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Open access

Nathaniel R. Ellens, Matthew C. Miller, Ismat Shafiq, Zoe R. Williams, and G. Edward Vates

BACKGROUND

Rathke cleft cyst (RCC) has a recurrence rate of 10% to 22%, and preventing recurrence is challenging. For patients who experience persistent recurrence of RCC, placement of steroid-eluting bioabsorbable intrasellar stents has been rarely described. However, recurrences are often delayed, suggesting that dissolvable stents may not be successful long-term. The release of steroids in close proximity to the pituitary gland may also unintentionally influence the hypothalamic-adrenal-pituitary axis.

OBSERVATIONS

The authors present a case of a 66-year-old woman with a persistently recurrent RCC who underwent drainage of her cyst with placement of a nonabsorbable intrasellar stent in the form of a tympanostomy tube. After repeat transsphenoidal drainage of her cyst, a tympanostomy T-tube was placed to stent open the dural aperture. Postoperatively, the patient’s condition showed improvement clinically and radiographically.

LESSONS

Placement of an intrasellar stent for recurrent RCC has rarely been described. Steroid-eluting bioabsorbable stents may dissolve before RCC recurrence and may have an unintentional effect on the hypothalamic-pituitary-adrenal axis. The authors present the first case of nonabsorbable stent placement in the form of a tympanostomy tube for recurrence of RCC. Additional studies and longer follow-up are necessary to evaluate the long-term efficacy of both absorbable and nonabsorbable stent placement.

Open access

Jakob V. E. Gerstl, Kristian Aquilina, and Jeffrey E. Florman

BACKGROUND

Colloid cysts of the posterior third ventricle are exceedingly rare. This location is a high-risk zone for colloid cysts because of potential obstruction of the cerebral aqueduct.

OBSERVATIONS

The authors report a case of a 57-year-old man who presented with a 6-month history of progressive headache, short-term memory loss, visual blurring, and an episode of double vision. Magnetic resonance imaging (MRI) revealed a colloid cyst, 22 mm in diameter, attached to the roof of the posterior third ventricle. The posterior third ventricle was obstructed, but both foramina of Monro were patent. The entirety of the cyst was resected via a right frontal parasagittal, interhemispheric, transventricular approach through the foramen of Monro. It was not contained within the velum interpositum. The patient did not experience any postoperative deficits.

LESSONS

This case supports the theoretical evidence that colloid cysts in the posterior zone of the third ventricle pose a risk for obstructive hydrocephalus. This report of a large cyst in a rare location describes a unique lesion and provides the first published MRI description.

Open access

Liqing Xu, Changwei Yuan, Yingjin Wang, Shengli Shen, and Hongzhou Duan

BACKGROUND

Superficial siderosis of the central nervous system (SSCNS) is a rare condition that results from hemosiderin deposition in the brain, brainstem, cerebellum, and spinal cord as a result of chronic, repeated, and recurrent subarachnoid hemorrhage. SSCNS that originates in the spinal cord is rarely reported, and epilepsy as a manifestation of such a case has not been reported before.

OBSERVATIONS

The authors reported a rare case of SSCNS with epilepsy originating from traumatic cervical injury and presented a literature review of all reported SSCNS cases that originated in the spine. The patient was a 29-year-old man with a 16-year history of progressive headache accompanied by seizures, ataxia, and sensorineural hearing loss. He had experienced a traumatic cervical injury at age 7. Magnetic resonance imaging revealed a characteristic hypointense rim around the pons and cervical spinal cord on susceptibility-weighted imaging scans. Cerebrospinal fluid examination during a headache episode confirmed subarachnoid hemorrhage and increased intracranial pressure. Surgical exploration revealed a C6 dural defect with bone spurs inserted into the dura mater. After the patient underwent dura mater repair and shunt implantation, his symptoms disappeared completely except for hearing loss.

LESSONS

This rare case indicated that symptomatic epilepsy followed by SSCNS can be eliminated by complete repair of the cervical dura mater.

Open access

Akira Sugie, Makoto Yamada, Kunio Yokoyama, Tomoaki Miyake, Yutaka Ito, Hidekazu Tanaka, Yukiya Nomura, Masutsugu Fujita, Toshio Nakatani, and Masahiro Kawanishi

BACKGROUND

Intravenous tissue plasminogen activator (IV t-PA) is effective for the treatment of distal artery occlusion. However, after the use of IV t-PA, vascular occlusion in unaffected territories may occur. Early recurrent ischemic stroke (ERIS) is defined as the occurrence of new neurological symptoms that suggest the involvement of initially unaffected vascular territories after intravenous thrombolysis (IVT). The authors reviewed the cases of ERIS that occurred within 24 hours after treatment with IVT.

OBSERVATIONS

A 75-year-old woman with occlusion in the M2 segment of the left middle cerebral artery (MCA) was treated with IV t-PA. However, 360 minutes later, the patient presented with occlusion in the M1 distal segment of the contralateral side, the right MCA, which was recanalized by endovascular treatment. Her modified Rankin Scale score was 4; however, aphasia was not observed. She was transferred to a rehabilitation hospital after 3 months.

LESSONS

ERIS is an extremely rare but catastrophic event. The underlying mechanism of ERIS most likely involves the disintegration and subsequent scattering of a preexisting intracardiac thrombus. Hence, caution must be used when managing not only hemorrhagic complications but also ischemic complications after IV t-PA. Endovascular management may be the only effective treatment for this type of large vessel occlusion.

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Florian Connolly, Joan Alsolivany, Marcus Czabanka, Peter Vajkoczy, Jose M. Valdueza, Jens E. Röhl, Eberhard Siebert, and Leon A. Danyel

OBJECTIVE

Superficial temporal artery–middle cerebral artery (STA-MCA) bypass surgery is an important therapy for symptomatic moyamoya disease. Its success depends on bypass function, which may be impaired by primary or secondary bypass insufficiency. Catheter angiography is the current gold standard to assess bypass function, whereas the diagnostic value of ultrasonography (US) has not been systematically analyzed so far.

METHODS

The authors analyzed 50 STA-MCA bypasses in 39 patients (age 45 ± 14 years [mean ± SD]; 26 female, 13 male). Bypass patency was evaluated by catheter angiography, which was performed within 24 hours after US. The collateral circulation through the bypass was classified into 4 types as follows: the bypass supplies more than two-thirds (type A); between one-third and two-thirds (type B); or less than one-third (type C) of the MCA territory; or there is bypass occlusion (type D). The authors assessed the mean blood flow velocity (BFV), the blood volume flow (BVF), and the pulsatility index (PI) in the external carotid artery and STA by duplex sonography. Additionally, they analyzed the flow direction of the MCA by transcranial color-coded sonography. US findings were compared between bypasses with higher (types A and B) and lower (types C and D) capacity.

RESULTS

Catheter angiography revealed high STA-MCA bypass capacity in 35 cases (type A: n = 22, type B: n = 13), whereas low bypass capacity was noted in the remaining 15 cases (type C: n = 12, type D: n = 3). The BVF values in the STA were 60 ± 28 ml/min (range 4–121 ml/min) in the former and 12 ± 4 ml/min (range 6–18 ml/min) in the latter group (p < 0.0001). Corresponding values of mean BFV and PI were 57 ± 21 cm/sec (range 16–100 cm/sec) versus 22 ± 8 cm/sec (range 10–38 cm/sec) (p < 0.0001) and 0.8 ± 0.2 (range 0.4–1.3) versus 1.4 ± 0.5 (range 0.5–2.4) (p < 0.0001), respectively. Differences in the external carotid artery were less distinct: BVF 217 ± 71 ml/min (range 110–425 ml/min) versus 151 ± 41 ml/min (range 87–229 ml/min) (p = 0.001); mean BFV 47 ± 17 cm/sec (range 24–108 cm/sec) versus 40 ± 7 cm/sec (range 26–50 cm/sec) (p = 0.15); PI 1.5 ± 0.4 (range 1.0–2.5) versus 1.9 ± 0.4 (range 1.2–2.6) (p = 0.009). A retrograde blood flow in the MCA was found in 14 cases (9 in the M1 and M2 segment; 5 in the M2 segment alone), and all of them showed a good bypass function (type A, n = 10; type B, n = 4). The best parameter (cutoff value) to distinguish bypasses with higher capacity from bypasses with lower capacity was a BVF in the STA ≥ 21 ml/min (sensitivity 100%, negative predictive value 100%, specificity 91%, positive predictive value 83%).

CONCLUSIONS

Duplex sonography is a suitable diagnostic tool to assess STA-MCA bypass function in moyamoya disease. Hemodynamic monitoring of the STA by US provides an excellent predictor of bypass patency.

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Allan R. Martin, Sukhvinder Kalsi-Ryan, Muhammad A. Akbar, Anna C. Rienmueller, Jetan H. Badhiwala, Jefferson R. Wilson, Lindsay A. Tetreault, Aria Nouri, Eric M. Massicotte, and Michael G. Fehlings

OBJECTIVE

Degenerative cervical myelopathy (DCM) is among the most common pathologies affecting the spinal cord but its natural history is poorly characterized. The purpose of this study was to investigate functional outcomes in patients with DCM who were managed nonoperatively as well as the utility of quantitative clinical measures and MRI to detect deterioration.

METHODS

Patients with newly diagnosed DCM or recurrent myelopathic symptoms after previous surgery who were initially managed nonoperatively were included. Retrospective chart reviews were performed to analyze clinical outcomes and anatomical MRI scans for worsening compression or increased signal change. Quantitative neurological assessments were collected prospectively, including modified Japanese Orthopaedic Association (mJOA) score; Quick-DASH; graded redefined assessment of strength, sensation, and prehension–myelopathy version (GRASSP–M: motor, sensory, and dexterity); grip dynamometer; Berg balance scale score; gait stability ratio; and gait variability index. A deterioration of 10% was considered significant (e.g., a 2-point decrease in mJOA score).

RESULTS

A total of 117 patients were included (95 newly diagnosed, 22 recurrent myelopathy), including 74 mild, 28 moderate, and 15 severe cases. Over a mean follow-up of 2.5 years, 57% (95% CI 46%–67%) of newly diagnosed patients and 73% (95% CI 50%–88%) of patients with recurrent DCM deteriorated neurologically. Deterioration was best detected with grip strength (60%), GRASSP dexterity (60%), and gait stability ratio (50%), whereas the mJOA score had low sensitivity (33%) in 50 patients. A composite score had a sensitivity of 81% and a specificity of 82%. The sensitivity of anatomical MRI was 28% (83 patients).

CONCLUSIONS

DCM appears to have a poor natural history; however, prospective studies are needed for validation. Serial assessments should include mJOA score, grip strength, dexterity, balance, and gait analysis. The absence of worsening on anatomical MRI or in mJOA scores is not sufficient to determine clinical stability.

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Anna Lindner, Verena Rass, Bogdan-Andrei Ianosi, Alois Josef Schiefecker, Mario Kofler, Max Gaasch, Alberto Addis, Paul Rhomberg, Bettina Pfausler, Ronny Beer, Erich Schmutzhard, Claudius Thomé, and Raimund Helbok

OBJECTIVE

Recent guidelines recommend targeting a systolic blood pressure (SBP) < 140 mm Hg in the early management of patients with spontaneous intracerebral hemorrhage (ICH). The optimal SBP targets for ICH patients after hematoma evacuation (HE) remain unclear. Here, the authors aimed to define the optimal SBP range based on multimodal neuromonitoring data.

METHODS

Forty poor-grade ICH patients who had undergone HE and then monitoring of intracerebral pressure, brain tissue oxygen tension (PbtO2), and cerebral metabolism (via cerebral microdialysis [CMD]) were prospectively included. Episodes of brain tissue hypoxia (BTH) (1-hour averaged PbtO2 < 20 mm Hg) and metabolic distress (CMD–lactate/pyruvate ratio [LPR] ≥ 40) were identified and linked to corresponding parameters of hemodynamic monitoring (SBP and cerebral perfusion pressure [CPP]). Multivariable regression analysis was performed using generalized estimating equations to identify associations between SBP levels, PbtO2, and brain metabolism.

RESULTS

The mean patient age was 60 (range 51–66) years and the median [IQR] initial ICH volume was 47 [29–60] ml. In multivariable models adjusted for Glasgow Coma Scale score, probe location, ICH volume, and age, lower SBP was independently associated with a higher risk of BTH (≤ 120 mm Hg: adjusted OR 2.9, p = 0.007; 120–130 mm Hg: adj OR 2.4, p = 0.002; 130–140 mm Hg: adj OR 1.6, p = 0.017) compared to a reference range of 140–150 mm Hg at the level of the foramen interventriculare Monroi, which corresponded to a CPP of 70–80 mm Hg and SBP levels between 150 and 160 mm Hg at the heart level. After exclusion of episodes with mitochondrial dysfunction, SBP targets < 140 mm Hg were associated with higher odds of cerebral metabolic distress (≤ 130 mm Hg: OR 2.5, p = 0.041; 130–140 mm Hg: OR 2.3, p = 0.033). Patients with a modified Rankin Scale score ≥ 5 at neurological ICU discharge more often exhibited BTH than patients with better outcomes (51% vs 10%, p = 0.003).

CONCLUSIONS

These data suggest that lower SPB and CPP levels are associated with a higher risk for BTH. Further studies are needed to evaluate whether a higher SPB target may prevent BTH and improve outcomes.

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Yuanzhi Xu, Ahmed Mohyeldin, Ayoze Doniz-Gonzalez, Vera Vigo, Felix Pastor-Escartin, Lingzhao Meng, Aaron A Cohen-Gadol, and Juan C Fernandez-Miranda

OBJECTIVE

The lateral posterior choroidal artery (LPChA) should be a major surgical consideration in the microsurgical management of lateral ventricular tumors. Here the authors aim to delineate the microsurgical anatomy of the LPChA by using anatomical microdissections. They describe the trajectory, segments, and variations of the LPChA and discuss the surgical implications when approaching the choroid plexus using different routes.

METHODS

Twelve colored silicone–injected, lightly fixed, postmortem human head specimens were prepared for dissection. The origin, diameter, trunk, course, segment, length, spatial relationships, and anastomosis of the LPChA were investigated. The surgical landmarks of 4 different approaches to the LPChA were also examined thoroughly.

RESULTS

The LPChA was present in 23 hemispheres (96%), and in 14 (61%) it originated from the posterior segment of the P2 (i.e., P2P); most commonly (61%) the LPChA had 2 trunks, and in 17 hemispheres (74%) it had a C-shaped trajectory. According to its course, the authors divided the LPChA into 3 segments: 1) cisternal, from PCA to choroidal fissure (length 10.6 ± 2.5 mm); 2) forniceal, starting at the choroidal fissure, 8.2 ± 5.7 mm posterior to the inferior choroidal point, and terminating at the posterior level of the choroidal fissure (length 28.7 ± 6.8 mm); and 3) pulvinar, starting at the posterior choroidal fissure and terminating in the pulvinar (length 5.9 ± 2.2 mm). The LPChA was divided into 3 patterns according to its entrance into the choroidal fissure: A (anterior) 78%; B (posterior) 13%; and C (mixed) 9%. The transsylvian trans–limen insulae approach provided the best exposure for cisternal and proximal forniceal segments; the lateral transtemporal approach facilitated a more direct approach to the forniceal segment, including cases with posterior entrance; the transparietal transcortical and contralateral posterior interhemispheric transfalcine transprecuneus approaches provided direct access to the pulvinar segment of the LPChA and to the posterior forniceal segment, including cases with posterior choroidal entrance.

CONCLUSIONS

The LPChA typically runs in the medial border of the choroid plexus, which may facilitate its recognition during surgery. The distance between the AChA at the inferior choroidal point and the LPChA is a valuable reference during surgery, but there are cases of posterior choroidal entrance. Most frequently, there are 2 or more LPChA trunks, which makes possible the sacrifice of one trunk feeding the tumor while preserving the other that provides supply to relevant structures. The intraventricular approaches can be selected based on the tumor location and the LPChA anatomy.

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Grégoire Boulouis, Sarah Stricker, Sandro Benichi, Jean-François Hak, Florent Gariel, Manoelle Kossorotoff, Nicolas Garcelon, Annie Harroche, Quentin Alias, Lorenzo Garzelli, Fanny Bajolle, Nathalie Boddaert, Philippe Meyer, Thomas Blauwblomme, and Olivier Naggara

OBJECTIVE

The clinical outcome of pediatric intracerebral hemorrhage (pICH) is rarely reported in a comprehensive way. In this cohort study, systematic review, and meta-analysis of patients with pICH, the authors aimed to describe the basic clinical outcomes of pICH.

METHODS

Children who received treatment for pICH at the authors’ institution were prospectively enrolled in the cohort in 2008; data since 2000 were retrospectively included, and data through October 2019 were analyzed. The authors then searched PubMed and conducted a systematic review of relevant articles published since 1990. Data from the identified populations and patients from the cohort study were pooled into a multicategory meta-analysis and analyzed with regard to clinical outcomes.

RESULTS

Among 243 children screened for inclusion, 231 patients were included. The median (IQR) age at ictus was 9.6 (4.6–12.5) years, and 128 patients (53%) were male. After a median (IQR) follow-up of 33 (13–63) months, 132 patients (57.4%) had a favorable clinical outcome, of whom 58 (44%) had no residual symptoms. Nineteen studies were included in the meta-analysis. Overall, the proportion of children with complete recovery was 27% (95% CI 19%–36%; Q = 49.6; I2 = 76%); of those with residual deficits, the complete recovery rate was 48.1% (95% CI 40%–57%; Q = 75.3; I2 = 81%). When pooled with the cohort study, the aggregate case-fatality rate at the last follow-up was 17.3% (95% CI 12%–24%; Q = 101.6; I2 = 81%).

CONCLUSIONS

Here, the authors showed that 1 in 6 children died after pICH, and the majority of children had residual neurological deficits at the latest follow-up. Results from the cohort study also indicate that children with vascular lesions as the etiology of pICH had significantly better clinical functional outcomes.

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Patrick J Grover, Lauren Harris, Ayman M Qureshi, Adam Rennie, Fergus Robertson, and Greg James

This is the eighth case report of a pediatric dissecting posterior inferior cerebellar artery aneurysm. The authors present the case of a 13-year-old boy who presented with posttraumatic posterior fossa subdural, subarachnoid, and intraventricular hemorrhage with hydrocephalus. Initial vascular imaging findings were negative; however, a high level of suspicion is necessary. The aneurysm was identified on day 20, after recurrence of hydrocephalus, and was treated with endovascular vessel sacrifice. The patient made a good recovery. It is important to consider arterial dissection in pediatric traumatic brain injury, especially with suspicious findings on initial CT scan and clinical presentation out of proportion to the mechanism of injury. Delayed vascular imaging is imperative for appropriate management.