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Ryan P. Lee, Kimberly A. Foster, Jock C. Lillard, Paul Klimo Jr., David W. Ellison, Brent Orr, and Frederick A. Boop

OBJECTIVE

Thalamopeduncular tumors are a group of pediatric low-grade gliomas that arise at the interface of the thalamus and brainstem peduncle. They typically occur within the first 2 decades of life, presenting with progressive spastic hemiparesis. Treatment strategies, including surgical intervention, have varied significantly. The authors present their experience in the treatment of 13 children, ages 2–15 years, with non-neurofibromatosis–related pilocytic astrocytomas located in the thalamopeduncular region.

METHODS

Between 2003 and 2016, 13 children presenting with progressive spastic hemiparesis due to a pilocytic astrocytoma at the interface of the thalamus and cerebral peduncles were identified. Medical records were reviewed retrospectively for clinical, radiological, pathological, and surgical data. Formalin-fixed, paraffin-embedded tissue was obtained for 12 cases and tested for KIAA1549-BRAF fusion and BRAF V600E point mutation.

RESULTS

On preoperative diffusion tensor imaging tractography (performed in 12 patients), the ipsilateral corticospinal tract was displaced laterally in 1 case (8.3%), medially in 1 case (8.3%), anterolaterally in 10 cases (83%), and posteriorly in no cases. Ten patients underwent resection via a transtemporal, transchoroidal approach, which was chosen to avoid further damage to motor function in cases of tumors that caused anterolateral or medial corticospinal tract displacement. With this approach, complications included hemianopia, oculomotor palsy, and tremor at a rate of 50%. Among the 12 patients with obtainable follow-up (mean 50.9 months), none received adjuvant therapy, and only 2 (17%) experienced recurrence or progression. KIAA1549-BRAF fusions were present in 10 cases (83%), while BRAF V600E was absent (0%). The 2 fusion-negative tumors had clinical features atypical for the series, including multi-focality and infiltration.

CONCLUSIONS

Transcortical, transchoroidal resection of thalamopeduncular tumors through the middle temporal gyrus allows for a high rate of gross-total resection and cure. Diffuse tensor tractography is a critical component of the preoperative planning process to determine the location of white matter tracts in proximity. Molecular status may correlate with clinical features, and the presence of BRAF lesions offers an additional target for future novel therapeutics.

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Michael DeCuypere, Michael S. Muhlbauer, Frederick A. Boop, and Paul Klimo Jr.

OBJECTIVE

Penetrating brain injury in civilians is much less common than blunt brain injury but is more severe overall. Gunshot wounds (GSWs) cause high morbidity and mortality related to penetrating brain injury; however, there are few reports on the management and outcome of intracranial GSWs in children. The goals of this study were to identify clinical and radiological factors predictive for death in children and to externally validate a recently proposed pediatric prognostic scale.

METHODS

The authors conducted a retrospective review of penetrating, isolated GSWs sustained in children whose ages ranged from birth to 18 years and who were treated at 2 major metropolitan Level 1 trauma centers from 1996 through 2013. Several standard clinical, laboratory, and radiological factors were analyzed for their ability to predict death in these patients. The authors then applied the St. Louis Scale for Pediatric Gunshot Wounds to the Head, a scoring algorithm that was designed to provide rapid prognostic information for emergency management decisions. The scale's sensitivity, specificity, and positive and negative predictability were determined, with death as the primary outcome.

RESULTS

Seventy-one children (57 male, 14 female) had a mean age of 14 years (range 19 months to 18 years). Overall mortality among these children was 47.9%, with 81% of survivors attaining a favorable clinical outcome (Glasgow Outcome Scale score ≥ 4). A number of predictors of mortality were identified (all p < 0.05): 1) bilateral fixed pupils; 2) deep nuclear injury; 3) transventricular projectile trajectory; 4) bihemispheric injury; 5) injury to ≥ 3 lobes; 6) systolic blood pressure < 100 mm Hg; 7) anemia (hematocrit < 30%); 8) Glasgow Coma Scale score ≤ 5; and 9) a blood base deficit < −5 mEq/L. Patient age, when converted to a categorical variable (0–9 or 10–18 years), was not predictive. Based on data from the 71 patients in this study, the positive predictive value of the St. Louis scale in predicting death (score ≥ 5) was 78%.

CONCLUSIONS

This series of pediatric cranial GSWs underscores the importance of the initial clinical exam and CT studies along with adequate resuscitation to make the appropriate management decision(s). Based on our population, the St. Louis Scale seems to be more useful as a predictor of who will survive than who will succumb to their injury.

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Joseph H. McAbee, Joseph Modica, Clinton J. Thompson, Alberto Broniscer, Brent Orr, Asim F. Choudhri, Frederick A. Boop, and Paul Klimo Jr.

OBJECT

Cervicomedullary tumors (CMTs) represent a heterogeneous group of intrinsic neoplasms that are typically low grade and generally carry a good prognosis. This single-institution study was undertaken to document the outcomes and current treatment philosophy for these challenging neoplasms.

METHODS

The charts of all pediatric patients with CMTs who received treatment at St. Jude Children’s Research Hospital between January 1988 and May 2013 were retrospectively reviewed. Demographic, surgical, clinical, radiological, pathological, and survival data were collected. Treatment-free survival and overall survival were estimated, and predictors of recurrence were analyzed.

RESULTS

Thirty-one children (16 boys, 15 girls) with at least 12 months of follow-up data were identified. The median age at diagnosis was 6 years (range 7 months-17 years) and the median follow-up was 4.3 years. Low-grade tumors (Grade I or II) were present in 26 (84%) patients. Thirty patients underwent either a biopsy alone or resection, with the majority of patients undergoing biopsy only (n = 12, 39%) or subtotal resection (n = 14, 45%). Only 4 patients were treated solely with resection; 21 patients received radiotherapy alone or in combination with other treatments. Recurrent tumor developed in 14 children (45%) and 4 died as a result of their malignancy. A high-grade pathological type was the only independent variable that predicted recurrence. The 5- and 10-year treatment-free survival estimates are 64.7% and 45.3%, respectively. The 5- and 10-year overall survival estimate is 86.7%.

CONCLUSIONS

Children with CMTs typically have low-grade neoplasms and consequently long-term survival, but high risk of recurrence. Therapy should be directed at achieving local tumor control while preserving and even restoring neurological function.

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Joseph H. McAbee, Brian T. Ragel, Shirley McCartney, G. Morgan Jones, L. Madison Michael II, Michael DeCuypere, Joseph S. Cheng, Frederick A. Boop, and Paul Klimo Jr.

OBJECT

The object of this study was to identify and quantify predictors of burnout and career satisfaction among US neurosurgeons.

METHODS

All US members (3247) of the American Association of Neurological Surgeons (AANS) were invited to participate in a survey between September and December 2012. Responses were evaluated through univariate analysis. Factors independently associated with burnout and career satisfaction were determined using multivariable logistic regression. Subgroup analysis of academic and nonacademic neurosurgeons was performed as well.

RESULTS

The survey response rate was 24% (783 members). The majority of respondents were male, 40–60 years old, in a stable relationship, with children, working in a group or university practice, and trained in a subspecialty. More than 80% of respondents reported being at least somewhat satisfied with their career, and 70% would choose a career in neurosurgery again; however, only 26% of neurosurgeons believed their professional lives would improve in the future, and 52% believed it would worsen. The overall burnout rate was 56.7%. Factors independently associated with both burnout and career satisfaction included achieving a balance between work and life outside the hospital (burnout OR 0.45, satisfaction OR 10.0) and anxiety over future earnings and/or health care reform (burnout OR 1.96, satisfaction OR 0.32). While the burnout rate for nonacademic neurosurgeons (62.9%) was higher than that for academic neurosurgeons (47.7%), academicians who had practiced for over 20 years were less likely to be satisfied with their careers.

CONCLUSIONS

The rates of burnout and career satisfaction were both high in this survey study of US neurosurgeons. The negative effects of burnout on the lives of surgeons, patients, and their families require further study and probably necessitate the development of interventional programs at local, regional, and even national levels.

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Frederick A. Boop, Berkeley Bate, Asim F. Choudhri, Brian Burkholder, and Paul Klimo Jr.

The development of high-quality intraoperative MRI (iMRI) capability has offered a major advance in the care of patients with complex intracranial disease. To date, this technology has been limited by the need for pin fixation of the calvaria. The authors report their preliminary experience with an MRI-compatible horseshoe headrest that allows for the following: 1) iMRI in patients too young for pin fixation; 2) iMRI in patients with large calvarial defects; 3) the ability to move the head during iMRI surgery; and 4) the use of neuronavigation in such cases. The authors report 2 cases of infants in whom the Visius Surgical Theatre horseshoe headrest (IMRIS Inc.) was used. Image quality was equivalent to that of pin fixation. The infants suffered no skin issues. The use of neuronavigation with the system remained accurate and could be updated with the new iMRI information. The Visius horseshoe headrest offers a technical advance in iMRI technology for infants, for patients with cranial defects or prior craniotomies in whom pin fixation may not be safe, or for patients in whom the need to move the head during surgery is required. The image quality of the system remains excellent, and the ability to merge new images to the neuronavigation system is helpful.

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Paul Klimo Jr., Garrett T. Venable, Frederick A. Boop, and Thomas E. Merchant

OBJECT

In this paper the authors present their experience treating children with recurrent craniopharyngioma who were initially managed with surgery followed by conformal radiation therapy (CRT).

METHODS

A departmental oncology information system was queried to identify all children (< 18 years old) who received CRT for a craniopharyngioma between 1998 and 2010 (inclusive) and specifically those who experienced tumor progression. For each patient, the authors recorded the type of recurrence (solid, cystic, or both), the time interval to first progression and each subsequent progression, the associated treatment complications, and disease status at last follow-up evaluation.

RESULTS

Among the 97 patients that met criteria for entry into this study, 18 (18.6%) experienced tumor progression (9 cystic, 3 solid, 6 cystic and solid). The median time to first recurrence was 4.62 years (range 1.81–9.11 years). The subgroup included 6 female and 12 male patients with a median age of 7.54 years (range 3.61–13.83 years). Ten patients experienced first progression within 5 years of CRT. The 5- and 10-year treatment-free survival rates for the entire cohort were 89.0% (95% confidence interval [CI] 80.5%–93.9%) and 76.2% (95% CI 64%–85%), respectively. Seven patients had a single episode of progression and 11 had more than 1. The time interval between each subsequent progression was progressively shorter. The 18 patients underwent 38 procedures. The median follow-up duration for this group was 9.32 years (range 4.04–19.0 years). Three patients died, including 1 from perioperative complications.

CONCLUSIONS

Craniopharyngioma progression after prior irradiation is exceedingly difficult to treat and local control is challenging despite repeated surgical procedures. Given our results, gross-total resection may need to be the surgical goal at the time of first recurrence, if possible. Decompressing new cyst formation alone has a low rate of long-term success.

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Cody L. Nesvick, Clinton J. Thompson, Frederick A. Boop, and Paul Klimo Jr.

Object

Observational studies, such as cohort and case-control studies, are valuable instruments in evidence-based medicine. Case-control studies, in particular, are becoming increasingly popular in the neurosurgical literature due to their low cost and relative ease of execution; however, no one has yet systematically assessed these types of studies for quality in methodology and reporting.

Methods

The authors performed a literature search using PubMed/MEDLINE to identify all studies that explicitly identified themselves as “case-control” and were published in the JNS Publishing Group journals (Journal of Neurosurgery, Journal of Neurosurgery: Pediatrics, Journal of Neurosurgery: Spine, and Neurosurgical Focus) or Neurosurgery. Each paper was evaluated for 22 descriptive variables and then categorized as having either met or missed the basic definition of a case-control study. All studies that evaluated risk factors for a well-defined outcome were considered true case-control studies. The authors sought to identify key features or phrases that were or were not predictive of a true case-control study. Those papers that satisfied the definition were further evaluated using the Strengthening the Reporting of Observational Studies in Epidemiology (STROBE) checklist.

Results

The search detected 67 papers that met the inclusion criteria, of which 32 (48%) represented true case-control studies. The frequency of true case-control studies has not changed with time. Use of odds ratios (ORs) and logistic regression (LR) analysis were strong positive predictors of true case-control studies (for odds ratios, OR 15.33 and 95% CI 4.52–51.97; for logistic regression analysis, OR 8.77 and 95% CI 2.69–28.56). Conversely, negative predictors included focus on a procedure/intervention (OR 0.35, 95% CI 0.13–0.998) and use of the word “outcome” in the Results section (OR 0.23, 95% CI 0.082–0.65). After exclusion of nested case-control studies, the negative correlation between focus on a procedure/intervention and true case-control studies was strengthened (OR 0.053, 95% CI 0.0064–0.44). There was a trend toward a negative association between the use of survival analysis or Kaplan-Meier curves and true case-control studies (OR 0.13, 95% CI 0.015–1.12). True case-control studies were no more likely than their counterparts to use a potential study design “expert” (OR 1.50, 95% CI 0.57–3.95). The overall average STROBE score was 72% (range 50–86%). Examples of reporting deficiencies were reporting of bias (28%), missing data (55%), and funding (44%).

Conclusions

The results of this analysis show that the majority of studies in the neurosurgical literature that identify themselves as “case-control” studies are, in fact, labeled incorrectly. Positive and negative predictors were identified. The authors provide several recommendations that may reverse the incorrect and inappropriate use of the term “case-control” and improve the quality of design and reporting of true case-control studies in neurosurgery.

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John H. Sampson and Fred G. Barker II

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Paul Klimo Jr., Clinton J. Thompson, Brian T. Ragel, and Frederick A. Boop

Object

Neurosurgeons are inundated with vast amounts of new clinical research on a daily basis, making it difficult and time-consuming to keep up with the latest literature. Meta-analysis is an extension of a systematic review that employs statistical techniques to pool the data from the literature in order to calculate a cumulative effect size. This is done to answer a clearly defined a priori question. Despite their increasing popularity in the neurosurgery literature, meta-analyses have not been scrutinized in terms of reporting and methodology.

Methods

The authors performed a literature search using PubMed/MEDLINE to locate all meta-analyses that have been published in the JNS Publishing Group journals (Journal of Neurosurgery, Journal of Neurosurgery: Pediatrics, Journal of Neurosurgery: Spine, and Neurosurgical Focus) or Neurosurgery. Accepted checklists for reporting (PRISMA) and methodology (AMSTAR) were applied to each meta-analysis, and the number of items within each checklist that were satisfactorily fulfilled was recorded. The authors sought to answer 4 specific questions: Are meta-analyses improving 1) with time; 2) when the study met their definition of a meta-analysis; 3) when clinicians collaborated with a potential expert in meta-analysis; and 4) when the meta-analysis was the only focus of the paper?

Results

Seventy-two meta-analyses were published in the JNS Publishing Group journals and Neurosurgery between 1990 and 2012. The number of published meta-analyses has increased dramatically in the last several years. The most common topics were vascular, and most were based on observational studies. Only 11 papers were prepared using an established checklist. The average AMSTAR and PRISMA scores (proportion of items satisfactorily fulfilled divided by the total number of eligible items in the respective instrument) were 31% and 55%, respectively. Major deficiencies were identified, including the lack of a comprehensive search strategy, study selection and data extraction, assessment of heterogeneity, publication bias, and study quality. Almost one-third of the papers did not meet our basic definition of a meta-analysis. The quality of reporting and methodology was better 1) when the study met our definition of a meta-analysis; 2) when one or more of the authors had experience or expertise in conducting a meta-analysis; 3) when the meta-analysis was not conducted alongside an evaluation of the authors' own data; and 4) in more recent studies.

Conclusions

Reporting and methodology of meta-analyses in the neurosurgery literature is excessively variable and overall poor. As these papers are being published with increasing frequency, neurosurgical journals need to adopt a clear definition of a meta-analysis and insist that they be created using checklists for both reporting and methodology. Standardization will ensure high-quality publications.