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Courtney S. McGuire, Kristin L. Sainani and Paul G. Fisher

Object

Previous small studies disagree about which clinical risk factors influence ependymoma incidence. The authors analyzed a large, population-based cancer registry to examine the relationship of incidence to patient age, sex, race, and tumor location, and to determine incidence trends over the past 3 decades.

Methods

Data were obtained from the Surveillance, Epidemiology, and End Results (SEER-9) study, which was conducted from 1973 to 2003. Histological codes were used to define ependymomas. Age-adjusted incidence rates were compared by confidence intervals in the SEER*Stat 6.2 program. Multiplicative Poisson regression and Joinpoint analysis were used to determine annual percentage change and to look for sharp changes in incidence, respectively.

Results

From the SEER database, 1402 patients were identified. The incidence rate per 100,000 person-years was significantly higher in male than in female patients (males 0.227 ± 0.029, females 0.166 ± 0.03). For children, the age at diagnosis differed significantly by tumor location, with the mean age for patients with infratentorial tumors calculated as 5 ± 0.4 years; for supratentorial tumors it was 7.77 ± 0.6 years, and for spinal lesions it was 12.16 ± 0.8 years. (Values are expressed as the mean ± standard error [SE].) Adults showed no difference in the mean age of incidence by location, although most tumors in this age group were spinal. Between 1973 and 2003, the incidence increased significantly among adults but not among children, and there were no sharp changes at any single year, both before and after age adjustment.

Conclusions

Males have a higher incidence of ependymoma than do females. A biological explanation remains elusive. Ependymoma occurs within the CNS at distinct locations at different ages, consistent with hypotheses postulating distinct populations of radial glial stem cells within the CNS. Ependymoma incidence appears to have increased over the past 3 decades, but only in adults.

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Benjamin S. Carson, Jon D. Weingart, Michael Guarnieri and Paul G. Fisher

✓ This 9-year-old boy with a history of behavioral problems and worsening psychosis responded initially to treatment with haloperidol. However, a magnetic resonance image obtained as part of his psychiatric evaluation revealed an anterior third ventricle tumor and mild-to-moderate hydrocephalus. The resected tumor was found on pathological examination to be a choroid plexus papilloma. The patient had an uneventful postoperative course and remained free of psychosis or mood disorder at 1-year follow-up examination.

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Y. Raja Rampersaud, Paul A. Anderson, John R. Dimar II, Charles G. Fisher and on behalf of the Spine Trauma Study Group and Degenerative Spine Study Group

OBJECTIVE

Reporting of adverse events (AEs) in spinal surgery uses inconsistent definitions and severity grading, making it difficult to compare results between studies. The Spinal Adverse Events Severity System, version 2 (SAVES-V2) aims to standardize the classification of spine surgery AEs; however, its inter- and intraobserver reliability are unknown. The objective of this study was to assess inter- and intraobserver reliability of the SAVES-V2 grading system for assessing AEs in spinal surgery.

METHODS

Two multinational, multicenter surgical study groups assessed surgical case vignettes (10 trauma and 12 degenerative cases) for AE occurrence by using SAVES-V2. Thirty-four members of the Spine Trauma Study Group (STSG) and 17 members of the Degenerative Spine Study Group (DSSG) participated in the first round of case vignettes. Six months later, the same case vignettes were randomly reorganized and presented in an otherwise identical manner. Inter- and intraobserver agreement on the presence, severity, number, and type of AE, as well as the impact of the AE on length of stay (LOS) were assessed using intraclass correlation (ICC), Cohen's kappa value, and the percentage of participants in agreement.

RESULTS

Agreement on the presence of AEs ranged from 97% to 100% in the 2 groups. Severity classification showed substantial interobserver (ICC = 0.75 for both groups) and intraobserver (ICC = 0.70 in DSSG, 0.71 in STSG) agreement. Judgments on the number of AEs showed high interobserver agreement and moderate intraobserver agreement in both groups. Both the STSG and DSSG had high intraobserver agreement on the type of AE; interobserver agreement for AE type was high in the STSG and fair in the DSSG. Agreement on impact of the AE on LOS was excellent in the DSSG and fair in the STSG.

CONCLUSIONS

There was good agreement on the presence, severity, and number of AEs in both trauma and degenerative cases in using the SAVES-V2. This grading system is a simple, reliable tool for identifying and capturing AEs in spinal surgery.

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Melanie G. Hayden Gephart, Robert M. Lober, Robert T. Arrigo, Corinna C. Zygourakis, Raphael Guzman, Maxwell Boakye, Michael S. B. Edwards and Paul G. Fisher

Object

Pediatric primary spinal cord tumors (PSCTs) are rare, with limited comprehensive data regarding incidence and patterns of diagnosis and treatment. The authors evaluated trends in the diagnosis and treatment of PSCTs using a nationwide database.

Methods

The Surveillance, Epidemiology, and End Results (SEER) registry was queried for the years 1975–2007, evaluating clinical patterns in 330 patients 19 years of age or younger in whom a pediatric PSCT had been diagnosed. Histological diagnoses were grouped into pilocytic astrocytoma, other low-grade astrocytoma, ependymoma, and high-grade glioma. Patient demographics, tumor pathology, use of external beam radiation (EBR), and overall survival were analyzed.

Results

The incidence of pediatric PSCT was 0.09 case per 100,000 person-years and did not change over time. Males were more commonly affected than females (58% vs 42%, respectively; p < 0.006). Over the last 3 decades, the specific diagnoses of pilocytic astrocytoma and ependymoma increased, whereas the use of EBR decreased (60.6% from 1975 to 1989 vs 31.3% from 1990 to 2007; p < 0.0001). The 5- and 10-year survival rates did not differ between these time periods.

Conclusions

While the incidence of pediatric PSCT has not changed over time, the pattern of pathological diagnoses has shifted, and pilocytic astrocytoma and ependymoma have been increasingly diagnosed. The use of EBR over time has declined. Relative survival of patients with low-grade PSCT has remained high regardless of the pathological diagnosis.

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Mohanpal S. Dulai, Dario V. Caccamo, Anita L. Briley, Michael S. B. Edwards, Paul G. Fisher and Norman L. Lehman

This 8-year-old girl presented with a papillary ependymoma in the thoracic spinal cord. Resection was followed by recurrence at the primary site and later in the lumbosacral thecal sac, followed by cerebrospinal fluid dissemination to the brain approximately 5 years after her initial presentation. The tumor showed cytological and immunohistochemical features overlapping those of classic ependymomas and choroid plexus tumors similar to those seen in uncommon supratentorial papillary ependymomas, also known as papillary tumors of the pineal region. The histopathological and clinical courses of this rare spinal papillary ependymoma exhibiting mixed ependymal and choroid plexus–like differentiation are discussed.

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Daniel M. Sciubba, Mohamed Macki, Mohamad Bydon, Niccole M. Germscheid, Jean-Paul Wolinsky, Stefano Boriani, Chetan Bettegowda, Dean Chou, Alessandro Luzzati, Jeremy J. Reynolds, Zsolt Szövérfi, Patti Zadnik, Laurence D. Rhines, Ziya L. Gokaslan, Charles G. Fisher and Peter Paul Varga

OBJECT

Clinical outcomes in patients with primary spinal osteochondromas are limited to small series and sporadic case reports. The authors present data on the first long-term investigation of spinal osteochondroma cases.

METHODS

An international, multicenter ambispective study on primary spinal osteochondroma was performed. Patients were included if they were diagnosed with an osteochondroma of the spine and received surgical treatment between October 1996 and June 2012 with at least 1 follow-up. Perioperative prognostic variables, including patient age, tumor size, spinal level, and resection, were analyzed in reference to long-term local recurrence and survival. Tumor resections were compared using Enneking appropriate (EA) or Enneking inappropriate surgical margins.

RESULTS

Osteochondromas were diagnosed in 27 patients at an average age of 37 years. Twenty-two lesions were found in the mobile spine (cervical, thoracic, or lumbar) and 5 in the fixed spine (sacrum). Twenty-three cases (88%) were benign tumors (Enneking tumor Stages 1–3), whereas 3 (12%) exhibited malignant changes (Enneking tumor Stages IA–IIB). Sixteen patients (62%) underwent en bloc treatment—that is, wide or marginal resection—and 10 (38%) underwent intralesional resection. Twenty-four operations (92%) followed EA margins. No one received adjuvant therapy. Two patients (8%) experienced recurrences: one in the fixed spine and one in the mobile spine. Both recurrences occurred in latent Stage 1 tumors following en bloc resection. No osteochondroma-related deaths were observed.

CONCLUSIONS

In the present study, most patients underwent en bloc resection and were treated as EA cases. Both recurrences occurred in the Stage 1 tumor cohort. Therefore, although benign in character, osteochondromas still require careful management and thorough follow-up.

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Tej D. Azad, Arjun V. Pendharkar, James Pan, Yuhao Huang, Amy Li, Rogelio Esparza, Swapnil Mehta, Ian D. Connolly, Anand Veeravagu, Cynthia J. Campen, Samuel H. Cheshier, Michael S. B. Edwards, Paul G. Fisher and Gerald A. Grant

OBJECTIVE

Pediatric spinal astrocytomas are rare spinal lesions that pose unique management challenges. Therapeutic options include gross-total resection (GTR), subtotal resection (STR), and adjuvant chemotherapy or radiation therapy. With no randomized controlled trials, the optimal management approach for children with spinal astrocytomas remains unclear. The aim of this study was to conduct a systematic review and meta-analysis on pediatric spinal astrocytomas.

METHODS

The authors performed a systematic review of the PubMed/MEDLINE electronic database to investigate the impact of histological grade and extent of resection on overall survival among patients with spinal cord astrocytomas. They retained publications in which the majority of reported cases included astrocytoma histology.

RESULTS

Twenty-nine previously published studies met the eligibility criteria, totaling 578 patients with spinal cord astrocytomas. The spinal level of intramedullary spinal cord tumors was predominantly cervical (53.8%), followed by thoracic (40.8%). Overall, resection was more common than biopsy, and GTR was slightly more commonly achieved than STR (39.7% vs 37.0%). The reported rates of GTR and STR rose markedly from 1984 to 2015. Patients with high-grade astrocytomas had markedly worse 5-year overall survival than patients with low-grade tumors. Patients receiving GTR may have better 5-year overall survival than those receiving STR.

CONCLUSIONS

The authors describe trends in the management of pediatric spinal cord astrocytomas and suggest a benefit of GTR over STR for 5-year overall survival.

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Ziya L. Gokaslan, Patricia L. Zadnik, Daniel M. Sciubba, Niccole Germscheid, C. Rory Goodwin, Jean-Paul Wolinsky, Chetan Bettegowda, Mari L. Groves, Alessandro Luzzati, Laurence D. Rhines, Charles G. Fisher, Peter Pal Varga, Mark B. Dekutoski, Michelle J. Clarke, Michael G. Fehlings, Nasir A. Quraishi, Dean Chou, Jeremy J. Reynolds, Richard P. Williams, Norio Kawahara and Stefano Boriani

OBJECT

A chordoma is an indolent primary spinal tumor that has devastating effects on the patient's life. These lesions are chemoresistant, resistant to conventional radiotherapy, and moderately sensitive to proton therapy; however, en bloc resection remains the preferred treatment for optimizing patient outcomes. While multiple small and largely retrospective studies have investigated the outcomes following en bloc resection of chordomas in the sacrum, there have been few large-scale studies on patients with chordomas of the mobile spine. The goal of this study was to review the outcomes of surgically treated patients with mobile spine chordomas at multiple international centers with respect to local recurrence and survival. This multiinstitutional retrospective study collected data between 1988 and 2012 about prognosis-predicting factors, including various clinical characteristics and surgical techniques for mobile spine chordoma. Tumors were classified according to the Enneking principles and analyzed in 2 treatment cohorts: Enneking-appropriate (EA) and Enneking-inappropriate (EI) cohorts. Patients were categorized as EA when the final pathological assessment of the margin matched the Enneking recommendation; otherwise, they were categorized as EI.

METHODS

Descriptive statistics were used to summarize the data (Student t-test, chi-square, and Fisher exact tests). Recurrence and survival data were analyzed using Kaplan-Meier survival curves, log-rank tests, and multivariate Cox proportional hazard modeling.

RESULTS

A total of 166 patients (55 female and 111 male patients) with mobile spine chordoma were included. The median patient follow-up was 2.6 years (range 1 day to 22.5 years). Fifty-eight (41%) patients were EA and 84 (59%) patients were EI. The type of biopsy (p < 0.001), spinal location (p = 0.018), and if the patient received adjuvant therapy (p < 0.001) were significantly different between the 2 cohorts. Overall, 58 (35%) patients developed local recurrence and 57 (34%) patients died. Median survival was 7.0 years postoperative: 8.4 years postoperative for EA patients and 6.4 years postoperative for EI patients (p = 0.023). The multivariate analysis showed that the EI cohort was significantly associated with an increased risk of local recurrence in comparison with the EA cohort (HR 7.02; 95% CI 2.96–16.6; p < 0.001), although no significant difference in survival was observed.

CONCLUSIONS

EA resection plays a major role in decreasing the risk for local recurrence in patients with chordoma of the mobile spine.

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Charles G. Fisher, Tony Goldschlager, Stefano Boriani, Peter Paul Varga, Laurence D. Rhines, Michael G. Fehlings, Alessandro Luzzati, Mark B. Dekutoski, Jeremy J. Reynolds, Dean Chou, Sigurd H. Berven, Richard P. Williams, Nasir A. Quraishi, Chetan Bettegowda and Ziya L. Gokaslan

Object

The National Institutes of Health recommends strategies to obtain evidence for the treatment of rare conditions such as primary tumors of the spine (PTSs). These tumors have a low incidence and are pathologically heterogeneous, and treatment approaches are diverse. Appropriate evidence-based care is imperative. Failure to follow validated oncological principles may lead to unnecessary mortality and profound morbidity. This paper outlines a scientific model that provides significant evidence guiding the treatment of PTSs.

Methods

A four-stage approach was used: 1) planning: data from large-volume centers were reviewed to provide insight; 2) recruitment: centers were enrolled and provided the necessary infrastructure; 3) retrospective stage: existing medical records were reviewed and completed with survival data; and 4) prospective stage: prospective data collection has been implemented. The AOSpine Knowledge Forum Tumor designed six modules: demographic, clinical, diagnostic, therapeutic, local recurrence, survival, and perioperative morbidity data fields and provided funding.

Results

It took 18 months to implement Stages 1–3, while Stage 4 is ongoing. A total of 1495 tumor cases were captured and diagnosed as one of 18 PTS histotypes. In addition, a PTS biobank network has been created to link clinical data with tumor pathology and molecular analysis.

Conclusions

This scientific model has not only aggregated a large amount of PTS data, but has also established an international collaborative network of spine oncology centers. Access to large volumes of data will generate further research to guide and enhance PTS clinical management. This model could be applied to other rare neoplastic conditions. Clinical trial registration no.: NCT01643174 (ClinicalTrials.gov).

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James S. Harrop, Alexander R. Vaccaro, R. John Hurlbert, Jared T. Wilsey, Eli M. Baron, Christopher I. Shaffrey, Charles G. Fisher, Marcel F. Dvorak, F. C. Öner, Kirkham B. Wood, Neel Anand, D. Greg Anderson, Moe R. Lim, Joon Y. Lee, Christopher M. Bono, Paul M. Arnold, Y. Raja Rampersaud, Michael G. Fehlings and The Spine Trauma Study Group

Object

A new classification and treatment algorithm for thoracolumbar injuries was recently introduced by Vaccaro and colleagues in 2005. A thoracolumbar injury severity scale (TLISS) was proposed for grading and guiding treatment for these injuries. The scale is based on the following: 1) the mechanism of injury; 2) the integrity of the posterior ligamentous complex (PLC); and 3) the patient’s neurological status. The reliability and validity of assessing injury mechanism and the integrity of the PLC was assessed.

Methods

Forty-eight spine surgeons, consisting of neurosurgeons and orthopedic surgeons, reviewed 56 clinical thoracolumbar injury case histories. Each was classified and scored to determine treatment recommendations according to a novel classification system. After 3 months the case histories were reordered and the physicians repeated the exercise. Validity of this classification was good among reviewers; the vast majority (> 90%) agreed with the system’s treatment recommendations. Surgeons were unclear as to a cogent description of PLC disruption and fracture mechanism.

Conclusions

The TLISS demonstrated acceptable reliability in terms of intra- and interobserver agreement on the algorithm’s treatment recommendations. Replacing injury mechanism with a description of injury morphology and better definition of PLC injury will improve inter- and intraobserver reliability of this injury classification system.