy, smoking and thrombophilic conditions [1,3]. The recommended management is anticoagulant therapy. However, this fails in a proportion of patients (5-10%). Based on expert consensus, endovascular treatment is then recommended. The prognosis of the disease is good (80-90%). A severe course is reported in 10-20% of cases [1,4,5] and may be infaustive despite treatment [1,2,4-6]. We present a case report of a patient with cerebral plexus thrombosis with a complicated course. The procedure is specific combining systemic anticoagulation, local thrombolytic and endovascular treatment in multiple sessions.

A woman (45 years old) was admitted after a seizure. She had a history of catatonic schizophrenia, conversion mixed sensorimotor seizures and collapse states. She had suffered from cephalea for the last 3 days.

At the ictal center, neurological examination revealed somnolence and mild right-sided hemiparesis. Native CT scan showed multiple subarachnoid (SAH) (Hunt-Hess score 3, Fischer scale 4) and intracerebral hemorrhages (ICH) (subcortical parietal left, SAH temporal right, SAH in the area of the sternotemporal angle left) (Figure 1) and suspected thrombosis of the sinus transversus right and s. sagitalis superior. Supplemented CT venography diagnosed thrombosis of the s. sigmoideus, s. transversus on the right and s. sagitalis superior and, more recently, minor haematocephalus. Anticoagulation therapy with low molecular weight heparin (Clexane 0.8 ml twice daily) was started.

The next day, the patient stopped communicating and right-sided hemiparesis persisted. She was then transferred to a comprehensive cerebrovascular center, when she was already soporific with a severe fatal lesion -⁠ only sounds. The patient was transferred to the angiography suite where she was intubated (orotracheal intubation [OTI]).

Under general anesthesia, after verifying the absence of filling of almost all the rafts, closed rafts were catheterized with a Vasco+21 microcatheter (Balt, CA, USA) and an Agility guidewire (Cerenovus, CA, USA) via the vena jugularis interna on the right via an 8F Cerebase guiding catheter (Cerenovus, CA, USA). After a combination of endovascular techniques using the Embovac aspiration catheter (Cerenovus, CA, USA) and the CatchMAXI 5.5 × 50 mm stentriever (Balt, CA, USA), restoration of flow in the right sinus transversus and sigmoid sinus with bypassed thrombus was achieved. Subsequently, flow was restored in the sinus sagitalis superior using a similar procedure. At the end of the procedure, a microcatheter (Prowler Select Plus, Cerenovus, CA, USA) was inserted frontally into the sinus sagitalis superior and local thrombolysis was initiated with a bolus of 1 mg alteplase followed by a continuous infusion of 20 mg at a rate of 1 mg/h (Actilyse, Boehringer Ingelheim, Germany) (Figure 2). At the same time, full heparinization was initiated under laboratory control with a target level of 2-2.5× the APTT norm.

The following day, endovascular procedure was performed again (under general anaesthesia) combining aspiration and retraction of other thrombotic masses macerated by local action of alteplase. After the procedure, significant residual thrombosis persisted at the junction of the sinus transversus and sinus sigmoideus with a high risk of reocclusion. Local application of alteplase (1 mg/h) was continued, with concomitant heparinization. A third endovascular procedure was performed ten hours apart, with removal of residual thrombotic masses. Both thrombolysis and heparinization were terminated. At the end of each procedure, a follow-up cone beam CT was performed in the operating room to rule out complications. Further therapy included low molecular weight heparin (Clexane 0.8 ml twice daily) at a therapeutic dose.

The next day, a follow-up CT scan was performed with stable findings of SAH and multiple hematomas (subcortical parietal left, SAH temporal right, SAH in the area of the left costomedial angle) and a new small (13 × 30 mm) ischemia temporal left. A non-significant partial thrombosis of the sinus sigmoideus on the left remained. The patient has been sedated since intubation. Between procedures, sedation was reduced to the minimum necessary (to tolerate OTI) to allow clinical control. After the final endovascular procedure, the patient was extubated uncomplicated.

She was discharged home one week later with no neurological deficit. One month after the event, anticoagulation therapy was changed from low molecular weight heparin to per os therapy with dabigatran (150 mg twice daily) for one year. Follow-up MR imaging at 5 months interval showed full recanalization of the cerebral plexus.

Risk factors were mainly the patient's age (45 years) and use of hormonal contraceptives (replacement therapy).

Despite a comprehensive examination (standard thrombophilic examination, hepatitis panel, gynecological screening, lung X-ray, abdominal CT), the cause of extensive thrombosis was not identified. Whole-body PET/CT was added, which showed significant glucose hypermetabolism in the root and edge of the tongue extending through the m. sternohyoideus to the sternum. Further investigation suggested that this was probably latent inflammation. This finding may have been the origin of the thrombophilic condition that led to the massive thrombosis mentioned above. However, a definite link cannot be established.

Early initiation of anticoagulant therapy is the recommended treatment for cerebral pleural thrombosis (prevention of progression of residual thrombotic masses, promotion of further dissolution of thrombotic masses, prevention of new thrombotic events) [1,3]. The presence of ICH associated with thrombosis of the plexiform vessels is not a contraindication to anticoagulation therapy [1]. However, up to 15% of patients are refractory to anticoagulation therapy [4].

In a review article, Quealy et al. present the results of a synthesis of 124 publications (486 patients). Mechanical thrombectomy appears to be an effective and safe procedure. Concurrent local thrombolysis is effective according to this work [5].

Currently, recommendations for endovascular treatment of cerebral plexus are not based on randomized trials. Nor is there a clear conclusion as to which endovascular technique is recommended [1]. The heterogeneity of endovascular approaches with different safety profiles and clinical outcomes is also demonstrated by Batista et al. in their review [7].

Recent guidelines based on patient cohort and clinical experience recommend proceeding to endovascular surgery when the disease course is complicated and anticoagulation therapy fails [1,3]. In our opinion, we prefer to recommend earlier, at the first signs of impaired consciousness or significant neurological deficit or on the basis of imaging examinations in case of the first signs of developing edematous changes, progression of ischemic changes or progression of hemorrhagic complications.

In our opinion, the fractionated endovascular procedure with continuous maceration of thrombotic masses by local thrombolytic treatment may be a more effective procedure than the standard single procedure. Compared with the arterial procedure, there is a relative time window after partial patency of the grafts for a slower and possibly safer procedure. Nevertheless, the prognosis of the patient may depend on the radicality and timeliness of the first procedure. A limitation of this communication is that it is a case report. However, this procedure is not described even in small case series or case reports.

The use of direct anticoagulants in long-term therapy appears to be a safe and effective alternative to warfarin [1,3,8].

In conclusion, the chosen approach led to an excellent clinical outcome when standard anticoagulation therapy failed. The combination of local thrombolytic and systemic anticoagulation therapy did not cause the development of hemorrhagic complications. The duration for which the therapy was administered and the initial CT findings (extensive hemorrhagic complications) indicate the relative safety of this procedure. Clear recommendations for endovascular treatment in this area are still lacking. In our opinion, fractionated endovascular procedure combined with local thrombolytic treatment may be more effective and safer compared to a single procedure. However, a larger randomized trial would be appropriate to verify this. In the search for the etiology, we recommend the addition of PET/CT in case of failure of other investigations.

Grant support

Supported in part by grant IGA-KZ-2021-1-17 from the Regional Health Association.

Conflict of interest

The authors declare that they have no conflict of interest in relation to the subject of the study.

This is an unauthorised machine translation into English made using the DeepL Translate Pro translator. The editors do not guarantee that the content of the article corresponds fully to the original language version.