Publications by authors named "Anastasia Arynchyna"

28 Publications

  • Page 1 of 1

Early implementation of stereoelectroencephalography in children: a multiinstitutional case series.

J Neurosurg Pediatr 2021 Sep 3:1-8. Epub 2021 Sep 3.

1Department of Neurological Surgery, University of Alabama at Birmingham, Alabama.

Objective: Pediatric stereoelectroencephalography (SEEG) has been increasingly performed in the United States, with published literature being limited primarily to large single-center case series. The purpose of this study was to evaluate the experience of pediatric epilepsy centers, where the technique has been adopted in the last several years, via a multicenter case series studying patient demographics, outcomes, and complications.

Methods: A retrospective cohort methodology was used based on the STROBE criteria. ANOVA was used to evaluate for significant differences between the means of continuous variables among centers. Dichotomous outcomes were assessed between centers using a univariate and multivariate logistic regression.

Results: A total of 170 SEEG insertion procedures were included in the study from 6 different level 4 pediatric epilepsy centers. The mean patient age at time of SEEG insertion was 12.3 ± 4.7 years. There was no significant difference between the mean age at the time of SEEG insertion between centers (p = 0.3). The mean number of SEEG trajectories per patient was 11.3 ± 3.6, with significant variation between centers (p < 0.001). Epileptogenic loci were identified in 84.7% of cases (144/170). Patients in 140 cases (140/170, 82.4%) underwent a follow-up surgical intervention, with 47.1% (66/140) being seizure free at a mean follow-up of 30.6 months. An overall postoperative hemorrhage rate of 5.3% (9/170) was noted, with patients in 4 of these cases (4/170, 2.4%) experiencing a symptomatic hemorrhage and patients in 3 of these cases (3/170, 1.8%) requiring operative evacuation of the hemorrhage. There were no mortalities or long-term complications.

Conclusions: As the first multicenter case series in pediatric SEEG, this study has aided in establishing normative practice patterns in the application of a novel surgical technique, provided a framework for anticipated outcomes that is generalizable and useful for patient selection, and allowed for discussion of what is an acceptable complication rate relative to the experiences of multiple institutions.
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http://dx.doi.org/10.3171/2021.5.PEDS20923DOI Listing
September 2021

Comparison of aesthetic outcomes between open and endoscopically treated sagittal craniosynostosis.

J Neurosurg Pediatr 2021 Jul 30:1-7. Epub 2021 Jul 30.

Departments of1Neurological Surgery and.

Objective: In the last several decades, there has been much debate regarding the ideal treatment for sagittal synostosis. The purpose of this study was to compare perioperative, anthropometric, and subjective assessments of cosmetic outcomes between open and endoscopic management of isolated sagittal synostosis.

Methods: At their routine postoperative follow-up, pediatric patients with sagittal craniosynostosis were recruited to undergo digital cranial measurement and standardized photography for objective and subjective assessments of perioperative outcomes. Age-normalized z-scores for cephalic index, head circumference, euryon-euryon diameter (Eu-Eu), and glabella-opisthocranion diameter (G-Op) were calculated for each patient. Faculty surgeons, surgical trainees, nurses, and laypersons were asked to rate the normalcy of craniofacial appearances using a 5-point Likert scale. Outcomes were compared between patients treated with endoscopic correction and those treated with open repair.

Results: A total of 50 patients were included in the study. Thirty-one had undergone open surgical correction, and 19 had undergone endoscopic treatment. Endoscopic repair involved significantly lower operative time, blood loss, transfusion rate, and hospital length of stay than those with open repair (p < 0.001). There was no significant difference between groups in terms of z-scores for head circumference (p = 0.22), cephalic index (p = 0.25), or Eu-Eu (p = 0.38). Endoscopic treatment was associated with a significantly lower G-Op (p = 0.009). Additionally, the average subjective rating of head shape was higher for endoscopic treatment when corrected for age, gender, and ethnicity (p = 0.02).

Conclusions: The study findings suggest that patients who are treated endoscopically may have an overall more normal appearance in skull morphology and cosmesis, although these results are limited by poor reliability.
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http://dx.doi.org/10.3171/2021.3.PEDS20894DOI Listing
July 2021

Incidental Pituitary Cysts in Children: Does Growth Hormone Treatment Affect Cyst Size?

Endocr Pract 2021 Jun 17. Epub 2021 Jun 17.

University of Alabama at Birmingham, Birmingham, Alabama. Electronic address:

Objective: To evaluate the response of incidentally discovered pituitary cysts to growth hormone (GH) treatment.

Methods: A retrospective chart review was performed of children with pituitary cysts on magnetic resonance imaging (MRI) over a 5-year period. Records and images were reviewed, and the results were analyzed using descriptive statistics. Children with pituitary cysts who received GH treatment were compared with those without.

Results: We identified 109 children with pituitary cysts, 24 were treated with GH therapy. The average age was 8.5 ± 5.1 years. Children whose initial MRI scan was to evaluate growth hormone deficiency were more commonly male and non-Hispanic White compared with those with scans for other indications (male, 18 of 24 vs 35 of 85, P = .003; White, 23 of 24 vs 58 of 85, P = .004). Among patients who received GH treatment, 12 had follow-up MRI. Six had no change in cyst size and 6 had a decrease in cyst size. We observed no difference in the likelihood of cyst growth between those who received GH and those who did not (0 of 12 cysts with GH vs 1 of 15 cysts without GH showed growth at follow-up). No patient had neurologic deficits attributable to the pituitary cyst at any time.

Conclusion: In a single-institution, retrospective study, we find no evidence of growth in pituitary cysts in response to GH therapy.
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http://dx.doi.org/10.1016/j.eprac.2021.04.887DOI Listing
June 2021

Imaging characteristics associated with surgery in Chiari malformation type I.

J Neurosurg Pediatr 2021 Apr 23:1-9. Epub 2021 Apr 23.

1Division of Pediatric Neurosurgery, Department of Neurosurgery; and.

Objective: In Chiari malformation type I (CM-I), a variety of imaging findings have been purported to be important; however, results have been inconclusive, inconsistent, or not replicated in independent studies. The purpose of this study was to report imaging characteristics for a large cohort of patients with CM-I and identify the imaging findings associated with surgical decompression.

Methods: Patients were identified using ICD-9 codes for CM-I for the period from 1996 to 2017. 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, or 3) did not have available preoperative MRI. Retrospective chart review was performed to collect demographic and clinical data. Imaging parameters were measured according to the Chiari I Malformation Common Data Elements.

Results: A total of 731 patients were included for analysis, having a mean follow-up duration of 25.5 months. The mean age at presentation was 8.5 years. The mean tonsil position was 11.4 mm below the foramen magnum, and 62.8% of patients had a pegged tonsil shape. Two hundred patients (27.4%) underwent surgery for life-dominating tussive headache, lower cranial nerve dysfunction, syrinx, and/or brainstem dysfunction. Surgical treatment was associated with a syrinx (OR 20.4, 95% CI 12.3-33.3, p < 0.0001), CM-1.5 (OR 1.797, 95% CI 1.08-2.98, p = 0.023), lower tonsil position (OR 1.130, 95% CI 1.08-1.18, p < 0.0001), and congenital fusion of cervical vertebrae (OR 5.473, 95% CI 1.08-27.8, p = 0.040). Among patients with benign CM-I, tonsil position was statistically significantly associated with future surgery.

Conclusions: Comprehensive imaging characteristics for a large cohort of patients with CM-I are reported. Analysis showed that a lower tonsillar position, a syrinx, and CM-1.5 were associated with undergoing posterior fossa decompression. This study demonstrates the importance of considering imaging findings in the context of patient symptomatology.
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http://dx.doi.org/10.3171/2020.9.PEDS20347DOI Listing
April 2021

Objective Craniometric Versus Subjective Outcome Ratings in Endoscopic and Open Sagittal Synostosis Surgery.

J Craniofac Surg 2021 Jan 28. Epub 2021 Jan 28.

Department of Neurosurgery Department of Surgery, Division of Plastic Surgery, University of Alabama at Birmingham, AL Department of Neurosurgery, Penn State College of Medicine, Hershey, PA.

Background: Despite advances in surgical treatments and assessments of objective outcomes in surgery for sagittal synostosis, there is no agreement regarding the optimal assessment of postoperative outcomes. Additionally, few studies have evaluated subjective assessments of cranial morphology after surgical correction. This study sought to evaluate the utility of subjective aesthetic outcome assessment and compare these assessments to established craniometric outcomes in patients undergoing surgery for isolated sagittal synostosis.

Methods: Nineteen raters (5 parents, 4 surgeons, 5 trainees and 5 nurses) evaluated fifty patients who underwent surgical correction of isolated sagittal synostosis using standardized postoperative patient photos and a five-point Likert scale. Previously established anthropomorphic measurements were recorded postoperatively in these same patients and comparisons were made between the objective anthropomorphic and subjective outcome evaluations.

Results: There were no statistically significant correlations between age-controlled cephalic index, head circumference, or euryon-euryon diameter and subjective aesthetic scores. Lay persons assigned a significantly lower proportion of scores (37.9%) as middle values (2, 3, or 4) compared with faculty (64.8%). There was a statistically significant association between high scores given by surgical faculty and laypersons (P < 0.001).

Conclusions: Subjective measurement of cosmetic outcome is a useful metric in surgical correction of craniosynostosis. Although no correlations were found between objective measurements and subjective aesthetic scores, cosmetic assessments by surgeons demonstrated strong correlation with lay perception, indicating that these ratings may be a good gauge of overall cosmetic outcome. When used in combination, objective and subjective measurements provide unique value to assess outcomes after surgery for craniosynostosis.
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http://dx.doi.org/10.1097/SCS.0000000000007500DOI Listing
January 2021

Prevalence of Sleep Disordered Breathing in Children With Myelomeningocele.

Neurosurgery 2021 03;88(4):785-790

Division of Pediatric Neurosurgery, Department of Neurosurgery, University of Alabama at Birmingham, Birmingham, Alabama.

Background: Retrospective studies have shown high rates of sleep disordered breathing in children with myelomeningocele. However, most patients included in those studies underwent polysomnography because of symptoms, so the prevalence of sleep disordered breathing in this population is unknown.

Objective: To determine the prevalence of sleep disordered breathing in children with myelomeningocele using screening polysomnography.

Methods: In this cross-sectional study, all children with myelomeningocele seen in a multi-disciplinary spina bifida clinic between 2016 and 2020 were referred for polysomnography regardless of clinical symptoms. Included children had not previously undergone polysomnography. The primary outcome for this study was presence of sleep disordered breathing, defined as Apnea-Hypopnea Index (AHI, number of apnea or hypopnea events per hour of sleep) greater than 2.5. Clinical and demographic variables relevant to myelomeningocele were also prospectively collected and tested for association with presence of sleep apnea.

Results: A total of 117 participants underwent polysomnography (age 1 mo to 21 yr, 49% male). The majority were white, non-Hispanic. Median AHI was 1.9 (interquartile range 0.6-4.2). A total of 49 children had AHI 2.5 or greater, yielding a sleep disordered breathing prevalence of 42% (95% CI 33%-51%). In multivariable logistic regression analysis, children with more rostral neurological lesion levels had higher odds of sleep disordered breathing (OR for thoracic, mid-lumbar, and low-lumbar: 7.34, 3.70, 4.04, respectively, compared to sacral level, P = .043).

Conclusion: Over 40% of a sample of children with myelomeningocele, who underwent screening polysomnography, had significant sleep disordered breathing. Routine screening polysomnography may be indicated in this population.
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http://dx.doi.org/10.1093/neuros/nyaa507DOI Listing
March 2021

Shunt failure clusters: an analysis of multiple, frequent shunt failures.

J Neurosurg Pediatr 2020 Dec 18:1-7. Epub 2020 Dec 18.

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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http://dx.doi.org/10.3171/2020.7.PEDS20199DOI Listing
December 2020

Post-Traumatic Stress Symptoms in Caregivers and Children with Hydrocephalus.

World Neurosurg 2021 04 9;148:e66-e73. Epub 2020 Dec 9.

Department of Neurosurgery, Division of Pediatrics, University of Alabama at Birmingham, Birmingham, Alabama, USA.

Objective: Hydrocephalus is a disorder of cerebrospinal fluid dynamics, traditionally treated by placement of a ventricular shunt. Shunts are effective but imperfect as they fail in an unpredictable pattern, and the patient's well-being is dependent on adequate shunt function. The omnipresent threat of shunt failure along with the potential need for invasive investigations can be stressful for patients and caregivers. Our objective was to measure post-traumatic stress symptoms (PTSS) in children with hydrocephalus and their caregivers.

Methods: A cross-sectional analysis of children with hydrocephalus and their caregivers was conducted. Caregivers completed a measure of their own PTSS (the Post-Traumatic Stress Disorders Checklist for the Diagnostic and Statistical Manual of Mental Disorders-V) and resilience (the Connor Davidson Resilience Scale). Pediatric patients rated their own PTSS and resilience using the Acute Stress Checklist for Kids and Connor Davidson Resilience Scale.

Results: Ninety-one caregivers completed the Post-Traumatic Stress Disorders Checklist for the Diagnostic and Statistical Manual of Mental Disorders-V. Mean score was 17.0 (standard deviation 15.7; median 13.0). Fourteen percent scored above 33, the threshold suggestive of a preliminary diagnosis of post-traumatic stress disorder. There was a statistically significant association between caregiver post-traumatic stress and marital status, child's race, and caregiver education. More than half (52%) of caregivers reported their child's hydrocephalus as the most significant source of their PTSS. Children did not have markedly elevated levels of PTSS. Forty-one percent of caregivers and 60% of children scored in the lowest resilience quartile compared with the general population.

Conclusions: Results from this study suggest that post-traumatic stress affects caregivers with hydrocephalus, yet levels of resilience for caregivers and pediatric patients are low.
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http://dx.doi.org/10.1016/j.wneu.2020.12.008DOI Listing
April 2021

Anxiety, depression, fatigue, and headache burden in the pediatric hydrocephalus population.

J Neurosurg Pediatr 2020 Jul 24:1-7. Epub 2020 Jul 24.

1Department of Neurosurgery, Division of Pediatrics; Departments of.

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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http://dx.doi.org/10.3171/2020.4.PEDS19697DOI Listing
July 2020

Hydrocephalus-related quality of life as assessed by children and their caregivers.

J Neurosurg Pediatr 2020 Jul 10:1-11. Epub 2020 Jul 10.

1Department of Neurosurgery, Division of Pediatric Neurosurgery.

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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http://dx.doi.org/10.3171/2020.4.PEDS19660DOI Listing
July 2020

Caregiver-provider communication after resection of pediatric brain tumors.

J Neurosurg Pediatr 2020 May 29:1-7. Epub 2020 May 29.

2Department of Neurosurgery, Division of Pediatrics.

Objective: Because caregivers are a crucial part of a child's medical care, it is important to understand how to best communicate with them during hospitalization. Qualitative research can elucidate the best strategies for effective parent-provider communication. This study aims to reveal communication styles of neurosurgery team members, and to identify areas for improvement in the future.

Methods: Caregivers of children with a newly diagnosed brain tumor requiring neurosurgery were enrolled during their child's hospitalization. During routine follow-up clinic visits within 3 months after diagnosis and tumor resection, caregivers participated in a semistructured interview, which assessed the quality of communication with the neurosurgery and oncology teams during hospitalization. Interviews were audio-recorded, transcribed, and coded for common themes until thematic saturation was reached.

Results: During caregiver interviews (N = 22), several domains were discussed including communicating the diagnosis to the patient and siblings, to the rest of the family/support network, and with the neurosurgery team. Regarding parent-neurosurgeon communication, 82% of caregivers identified at least one positive aspect and 55% identified at least one negative aspect of communication. Caregivers who provided positive feedback appreciated that their neurosurgeon was thorough (73%), direct (27%), or compassionate (14%). They also valued when providers would speak "on my level" (18%) and would speak directly to the patient (27%). In terms of negative feedback, caregivers identified miscommunications (32%), discussing the diagnosis in front of the child before feeling prepared to do so (14%), and a lack of clarity about expectations, medications, or treatment (32%).

Conclusions: These data provide specific ways in which neurosurgery providers have communicated effectively with caregivers and identify areas for improvement. Results have been used to develop a navigator-led intervention geared toward improving parent-provider communication during hospitalization for resection of a brain tumor.
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http://dx.doi.org/10.3171/2020.4.PEDS19696DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8099025PMC
May 2020

Functional outcomes at 2 years of age following treatment for posthemorrhagic hydrocephalus of prematurity: what do we know at the time of consult?

J Neurosurg Pediatr 2020 Feb 14:1-9. Epub 2020 Feb 14.

1Department of Neurosurgery, Division of Pediatric Neurosurgery.

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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http://dx.doi.org/10.3171/2019.12.PEDS19381DOI Listing
February 2020

Pediatric herniated lumbar disc: a population-based risk factor analysis.

J Neurosurg Pediatr 2019 Nov 29:1-8. Epub 2019 Nov 29.

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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http://dx.doi.org/10.3171/2019.9.PEDS19167DOI Listing
November 2019

Care management and contemporary challenges in spina bifida: a practice preference survey of the American Society of Pediatric Neurosurgeons.

J Neurosurg Pediatr 2019 Aug 30:1-10. Epub 2019 Aug 30.

1Department of Neurosurgery, University of Alabama at Birmingham.

Objective: Neurosurgical management preferences related to myelomeningocele (MMC) care demonstrate significant variability. The authors sought to evaluate variability in practice patterns across a group of senior pediatric neurosurgeons. The purpose of this study was to identify the extent of variability and of consensus with regard to neurosurgical management of MMC and associated hydrocephalus, Chiari II malformation, and tethered spinal cord.

Methods: A 43-question survey was distributed electronically to the members of the American Society of Pediatric Neurosurgeons (ASPN). The survey covered domains such as clinic case volume, newborn management, hydrocephalus management, transition to adulthood, clinical indications for shunt revision, Chiari II malformation decompression (C2MD), and tethered cord release (TCR). Ninety responses were received from 200 active ASPN members, for an overall response rate of 45%.

Results: The majority (58%) of respondents closed 5-15 new cases of open MMC per year. Nearly all (98%) respondents perform back closure within 48 hours of birth, with the majority imbricating the placode and striving for a 3- to 4-layer closure. The most consistent indications for surgical intervention in early hydrocephalus were CSF leak from the back (92%), progressive ventricular enlargement (89%), and brainstem symptoms, including apnea/bradycardia (81%), stridor (81%), and dysphagia (81%). Eighty percent of respondents indicated that spina bifida care is delivered through multidisciplinary clinics, with neurosurgery, orthopedic surgery, urology, physical therapy, and social work as the most common disciplines included. One-third of clinics see both pediatric and adult patients, one-third offer a formal transition program to adult care, and one-third have no transition program. The vast majority of respondents offer prenatal counseling (95%), referral for in utero closure (66%), and endoscopic third ventriculostomy/choroid plexus cauterization (72%). Respondents were more willing to perform shunt revision for symptoms alone than for image changes alone. An asymptomatic broken shunt without ventricular enlargement produced responses evenly divided between observation, intervention, and further investigation. Operative shunt exploration was always performed before C2MD by 56% of respondents and performed sometimes by 40% of respondents. Symptoms of brainstem dysfunction were the strongest clinical triggers reported for C2MD, while declines in urinary continence, leg strength or sensation, or ambulation were the most consistent thresholds for TCR.

Conclusions: Significant disparities exist surrounding key areas of decision making regarding treatment for patients with MMC, though there are central areas of agreement among ASPN members. Additionally, there is significant variation in the clinical management of chronic hydrocephalus, C2MD, and TCR, underscoring the need for further research into these specific areas.
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http://dx.doi.org/10.3171/2019.5.PEDS18738DOI Listing
August 2019

Program Evaluation of Camp V.I.P: Promoting Self-confidence and Independence for Patients with Spina Bifida.

J Pediatr Nurs 2019 Jul - Aug;47:30-35. Epub 2019 Apr 23.

University of Alabama at Birmingham, Department of Neurosurgery, Section of Pediatric Neurosurgery, United States of America; Children's of Alabama, United States of America. Electronic address:

Purpose: Camp V.I·P (Victory, Independence, Possibilities) was established by the Spina Bifida Association of Alabama in 2012. The goal is to provide children with spina bifida (SB) and their families a traditional-style summer camp focusing on self-empowerment and fostering independence in medical self-management.

Design And Methods: Part 1 of this study describes Camp V.I·P. structure. Part 2 describes an online survey to caregivers of campers gauging camp effectiveness and value and evaluating perceptions of confidence/independence and family connections. Camper characteristics were compared to the overall SB clinic population.

Results: 63 children with SB, 81 siblings, and over 100 caregivers have attended camp. Campers are similar to the overall clinic population in race, gender, and ambulation status. At camp, there are significantly more children with myelomeningocele (versus other spinal dysraphism) and lumbar functional level (versus thoracic). Survey results show that confidence grew in 93% of campers, and 86% gained greater independence. Parents stated that they created lifelong friendships. The majority of caregivers (91%) indicated "certainty" that they would attend camp again.

Conclusion: Camp V.I·P is a family-centered environment that emphasizes respite, nurture, and the continuum between family and clinic. Our research shows that camp can be a useful tool to aid in independence, confidence, and social adaption.

Practice Implications: Camp has become an integral part of our comprehensive care of children with SB and has been shown to reinforce what is taught through the clinic by creating trust between the care team and campers.
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http://dx.doi.org/10.1016/j.pedn.2019.04.014DOI Listing
January 2020

Predictors of endoscopic third ventriculostomy ostomy status in patients who experience failure of endoscopic third ventriculostomy with choroid plexus cauterization.

J Neurosurg Pediatr 2019 04;24(1):41-46

1Surgical Outcomes Center for Kids, Monroe Carell Jr. Children's Hospital at Vanderbilt, Nashville.

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.
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http://dx.doi.org/10.3171/2019.2.PEDS18743DOI Listing
April 2019

Patients with "benign" Chiari I malformations require surgical decompression at a low rate.

J Neurosurg Pediatr 2019 01;23(4):498-506

1Department of Neurosurgery and Division of Pediatric Neurosurgery, University of Alabama at Birmingham, Alabama; and.

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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http://dx.doi.org/10.3171/2018.10.PEDS18407DOI Listing
January 2019

Skin breakdown of the feet in patients with spina bifida: Analysis of risk factors.

J Pediatr Rehabil Med 2018 ;11(4):237-241

Department of Neurosurgery, University of Alabama at Birmingham, Birmingham, AL, USA.

Purpose: Recent studies on patients with spina bifida have noted an increased incidence of skin breakdown with more proximal functional neurologic level. We hypothesized that there would be an inverse relationship between skin breakdown of the foot and severity of functional level of lesion, because patients with more caudal levels of lesion spend more time ambulating.

Methods: The National Spina Bifida Patient Registry (NSBPR) at Children's of Alabama was queried for the presence of skin breakdown of the foot, ambulatory status, functional neurologic level, and diagnosis of myelomeningocoele (MMC) vs. non-myelomeningocoele (non-MMC). Univariate and multivariate analysis were performed.

Results: Of 491 total patients, 378 were MMC and 113 were non-MMC. Eighty-five of 378 (22.5%) patients with MMC and 5 of 113 (4.4%) non-MMC patients reported skin breakdown (p= 0.009). Thoracic and lumbar levels were compared to the sacral level for statistical analysis. Skin breakdown occurred in 26.2% of thoracic (p= 0.001), 33.3% of high-lumbar (p= 0.001), 21.5% of mid-lumbar (p= 0.008), 26.2% of low-lumbar (p= 0.001), and 6.1% of sacral level patients. Ambulatory status was not significant on multivariate analysis.

Conclusion: A diagnosis of MMC is a significant, independent risk factor for skin breakdown of the foot. Compared to sacral level, thoracic and lumbar levels of function were also independently significant. Ambulatory status was not significant.
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http://dx.doi.org/10.3233/PRM-170520DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6953480PMC
October 2019

Distress and psychosocial risk in families with newly diagnosed pediatric brain tumors.

J Neurosurg Pediatr 2018 10;23(1):40-47

4Division of Hematology/Oncology, Department of Pediatrics, University of Alabama at Birmingham, Alabama.

In BriefThe investigators administered surveys to parents whose children were hospitalized with a brain tumor. They learned that parents in this situation are highly distressed, and that families have multiple sources of stress when a child is hospitalized. The investigators used this information to improve the psychosocial supportive care provided at their hospital. In general, these findings have the potential to improve the patient's and family's experience with a new brain tumor by guiding teams to address family needs as well as the child's acute medical needs.
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http://dx.doi.org/10.3171/2018.7.PEDS18297DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6944277PMC
October 2018

Coping strategies used by caregivers of children with newly diagnosed brain tumors.

J Neurosurg Pediatr 2018 10;23(1):30-39

2Division of Pediatrics, Department of Neurosurgery, University of Alabama at Birmingham.

In BriefThe authors interviewed families whose children had recently been hospitalized with a new brain tumor. From these interviews, they identified parents' coping strategies for handling the stress of having a child with a newly diagnosed tumor. Some strategies are considered "adaptive" and help parents deal with the stress better. Others are "maladaptive," leading to worse outcomes. Parents of children with brain tumors are at risk for maladaptive coping. Efforts to teach parents how to cope effectively with the stress of a sick child have the potential to improve outcomes.
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http://dx.doi.org/10.3171/2018.7.PEDS18296DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6944290PMC
October 2018

Reducing inequities in preventable neural tube defects: the critical and underutilized role of neurosurgical advocacy for folate fortification.

Neurosurg Focus 2018 10;45(4):E20

1Department of Neurosurgery, University of Alabama at Birmingham, Alabama.

Neural tube defects (NTDs) are one of the greatest causes of childhood mortality and disability-adjusted life years worldwide. Global prevalence at birth is approximately 18.6 per 10,000 live births, with more than 300,000 infants with NTDs born every year. Substantial strides have been made in understanding the genetics, pathophysiology, and surgical treatment of NTDs, yet the natural history remains one of high morbidity and profound impairment of quality of life. Direct and indirect costs of care are enormous, which ensures profound inequities and disparities in the burden of disease in countries of low and moderate resources. All indices of disease burden are higher for NTDs in developing countries. The great tragedy is that the majority of NTDs can be prevented with folate fortification of commercially produced food. Unequivocal evidence of the effectiveness of folate to reduce the incidence of NTDs has existed for more than 25 years. Yet, the most comprehensive surveys of effectiveness of implementation strategies show that more than 100 countries fail to fortify, and consequently only 13% of folate-preventable spina bifida is actually prevented. Neurosurgeons harbor a disproportionate, central, and fundamental role in the management of NTDs and enjoy high standing in society. No organized group in medicine can speak as authoritatively or convincingly. As a result, neurosurgeons and organized neurosurgery harbor disproportionate potential to advocate for more comprehensive folate fortification, and thereby prevent the most common and severe birth defect to impact the human nervous system. Assertive, proactive, informed advocacy for folate fortification should be a central and integral part of the neurosurgical approach to NTDs. Only by making the prevention of dysraphism a priority can we best address the inequities often observed worldwide.
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http://dx.doi.org/10.3171/2018.7.FOCUS18231DOI Listing
October 2018

Surveillance survey of family history in children with neural tube defects.

J Neurosurg Pediatr 2017 Jun 31;19(6):690-695. Epub 2017 Mar 31.

Department of Neurosurgery and.

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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http://dx.doi.org/10.3171/2016.12.PEDS1668DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5640157PMC
June 2017

Risk factors for surgical site infection following nonshunt pediatric neurosurgery: a review of 9296 procedures from a national database and comparison with a single-center experience.

J Neurosurg Pediatr 2017 04 10;19(4):407-420. Epub 2017 Feb 10.

Department of Neurosurgery, Division of Pediatric Neurosurgery, The University of Alabama at Birmingham and Children's Hospital of Alabama, Birmingham, Alabama

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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http://dx.doi.org/10.3171/2016.11.PEDS16454DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5450913PMC
April 2017

A comparison of the MOMS trial results to a contemporaneous, single-institution, postnatal closure cohort.

Childs Nerv Syst 2017 Apr 27;33(4):639-646. Epub 2016 Dec 27.

Department of Neurological Surgery, Division of Pediatric Neurosurgery, The University of Alabama at Birmingham, 1600 7th Ave. S, Lowder 400, Birmingham, AL, 35233, USA.

Purpose: We evaluate a single-institution cohort of mothers contemporaneous with the Management of Myelomeningocele Study (MOMS) trial to determine the generalizability of MOMS results and compare shunt rates.

Methods: A retrospective chart review identified patients with myelomeningocele born between 2003 and 2009. We applied MOMS eligibility criteria and compared sociodemographic variables between patients at our institution who would have been eligible or ineligible and MOMS participants. Finally, we applied the original MOMS primary outcome and the revised primary outcome to our cohort.

Results: Of the 78 patients, 55 (70.5%) were eligible for the MOMS trial. Mean maternal age, race, and marital status were different from both MOMS groups. Comparing our series to MOMS postnatal shows fewer female infants (44.9 vs. 63.8%, p = 0.017) and more thoracic lesions (12.8 vs. 3.8%, p = 0.038). Shunt rates in our cohort (84.6%) were higher than MOMS prenatal and similar to MOMS postnatal (44.0 and 83.7%, respectively). Fewer children met the original primary outcome than the postnatal group (84.6 vs. 97.8%, p = 0.002). There was no significant difference between our cohort and the prenatal group (84.6 vs. 72.5%, p = 0.058). When applying the revised criteria, we find the opposite: a significant difference between local and MOMS prenatal (84.6 vs. 49.5%, p < 0.001) but no difference between the local group and MOMS postnatal (84.6 vs. 87.0%, p = 0.662).

Conclusions: Mothers in our cohort differ from mothers enrolled in MOMS via several sociodemographic factors. Baseline fetal characteristics show a significantly higher functional lesion level in between our cohort and MOMS. Treatment of hydrocephalus in our series tracks almost identically with original MOMS shunt criteria. Revision of the criteria led to greater concordance between meeting criteria and receiving a shunt in MOMS patients, but changes the results in our series.
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http://dx.doi.org/10.1007/s00381-016-3328-3DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5383528PMC
April 2017

Soluble membrane attack complex is diagnostic for intraventricular shunt infection in children.

JCI Insight 2016 07 7;1(10):e87919. Epub 2016 Jul 7.

Division of Pediatric Neurosurgery, Department of Neurosurgery, Children's of Alabama.

Background: Children treated with cerebrospinal fluid (CSF) shunts to manage hydrocephalus frequently develop shunt failure and/or infections, conditions that present with overlapping symptoms. The potential life-threatening nature of shunt infections requires rapid diagnosis; however, traditional microbiology is time consuming, expensive, and potentially unreliable. We set out to identify a biomarker that would identify shunt infection.

Methods: CSF was assayed for the soluble membrane attack complex (sMAC) by ELISA in patients with suspected shunt failure or infection. CSF was obtained at the time of initial surgical intervention. Statistical analysis was performed to assess the diagnostic potential of sMAC in pyogenic-infected versus noninfected patients.

Results: Children with pyogenic shunt infection had significantly increased sMAC levels compared with noninfected patients (3,211 ± 1,111 ng/ml vs. 26 ± 3.8 ng/ml, = 0.0001). In infected patients undergoing serial CSF draws, sMAC levels were prognostic for both positive and negative clinical outcomes. Children with delayed, broth-only growth of commensal organisms (. , . , etc.) had the lowest sMAC levels (7.96 ± 1.7 ng/ml), suggesting contamination rather than shunt infection.

Conclusion: Elevated CSF sMAC levels are both sensitive and specific for diagnosing pyogenic shunt infection and may serve as a useful prognostic biomarker during recovery from infection.

Funding: This work was supported in part by the Impact Fund of Children's of Alabama.
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http://dx.doi.org/10.1172/jci.insight.87919DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5033849PMC
July 2016

Radiographic markers of clinical outcomes after endoscopic third ventriculostomy with choroid plexus cauterization: cerebrospinal fluid turbulence and choroid plexus visualization.

J Neurosurg Pediatr 2016 Sep 13;18(3):287-95. Epub 2016 May 13.

Division of Pediatric Neurosurgery, Children's of Alabama and University of Alabama at Birmingham, Alabama.

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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http://dx.doi.org/10.3171/2016.3.PEDS15364DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5476945PMC
September 2016

Sleep-disordered breathing in patients with myelomeningocele.

J Neurosurg Pediatr 2015 Jul 3;16(1):30-5. Epub 2015 Apr 3.

Department of Neurosurgery;

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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http://dx.doi.org/10.3171/2014.11.PEDS14314DOI Listing
July 2015

Assessing health-related quality of life in children with spina bifida.

J Neurosurg Pediatr 2015 Feb 21;15(2):144-9. Epub 2014 Nov 21.

Pediatric Neurosurgery, Department of Neurosurgery, University of Alabama at Birmingham, Alabama; and.

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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http://dx.doi.org/10.3171/2014.10.PEDS1441DOI Listing
February 2015
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