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Syed Hassan A. Akbari, Gabriela R. Oates, Irina Gonzalez-Sigler, Anastasia A. Arynchyna, Justin McCroskey, Elizabeth N. Alford, Tofey J. Leon, Sarah Rutland, James M. Johnston, Jeffrey P. Blount, Curtis J. Rozzelle, and Brandon G. Rocque

OBJECTIVE

There is little research on the effect of social determinants of health on Chiari malformation type I (CM-I). The authors analyzed data on all children evaluated for CM-I at a single institution to assess how socioeconomic factors and race affect the surgical treatment of this population.

METHODS

Medical records of patients treated for CM-I at the authors’ institution between 1992 and 2017 were reviewed. Area Deprivation Index (ADI) and Rural-Urban Commuting Area (RUCA) codes for each patient were used to measure neighborhood disadvantage. Non-Hispanic White patients were compared to non-White patients and Hispanic patients of any race (grouped together as non-White in this study) in terms of insurance status, ADI, and RUCA. Patients with initially benign CM-I, defined as not having undergone surgery within 9 months of their initial visit, were then stratified by having delayed symptom presentation or not, and compared on these same measures.

RESULTS

The sample included 665 patients with CM-I: 82% non-Hispanic White and 18% non-White. The non-White patients were more likely to reside in disadvantaged (OR 3.4, p < 0.001) and urban (OR 4.66, p < 0.001) neighborhoods and to have public health insurance (OR 3.11, p < 0.001). More than one-quarter (29%) of patients underwent surgery. The non-White and non-Hispanic White patients had similar surgery rates (29.5% vs 28.9%, p = 0.895) at similar ages (8.8 vs 9.7 years, p = 0.406). There were no differences by race/ethnicity for symptoms at presentation. Surgical and nonsurgical patients had similar ADI scores (3.9 vs 4.2, p = 0.194), RUCA scores (2.1 vs 2.3, p = 0.252), and private health insurance rates (73.6% vs 74.2%, p = 0.878). A total of 153 patients underwent surgery within 9 months of their initial visit. The remaining 512 were deemed to have benign CM-I. Of these, 40 (7.8%) underwent decompression surgery for delayed symptom presentation. Patients with delayed symptom presentation were from less disadvantaged (ADI 3.2 vs 4.2; p = 0.025) and less rural (RUCA 1.8 vs 2.3; p = 0.023) areas than those who never underwent surgery.

CONCLUSIONS

Although non-White patients were more likely to be socioeconomically disadvantaged, race and socioeconomic disadvantage were not associated with undergoing surgical treatment. However, among patients with benign CM-I, those undergoing decompression for delayed symptom presentation resided in more affluent and urban areas.

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Benjamin C. Kennedy, Joshua Katz, Jacob Lepard, and Jeffrey P. Blount

OBJECTIVE

Stereoelectroencephalography (SEEG) has become widespread in the United States during the past decade. Many pediatric neurosurgeons practicing SEEG may not have had experience with this technique during their formal training, and the literature is mostly limited to single-center series. As a result, implementation of this relatively new technique may vary at different institutions. The authors hypothesized that aspects of SEEG experience, techniques, and outcomes would vary widely among programs across the country.

METHODS

An electronic survey with 35 questions addressing the categories of training and experience, technique, electrode locations, and outcomes was sent to 128 pediatric epilepsy surgeons who were potential SEEG users.

RESULTS

Sixty-one pediatric fellowship-trained epilepsy surgeons in the United States responded to the survey. Eighty-nine percent were actively using SEEG in their practice. Seventy-two percent of SEEG programs were in existence for less than 5 years, and 68% were using SEEG for > 70% of their invasive monitoring. Surgeons at higher-volume centers operated on younger patients (p < 0.001). Most surgeons (70%) spent 1–3 hours per case planning electrode trajectories. Two-thirds of respondents reported a median implant duration of 5–7 days, but 16% reported never having an implant duration > 5 days, and 16% reported having had implants stay in place for > 4 weeks. The median response for the median number of electrodes initially implanted was 12 electrodes, although 19% of respondents reported median implants of 5–8 electrodes and 17% reported median implants of 15–18 electrodes. Having a higher volume of SEEG cases per year was associated with a higher median number of electrodes implanted (p < 0.001). Most surgeons found SEEG helpful in defining an epileptic network and reported that most of their SEEG patients undergo focal surgical treatment.

CONCLUSIONS

SEEG has been embraced by the pediatric epilepsy surgery community. Higher case volume is correlated with a tendency to place more electrodes and operate on younger patients. For most parameters addressed in the survey, responses from surgeons clustered around a norm, though additional findings of substantial variations highlight differences in implementation and philosophy among pediatric epilepsy programs.

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Benjamin C. Kennedy, Joshua Katz, Jacob Lepard, and Jeffrey P. Blount

OBJECTIVE

Stereoelectroencephalography (SEEG) has become widespread in the United States during the past decade. Many pediatric neurosurgeons practicing SEEG may not have had experience with this technique during their formal training, and the literature is mostly limited to single-center series. As a result, implementation of this relatively new technique may vary at different institutions. The authors hypothesized that aspects of SEEG experience, techniques, and outcomes would vary widely among programs across the country.

METHODS

An electronic survey with 35 questions addressing the categories of training and experience, technique, electrode locations, and outcomes was sent to 128 pediatric epilepsy surgeons who were potential SEEG users.

RESULTS

Sixty-one pediatric fellowship-trained epilepsy surgeons in the United States responded to the survey. Eighty-nine percent were actively using SEEG in their practice. Seventy-two percent of SEEG programs were in existence for less than 5 years, and 68% were using SEEG for > 70% of their invasive monitoring. Surgeons at higher-volume centers operated on younger patients (p < 0.001). Most surgeons (70%) spent 1–3 hours per case planning electrode trajectories. Two-thirds of respondents reported a median implant duration of 5–7 days, but 16% reported never having an implant duration > 5 days, and 16% reported having had implants stay in place for > 4 weeks. The median response for the median number of electrodes initially implanted was 12 electrodes, although 19% of respondents reported median implants of 5–8 electrodes and 17% reported median implants of 15–18 electrodes. Having a higher volume of SEEG cases per year was associated with a higher median number of electrodes implanted (p < 0.001). Most surgeons found SEEG helpful in defining an epileptic network and reported that most of their SEEG patients undergo focal surgical treatment.

CONCLUSIONS

SEEG has been embraced by the pediatric epilepsy surgery community. Higher case volume is correlated with a tendency to place more electrodes and operate on younger patients. For most parameters addressed in the survey, responses from surgeons clustered around a norm, though additional findings of substantial variations highlight differences in implementation and philosophy among pediatric epilepsy programs.

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Mark S. Dias, Ming Wang, Elias B. Rizk, Robin Bowman, Michael D. Partington, Jeffrey P. Blount, Brandon G. Rocque, Betsy Hopson, Daria Ettinger, Amy Lee, William O. Walker, and on behalf of the National Spina Bifida Patient Registry Group

OBJECTIVE

The aims of this study were to review the National Spina Bifida Patient Registry (NSBPR) data set to study the rates of tethered spinal cord release (TCR) among patients with myelomeningocele and variability between centers, to compare TCR rates between males and females, and to study the relationships between TCR rates and other condition-specific characteristics.

METHODS

The NSBPR registry was queried to identify all patients with myelomeningocele. TCR rates were calculated over time using survival analyses; rates between centers and between males and females were compared. Cox proportional hazards models were constructed to identify relationships between TCR rates and sex, functional lesion level, ambulation status, treated hydrocephalus, and prior Chiari decompression.

RESULTS

Of 6339 patients with information about their operations, 1366 (21.5%) underwent TCR, with significant variability between centers. The majority (75.8%) underwent a single TCR. The annual TCR rate was linear between birth and 13 years (1.8%/year) but declined sharply from 14 to 21 years (0.7%/year). There was no period of time at which the TCR rate accelerated. There were no significant differences in TCR rates between males and females. TCR rate was not related to functional lesion level but was lower among nonambulators compared with community ambulators (p = 0.005) and among those with treated hydrocephalus (HR 0.30, p < 0.001), and higher among those having prior Chiari decompression (HR 1.71, p < 0.001).

CONCLUSIONS

These results extend the results of prior single-institution studies, demonstrate significant treatment variability between institutions, and challenge the traditional concept that tethering is related to spinal cord stretching due to spinal growth.

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Brandon G. Rocque, Raymond P. Waldrop, Isaac Shamblin, Anastasia A. Arynchyna, Betsy Hopson, Tammie Kerr, James M. Johnston, Curtis J. Rozzelle, and Jeffrey P. Blount

OBJECTIVE

Repeated failure of ventriculoperitoneal shunts (VPSs) is a problem familiar to pediatric neurosurgeons and patients. While there have been many studies to determine what factors are associated with the first shunt failure, studies of subsequent failures are much less common. The purpose of this study was to identify the prevalence and associated risk factors of clustered shunt failures (defined as 3 or more VPS operations within 3 months).

METHODS

The authors reviewed prospectively collected records from all patients who underwent VPS surgery from 2008 to 2017 at their institution and included only those children who had received all of their hydrocephalus care at that institution. Demographics, etiology of hydrocephalus, history of endoscopic third ventriculostomy or temporizing procedure, initial valve type, age at shunt placement, and other factors were analyzed. Logistic regression was used to test for the association of each variable with a history of shunt failure cluster.

RESULTS

Of the 465 included children, 28 (6.0%) had experienced at least one cluster of shunt failures. Among time-independent variables, etiology of hydrocephalus (OR 0.27 for non–intraventricular hemorrhage [IVH], nonmyelomeningocele, nonaqueductal stenosis etiology vs IVH, 95% CI 0.11–0.65; p = 0.003), younger gestational age at birth (OR 0.91, 95% CI 0.85–0.97; p = 0.003), history of a temporizing procedure (OR 2.77, 95% CI 1.12–6.85; p = 0.028), and smaller head circumference at time of initial shunt placement (OR 0.91, 95% CI 0.84–0.99; p = 0.044) showed significant association with shunt failure cluster on univariate analysis. None of these variables maintained significance in a multivariate model. Among children with a history of a shunt failure cluster, 21 (75%) had a shunt infection either prior to or during the shunt failure cluster. A comparison of the infecting organism between these children and 62 children with a history of infection but without a shunt failure cluster showed an association of cluster with gram-negative rod species.

CONCLUSIONS

Six percent of children in this institutional sample had at least one shunt failure cluster. These children accounted for 30% of the total shunt revisions in the sample. Shunt infection is an important factor associated with shunt failure cluster. Children with a history of prematurity and IVH may have a higher risk for failure cluster.

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Kathrin Zimmerman, Bobby May, Katherine Barnes, Anastasia Arynchyna, Elizabeth N. Alford, Caroline Arata Wessinger, Laura Dreer, Inmaculada Aban, James M. Johnston, Curtis J. Rozzelle, Jeffrey P. Blount, and Brandon G. Rocque

OBJECTIVE

Childhood hydrocephalus is a common chronic medical condition. However, little is known about the burden of headache and psychological comorbidities in children living with hydrocephalus. The purpose of this study was to determine the prevalence and severity of these conditions among the pediatric hydrocephalus population.

METHODS

During routine neurosurgery clinic visits from July 2017 to February 2018, the authors administered four surveys to children ages 7 years and older: Pediatric Migraine Disability Assessment (PedMIDAS), Patient-Reported Outcomes Measurement Information System (PROMIS) Anxiety, PROMIS Depression, and PROMIS Fatigue. The PedMIDAS is an assessment of headache disability in pediatric and adolescent patients. The PROMIS measures are pediatric self-reported instruments to assess social and emotional health. PROMIS measures utilize T-scores (mean 50, SD 10) to compare anxiety, depression, and fatigue in specific populations to those in the US general population. Clinical and demographic data were collected from the medical record (hydrocephalus etiology, shunt infection, race, etc.) and tested for associations with survey measure scores.

RESULTS

Forty children completed the PedMIDAS. Ten percent of them were in the severe headache range, 5% were in the moderate range, and 5% were in the mild range. There was a statistically significant association between undergoing a cluster of shunt operations and headache burden (p = 0.003).

Forty children completed all three PROMIS measures. The mean anxiety score was 45.8 (SD 11.7), and 2.5% of children scored in the severe anxiety range, 17.5% in the moderate range, and 20% in the mild range. The mean depression score was 42.7 (SD 10.0), with 2.5% of children scoring in the severe depression range, 5% in the moderate range, and 12.5% in the mild range. The mean fatigue score was 45.1 (SD 16.4), with 15% percent of children scoring in the severe fatigue range, 10% in the moderate range, and 7.5% in the mild range. There were no statistically significant associations between child anxiety, depression, or fatigue and clinical or demographic variables.

CONCLUSIONS

Children with hydrocephalus have an average burden of headache, anxiety, depression, and fatigue as compared to the general population overall. Having a cluster of shunt operations correlates with a higher headache burden, but no clinical or demographic variable is associated with anxiety, depression, or fatigue.

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Kathrin Zimmerman, Bobby May, Katherine Barnes, Anastasia Arynchyna, Elizabeth N. Alford, Gustavo Chagoya, Caroline Arata Wessinger, Laura E. Dreer, Inmaculada Aban, James M. Johnston, Curtis J. Rozzelle, Jeffrey P. Blount, and Brandon G. Rocque

OBJECTIVE

Hydrocephalus is a chronic medical condition that has a significant impact on children and their caregivers. The objective of this study was to measure the quality of life (QOL) of children with hydrocephalus, as assessed by both caregivers and patients.

METHODS

Pediatric patients with hydrocephalus and their caregivers were enrolled during routine neurosurgery clinic visits. The Hydrocephalus Outcomes Questionnaire (HOQ), a report of hydrocephalus-related QOL, was administered to both children with hydrocephalus (self-report) and their caregivers (proxy report about the child). Patients with hydrocephalus also completed measures of anxiety, depression, fatigue, traumatic stress, and headache. Caregivers completed a proxy report of child traumatic stress and a measure of caregiver burden. Demographic information was collected from administration of the Psychosocial Assessment Tool (version 2.0) and from the medical record. Child and caregiver HOQ scores were analyzed and correlated with clinical, demographic, and psychological variables.

RESULTS

The mean overall HOQ score (parent assessment of child QOL) was 0.68. HOQ Physical Health, Social-Emotional Health, and Cognitive Health subscore averages were 0.69, 0.73, and 0.54, respectively. The mean overall child self-assessment (cHOQ) score was 0.77, with cHOQ Physical Health, Social-Emotional Health, and Cognitive Health subscore means of 0.84, 0.79, and 0.66, respectively. Thirty-nine dyads were analyzed, in which both a child with hydrocephalus and his or her caregiver completed the cHOQ and HOQ. There was a positive correlation between parent and child scores (p < 0.004 for all subscores). Child scores were consistently higher than parent scores. Variables that showed association with caregiver-assessed QOL in at least one domain included child age, etiology of hydrocephalus, and history of endoscopic third ventriculostomy. There was a significant negative relationship (rho −0.48 to −0.60) between child-reported cHOQ score and child-reported measures of posttraumatic stress, anxiety, depression, and fatigue. There was a similar significant relationship between caregiver report of child’s QOL (HOQ) and caregiver assessment of the child’s posttraumatic stress symptoms as well as their assessment of burden of care (rho = −0.59 and rho = −0.51, respectively). No relationship between parent-reported HOQ and child-reported psychosocial factors was significant. No clinical or demographic variables were associated with child self-assessed cHOQ.

CONCLUSIONS

Pediatric patients with hydrocephalus consistently rate their own QOL higher than their caregivers do. Psychological factors such as anxiety and posttraumatic stress may be associated with lower QOL. These findings warrant further exploration.

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Howard L. Weiner, P. David Adelson, Douglas L. Brockmeyer, Cormac O. Maher, Nalin Gupta, Matthew D. Smyth, Andrew Jea, Jeffrey P. Blount, Jay Riva-Cambrin, Sandi K. Lam, Edward S. Ahn, Gregory W. Albert, and Jeffrey R. Leonard

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Pongkiat Kankirawatana, Ismail S. Mohamed, Jason Lauer, Inmaculada Aban, Hyunmi Kim, Rong Li, Allan Harrison, AS, Monisha Goyal, Curtis J. Rozzelle, Robert Knowlton, and Jeffrey P. Blount

OBJECTIVE

The goal of this study was to evaluate the predictive value and relative contribution of noninvasive presurgical functional imaging modalities based on the authors’ institutional experience in pursuing seizure-free surgical outcomes in children with medically refractory epilepsy.

METHODS

This was a retrospective, single-institution, observational cohort study of pediatric patients who underwent evaluation and surgical treatment for medically refractory partial epilepsy between December 2003 and June 2016. During this interval, 108 children with medically refractory partial epilepsy underwent evaluation for localization and resective epilepsy surgery. Different noninvasive functional imaging modalities, including ictal SPECT, FDG-PET, and magnetoencephalography–magnetic source imaging, were utilized to augment a standardized paradigm (electroencephalography/semiology, MRI, and neuropsychology findings) for localization. Outcomes were evaluated at a minimum of 2 years (mean 7.5 years) utilizing area under the receiver operating characteristic curve analysis. Localizing modalities and other clinical covariates were examined in relation to long-term surgical outcomes.

RESULTS

There was variation in the contribution of each test, and no single presurgical workup modality could singularly and reliably predict a seizure-free outcome. However, concordance of presurgical modalities yielded a high predictive value. No difference in long-term outcomes between inconclusive (normal or diffusely abnormal) and abnormal focal MRI results were found. Long-term survival analyses revealed a statistically significant association between seizure freedom and patients with focal ictal EEG, early surgical intervention, and no history of generalized convulsions.

CONCLUSIONS

Comprehensive preoperative evaluation utilizing multiple noninvasive functional imaging modalities is not redundant and can improve pediatric epilepsy surgical outcomes.