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Gregory W. Albert and Murat Gokden

Solitary fibrous tumors of the spine are rare lesions. Their description in the literature is limited to case reports and small case series. While generally benign lesions, they can recur and occasionally occur as malignancies. Here the authors present the case of a 10-year-old boy, the youngest patient and first preadolescent reported thus far, with this condition. In addition, they perform a comprehensive review of all previously published cases of spinal solitary fibrous tumors.

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Gregory W. Albert and Arnold H. Menezes

Object

The objective of this study was to describe a single surgeon's experience managing craniovertebral junction (CVJ) disease due to ankylosing spondylitis.

Methods

The authors undertook a retrospective review of the records of patients with CVJ disease due to ankylosing spondylitis who were evaluated and treated by the senior author. Charts were reviewed for symptoms and signs at presentation, radiography results, treatment, and outcome. In addition, some of the patients had pathology reports available for review.

Results

Eight patients with CVJ disease due to ankylosing spondylitis were identified who were evaluated by the senior author in the years 1990–2008. The most common presenting symptoms were neck pain (37.5%), cranial neuropathy (37.5%), and sensory disturbance (62.5%). On examination, the most common findings were limited cervical range of motion (37.5%), weakness (50%), and myelopathy (75%). Radiographic evaluation revealed atlantoaxial subluxation, retroodontoid pannus formation, basilar invagination, and bone erosion. Surgery was offered to all of the patients, 7 of whom underwent operations. In most cases, the treatment was transoral–transpalatopharyngeal decompression followed by occipitocervical fusion. One patient with a reducible lesion underwent dorsal fusion alone. Neurological outcomes were favorable overall.

Conclusions

The incidence of CVJ disease in patients with ankylosing spondylitis varies among reports. These cases are rare in most neurosurgery clinics. It is important to recognize that patients with ankylosing spondylitis are at risk for CVJ disease, similar to patients with rheumatoid arthritis. Principles common to the management of other CVJ pathologies apply to these patients as well.

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Angela W. Palmer and Gregory W. Albert

OBJECTIVE

Various surgical techniques have been described to treat subdural fluid collections in infants, including transfontanelle aspiration, burr holes, subdural drain, subduroperitoneal shunt, and minicraniotomy. The purpose of this study was to describe a modification of the minicraniotomy technique that avoids the implantation of external drainage catheters and potentially carries a higher success rate.

METHODS

In this retrospective study, the authors describe 11 cases involving pediatric patients who underwent parietal minicraniotomies for the evacuation of subdural fluid collections. In contrast to cases previously described in the literature, no patient received a drain; instead, a subgaleal pocket was created such that the fluid could flow from the subdural to the subgaleal space. Preoperative and postoperative data were reviewed, including neurological examination findings, radiological findings, complications, hospital length of stay, and findings on follow-up examinations and imaging. The primary outcome was failure of the treatment strategy, defined as an increase in subdural fluid collection requiring further intervention.

RESULTS

Eleven patients (8 boys and 3 girls, median age 4.5 months) underwent the described procedure. Eight of the patients had complete resolution of the subdural collection on follow-up imaging, and 2 had improvement. One patient had a new subdural collection due to a second injury. Only 1 patient underwent aspiration and subsequent surgical repair of a pseudomeningocele after the initial surgery. Notably, no patients required subduroperitoneal shunt placement.

CONCLUSIONS

The authors describe a new surgical option for subdural fluid collections in infants that allows for more aggressive evacuation of the subdural fluid and eliminates the need for a drain or shunt placement. Further work with more patients and direct comparison to other alternative therapies is necessary to fully evaluate the efficacy and safety of this new technique.

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Gregory W. Albert, Nader S. Dahdaleh, Chandan Reddy, Daniel R. Hansen, Timothy W. Vogel, Hiroto Kawasaki and Matthew A. Howard III

Object

In this study the authors sought to determine whether any correlations existed between postimplantation head CT findings and the need to perform decompression surgery in patients with grid electrodes.

Methods

The authors identified 74 patients who underwent intracranial electrode monitoring for medically refractory epilepsy from January 2000 through June 2008. Only the 46 patients who had head CT scans available for review were included in the study. The authors were able to determine the number and types of electrodes placed as well as complications experienced. They reviewed the CT scans for abnormal findings including extraaxial fluid collections, intracranial hemorrhages, and signs of mass effect.

Results

All patients developed some degree of extraaxial fluid collection following the placement of intracranial electrodes. The maximum width of the extraaxial fluid collection and the degree of midline shift were not predictive of the need for decompressive surgery. The presence, but not degree, of midline shift was associated with the need for decompressive surgery. Likewise, the presence of ventricular asymmetry was correlated with the need for removal of the electrodes and bone flap. Patients without midline shift or ventricular asymmetry on CT did not require decompressive surgery.

Conclusions

After undergoing placement of intracranial electrodes all patients develop extraaxial fluid collections. In addition, many patients develop signs of mass effect including midline shift and ventricular asymmetry. When these findings are absent it is highly unlikely that surgical decompression is required.

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Gregory W. Albert, Arnold H. Menezes, Daniel R. Hansen, Jeremy D. W. Greenlee and Stuart L. Weinstein

Object

The authors conducted a study to evaluate the unique presenting signs and symptoms of Chiari malformation Type I (CM-I) in children younger than 6 years of age and highlight the benefits of early surgical treatment in this patient population.

Methods

The authors reviewed the medical records of patients who presented to the neurosurgery department before their 6th birthday and subsequently underwent surgery for CM-I. They identified 39 patients who had been evaluated between 1984 and 2007 and examined the medical records for presentation, surgical intervention, and outcome.

Results

Children aged 0–2 years commonly presented with oropharyngeal dysfunction (77.8%). Children aged 3–5 years more frequently presented with syringomyelia (85.7%), scoliosis (38.1%), and/or headache (57.1%). All patients underwent posterior fossa craniectomy. Additionally, in many patients cervical laminectomy and/or duraplasty was performed. A few patients required transoral decompression and occipitocervical fusion. In most cases, surgery led to resolution or dramatic improvement of initial symptoms.

Conclusions

Early recognition and surgical treatment of CM-I in young children leads to good outcomes in the majority of patients. Additional therapies for oropharyngeal dysfunction, syringomyelia, and scoliosis can frequently be avoided.

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Gregory W. Albert, George M. Ibrahim, Hiroshi Otsubo, Ayako Ochi, Cristina Y. Go, O. Carter Snead III, James M. Drake and James T. Rutka

Object

Resective surgery is increasingly used in the management of pediatric epilepsy. Frequently, invasive monitoring with subdural electrodes is required to adequately map the epileptogenic focus. The risks of invasive monitoring include the need for 2 operations, infection, and CSF leak. The aim of this study was to evaluate the feasibility and outcomes of resective epilepsy surgery guided by magnetoencephalography (MEG) in children who would have otherwise been candidates for electrode implantation.

Methods

The authors reviewed the records of patients undergoing resective epilepsy surgery at the Hospital for Sick Children between 2001 and 2010. They identified cases in which resections were based on MEG data and no intracranial recordings were performed. Each patient's chart was reviewed for presentation, MRI findings, MEG findings, surgical procedure, pathology, and surgical outcome.

Results

Sixteen patients qualified for the study. All patients had localized spike clusters on MEG and most had abnormal findings on MRI. Resection was carried out in each case based on the MEG data linked to neuronavigation and supplemented with intraoperative neuromonitoring. Overall, 62.5% of patients were seizure free following surgery, and 20% of patients experienced an improvement in seizures without attaining seizure freedom. In 2 cases, additional surgery was performed subsequently with intracranial monitoring in attempts to obtain seizure control.

Conclusions

MEG is a viable alternative to invasive monitoring with intracranial electrodes for planning of resective surgery in carefully selected pediatric patients with localization-related epilepsy. Good candidates for this approach include patients who have a well-delineated, localized spike cluster on MEG that is concordant with findings of other preoperative evaluations and patients with prior brain pathologies that make the implantation of subdural and depth electrodes somewhat problematic.

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Blake C. Phillips, Michael Gelsomino, Ambre' L. Pownall, Eylem Ocal, Horace J. Spencer, Mark S. O'Brien and Gregory W. Albert

Object

Many patients with myelomeningocele (MMC) develop hydrocephalus, and most will undergo CSF diversion. The goal of this retrospective study was to determine whether there was a change in the shunt rate over the 7 consecutive years of the study. The authors will also identify the criteria used to determine the need for shunt placement.

Methods

During a 7-year period, 73 patients underwent MMC closure at Arkansas Children's Hospital. The shunt rate for each year was calculated. Clinical characteristics were evaluated, including apneic and bradycardic spells, CSF leak, level of the MMC, head circumference, and rate of head growth. In addition, radiological images were reviewed, and the frontooccipital horn ratio (FOHR), ventricular index (VI), and thalamooccipital distance (TOD) were calculated. Comparisons were made between those patients who underwent shunt placement and those who did not.

Results

One patient was excluded due to death in the perinatal period. Of the 72 remaining patients, 54 (75%) underwent placement of a ventriculoperitoneal shunt. This rate did not change significantly over time. Between the cohorts with and without a shunt there was no significant difference in age, sex, or race. There was no significant difference in apneic episodes or bradycardic episodes. There was a statistically significant difference in fontanelle characteristics, head circumference at birth, and rate of head growth. Patients who required CSF diversion had a mean head growth of 0.32 cm/day compared with those who did not receive a shunt (0.13 cm/day; p < 0.05). All radiological parameters were found to be statistically significant.

Conclusions

In this study, several classic indicators of hydrocephalus in the neonate were not found to be significantly associated with the need for CSF diversion. Fontanelle characteristics, head circumference at birth, and head growth velocity were associated with the need for shunt placement. Imaging information including the VI, TOD, and FOHR are statistically significant measures to evaluate prior to placement of a ventriculoperitoneal shunt. The optimal patient with MMC for CSF diversion will have full to tense fontanelle, increasing head circumference of more than 3 mm/day, and radiological evidence of an elevated VI, TOD, and/or FOHR.

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Andrew T. Hale, P. David Adelson, Gregory W. Albert, Philipp R. Aldana, Tord D. Alden, Richard C. E. Anderson, David F. Bauer, Christopher M. Bonfield, Douglas L. Brockmeyer, Joshua J. Chern, Daniel E. Couture, David J. Daniels, Susan R. Durham, Richard G. Ellenbogen, Ramin Eskandari, Timothy M. George, Gerald A. Grant, Patrick C. Graupman, Stephanie Greene, Jeffrey P. Greenfield, Naina L. Gross, Daniel J. Guillaume, Gregory G. Heuer, Mark Iantosca, Bermans J. Iskandar, Eric M. Jackson, James M. Johnston, Robert F. Keating, Jeffrey R. Leonard, Cormac O. Maher, Francesco T. Mangano, J. Gordon McComb, Thanda Meehan, Arnold H. Menezes, Brent O’Neill, Greg Olavarria, Tae Sung Park, John Ragheb, Nathan R. Selden, Manish N. Shah, Matthew D. Smyth, Scellig S. D. Stone, Jennifer M. Strahle, Scott D. Wait, John C. Wellons, William E. Whitehead, Chevis N. Shannon, David D. Limbrick Jr. and for the Park-Reeves Syringomyelia Research Consortium Investigators

OBJECTIVE

Factors associated with syrinx size in pediatric patients undergoing posterior fossa decompression (PFD) or PFD with duraplasty (PFDD) for Chiari malformation type I (CM-I) with syringomyelia (SM; CM-I+SM) are not well established.

METHODS

Using the Park-Reeves Syringomyelia Research Consortium registry, the authors analyzed variables associated with syrinx radiological outcomes in patients (< 20 years old at the time of surgery) with CM-I+SM undergoing PFD or PFDD. Syrinx resolution was defined as an anteroposterior (AP) diameter of ≤ 2 mm or ≤ 3 mm or a reduction in AP diameter of ≥ 50%. Syrinx regression or progression was defined using 1) change in syrinx AP diameter (≥ 1 mm), or 2) change in syrinx length (craniocaudal, ≥ 1 vertebral level). Syrinx stability was defined as a < 1-mm change in syrinx AP diameter and no change in syrinx length.

RESULTS

The authors identified 380 patients with CM-I+SM who underwent PFD or PFDD. Cox proportional hazards modeling revealed younger age at surgery and PFDD as being independently associated with syrinx resolution, defined as a ≤ 2-mm or ≤ 3-mm AP diameter or ≥ 50% reduction in AP diameter. Radiological syrinx resolution was associated with improvement in headache (p < 0.005) and neck pain (p < 0.011) after PFD or PFDD. Next, PFDD (p = 0.005), scoliosis (p = 0.007), and syrinx location across multiple spinal segments (p = 0.001) were associated with syrinx diameter regression, whereas increased preoperative frontal-occipital horn ratio (FOHR; p = 0.007) and syrinx location spanning multiple spinal segments (p = 0.04) were associated with syrinx length regression. Scoliosis (HR 0.38 [95% CI 0.16–0.91], p = 0.03) and smaller syrinx diameter (5.82 ± 3.38 vs 7.86 ± 3.05 mm; HR 0.60 [95% CI 0.34–1.03], p = 0.002) were associated with syrinx diameter stability, whereas shorter preoperative syrinx length (5.75 ± 4.01 vs 9.65 ± 4.31 levels; HR 0.21 [95% CI 0.12–0.38], p = 0.0001) and smaller pB-C2 distance (6.86 ± 1.27 vs 7.18 ± 1.38 mm; HR 1.44 [95% CI 1.02–2.05], p = 0.04) were associated with syrinx length stability. Finally, younger age at surgery (8.19 ± 5.02 vs 10.29 ± 4.25 years; HR 1.89 [95% CI 1.31–3.04], p = 0.01) was associated with syrinx diameter progression, whereas increased postoperative syrinx diameter (6.73 ± 3.64 vs 3.97 ± 3.07 mm; HR 3.10 [95% CI 1.67–5.76], p = 0.003), was associated with syrinx length progression. PFD versus PFDD was not associated with syrinx progression or reoperation rate.

CONCLUSIONS

These data suggest that PFDD and age are independently associated with radiological syrinx improvement, although forthcoming results from the PFDD versus PFD randomized controlled trial (NCT02669836, clinicaltrials.gov) will best answer this question.

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Jennifer M. Strahle, Rukayat Taiwo, Christine Averill, James Torner, Chevis N. Shannon, Christopher M. Bonfield, Gerald F. Tuite, Tammy Bethel-Anderson, Jerrel Rutlin, Douglas L. Brockmeyer, John C. Wellons III, Jeffrey R. Leonard, Francesco T. Mangano, James M. Johnston, Manish N. Shah, Bermans J. Iskandar, Elizabeth C. Tyler-Kabara, David J. Daniels, Eric M. Jackson, Gerald A. Grant, Daniel E. Couture, P. David Adelson, Tord D. Alden, Philipp R. Aldana, Richard C. E. Anderson, Nathan R. Selden, Lissa C. Baird, Karin Bierbrauer, Joshua J. Chern, William E. Whitehead, Richard G. Ellenbogen, Herbert E. Fuchs, Daniel J. Guillaume, Todd C. Hankinson, Mark R. Iantosca, W. Jerry Oakes, Robert F. Keating, Nickalus R. Khan, Michael S. Muhlbauer, J. Gordon McComb, Arnold H. Menezes, John Ragheb, Jodi L. Smith, Cormac O. Maher, Stephanie Greene, Michael Kelly, Brent R. O’Neill, Mark D. Krieger, Mandeep Tamber, Susan R. Durham, Greg Olavarria, Scellig S. D. Stone, Bruce A. Kaufman, Gregory G. Heuer, David F. Bauer, Gregory Albert, Jeffrey P. Greenfield, Scott D. Wait, Mark D. Van Poppel, Ramin Eskandari, Timothy Mapstone, Joshua S. Shimony, Ralph G. Dacey Jr., Matthew D. Smyth, Tae Sung Park and David D. Limbrick Jr.

OBJECTIVE

Scoliosis is frequently a presenting sign of Chiari malformation type I (CM-I) with syrinx. The authors’ goal was to define scoliosis in this population and describe how radiological characteristics of CM-I and syrinx relate to the presence and severity of scoliosis.

METHODS

A large multicenter retrospective and prospective registry of pediatric patients with CM-I (tonsils ≥ 5 mm below the foramen magnum) and syrinx (≥ 3 mm in axial width) was reviewed for clinical and radiological characteristics of CM-I, syrinx, and scoliosis (coronal curve ≥ 10°).

RESULTS

Based on available imaging of patients with CM-I and syrinx, 260 of 825 patients (31%) had a clear diagnosis of scoliosis based on radiographs or coronal MRI. Forty-nine patients (5.9%) did not have scoliosis, and in 516 (63%) patients, a clear determination of the presence or absence of scoliosis could not be made. Comparison of patients with and those without a definite scoliosis diagnosis indicated that scoliosis was associated with wider syrinxes (8.7 vs 6.3 mm, OR 1.25, p < 0.001), longer syrinxes (10.3 vs 6.2 levels, OR 1.18, p < 0.001), syrinxes with their rostral extent located in the cervical spine (94% vs 80%, OR 3.91, p = 0.001), and holocord syrinxes (50% vs 16%, OR 5.61, p < 0.001). Multivariable regression analysis revealed syrinx length and the presence of holocord syrinx to be independent predictors of scoliosis in this patient cohort. Scoliosis was not associated with sex, age at CM-I diagnosis, tonsil position, pB–C2 distance (measured perpendicular distance from the ventral dura to a line drawn from the basion to the posterior-inferior aspect of C2), clivoaxial angle, or frontal-occipital horn ratio. Average curve magnitude was 29.9°, and 37.7% of patients had a left thoracic curve. Older age at CM-I or syrinx diagnosis (p < 0.0001) was associated with greater curve magnitude whereas there was no association between syrinx dimensions and curve magnitude.

CONCLUSIONS

Syrinx characteristics, but not tonsil position, were related to the presence of scoliosis in patients with CM-I, and there was an independent association of syrinx length and holocord syrinx with scoliosis. Further study is needed to evaluate the nature of the relationship between syrinx and scoliosis in patients with CM-I.