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Jonathan Pindrik, Brandon G. Rocque, Anastasia A. Arynchyna, James M. Johnston and Curtis J. Rozzelle

OBJECTIVE

Endoscopic third ventriculostomy (ETV) with choroid plexus (CP) cauterization (CPC) represents a viable treatment option for congenital hydrocephalus in infants younger than 2 years. Imaging studies complement clinical data in the evaluation of treatment success or failure. The objectives of this study were to investigate novel radiographic markers—cerebrospinal fluid (CSF) turbulence and CP visualization—and their ability to reflect or predict clinical outcomes following ETV/CPC.

METHODS

Hydrocephalic patients younger than 2 years who were initially treated by ETV/CPC at the senior authors' institution between March 2013 and February 2014 were retrospectively reviewed. Clinical data, as well as the visualization of CSF turbulence and CP on pre- and postoperative fast-sequence MRI, were recorded. Radiographic images were reviewed by a blinded observer based on specific criteria for the visualization of CSF turbulence and CP. Data were collected and analyzed using descriptive statistics, including Fisher's exact test for comparisons. The research team obtained appropriate institutional review board approval for this study, without the need for informed consent.

RESULTS

Among the 32 patients (53% male and 47% female) studied, 18 of 32 (56%) responded favorably to initial or repeat ETV/CPC, with 13 of 32 (41%) patients requiring 1 surgery. Of the 19 (59%) patients whose initial ETV/CPC failed, 8 of 19 (42%) patients underwent repeat ETV/CPC, with 5 of 8 (63%) patients responding favorably. Radiographic CSF turbulence appeared more frequently following ETV/CPC failure than after ETV/CPC success (55% vs 18%, respectively; p = 0.02). The sensitivity and specificity of CSF turbulence as a radiographic marker for ETV/CPC failure were 80% and 58%, respectively. The radiographic depiction of CP disappearance following ETV/CPC from pre- to postoperative imaging occurred in 20 of 30 patients (67%). Among the patients who responded unsuccessfully to ETV/CPC and ultimately required secondary shunt insertion, 71% (10 of 14 patients) demonstrated CP persistence on postoperative imaging. In contrast, 6% (1 of 18) of patients who were treated successfully by ETV/CPC demonstrated the presence of CP on follow-up imaging. This difference reached statistical significance (p = 0.0001). The visualization of CP persistence despite ETV/CPC reflected treatment failure with 91% sensitivity and 81% specificity. The sensitivity of either or both radiographic markers to suggest ETV/CPC failure was 77%, while their specificity (both markers absent, thereby indicating ETV/CPC success) was 81%.

CONCLUSIONS

Radiographic markers correlate with clinical outcomes following the treatment of infantile hydrocephalus with ETV/CPC. Specifically, CSF turbulence may indicate ongoing pathological CSF flow dynamics, while CP absence following ETV/CPC may predict shunt independence. Future studies that incorporate prospective review and formal intra- and interobserver reliability estimates may help corroborate the utility of these radiographic markers.

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Brandon A. Sherrod, Anastasia A. Arynchyna, James M. Johnston, Curtis J. Rozzelle, Jeffrey P. Blount, W. Jerry Oakes and Brandon G. Rocque

OBJECTIVE

Surgical site infection (SSI) following CSF shunt operations has been well studied, yet risk factors for nonshunt pediatric neurosurgery are less well understood. The purpose of this study was to determine SSI rates and risk factors following nonshunt pediatric neurosurgery using a nationwide patient cohort and an institutional data set specifically for better understanding SSI.

METHODS

The authors reviewed the American College of Surgeons National Surgical Quality Improvement Program–Pediatric (ACS NSQIP-P) database for the years 2012–2014, including all neurosurgical procedures performed on pediatric patients except CSF shunts and hematoma evacuations. SSI included deep (intracranial abscesses, meningitis, osteomyelitis, and ventriculitis) and superficial wound infections. The authors performed univariate analyses of SSI association with procedure, demographic, comorbidity, operative, and hospital variables, with subsequent multivariate logistic regression analysis to determine independent risk factors for SSI within 30 days of the index procedure. A similar analysis was performed using a detailed institutional infection database from Children's of Alabama (COA).

RESULTS

A total of 9296 nonshunt procedures were identified in NSQIP-P with an overall 30-day SSI rate of 2.7%. The 30-day SSI rate in the COA institutional database was similar (3.3% of 1103 procedures, p = 0.325). Postoperative time to SSI in NSQIP-P and COA was 14.6 ± 6.8 days and 14.8 ± 7.3 days, respectively (mean ± SD). Myelomeningocele (4.3% in NSQIP-P, 6.3% in COA), spine (3.5%, 4.9%), and epilepsy (3.4%, 3.1%) procedure categories had the highest SSI rates by procedure category in both NSQIP-P and COA. Independent SSI risk factors in NSQIP-P included postoperative pneumonia (OR 4.761, 95% CI 1.269–17.857, p = 0.021), immune disease/immunosuppressant use (OR 3.671, 95% CI 1.371–9.827, p = 0.010), cerebral palsy (OR 2.835, 95% CI 1.463–5.494, p = 0.002), emergency operation (OR 1.843, 95% CI 1.011–3.360, p = 0.046), spine procedures (OR 1.673, 95% CI 1.036–2.702, p = 0.035), acquired CNS abnormality (OR 1.620, 95% CI 1.085–2.420, p = 0.018), and female sex (OR 1.475, 95% CI 1.062–2.049, p = 0.021). The only COA factor independently associated with SSI in the COA database included clean-contaminated wound classification (OR 3.887, 95% CI 1.354–11.153, p = 0.012), with public insurance (OR 1.966, 95% CI 0.957–4.041, p = 0.066) and spine procedures (OR 1.982, 95% CI 0.955–4.114, p = 0.066) approaching significance. Both NSQIP-P and COA multivariate model C-statistics were > 0.7.

CONCLUSIONS

The NSQIP-P SSI rates, but not risk factors, were similar to data from a single center.

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Daxa M. Patel, Brandon G. Rocque, Betsy Hopson, Anastasia Arynchyna, E. Ralee’ Bishop, David Lozano and Jeffrey P. Blount

OBJECT

A paucity of literature examines sleep apnea in patients with myelomeningocele, Chiari malformation Type II (CM-II), and related hydrocephalus. Even less is known about the effect of hydrocephalus treatment or CM-II decompression on sleep hygiene. This study is an exploratory analysis of sleep-disordered breathing in patients with myelomeningocele and the effects of neurosurgical treatments, in particular CM-II decompression and hydrocephalus management, on sleep organization.

METHODS

The authors performed a retrospective review of all patients seen in their multidisciplinary spina bifida clinic (approximately 435 patients with myelomeningocele) to evaluate polysomnographs obtained between March 1999 and July 2013. They analyzed symptoms prompting evaluation, results, and recommended interventions by using descriptive statistics. They also conducted a subset analysis of 9 children who had undergone polysomnography both before and after neurosurgical intervention.

RESULTS

Fifty-two patients had polysomnographs available for review. Sleep apnea was diagnosed in 81% of these patients. The most common presenting symptom was “breathing difficulties” (18 cases [43%]). Mild sleep apnea was present in 26 cases (50%), moderate in 10 (19%), and severe in 6 (12%). Among the 42 patients with abnormal sleep architecture, 30 had predominantly obstructive apneas and 12 had predominantly central apneas. The most common pulmonology-recommended intervention was adjustment of peripheral oxygen supplementation (24 cases [57%]), followed by initiation of peripheral oxygen (10 cases [24%]).

In a subset analysis of 9 patients who had sleep studies before and after neurosurgical intervention, there was a trend toward a decrease in the mean number of respiratory events (from 34.8 to 15.9, p = 0.098), obstructive events (from 14.7 to 13.9, p = 0.85), and central events (from 20.1 to 2.25, p = 0.15) and in the apnea-hypopnea index (from 5.05 to 2.03, p = 0.038, not significant when corrected for multiple measures).

CONCLUSIONS

A large proportion of patients with myelomeningocele who had undergone polysomnography showed evidence of disordered sleep on an initial study. Furthermore, 31% of patients had moderate or severe obstructive sleep apnea. Myelomeningocele patients with an abnormal sleep structure who had undergone nonoperative treatment with peripheral oxygen supplementation showed improvement in the apnea-hypopnea index. Results in this study suggested that polysomnography in patients with myelomeningocele may present an opportunity to detect and classify sleep apnea, identify low-risk interventions, and prevent future implications of sleep-disordered breathing.

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Esther B. Dupépé, Daxa M. Patel, Brandon G. Rocque, Betsy Hopson, Anastasia A. Arynchyna, E. Ralee' Bishop and Jeffrey P. Blount

OBJECTIVE

Although there are known risk factors for the development of neural tube defects (NTDs), little is known regarding the role of family history. The authors' goal in this study is to describe the family history in their population of patients with NTDs.

METHODS

Surveys were completed for 254 patients who were accompanied by their biological mother during their annual visit to the multidisciplinary Spina Bifida Clinic at Children's of Alabama. An NTD has been diagnosed in all patients who are seen in this clinic (myelomeningocele, lipomeningocele, split cord malformation, and congenital dermal sinus tract). Each mother answered questions regarding known NTD risk factors and their pregnancy, as well as the family history of NTDs, other CNS disorders, and birth defects.

RESULTS

The overall prevalence of family history of NTDs in children with an NTD was 16.9% (n = 43), of which 3.1% (n = 8) were in first-degree relatives. In patients with myelomeningocele, 17.7% (n = 37) had a positive family history for NTDs, with 3.8% in first-degree relatives. Family history in the paternal lineage for all NTDs was 8.7% versus 10.6% in the maternal lineage. Twenty-two patients (8.7%) had a family history of other congenital CNS disorders. Fifteen (5.9%) had a family history of Down syndrome, 12 (4.7%) had a family history of cerebral palsy, and 13 (5.1%) patients had a family history of clubfoot. Fourteen (5.5%) had a family history of cardiac defect, and 13 (5.1%) had a family history of cleft lip or palate.

CONCLUSIONS

The family history of NTDs was 16.9% in children with NTD without a difference between maternal and paternal lineage. This high rate of positive family history suggests that genetics and epigenetics may play a larger role in the pathogenesis of NTD in the modern era of widespread folate supplementation.

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Alexandra Cutillo, Kathrin Zimmerman, Susan Davies, Avi Madan-Swain, Wendy Landier, Anastasia Arynchyna and Brandon G. Rocque

OBJECTIVE

The goal of this study was to determine what strategies caregivers use to cope with the stress of a child who has recently undergone surgical treatment for a newly diagnosed brain tumor. Results will be used to improve psychosocial assessments and treatments provided to these families during initial hospitalization.

METHODS

Caregivers of children with newly diagnosed brain tumors admitted to Children’s of Alabama were enrolled during the child’s initial hospitalization for surgical treatment from April 2016 to August 2017. The single-item, National Comprehensive Cancer Network visual analog Distress Thermometer (DT) was administered. Clinical and demographic variables were collected from the medical record. Approximately 1 month after patients were discharged from the hospital, caregivers participated in a semistructured interview that included questions about parent and family coping strategies. Broad questions about stress management since diagnosis were followed by specific questions about individual coping strategies. Interviews were recorded, transcribed, and coded for common themes. Themes were broadly characterized as adaptive versus maladaptive coping. Analysis was then performed to determine if DT scores or clinical or demographic factors were associated with the presence of maladaptive coping using a concurrent triangulation mixed-methods design.

RESULTS

Caregivers identified several adaptive coping strategies, such as active coping, acceptance coping, emotion-focused coping, spiritual coping, social support, and posttraumatic growth. Maladaptive coping strategies were mostly categorized as avoidant coping. Caregivers endorsed multiple different coping strategies (mean of 4.59 strategies per caregiver). No clinical or demographic variables, whether the tumor was benign or malignant, or the DT score correlated with caregiver endorsement of maladaptive coping strategies.

CONCLUSIONS

Caregivers of children newly diagnosed with brain tumors employ a number of coping strategies, some of which are maladaptive. The authors have not identified any method for predicting who is at risk for maladaptive coping. Assessment of coping skills and education about coping have the potential to improve overall care delivery to these families.

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Brandon G. Rocque, Alexandra Cutillo, Kathrin Zimmerman, Anastasia Arynchyna, Susan Davies, Wendy Landier and Avi Madan-Swain

OBJECTIVE

Hospitalization for a newly diagnosed pediatric brain tumor is an extremely stressful time for a family, but this period has not been the focus of rigorous study. The purpose of this study was to quantify distress and psychosocial risk in this population to improve psychosocial care delivery.

METHODS

The authors administered the National Comprehensive Cancer Network Distress Thermometer (DT) and the Psychosocial Assessment Tool 2.0 (PAT) to primary caregivers of all children admitted to Children’s of Alabama with a new brain tumor between April 2016 and August 2017. The DT is a single-item measure of distress (scale range 0–10). The PAT (range 0–7) stratifies families by risk level: a score less than 1.0 indicates universal risk level (risk typically experienced during hospitalization); a score of 1.0–2.0 indicates targeted risk (specific psychosocial difficulties that impact medical treatment); and a score higher than 2.0 indicates clinically significant risk. Demographic and clinical information was abstracted from each child’s medical record. A correlation matrix using nonparametric statistics was created between abstracted data and the DT and PAT scores.

RESULTS

Forty primary caregivers were enrolled (of 49 eligible), with the patient age ranging from newborn to 17 years (mean 7.7 years). Twenty-five (63%) of the children were male, and 24 (60%) were white, non-Hispanic. Mean and median DT scores were 7.2 (SD 2.6) and 7, respectively. However, 12 (30%) rated their distress 10/10, and 85% rated distress 5 or greater. PAT scores ranged from 0.0 to 2.36 with mean and median scores of 0.89 (SD 0.50) and 0.86, respectively. PAT results for 16 (40%) families were in the targeted or clinical range, indicating psychosocial factors that have the potential to interfere with medical treatment. No clinical or demographic variable correlated significantly with the DT or PAT score.

CONCLUSIONS

Families of children with newly diagnosed brain tumors experience high levels of distress and psychosocial risk. This work will serve as the foundation for efforts to standardize psychosocial evaluation for newly diagnosed pediatric neurosurgical patients, and to create protocols that organize existing hospital-based psychosocial support services. These efforts have the potential to improve patient and family satisfaction as well as treatment outcomes.

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Jacob R. Lepard, Kathrin D. Zimmerman, Anastasia A. Arynchyna, Jessica A. Gutman, Arsalaan A. Salehani, Brandon G. Rocque and Curtis J. Rozzelle

OBJECTIVE

Surgical treatment of herniated lumbar disc (HLD) remains rare in children. The purpose of this study was to evaluate for potential disease risk factors leading to surgery based on a large single-center experience.

METHODS

Data for all patients who had undergone surgical treatment for HLD between December 2008 and December 2016 at a single pediatric tertiary care referral center were collected and compared to data for a healthy control population obtained through a Youth Risk Behavior Surveillance System (YRBSS) survey in order to determine relevant disease risk factors. Univariate and multivariate logistic regression were used to determine the effect of potential risk factors.

RESULTS

Twenty-seven patients in the disease cohort and 5212 healthy controls from the general population were included in the risk factor analysis. The mean body mass index was significantly higher in the disease population (30.2 vs 24.0 kg/m2, p < 0.0001). Children who had undergone microdiscectomy were more likely to be obese (OR 7.4, 95% CI 3.46–15.8, p < 0.001). No association was found between lumbar microdiscectomy and sports participation (OR 1.0, 95% CI −0.002 to 0.005, p = 0.37).

CONCLUSIONS

Microdiscectomy remains a viable and safe option in the setting of failed conservative management for pediatric HLD. Childhood obesity is a risk factor for HLD and many other diseases, which increases its importance as a public health priority.

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Brandon G. Rocque, E. Ralee' Bishop, Mallory A. Scogin, Betsy D. Hopson, Anastasia A. Arynchyna, Christina J. Boddiford, Chevis N. Shannon and Jeffrey P. Blount

OBJECT

The purpose of this study is to explore various aspects of health-related quality of life (HRQOL) in children with spinal dysraphism.

METHODS

The authors enrolled a prospective cohort of 159 patients from the multidisciplinary spina bifida clinic. Surveys were distributed to caregivers of patients with spina bifida who were 5 years old and older. Data were collected using the Health Utilities Index Mark 3 focusing on vision, speech, hearing, dexterity, ambulation, cognition, emotions, and pain. Each participant received an overall HRQOL utility score and individual domain subscores. These were correlated with demographic and treatment variables. Analysis was done using SPSS statistics (version 21).

RESULTS

There were 125 patients with myelomeningocele, 25 with lipomyelomeningocele, and 9 with other dysraphisms. Among patients with myelomeningocele, 107 (86%) had CSF shunts in place, 14 (11%) had undergone Chiari malformation Type II decompression, 59 (47%) were community ambulators, and 45 (36%) were nonambulatory. Patients with myelomeningocele had significantly lower overall HRQOL scores than patients with closed spinal dysraphism. Among patients with myelomeningocele, younger patients had higher HRQOL scores. Patients with impaired bowel continence had lower overall HRQOL scores. History of a ventriculoperitoneal shunt was associated with worse HRQOL (overall score, ambulation, and cognition subscores). History of Chiari malformation Type II decompression was associated with worse overall, speech, and cognition scores. Patients who could ambulate in the community had higher overall and ambulation scores. A history of tethered cord release was correlated with lower pain subscore. No association was found between sex, race, insurance type, or bladder continence and HRQOL.

CONCLUSIONS

Patients with myelomeningocele have significantly lower HRQOL scores than those with other spinal dysraphisms. History of shunt treatment and Chiari decompression correlate with lower HRQOL scores.

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Tofey J. Leon, Elizabeth N. Kuhn, Anastasia A. Arynchyna, Burkely P. Smith, R. Shane Tubbs, James M. Johnston, Jeffrey P. Blount, Curtis J. Rozzelle, W. Jerry Oakes and Brandon G. Rocque

OBJECTIVE

There are sparse published data on the natural history of “benign” Chiari I malformation (CM-I)—i.e., Chiari with minimal or no symptoms at presentation and no imaging evidence of syrinx, hydrocephalus, or spinal cord signal abnormality. The purpose of this study was to review a large cohort of children with benign CM-I and to determine whether these children become symptomatic and require surgical treatment.

METHODS

Patients were identified from institutional outpatient records using International Classification of Diseases, 9th Revision, diagnosis codes for CM-I from 1996 to 2016. After review of the medical records, patients were excluded if they 1) did not have a diagnosis of CM-I, 2) were not evaluated by a neurosurgeon, 3) had previously undergone posterior fossa decompression, or 4) had imaging evidence of syringomyelia at their first appointment. To include only patients with benign Chiari (without syrinx or classic Chiari symptoms that could prompt immediate intervention), any patient who underwent decompression within 9 months of initial evaluation was excluded. After a detailed chart review, patients were excluded if they had classical Chiari malformation symptoms at presentation. The authors then determined what changes in the clinical picture prompted surgical treatment. Patients were excluded from the multivariate logistic regression analysis if they had missing data such as race and insurance; however, these patients were included in the overall survival analysis.

RESULTS

A total of 427 patients were included for analysis with a median follow-up duration of 25.5 months (range 0.17–179.1 months) after initial evaluation. Fifteen patients had surgery at a median time of 21.0 months (range 11.3–139.3 months) after initial evaluation. The most common indications for surgery were tussive headache in 5 (33.3%), syringomyelia in 5 (33.3%), and nontussive headache in 5 (33.3%). Using the Kaplan-Meier method, rate of freedom from posterior fossa decompression was 95.8%, 94.1%, and 93.1% at 3, 5, and 10 years, respectively.

CONCLUSIONS

Among a large cohort of patients with benign CM-I, progression of imaging abnormalities or symptoms that warrant surgical treatment is infrequent. Therefore, these patients should be managed conservatively. However, clinical follow-up of such individuals is justified, as there is a low, but nonzero, rate of new symptom or syringomyelia development. Future analyses will determine whether imaging or clinical features present at initial evaluation are associated with progression and future need for treatment.

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Andrew T. Hale, Amanda N. Stanton, Shilin Zhao, Faizal Haji, Stephen R. Gannon, Anastasia Arynchyna, John C. Wellons, Brandon G. Rocque and Robert P. Naftel

OBJECTIVE

At failure of endoscopic third ventriculostomy (ETV) with choroid plexus cauterization (CPC), the ETV ostomy may be found to be closed or open. Failure with a closed ostomy may indicate a population that could benefit from evolving techniques to keep the ostomy open and may be candidates for repeat ETV, whereas failure with an open ostomy may be due to persistently abnormal CSF dynamics. This study seeks to identify clinical and radiographic predictors of ostomy status at the time of ETV/CPC failure.

METHODS

The authors conducted a multicenter, retrospective cohort study on all pediatric patients with hydrocephalus who failed initial ETV/CPC treatment between January 2013 and October 2016. Failure was defined as the need for repeat ETV or ventriculoperitoneal (VP) shunt placement. Clinical and radiographic data were collected, and ETV ostomy status was determined endoscopically at the subsequent hydrocephalus procedure. Statistical analysis included the Mann-Whitney U-test, Wilcoxon rank-sum test, t-test, and Pearson chi-square test where appropriate, as well as multivariate logistic regression.

RESULTS

Of 72 ETV/CPC failures, 28 patients (39%) had open-ostomy failure and 44 (61%) had closed-ostomy failure. Patients with open-ostomy failure were older (median 5.1 weeks corrected age for gestation [interquartile range (IQR) 0.9–15.9 weeks]) than patients with closed-ostomy failure (median 0.2 weeks [IQR −1.3 to 4.5 weeks]), a significant difference by univariate and multivariate regression. Etiologies of hydrocephalus included intraventricular hemorrhage of prematurity (32%), myelomeningocele (29%), congenital communicating (11%), aqueductal stenosis (11%), cyst/tumor (4%), and other causes (12%). A wider baseline third ventricle was associated with open-ostomy failure (median 15.0 mm [IQR 10.3–18.5 mm]) compared to closed-ostomy failure (median 11.7 mm [IQR 8.9–16.5 mm], p = 0.048). Finally, at the time of failure, patients with closed-ostomy failure had enlargement of their ventricles (frontal and occipital horn ratio [FOHR], failure vs baseline, median 0.06 [IQR 0.00–0.11]), while patients with open-ostomy failure had no change in ventricle size (median 0.01 [IQR −0.04 to 0.05], p = 0.018). Previous CSF temporizing procedures, intraoperative bleeding, and time to failure were not associated with ostomy status at ETV/CPC failure.

CONCLUSIONS

Older corrected age for gestation, larger baseline third ventricle width, and no change in FOHR were associated with open-ostomy ETV/CPC failure. Future studies are warranted to further define and confirm features that may be predictive of ostomy status at the time of ETV/CPC failure.