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Endodermal cyst: rare intraaxial brainstem location. Illustrative cases

Atul Goel, Dikpal Jadhav, Abhidha Shah, Asha Shenoy, and Naina Goel

BACKGROUND

This report detailed an extremely rare location of an endodermal cyst. Such location of the tumor can pose radiological confusion and a therapeutic dilemma. However, when identified, it can be a pleasant surprise for the surgeon and provide the possibility of a symptom-free long life for the patient.

OBSERVATIONS

This report discussed two young patients who presented with relatively short-duration reports of ataxia and diplopia. Investigations revealed intraaxial brainstem lesion. During surgery, thick, pus-like fluid was evacuated and part of a wall was resected. Histology revealed that the lesion was an endodermal cyst. Both patients are well and are lesion- and symptom-free for 24 and 72 months.

LESSONS

Endodermal cysts are some of the few long-standing and benign intraaxial brainstem lesions.

In Journal of Neurosurgery: Case Lessons Volume 2 (2021): Issue 11 (Sep 2021)

Open access

Multifocal metastases to choroid plexus from papillary thyroid carcinoma: illustrative case

Isidora R. Beach, Adam M. Olszewski, Alissa A. Thomas, John C. DeWitt, and Brandon D. Liebelt

BACKGROUND

Choroid plexus metastases are extremely rare from all types of malignancy, with only 42 cases reported in the literature thus far. Most of these originate from renal cell carcinoma and present as a solitary choroid plexus lesion; only two cases of multifocal choroid plexus metastases have been reported to date.

OBSERVATIONS

The authors report the third case of multifocal metastases to the choroid plexus, that of a 75-year-old man who developed three measurable choroid plexus lesions approximately 3.5 years after undergoing total thyroidectomy and chemotherapy for papillary thyroid carcinoma. He underwent intraventricular biopsy of the largest lesion and subsequently died of hydrocephalus after opting for comfort care only.

LESSONS

This is the third case of multifocal choroid plexus metastasis in the literature and the second case of multifocal metastasis from thyroid carcinoma. As such, the natural disease course is not well characterized. This case is compared with the previous eight reports of choroid plexus metastases from thyroid carcinoma, seven of which involved solitary lesions. The eight prior cases are evaluated with attention to treatment modalities used and factors potentially influencing prognosis, specifically those that might contribute to hydrocephalus, a reported complication for this pathology.

In Journal of Neurosurgery: Case Lessons Volume 2 (2021): Issue 11 (Sep 2021)

Open access

Multiple VHL-related hemangioblastomas and holocord syrinx: identifying the causative lesion. Illustrative case

Armin Mortazavi, Diana Nwokoye, David T. Asuzu, Gretchen Scott, Panagiotis Mastorakos, and Prashant Chittiboina

BACKGROUND

Brainstem and spinal cord hemangioblastomas are a common manifestation of von Hippel–Lindau (VHL) disease. Cysts and associated syringes are the most common cause of significant morbidity in these patients. Surgical treatment of symptomatic hemangioblastomas are often complicated by the presence of multiple potential lesions, leading to cyst and syrinx formation.

OBSERVATIONS

The authors present a case of a patient with multiple VHL-related hemangioblastomas who presented with syringobulbia and holocord syrinx. Resection of two cyst wall hemangioblastomas and one cervical hemangioblastoma only transiently improved syringobulbia. Eventual resolution of syringobulbia and collapse of the holocord syrinx only occurred following removal of a large lower thoracic hemangioblastoma.

LESSONS

Surgical management of hemangioblastomas and associated cysts in patients with VHL should only target lesions most likely contributing to neurological deficits as excess surgical intervention risks treatment-related morbidity. The authors illustrate how anatomical and pathophysiological considerations as well as patient symptoms are key to identifying target lesions for resection and developing deliberate treatment plans.

In Journal of Neurosurgery: Case Lessons Volume 2 (2021): Issue 11 (Sep 2021)

Open access

Sinking skin flap syndrome in a patient with bone resorption after cranioplasty and ventriculoperitoneal shunt placement: illustrative case

Camryn R. Rohringer, Taryn J. Rohringer, Sumit Jhas, and Mehdi Shahideh

BACKGROUND

Sinking skin flap syndrome (SSFS) is an uncommon complication that can follow decompressive craniectomy. Even less common is the development of SSFS following bone resorption after cranioplasty with exacerbation by a ventriculoperitoneal (VP) shunt.

OBSERVATIONS

A 56-year-old male sustained a severe traumatic brain injury and subsequently underwent an emergent decompressive craniectomy. After craniectomy, a cranioplasty was performed, and a VP shunt was placed. The patient returned to the emergency department 5 years later with left-sided hemiplegia and seizures. His clinical presentation was attributed to complete bone flap resorption (BFR) complicated by SSFS likely exacerbated by his VP shunt and the resultant mass effect on the underlying brain parenchyma. The patient underwent surgical intervention via synthetic bone flap replacement. Within 6 days, he recovered to his baseline neurological status.

LESSONS

SSFS after complete BFR is a rare complication following cranioplasty. To the authors’ knowledge, having a VP shunt in situ to exacerbate the clinical picture has yet to be reported in the literature. In addition to presenting the case, the authors also describe an effective treatment strategy of decompressing the brain and elevating the scalp flap while addressing the redundant tissue, then using a synthetic mesh to reconstruct the calvarial defect while keeping the shunt in situ.

In Journal of Neurosurgery: Case Lessons Volume 2 (2021): Issue 11 (Sep 2021)

Open access

Successful management of spinal cord ischemia in a pediatric patient with fibrocartilaginous embolism: illustrative case

Augustinas Fedaravičius, Yael Feinstein, Isaac Lazar, Micky Gidon, Ilan Shelef, Elad Avraham, Arimantas Tamašauskas, and Israel Melamed

BACKGROUND

Fibrocartilaginous embolism (FCE) is a rare cause of ischemic myelopathy that occurs when the material of the nucleus pulposus migrates into vessels supplying the spinal cord. The authors presented a case of pediatric FCE that was successfully managed by adapting evidence-based recommendations used for spinal cord neuroprotection in aortic surgery.

OBSERVATIONS

A 7-year-old boy presented to the emergency department with acute quadriplegia and hemodynamic instability that quickly progressed to cardiac arrest. After stabilization, the patient regained consciousness but remained in a locked-in state with no spontaneous breathing. The patient presented a diagnostic challenge. Traumatic, inflammatory, infectious, and ischemic etiologies were considered. Eventually, the clinical and radiological findings led to the presumed diagnosis of FCE. Treatment with continuous cerebrospinal fluid drainage (CSFD), pulse steroids, and mean arterial pressure augmentation was applied, with subsequent considerable and consistent neurological improvement.

LESSONS

The authors proposed consideration of the adaptation of spinal cord neuroprotection principles used routinely in aortic surgery for the management of traumatic spinal cord ischemia (FCE-related in particular), namely, permissive arterial hypertension and CSFD. This is hypothesized to allow for the maintenance of sufficient spinal cord perfusion until adequate physiological blood perfusion is reestablished (remodeling of the collateral arterial network and/or clearing/absorption of the emboli).

In Journal of Neurosurgery: Case Lessons Volume 2 (2021): Issue 11 (Sep 2021)

Open access

Syringomyelia intermittens: highlighting the complex pathophysiology of syringomyelia. Illustrative case

Jorn Van Der Veken, Marguerite Harding, Saba Hatami, Marc Agzarian, and Nick Vrodos

BACKGROUND

Chiari Type I malformation (CM1) is a disorder recognized by caudal displacement of the cerebellar tonsils through the foramen magnum and into the cervical canal. Syringomyelia is frequently found in patients with CM1, but the pathophysiology of syringomyelia remains an enigma. As a general consensus, symptomatic patients should be treated and asymptomatic patients without a syrinx should not be treated. Mildly symptomatic patients or asymptomatic patients with a syrinx, on the other hand, pose a more challenging dilemma, as the natural evolution is uncertain. For many surgeons, the presence of a syrinx is an indication to offer surgery even if the patient is asymptomatic or mildly symptomatic.

OBSERVATIONS

The authors describe an illustrative case of a 31-year-old female with an incidental finding of a CM1 malformation and cervical syrinx in 2013. Conservative management was advocated as the patient was asymptomatic. Monitoring of the syrinx over a course of 8 years showed resolution, followed by reappearance and finally a complete resolution in 2021. A review of the literature and the possible pathophysiology is discussed.

LESSONS

The unusual course of this patient highlights the importance of guiding treatment by clinical symptoms, not radiological findings. Furthermore it reflects the complexity of the pathophysiology and the uncertain natural history of syringomyelia.

In Journal of Neurosurgery: Case Lessons Volume 2 (2021): Issue 11 (Sep 2021)

Restricted access

Assessing the rate, natural history, and treatment trends of intracranial aneurysms in patients with intracranial dural arteriovenous fistulas: a Consortium for Dural Arteriovenous Fistula Outcomes Research (CONDOR) investigation

Isaac Josh Abecassis, R. Michael Meyer, Michael R. Levitt, Jason P. Sheehan, Ching-Jen Chen, Bradley A. Gross, Ashley Lockerman, W. Christopher Fox, Waleed Brinjikji, Giuseppe Lanzino, Robert M. Starke, Stephanie H. Chen, Adriaan R. E. Potgieser, J. Marc C. van Dijk, Andrew Durnford, Diederik Bulters, Junichiro Satomi, Yoshiteru Tada, Amanda Kwasnicki, Sepideh Amin-Hanjani, Ali Alaraj, Edgar A. Samaniego, Minako Hayakawa, Colin P. Derdeyn, Ethan Winkler, Adib Abla, Pui Man Rosalind Lai, Rose Du, Ridhima Guniganti, Akash P. Kansagra, Gregory J. Zipfel, and Louis J. Kim

OBJECTIVE

There is a reported elevated risk of cerebral aneurysms in patients with intracranial dural arteriovenous fistulas (dAVFs). However, the natural history, rate of spontaneous regression, and ideal treatment regimen are not well characterized. In this study, the authors aimed to describe the characteristics of patients with dAVFs and intracranial aneurysms and propose a classification system.

METHODS

The Consortium for Dural Arteriovenous Fistula Outcomes Research (CONDOR) database from 12 centers was retrospectively reviewed. Analysis was performed to compare dAVF patients with (dAVF+ cohort) and without (dAVF-only cohort) concomitant aneurysm. Aneurysms were categorized based on location as a dAVF flow-related aneurysm (FRA) or a dAVF non–flow-related aneurysm (NFRA), with further classification as extra- or intradural. Patients with traumatic pseudoaneurysms or aneurysms with associated arteriovenous malformations were excluded from the analysis. Patient demographics, dAVF anatomical information, aneurysm information, and follow-up data were collected.

RESULTS

Of the 1077 patients, 1043 were eligible for inclusion, comprising 978 (93.8%) and 65 (6.2%) in the dAVF-only and dAVF+ cohorts, respectively. There were 96 aneurysms in the dAVF+ cohort; 10 patients (1%) harbored 12 FRAs, and 55 patients (5.3%) harbored 84 NFRAs. Dural AVF+ patients had higher rates of smoking (59.3% vs 35.2%, p < 0.001) and illicit drug use (5.8% vs 1.5%, p = 0.02). Sixteen dAVF+ patients (24.6%) presented with aneurysm rupture, which represented 16.7% of the total aneurysms. One patient (1.5%) had aneurysm rupture during follow-up. Patients with dAVF+ were more likely to have a dAVF located in nonconventional locations, less likely to have arterial supply to the dAVF from external carotid artery branches, and more likely to have supply from pial branches. Rates of cortical venous drainage and Borden type distributions were comparable between cohorts. A minority (12.5%) of aneurysms were FRAs. The majority of the aneurysms underwent treatment via either endovascular (36.5%) or microsurgical (15.6%) technique. A small proportion of aneurysms managed conservatively either with or without dAVF treatment spontaneously regressed (6.2%).

CONCLUSIONS

Patients with dAVF have a similar risk of harboring a concomitant intracranial aneurysm unrelated to the dAVF (5.3%) compared with the general population (approximately 2%–5%) and a rare risk (0.9%) of harboring an FRA. Only 50% of FRAs are intradural. Dural AVF+ patients have differences in dAVF angioarchitecture. A subset of dAVF+ patients harbor FRAs that may regress after dAVF treatment.

In Journal of Neurosurgery Publish Before Print

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Bilateral open sacroiliac joint fusion during adult spinal deformity surgery using triangular titanium implants: technique description and presentation of 21 cases

Christopher T. Martin, Kenneth J. Holton, Kristen E. Jones, Jonathan N. Sembrano, and David W. Polly Jr.

OBJECTIVE

Pelvic fixation enhances long constructs during deformity surgery. Subsequent loosening of iliac screws and pain at the pelvis occur in as many as 29% of patients. Concomitant sacroiliac (SI) fusion may prevent potential pain and failure. The objective of this study was to describe a novel surgical technique and a single institution’s experience using bilateral SI fusion during adult deformity surgery with S2-alar-iliac (S2AI) screws and triangular titanium rods (TTRs) placed with navigation.

METHODS

The authors reviewed open SI joint fusions with TTR performed between August 2019 and March 2020. All patients underwent lumbosacral fusion through a midline approach and bilateral S2AI pelvic fixation in the caudal teardrop, followed by TTR placement just proximal and cephalad to the S2AI screws using intraoperative CT imaging guidance.

RESULTS

Twenty-one patients were identified who received 42 TTRs, ranging in size from 7.0 × 65 mm to 7.0 × 90 mm. Three TTRs (7%) were malpositioned intraoperatively, and each was successfully repositioned during index surgery without negative sequelae. All breaches occurred in a medial and cephalad direction into the pelvis. Incremental operative time for adding TTR averaged 8 minutes and 33 seconds per implant.

CONCLUSIONS

Image-guided open SI joint fusion with TTR during lumbosacral fusion is technically feasible. The bony corridor for implant placement is narrower cephalad, and implants tend to deviate medially into the pelvis. Detection of malpositioned implant is aided with intraoperative CT, but this can be salvaged. A prospective randomized clinical trial is underway that will better inform the impact of this technique on patient outcomes.

In Journal of Neurosurgery: Spine Publish Before Print

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Comparative analysis of the combined petrosal and the pretemporal transcavernous anterior petrosal approach to the petroclival region

Mohamed A. Labib, Xiaochun Zhao, Lena Mary Houlihan, Irakliy Abramov, Joshua S. Catapano, Komal Naeem, Mark C. Preul, A. Samy Youssef, and Michael T. Lawton

OBJECTIVE

The combined petrosal (CP) approach has been traditionally used to resect petroclival meningioma (PCM). The pretemporal transcavernous anterior petrosal (PTAP) approach has emerged as an alternative. A quantitative comparison of both approaches has not been made. This anatomical study compared the surgical corridors afforded by both approaches and identified key elements of the approach selection process.

METHODS

Twelve cadaveric specimens were dissected, and 10 were used for morphometric analysis. Groups A and B (n = 5 in each) underwent the CP and PTAP approaches, respectively. The area of drilled clivus, lengths of cranial nerves (CNs) II–X, length of posterior circulation vessels, surgical area of exposure of the brainstem, and angles of attack anterior and posterior to a common target were measured and compared.

RESULTS

The area of drilled clivus was significantly greater in group A than group B (mean ± SD 88.7 ± 17.1 mm2 vs 48.4 ± 17.9 mm2, p < 0.01). Longer segments of ipsilateral CN IV (52.4 ± 2.33 mm vs 46.5 ± 3.71 mm, p < 0.02), CN IX, and CN X (9.91 ± 3.21 mm vs 0.00 ± 0.00 mm, p < 0.01) were exposed in group A than group B. Shorter portions of CN II (9.31 ± 1.28 mm vs 17.6 ± 6.89 mm, p < 0.02) and V1 (26.9 ± 4.62 mm vs 32.4 ± 1.93 mm, p < 0.03) were exposed in group A than group B. Longer segments of ipsilateral superior cerebellar artery (SCA) were exposed in group A than group B (36.0 ± 4.91 mm vs 25.8 ± 3.55 mm, p < 0.02), but there was less exposure of contralateral SCA (0.00 ± 0.00 mm vs 7.95 ± 3.33 mm, p < 0.01). There was no statistically significant difference between groups with regard to the combined area of the exposed cerebral peduncles and pons (p = 0.75). Although exposure of the medulla was limited, group A had significantly greater exposure of the medulla than group B (p < 0.01). Finally, group A had a smaller anterior angle of attack than group B (24.1° ± 5.62° vs 34.8° ± 7.51°, p < 0.03).

CONCLUSIONS

This is the first study to quantitatively identify the advantages and limitations of the CP and PTAP approaches from an anatomical perspective. Understanding these data will aid in designing maximally effective yet minimally invasive approaches to PCM.

In Journal of Neurosurgery Publish Before Print

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Consortium for Dural Arteriovenous Fistula Outcomes Research (CONDOR): rationale, design, and initial characterization of patient cohort

Ridhima Guniganti, Enrico Giordan, Ching-Jen Chen, Isaac Josh Abecassis, Michael R. Levitt, Andrew Durnford, Jessica Smith, Edgar A. Samaniego, Colin P. Derdeyn, Amanda Kwasnicki, Ali Alaraj, Adriaan R. E. Potgieser, Samir Sur, Stephanie H. Chen, Yoshiteru Tada, Ethan Winkler, Ryan R. L. Phelps, Pui Man Rosalind Lai, Rose Du, Adib Abla, Junichiro Satomi, Robert M. Starke, J. Marc C. van Dijk, Sepideh Amin-Hanjani, Minako Hayakawa, Bradley A. Gross, W. Christopher Fox, Diederik Bulters, Louis J. Kim, Jason Sheehan, Giuseppe Lanzino, Jay F. Piccirillo, Akash P. Kansagra, and Gregory J. Zipfel

OBJECTIVE

Cranial dural arteriovenous fistulas (dAVFs) are rare lesions, hampering efforts to understand them and improve their care. To address this challenge, investigators with an established record of dAVF investigation formed an international, multicenter consortium aimed at better elucidating dAVF pathophysiology, imaging characteristics, natural history, and patient outcomes. This report describes the design of the Consortium for Dural Arteriovenous Fistula Outcomes Research (CONDOR) and includes characterization of the 1077-patient cohort.

METHODS

Potential collaborators with established interest in the field were identified via systematic review of the literature. To ensure uniformity of data collection, a quality control process was instituted. Data were retrospectively obtained.

RESULTS

CONDOR comprises 14 centers in the United States, the United Kingdom, the Netherlands, and Japan that have pooled their data from 1077 dAVF patients seen between 1990 and 2017. The cohort includes 359 patients (33%) with Borden type I dAVFs, 175 (16%) with Borden type II fistulas, and 529 (49%) with Borden type III fistulas. Overall, 852 patients (79%) presented with fistula-related symptoms: 427 (40%) presented with nonaggressive symptoms such as tinnitus or orbital phenomena, 258 (24%) presented with intracranial hemorrhage, and 167 (16%) presented with nonhemorrhagic neurological deficits. A smaller proportion (224 patients, 21%), whose dAVFs were discovered incidentally, were asymptomatic. Many patients (85%, 911/1077) underwent treatment via endovascular embolization (55%, 587/1077), surgery (10%, 103/1077), radiosurgery (3%, 36/1077), or multimodal therapy (17%, 184/1077). The overall angiographic cure rate was 83% (758/911 treated), and treatment-related permanent neurological morbidity was 2% (27/1467 total procedures). The median time from diagnosis to follow-up was 380 days (IQR 120–1038.5 days).

CONCLUSIONS

With more than 1000 patients, the CONDOR registry represents the largest registry of cranial dAVF patient data in the world. These unique, well-annotated data will enable multiple future analyses to be performed to better understand dAVFs and their management.

In Journal of Neurosurgery Publish Before Print