Eagle jugular syndrome accompanied by de novo brainstem cavernous malformation: a case-based systematic review.

BACKGROUND: Eagle jugular syndrome (EJS), recently identified as a cause of cerebrovascular disease (CVD) due to venous obstruction by an elongated styloid process (SP), is reported here alongside a case of concurrent de novo cerebral cavernous malformation (CCM). This study aims to explore the potential causal relationship between EJS and de novo CCM through a comprehensive literature review. METHOD: Systematic literature reviews, spanning from 1995 to 2023, focused on EJS cases with definitive signs and symptoms and de novo CCM cases with detailed clinical characteristics. Data on the pathophysiology and clinical manifestations of EJS, as well as potential risk factors preceding de novo CCM, were collected to assess the relationship between the two conditions. RESULT: Among 14 patients from 11 articles on EJS, the most common presentation was increased intracranial hypertension (IIH), observed in 10 patients (71.4%), followed by dural sinus thrombosis in four patients (28.6%). In contrast, 30 patients from 28 articles were identified with de novo CCM, involving 37 lesions. In these cases, 13 patients developed CCM subsequent to developmental venous anomalies (43%), seven following dural arteriovenous fistula (dAVF) (23%), and two after sinus thrombosis (6%). In a specific case of de novo brainstem CCM, the development of an enlarged condylar emissary vein, indicative of venous congestion due to IJV compression by the elongated SP, was noted before the emergence of CCM. CONCLUSION: This study underscores that venous congestion, a primary result of symptomatic EJS, might lead to the development of de novo CCM. Thus, EJS could potentially be an indicator of CCM development. Further epidemiological and pathophysiological investigations focusing on venous circulation are necessary to clarify the causal relationship between EJS and CCM.

[A Case of a Pituitary Adenoma Diagnosed as Carney Complex Syndrome in an Older Female Patient].

Carney complex syndrome is an autosomal dominant familial tumor syndrome first described by Carney et al. in 1985. The diagnostic criteria include endocrine hyperactivity and spotty skin pigmentation. A 73-year-old woman with cerebral infarction was referred to our department because her brain magnetic resonance imaging(MRI)revealed a pituitary tumor. Her blood tests revealed elevated levels of growth hormone(GH), thyroid stimulating hormone(TSH), and insulin-like growth factor-1(IGF-1). We suspected the presence of a GH-secreting tumor and performed the operation. The pathological finding was a TSH-positive pituitary adenoma. Her cervical computed tomography(CT)revealed a thyroid tumor and the tumor removal was performed. The pathological diagnosis was papillary carcinoma. She had skin pigmentation bilaterally on her face, forearms, hands, and legs. We diagnosed this case as Carney complex syndrome based on these findings.

Covered stent deployment for a recurrent cervical internal carotid artery aneurysm referencing angioscopy: illustrative case.

BACKGROUND: Extracranial carotid artery aneurysms (ECAAs) are rare, and treatment guidelines are lacking. Few reports on endovascular treatments performed for ECAAs exist. OBSERVATIONS: A 73-year-old woman with a left giant cervical internal carotid artery aneurysm was treated with overlapping closed-cell stents. The aneurysm regrew 1 year after the treatment, and then a covered stent was deployed. Angioscopy was performed to confirm neointimal development to determine the appropriate stent position before the retreatment, and it revealed that the stent struts were embedded in thick neointima for the most part but that the neointima was thin around the aneurysm neck. Multiple holes connecting to the aneurysm were observed between the stent struts. A covered stent overlapped inside the closed-cell stents, and blood flow into the aneurysm completely disappeared. LESSONS: When deploying the covered stent for recurrent aneurysms, angioscopy is useful for confirming neointimal development and determining the appropriate stent length and position. Angioscopic observations suggest that using stents with a higher mesh density and smaller pore size can reduce the neck hole size of the aneurysm and may achieve complete occlusion of the aneurysm. https://thejns.org/doi/10.3171/CASE24383.

Syringosubarachnoid shunting with augmented reality navigation through a keyhole laminectomy of the upper thoracic spine: illustrative case.

BACKGROUND: A syringosubarachnoid (SS) shunt combined with keyhole hemilaminectomy is a beneficial procedure that can reduce the size of the skin incision and the risk of complications. However, ingenuity is needed to confirm the position of the syrinx during surgery. The authors present a case in which they treated syringomyelia in the upper thoracic spine using augmented reality (AR) to confirm syrinx formation, bone resection, and skin incision. OBSERVATIONS: Microscope-based AR was an appropriate and practical choice in this case. By placing the reference array at the Mayfield clamp, it was possible to use AR from the point of skin incision. Under AR navigation, an SS shunt tube can be placed in the short syrinx. LESSONS: AR navigation enables pinpoint SS shunt tube insertion with minimal skin incision and bone resection. It is particularly useful for upper thoracic and small syrinx lesions. https://thejns.org/doi/10.3171/CASE24130.

Case report: Usefulness of angioscopy in determining antiplatelet drug reduction after carotid artery stenting.

We report a case in which neointima was confirmed by angioscopy and antiplatelet drug administration was reduced 2 months after carotid artery stenting (CAS). A patient in their 80s was scheduled to undergo resection for renal cancer; however, he also had right cervical internal carotid artery stenosis. Because this was a risk for general anesthesia, CAS was performed after first starting dual antiplatelet therapy. Urologically, early reduction of antiplatelet drugs was necessary for a nephrectomy. Although no obvious neointima could be identified on ultrasound 2 months after CAS, thin neointima was observed using angioscopy. Based on the above results, we reduced the antiplatelet drug administration, and then the nephrectomy was performed. Ultimately, no cerebral infarction occurred in the perioperative or postoperative periods. Angioscopy allows for visual confirmation of thin neointima. If sufficient neointima can be confirmed, antiplatelet drug reduction can be performed more safely and reliably.

Comparison Between Cz-C3/C4 and C3-C4 Montages to Protect Against Peripheral Stimulation in Transcranial Facial Motor-Evoked Potential Monitoring.

INTRODUCTION: In facial motor-evoked potential monitoring, efforts to reduce peripheral stimulation are necessary because it can cause false-negatives. The effects of peripheral stimulation on Cz-C3/C4 and C3-C4 montages were compared. METHODS: Facial motor-evoked potentials were recorded from bilateral orbicularis oculi (Oculi) and oris (Oris) muscles. The double-train approach combining single-pulse and five-train pulse stimulation was used to determine the effect of peripheral stimulation. If the five-train pulse produced a significant waveform, it was defined as "total success." In total success cases, "true success" was defined as a case in which no waveform appeared after the single pulse at the threshold level of the five-train pulse. The total and true success rates and the threshold value of Oculi and Oris were compared between Cz-C3/C4 and C3-C4 montages. RESULTS: Thirty-six muscles each of Oculi and Oris of 18 patients were used for the analysis. True success was more likely to be obtained by the Cz-C3/C4 montage than the C3-C4 montage in Oculi (42% vs. 22%, p = 0.039). Both Oculi and Oris had higher thresholds to elicit facial motor-evoked potentials with the Cz-C3/C4 montage (Oculi: 101.7 vs. 71.4 mA, p = 0.038; Oris: 94.8 vs. 73.1 mA, p = 0.016). CONCLUSIONS: Cz-C3/4 montage is more effective at reducing peripheral stimulation compared with the C3-4 montage. This effect was primarily seen in the orbicularis oculi muscle. It should be noted that the Cz-C3/C4 montage has a higher threshold than the C3-C4 montage in facial muscles. In facial motor-evoked potential monitoring, the Cz-C3/C4 montage may be more suitable to eliminate peripheral stimulation.

A Case of Concomitant Lip Injury and Facial Pressure Ulcer in Lumbar Intradural Tumor Surgery With Repeated Transcranial Electrical Stimulations.

Transcranial motor evoked potential (MEP) is a common method in spinal surgery but requires strong electrical stimulation. Frequent transcranial stimulations can cause bite injury. In addition, a facial pressure ulcer is a problem in spinal surgery requiring prone positioning. We present a case of bite injury and facial pressure ulcer in prolonged lumbar tumor surgery with repeated transcranial stimulations. A 74-year-old woman developed left lower limb and low back pain. MRI revealed an intradural extramedullary tumor at L1. We performed tumor resection surgery. A silicon bite block was used, and the patient's head was placed on a sponge headrest. The tumor was a schwannoma originating from the nerve root that innervated the left anal sphincter. Intracapsular resection was performed while referring to the frequent transcranial MEP monitoring. The left lower limb and low back pain improved after surgery; however, lip injury and facial skin ulcer occurred. The face showed marked swelling and was painful, so oral intake was difficult for a week. Wound healing was obtained three months postoperatively, but hypoesthesia remained. When using MEP in prolonged spine surgery with a headrest, it is necessary to pay attention to both bite injury and facial pressure ulcer. Intraoperative assessment of the face, number of transcranial stimulations, types of a bite block, and headrest may be important.

A Novel Approach for Transvenous Embolization of Dural Arteriovenous Fistula Using a Balloon and a Coil as Walls: Case Presentation.

BACKGROUND: Transvenous embolization (TVE) for dural arteriovenous fistula (DAVF) is difficult depending on an accessible route. Reported herein is a case of transvenous embolization using a balloon and a coil as "walls." Case Description. A 56-year-old male patient presented with a 1-month history of mild motor aphasia. The magnetic resonance imaging showed a hemorrhagic lesion in his left temporal lobe, and the cerebral angiography showed a DAVF, with parasinus shunt points near the torcula and the left transverse sinus. Access to the shunt point was very difficult; however, TVE was performed using a balloon as a wall. Furthermore, all lesion embolization was possible using a coil as a wall. CONCLUSIONS: Using a balloon or coil as a wall during a TVE is useful.

Cerebral vasospasm after coil embolization for unruptured anterior communicating artery aneurysm: illustrative case.

BACKGROUND: Compared with several reports of cerebral vasospasm after clipping for unruptured cerebral aneurysm, only one study to date has reported cerebral vasospasm after coil embolization. Herein, the authors report a rare case of cerebral vasospasm after coil embolization for unruptured cerebral aneurysm. OBSERVATIONS: A 58-year-old woman with an unruptured anterior communicating artery aneurysm was referred to our department. Stent-assisted coil embolization was performed for the aneurysm, and no obvious adverse events were observed on cerebral angiography obtained immediately after the operation. However, the patient developed mild headache and slight restlessness soon after the operation and new-onset disorientation, left hemispatial neglect, and left hemiplegia the day after the operation. Emergency brain magnetic resonance imaging and cerebral angiography indicated vasospasm in the right middle cerebral artery, and intra-arterial injection of fasudil hydrochloride hydrate was performed to dilate the middle cerebral artery. Blood flow in the middle cerebral artery immediately improved, and she was discharged without neurological deficits 8 days after the operation. LESSONS: Immediate intervention is necessary to prevent cerebral infarction in patients with cerebral vasospasm, which may occur even after coil embolization for unruptured cerebral aneurysm.

Navigation-assisted full-endoscopic spine surgery: a technical note.

BACKGROUND: Full-endoscopic spine surgery (FESS) necessitates the use of X-ray fluoroscopy for intraoperative guidance and orientation. However, the two-dimensional X-ray fluoroscopic images do not provide real-time guidance. The authors developed a new real-time three-dimensional (3D) navigation technique for FESS that entails the use of intraoperative cone beam computed tomography (CBCT) in a hybrid operating room (OR). METHODS: A total of 23 patients undergoing FESS using real-time 3D navigation system were enrolled. Preoperative and intraoperative CBCT data were registered in the navigation system. The 3D navigation was used to intraoperatively determine the trajectory and obtain position information. The feasibility and usefulness of the navigation system were retrospectively analyzed. RESULTS: Twenty patients had lumbar spine disease, whereas three patients had cervical spine disease. The 3D navigation was successfully used for intraoperative guidance and provided accurate information in all patients. X-ray fluoroscopy was not required in any of the patients. No complications associated with the use of 3D navigation system were encountered. CONCLUSIONS: The use of real-time 3D navigation system in the hybrid OR was found to be safe and effective in providing intraoperative guidance for FESS.

Intradural Disk Herniation Mimicking a Spinal Tumor: Radiologic Imaging, Pathogenesis, and Operative Management.

Intradural disk herniation (IDH) is a rare condition, occurring more often at the L4-5 level. We examined a case of an IDH at the L1-2 level mimicking an intradural spinal tumor. A 71-year-old woman with a long history of backache and pain radiating down the left leg was admitted to our hospital with the worsening of these symptoms. Magnetic resonance imaging and computed tomographic myelography demonstrated an intradural mass at the L1-2 level. Given the radiologic findings and the location of the mass, the preoperative differential diagnosis centered on intradural spinal tumors. Dural incision was performed using a surgical microscope to resect the mass. Contrary to our expectation, the diagnosis made during the surgery was IDH. Despite advances in imaging techniques, IDH could not be definitively diagnosed preoperatively. The pathogenesis of IDH remains unclear. In our patient, the ventral dural defect was smooth and round, and the dural tissue around the defect was thickened. These intraoperative findings suggested that the patient's IDH resulted not from an acute new event but from a chronic process. We recommend dural incision using a surgical microscope for treating IDH because it provides a clear visual field.

Expression of angiogenic growth factor in the rat DAVF model.

OBJECTIVE: The precise mechanisms responsible for the development and growth of dural arteriovenous fistula (DAVF) remain unclear, but it has been hypothesized that vascular endothelial growth factor (VEGF) might be involved in the pathogenesis. The aim of this study was to examine the expression of VEGF in the rat DAVF model. METHODS: Forty-five Sprague-Dawley rats were used in two experiments. In Experiment 1 (n = 20, including sham-operated controls), VEGF expression was analysed by Western blots in three different rat DAVF models: model I: common carotid artery-external jugular vein (CCA-EJV) anastomosis (n = 5); model II: sagittal sinus thrombosis and bipolar coagulation of the vein draining the transverse sinus (n = 5); model III: CCA-EJV anastomosis and bipolar coagulation of the vein draining the transverse sinus and sagittal sinus thrombosis to induce venous hypertension (n = 5). Based on the results of Experiment 1, Western blots were performed at weekly intervals 1, 2 and 3 weeks in Experiment 2 following induction of venous hypertension in model III (n = 5 at each time point and n = 5 sham controls); in addition, VEGF expression was immunohistochemically examined in the dura and the brain near the occluded sinus in five model III animals after 1 week. RESULTS: In Experiment 1, Western blot analysis showed barely detectable bands with molecular weights of 45 kD, corresponding to VEGF, in the sham group, but the highest level of VEGF was induced in model III, followed by models I and II (model III>model I>model II). In Experiment 2, the expression of VEGF peaked 1 week after induction of venous hypertension in model III, decreasing in a linear fashion over 2 and 3 weeks (week 1>weeks 2 and 3). The expression of immunoreactive VEGF was restricted in the connective tissue and the endothelial layer of the dura matter, cerebral cortical tissue and neurons of the basal ganglia. CONCLUSION: Our results strongly suggest a possible contribution of an angiogenic factor to the growth of DAVF. Venous ischemia by venous hypertension might be a mechanism for inducing up-regulation of angiogenic factor expression.

Intradural extramedullary ganglioneuroma associated with multiple hamartoma syndrome.

A 51-year-old woman presented with a rare completely intradural and extramedullary spinal ganglioneuroma associated with multiple hamartoma syndrome and manifesting as complaints of neck pain and dizziness persisting for 8 months. Magnetic resonance imaging of the spinal cord revealed an intradural extramedullary lesion at the C1 level. She underwent right suboccipital craniectomy and C1-2 hemilaminectomy to remove the tumor. Histological examination confirmed ganglioneuroma. She also suffered from multiple facial trichilemmomas, thyroid goiter, multiple polyposis of the gastrointestinal tract, and pulmonary hamartoma indicating multiple hamartoma syndrome. These benign neoplasms were treated conservatively.

Dural repair for intraspinal extradural meningeal cyst.

A 52-year-old woman presented with an intraspinal extradural meningeal cyst in the thoracolumbar region manifesting as progressive sensory disturbance of the bilateral lower extremities. Magnetic resonance imaging and computed tomography myelography showed an extradural meningeal cyst extending from the T-12 to L-4 levels in the thoracolumbar region with a dural defect and a valve-like mechanism developed in the enlarging cyst. Operative findings revealed a dural defect that allowed communication between the extradural cyst cavity and the subarachnoid space. Surgical resection of the cyst wall and repair of the dural defect resolved the symptoms.

Long-term outcome of unruptured giant cerebral aneurysms.

The long-term outcome of 39 patients with unruptured giant aneurysm (>2.5 cm) treated during the last 12 years was retrospectively reviewed. The 7 male and 32 female patients, aged 32 to 81 years, presented with symptoms related to compression of the surrounding structures by the aneurysm in 28 cases, cerebral infarction in one, and asymptomatic in 10. The locations were the internal carotid artery (ICA) in 27 cases, middle cerebral artery in three, anterior cerebral artery in one, and basilar artery in eight. Therapeutic modalities were direct clipping in 11 patients, ICA occlusion combined with extracranial-intracranial bypass in 15, and conservative treatment in 13. The follow-up period ranged from 16 to 128 months (mean 54.0 months). The mortality was 9% (1/11), 0% (0/15), and 31% (4/13), and morbidity was 18% (2/11), 20% (3/15), and 8% (1/13), respectively. Surgery reduced the mortality (from 31% to 4%) but increased the morbidity (from 8% to 19%) as compared with conservatively treated patients (p < 0.05). Giant intracranial aneurysm has a poor prognosis if left untreated, but these lesions are difficult to treat with the present management options.

Safety and tolerability of navigated TMS for preoperative mapping in neurosurgical patients.

OBJECTIVE: Navigated transcranial magnetic stimulation (nTMS) is a non-invasive technique for pre-surgical motor and language mapping in patients with brain lesions. This study examines the safety and tolerability of nTMS in a large, multi-center cohort of neurosurgical patients. METHODS: Functional mapping with monopulse and repetitive nTMS was performed in 733 patients. In this cohort, 57% of patients had left-sided tumors, 50% had frontal tumors, and 50% had seizures secondary to the lesion. Side effects and pain intensity related to the procedure were documented. RESULTS: Patients undergoing monopulse stimulation underwent an average of 490 pulses while those undergoing repetitive stimulation received an average of 2268 pulses. During monopulse stimulation, 5.1% reported discomfort (VAS 1-3), and 0.4% reported pain (VAS>3). During repetitive stimulation, 23.4% reported discomfort and 69.5% reported pain. No seizures or other adverse events were observed. CONCLUSIONS: nTMS is safe and well-tolerated in neurosurgical patients. Clinicians should consider expanding nTMS to patients with frequent seizures, but more evaluation is necessary to evaluate this risk fully. SIGNIFICANCE: nTMS is safe and well-tolerated, even in neurosurgical patients with persistent occasional seizure secondary to a lesion. It should be considered in any patient with a lesion in a presumed peri-eloquent or eloquent brain region.

Cerebral venous ischemia by dural arteriovenous fistulas.

Intracranial dural arteriovenous fistulas (AVFs) are potentially at risk for hemorrhage, and their symptoms and prognosis are highly variable. We present 7 surgical cases with the initial symptoms of venous ischemia by dural AVF. The series comprises 3 male and 4 female, ranging in age from 37 to 76 years (mean age, 61.1 years). Initial symptoms were dizziness in 3 cases, headache in 2 cases, unconsciousness in 1 case, and hemiparesis in 1 case. The locations included the superior sagittal sinus in 3 cases and the transverse-sigmoid sinus in 4 cases. Computed tomography with contrast media and magnetic resonance imaging revealed abnormal vessels. In all cases, retrograde feeding into the cortical veins was observed. On angiography, multiple retrograde venous drainage into the cortical veins were observed in all cases. Single photon emission computed tomography (SPECT) demonstrated apparent hypoperfusion in all 7 cases and further decrease by diamox challenging test in 4 cases. The dural AVFs were removed, and the symptoms disappeared in all cases, although transient aphasia was observed in a single case postoperatively. Postoperative SPECT showed improvement of cerebral blood flow in 4 and no change in 2 of 6 follow-up cases. Cerebral ischemia was induced by venous hypertension, and the hypoperfused brain improved immediately after the operation. Cerebral venous ischemia is a reversible condition that can be improved by appropriate early-stage treatment.

[Extracranial vertebral artery dissection repeating distal embolism: case report].

We report a case of vertebral artery (VA) dissection presenting with repeated distal embolism. A 48-year-old man was admitted to our institution with neck pain and vertigo of two day duration. MRI on arrival showed infarction of the left cerebellar hemisphere and the right occipital lobe. Emergency angiography revealed left VA dissection and occlusion of the left posterior inferior cerebellar artery and right calcaline artery. Six hours after admission, he manifested cortical blindness due to embolism of the contralateral left calcaline artery despite giving heparin and aspirin. Thrombolysis was performed but was not successful. We embolized the affected VA to prevent further embolism. We discuss here the indication and timing of treatment for VA dissection presenting with ischemic symptom.

[Vascular endothelial growth factor expression in the rat dural arteriovenous fistula model].

UNLABELLED: Although various mechanisms of the development of dural arteriovenous fistula (DAVF) have been proposed, the pathogenesis of these lesions are still unclear. Recent experimental evidence suggested a role of angiogenic growth factors in the genesis of vascular malformations of the central nervous system. To further investigate the pathogenesis of DAVF, we examined the expression of the angiogenic growth factor, vascular endothelial growth factor (VEGF), in rat DAVF model. MATERIAL AND METHODS: Male Wistar rats (weighting 280 to 300 g, n = 40) were used. Each rat was mounted on a stereotaxic frame under general anesthesia. DAVF model (Spetzler et al.) was made by common carotid artery-external jugular vein anastomosis, bipolar coagulation of the vein draining the transverse sinus, and superior sagittal sinus thrombosis (SSS). SSS was thrombosed by a hemostatic agent through 16-gauge needle. Venous hypertension was induced in 30 rats, which were divided into two experimental groups; (1) immunohistological study group (n = 15) and (2) angiography group (n = 15). Immunohistological analysis was performed by VEGF antibody 1 week after, and angiography was done 90 days after the surgery. Developing of angiographical DAVF was observed with the magnifying X-ray camera. Each 5 rats served as sham-operated controls, which received a similar surgery without induction of venous hypertension. RESULTS: VEGF expression and DAVF were not observed in sham group. In immunohistological study group, VEGF expression in the endothelium and the connective tissues of the dura mater in the five rats (33%) and in the neurons in the eleven rats (73%) of the cerebral cortex and the basal ganglia were identified. In angiography group, DAVF formed in 6 among 15 rats (40%). CONCLUSION: The findings of this study provide the first experimental evidence that angiogenic growth factors VEGF may participate in the genesis of DAVF. These results suggest a novel strategy for the management and prevention of DAVF and related disorders.