✓ Intracranial dural arteriovenous (AV) fistulas with spinal perimedullary venous drainage are rare lesions that have distinctive clinical, radiological, and therapeutic aspects. Five patients presented with an ascending myelopathy, which extended to involve the brain stem in three cases. Myelography and magnetic resonance imaging showed slightly dilated spinal perimedullary vessels. Spinal angiograms were normal in the arterial phase. Diagnosis was only possible after cerebral angiography, which demonstrated posterior fossa AV fistulas fed by meningeal arteries and draining into spinal perimedullary veins. Endovascular treatment alone resulted in angiographic obliteration of the lesion in three patients. Two patients required surgery in addition to endovascular therapy. One patient died postoperatively, and in one a transient complication of embolization was observed. Improvement after treatment was good in two cases and fair in two. Transverse sinus thrombosis was observed in three cases and was probably the cause of the aberrant venous drainage of the fistula into the spinal perimedullary veins. The pathophysiology is related to spinal cord venous hypertension. These lesions were classified as Type 5 in the Djindjian and Merland classification of dural intracranial AV fistulas. Endovascular therapy is a safe effective method in the treatment of these fistulas and should be tried first.
Y. Pierre Gobin, Andre Rogopoulos, Armand Aymard, Mazen Khayata, Daniel Reizine, Jacques Chiras and Jean-Jacques Merland
Joseph Gabrieli, Frédéric Clarençon, Federico Di Maria, Robert Fahed, Anne-Laure Boch, Vincent Degos, Jacques Chiras and Nader-Antoine Sourour
Intracranial aneurysms are relatively frequently encountered in patients with brain arteriovenous malformations (BAVMs). They may be located on the circle of Willis, on arterial feeders, or even inside the nidus. Because BAVM-associated aneurysms represent a risk factor of bleeding, the question of the timing and modality of their management remains a matter of debate in unruptured BAVMs. The authors present a case of fatal periprocedural rupture of a flow-related aneurysm (FRA) during the removal of the microcatheter after injection of a liquid embolic agent. A 40-year-old man was treated at the authors' institution for the management of a Spetzler-Martin Grade III left unruptured frontal BAVM, revealed by seizures and a focal neurological deficit attributed to flow steal phenomenon. After a multidisciplinary meeting, endovascular treatment was considered to reduce the flow of the BAVM. A proximal FRA located on the feeding internal carotid artery (ICA) was purposely left untreated because it did not meet the criteria of the authors' institution for preventative treatment (i.e., small size [2.5 mm]). During embolization, at the time of microcatheter retrieval, and after glue injection, the aneurysm unexpectedly ruptured. The aneurysm's rupture was attributed to the stress (torsion/flexion) on the ICA caused by the microcatheter removal. Despite the attempts to manage the bleeding, the patient eventually died of the acute increase of intracranial pressure related to the massive subarachnoid hemorrhage. This case highlights a previously unreported mechanism of FRA rupture during BAVM embolization: the stress transmitted to the parent artery during the removal of the microcatheter.
Robert Fahed, Frédéric Clarençon, Nader-Antoine Sourour, Dorian Chauvet, Lise Le Jean, Jacques Chiras and Federico Di Maria
One of the procedural risks in arteriovenous malformation (AVM) embolization is possible migration of the embolic agent into the venous drainage with an incomplete nidus occlusion, which may lead to severe hemorrhagic complications.
This report presents the case of a 29-year-old man who presented with a deep intraparenchymal hematoma on the left side secondary to the spontaneous rupture of a claustral AVM. Upon resorption of the hematoma, the patient underwent an initial therapeutic session of N-butyl-2 cyanoacrylate endovascular embolization, with the purpose of reducing the AVM volume and flow before performing Gamma Knife radiosurgery. After glue injection into one of the arterial feeders, the control angiography showed a partial migration of the glue cast into the straight sinus, with most of the nidus still visible. Because of the bleeding risk due to possible venous hypertension, it was decided to try to retrieve the glue from the vein by using a stent retriever via jugular access. This maneuver allowed a nearly complete removal of the glue cast, thereby restoring normal venous flow drainage. The patient showed no clinical worsening after the procedure.
To the authors’ knowledge, this is the first report of the use of the Solitaire FR device as a rescue glue retriever. This method should be considered by physicians in cases of unintended glue migration into the venous circulation during AVM embolization.
Amr Abdel Kerim, Fabrice Bonneville, Betty Jean, Philippe Cornu, Lise LeJean and Jacques Chiras
The authors report a novel technique of balloon-assisted embolization of a skull base meningioma supplied by a branch of the cavernous segment of the internal carotid artery using liquid embolic agent. A temporarily inflated balloon distal to the meningioma's feeding vessel may improve the access to this small branch and may reduce the chances of unintended reflux during delivery of the liquid embolic agent.
Joseph Gabrieli, Nader-Antoine Sourour, Dorian Chauvet, Federico Di Maria, Jacques Chiras and Frédéric Clarençon
The posterior inferior cerebellar artery (PICA) is a vessel located between the intra- and extracranial circulation. The artery is characterized by a complex embryological development and numerous anatomical variants. The authors present a case of the PICA supplied by both a hypertrophic anterior spinal artery and a hypoplastic bulbar artery. This unusual arrangement somehow completes the list of previously published variants, and the spontaneous rupture of a related aneurysm confirmed the fragility of this network. The authors discuss anatomical and treatment considerations.
Alexis Guédon, Frédéric Clarençon, Federico Di Maria, Charlotte Rosso, Alessandra Biondi, Joseph Gabrieli, Patricia Rojas, Jacques Chiras and Nader Sourour
The authors evaluate the rate and discuss the pathomechanisms of very late (≥ 4-month) ischemic complications after flow-diverter stent (FDS) placement for intracranial aneurysms.
The authors retrospectively reviewed the clinical data of the patients treated at Pitié-Salpêtrière Hospital between January 2010 and September 2014, who underwent FDS placement for intracranial aneurysm. The patients received dual-antiplatelet therapy (clopidogrel and aspirin) 5 days before and 3–6 months after the procedure and then aspirin alone for 6–9 months. An ischemic complication was defined as a sudden focal neurological deficit documented on diffusion-weighted images.
Eighty-six consecutive patients were included. Three (3.5%) patients treated with the Pipeline embolization device experienced a delayed acute ischemic stroke (2 cases of perforator/side-wall branch infarction and 1 case of thromboembolic stroke) with an average delay of 384 days (4 months, 20 months, and 13 months, respectively). The aneurysm locations were the left superior cerebellar artery, the right anterior choroid artery, and the left internal carotid artery (paraclinoid segment), respectively. The complications occurred after the patients had completed the antiaggregation protocol, except for Patient 1, who was receiving aspirin alone because of a spontaneous hematoma. At the acute phase, no in-stent thromboses were found on digital subtraction angiography. In Patient 2, the treated anterior choroid artery was occluded 20 months after the procedure. In Patient 3, a focal stenosis (approximately 40%) of the distal aspect of the FDS, probably caused by intimal hyperplasia, was seen.
Very late ischemic complications after FDS treatment were observed in 3.5% of the cases in the authors' series, some of which occurred as late as more than 1 year after placement.
Delphine Leclercq, Hugues Duffau, Christine Delmaire, Laurent Capelle, Peggy Gatignol, Mathieu Ducros, Jacques Chiras and Stéphane Lehéricy
Diffusion tensor (DT) imaging tractography is increasingly used to map fiber tracts in patients with surgical brain lesions to reduce the risk of postoperative functional deficit. There are few validation studies of DT imaging tractography in these patients. The aim of this study was to compare DT imaging tractography of language fiber tracts by using intraoperative subcortical electrical stimulations.
The authors included 10 patients with low-grade gliomas or dysplasia located in language areas. The MR imaging examination included 3D T1-weighted images for anatomical coregistration, FLAIR, and DT images. Diffusion tensors and fiber tracts were calculated using in-house software. Four tracts were reconstructed in each patient including the arcuate fasciculus, the inferior occipitofrontal fasciculus, and 2 premotor fasciculi (the subcallosal medialis fiber tract and cortical fibers originating from the medial and lateral premotor areas). The authors compared fiber tracts reconstructed using DT imaging with those evidenced using intraoperative subcortical language mapping.
Seventeen (81%) of 21 positive stimulations were concordant with DT imaging fiber bundles (located within 6 mm of a fiber tract). Four positive stimulations were not located in the vicinity of a DT imaging fiber tract. Stimulations of the arcuate fasciculus mostly induced articulatory and phonemic/syntactic disorders and less frequently semantic paraphasias. Stimulations of the inferior occipitofrontal fasciculus induced semantic paraphasias. Stimulations of the premotor-related fasciculi induced dysarthria and articulatory planning deficit.
There was a good correspondence between positive stimulation sites and fiber tracts, suggesting that DT imaging fiber tracking is a reliable technique but not yet optimal to map language tracts in patients with brain lesions. Negative tractography does not rule out the persistence of a fiber tract, especially when invaded by the tumor. Stimulations of the different tracts induced variable language disorders that were specific to each fiber tract.
Eimad Shotar, Matthieu Debarre, Nader-Antoine Sourour, Federico Di Maria, Joseph Gabrieli, Aurélien Nouet, Jacques Chiras, Vincent Degos and Frédéric Clarençon
The authors aimed to design a score for stratifying patients with brain arteriovenous malformation (BAVM) rupture, based on the likelihood of a poor long-term neurological outcome.
The records of consecutive patients with BAVM hemorrhagic events who had been admitted over a period of 11 years were retrospectively reviewed. Independent predictors of a poor long-term outcome (modified Rankin Scale score ≥ 3) beyond 1 year after admission were identified. A risk stratification scale was developed and compared with the intracranial hemorrhage (ICH) score to predict poor outcome and inpatient mortality.
One hundred thirty-five patients with 139 independent hemorrhagic events related to BAVM rupture were included in this analysis. Multivariate logistic regression followed by stepwise analysis showed that consciousness level according to the Glasgow Coma Scale (OR 6.5, 95% CI 3.1–13.7, p < 10−3), hematoma volume (OR 1.8, 95% CI 1.2–2.8, p = 0.005), and intraventricular hemorrhage (OR 7.5, 95% CI 2.66–21, p < 10−3) were independently associated with a poor outcome. A 12-point scale for ruptured BAVM prognostication was constructed combining these 3 factors. The score obtained using this new scale, the ruptured AVM prognostic (RAP) score, was a stronger predictor of a poor long-term outcome (area under the receiver operating characteristic curve [AUC] 0.87, 95% CI 0.8–0.92, p = 0.009) and inpatient mortality (AUC 0.91, 95% CI 0.85–0.95, p = 0.006) than the ICH score. For a RAP score ≥ 6, sensitivity and specificity for predicting poor outcome were 76.8% (95% CI 63.6–87) and 90.8% (95% CI 81.9–96.2), respectively.
The authors propose a new admission score, the RAP score, dedicated to stratifying the risk of poor long-term outcome after BAVM rupture. This easy-to-use scoring system may help to improve communication between health care providers and consistency in clinical research. Only external prospective cohorts and population-based studies will ensure full validation of the RAP scores' capacity to predict outcome after BAVM rupture.
Frédéric Clarençon, Federico Di Maria, Evelyne Cormier, Nader-Antoine Sourour, Eric Enkaoua, Frédéric Sailhan, Christina Iosif, Lise Le Jean and Jacques Chiras
Presurgical devascularization of hypervascular spinal metastases has been shown to be effective in preventing major blood loss during open surgery. Most often, embolization can be performed using polyvinyl alcohol (PVA) microparticles. However, in some cases, the close relationship between the feeders of the metastases and the feeders of the anterior spinal artery (ASA) poses a risk of spinal cord ischemia when PVA microparticle embolization is performed.
The authors present their early experience in the treatment of spinal metastases close to the ASA; in 2 cases they injected Onyx-18, by direct puncture, into hypervascular posterior arch spinal metastases situated close to the ASA.
Two women, one 36 and the other 55 years of age, who presented with spinal lesions (at the posterior arch of C-4 and T-6, respectively) from thyroid and a kidney tumors, were sent to the authors' department to undergo presurgical embolization. After having performed a complete spinal digital subtraction angiography study, a regular angiography catheter was positioned at the ostium of the artery that mainly supplied the lesion. Then, with the patient in the left lateral decubitus position, direct puncture with 18-gauge needles of the lesion was performed using roadmap guidance. Onyx-18 was injected through the needles under biplanar fluoroscopy.
Satisfactory devascularization of the lesions was obtained; the ASA remained patent in both cases. The metastases were surgically removed in both cases within the 48 hours after the embolization and major blood loss did not occur.
Presurgical devascularization of hypervascular spinal metastases close the ASA by direct puncture with Onyx-18 seems to be an effective technique and appears to be safe in terms of the preserving the ASA's patency.
Isabelle Ract, Aurélie Drier, Delphine Leclercq, Nader Sourour, Joseph Gabrieli, Marion Yger, Aurélien Nouet, Didier Dormont, Jacques Chiras and Frédéric Clarençon
The authors report a very rare presentation of traumatic carotid-cavernous fistula (CCF) with extensive edema of the basal ganglia and brainstem because of an anatomical variation of the basal vein of Rosenthal (BVR). A 45-year-old woman was admitted to the authors' institution for left hemiparesis, dysarthria, and a comatose state caused by right orbital trauma from a thin metal rod. Brain MRI showed a right CCF and vasogenic edema of the right side of the brainstem, right temporal lobe, and basal ganglia. Digital subtraction angiography confirmed a high-flow direct CCF and revealed a hypoplastic second segment of the BVR responsible for the hypertension in inferior striate veins and venous congestion. Endovascular treatment was performed on an emergency basis. One month after treatment, the patient's symptoms and MRI signal abnormalities almost totally disappeared.
Basal ganglia and brainstem venous congestion may occur in traumatic CCF in cases of a hypoplastic or agenetic second segment of the BVR and may provoke emergency treatment.