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Irene Kim, Betsy Hopson, Inmaculada Aban, Elias B. Rizk, Mark S. Dias, Robin Bowman, Laurie L. Ackerman, Michael D. Partington, Heidi Castillo, Jonathan Castillo, Paula R. Peterson, Jeffrey P. Blount and Brandon G. Rocque

OBJECTIVE

The purpose of this study was to determine the rate of decompression for Chiari malformation type II in individuals with myelomeningocele in the National Spina Bifida Patient Registry (NSBPR). In addition, the authors explored the variation in rates of Chiari II decompression across NSBPR institutions, examined the relationship between Chiari II decompression and functional lesion level of the myelomeningocele, age, and need for tracheostomy, and they evaluated for temporal trends in rates of Chiari II decompression.

METHODS

The authors queried the NSBPR to identify all individuals with myelomeningocele between 2009 and 2015. Among these patients, they identified individuals who had undergone at least 1 Chiari II decompression as well as those who had undergone tracheostomy. For each participating NSBPR institution, the authors calculated the proportion of patients enrolled at that site who underwent Chiari II decompression. Logistic regression was performed to analyze the relationship between Chiari II decompression, functional lesion level, age at decompression, and history of tracheostomy.

RESULTS

Of 4448 individuals with myelomeningocele identified from 26 institutions, 407 (9.15%) had undergone at least 1 Chiari II decompression. Fifty-one patients had undergone tracheostomy. Logistic regression demonstrated a statistically significant relationship between Chiari II decompression and functional lesion level of the myelomeningocele, with a more rostral lesion level associated with a higher likelihood of posterior fossa decompression. Similarly, children born before 2005 and those with history of tracheostomy had a significantly higher likelihood of Chiari II decompression. There was no association between functional lesion level and need for tracheostomy. However, among those children who underwent Chiari II decompression, the likelihood of also undergoing tracheostomy increased significantly with younger age at decompression.

CONCLUSIONS

The rate of Chiari II decompression in patients with myelomeningocele in the NSBPR is consistent with that in previously published literature. There is a significant relationship between Chiari II decompression and functional lesion level of the myelomeningocele, which has not previously been reported. Younger children who undergo Chiari II decompression are more likely to have undergone tracheostomy. There appears to be a shift away from Chiari II decompression, as children born before 2005 were more likely to undergo Chiari II decompression than those born in 2005 or later.

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Irene Kim, Betsy Hopson, Inmaculada Aban, Elias B. Rizk, Mark S. Dias, Robin Bowman, Laurie L. Ackerman, Michael D. Partington, Heidi Castillo, Jonathan Castillo, Paula R. Peterson, Jeffrey P. Blount and Brandon G. Rocque

OBJECTIVE

Although the majority of patients with myelomeningocele have hydrocephalus, reported rates of hydrocephalus treatment vary widely. The purpose of this study was to determine the rate of surgical treatment for hydrocephalus in patients with myelomeningocele in the National Spina Bifida Patient Registry (NSBPR). In addition, the authors explored the variation in shunting rates across NSBPR institutions, examined the relationship between hydrocephalus, and the functional lesion level of the myelomeningocele, and evaluated for temporal trends in rates of treated hydrocephalus.

METHODS

The authors queried the NSBPR to identify all patients with myelomeningoceles. Individuals were identified as having been treated for hydrocephalus if they had undergone at least 1 hydrocephalus-related operation. For each participating NSBPR institution, the authors calculated the proportion of patients with treated hydrocephalus who were enrolled at that site. Logistic regression was performed to analyze the relationship between hydrocephalus and the functional lesion level of the myelomeningocele and to compare the rate of treated hydrocephalus in children born before 2005 with those born in 2005 or later.

RESULTS

A total of 4448 patients with myelomeningocele were identified from 26 institutions, of whom 3558 patients (79.99%) had undergone at least 1 hydrocephalus-related operation. The rate of treated hydrocephalus ranged from 72% to 96% among institutions enrolling more than 10 patients. This difference in treatment rates between centers was statistically significant (p < 0.001). Insufficient data were available in the NSBPR to analyze reasons for the different rates of hydrocephalus treatment between sites. Multivariate logistic regression demonstrated that more rostral functional lesion levels were associated with higher rates of treated hydrocephalus (p < 0.001) but demonstrated no significant difference in hydrocephalus treatment rates between children born before versus after 2005.

CONCLUSIONS

The rate of hydrocephalus treatment in patients with myelomeningocele in the NSBPR is 79.99%, which is consistent with the rates in previously published literature. The authors’ data demonstrate a clear association between functional lesion level of the myelomeningocele and the need for hydrocephalus treatment.

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Samuel G. McClugage III, Nicholas M. B. Laskay, Brian N. Donahue, Anastasia Arynchyna, Kathrin Zimmerman, Inmaculada B. Aban, Elizabeth N. Alford, Myriam Peralta-Carcelen, Jeffrey P. Blount, Curtis J. Rozzelle, James M. Johnston and Brandon G. Rocque

OBJECTIVE

Posthemorrhagic hydrocephalus of prematurity remains a significant problem in preterm infants. In the literature, there is a scarcity of data on the early disease process, when neurosurgeons are typically consulted for recommendations on treatment. Here, the authors sought to evaluate functional outcomes in premature infants at 2 years of age following treatment for posthemorrhagic hydrocephalus. Their goal was to determine the relationship between factors identifiable at the time of the initial neurosurgical consult and outcomes of patients when they are 2 years of age.

METHODS

The authors performed a retrospective chart review of premature infants treated for intraventricular hemorrhage (IVH) of prematurity (grade III and IV) between 2003 and 2014. Information from three time points (birth, first neurosurgical consult, and 2 years of age) was collected on each patient. Logistic regression analysis was performed to determine the association between variables known at the time of the first neurosurgical consult and each of the outcome variables.

RESULTS

One hundred thirty patients were selected for analysis. At 2 years of age, 16% of the patients had died, 88% had cerebral palsy/developmental delay (CP), 48% were nonverbal, 55% were nonambulatory, 33% had epilepsy, and 41% had visual impairment. In the logistic regression analysis, IVH grade was an independent predictor of CP (p = 0.004), which had an estimated probability of occurrence of 74% in grade III and 96% in grade IV. Sepsis at or before the time of consult was an independent predictor of visual impairment (p = 0.024), which had an estimated probability of 58%. IVH grade was an independent predictor of epilepsy (p = 0.026), which had an estimated probability of 18% in grade III and 43% in grade IV. The IVH grade was also an independent predictor of verbal function (p = 0.007), which had an estimated probability of 68% in grade III versus 41% in grade IV. A higher weeks gestational age (WGA) at birth was an independent predictor of the ability to ambulate (p = 0.0014), which had an estimated probability of 15% at 22 WGA and up to 98% at 36 WGA. The need for oscillating ventilation at consult was an independent predictor of death before 2 years of age (p = 0.001), which had an estimated probability of 42% in patients needing oscillating ventilation versus 13% in those who did not.

CONCLUSIONS

IVH grade was consistently an independent predictor of functional outcomes at 2 years. Gestational age at birth, sepsis, and the need for oscillating ventilation may also predict worse functional outcomes.

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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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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.

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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.