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Does subtotal resection ameliorate hypothalamic morbidity in pediatric craniopharyngioma? A 30-year retrospective cohort study

Omer K. Yousuf, Arsalaan Salehani, Kathrin Zimmerman, Dagoberto Estevez-Ordonez, Casey Madura, Anastasia Arynchyna-Smith, James M. Johnston, Curtis J. Rozzelle, Brandon G. Rocque, and Jeffrey P. Blount

OBJECTIVE

The optimal extent of resection of craniopharyngiomas to minimize the long-term risks of hypothalamic and endocrine dysfunction (obesity and panhypopituitarism) in children remains uncertain. The purpose of this study was to report long-term outcomes of pediatric patients with craniopharyngioma undergoing surgical treatment and to study rates of endocrinological and hypothalamic dysfunction in association with extent of resection.

METHODS

This retrospective study was performed in a cohort of children who underwent resection for craniopharyngioma at Children’s of Alabama between 1990 and 2020. The primary outcome was hypothalamic dysfunction defined as a 0.5 increase in body mass index (BMI) Z-score and as a BMI > 2 SDs with or without psychiatric disturbances. Univariable analysis was performed using ANOVA, Wilcoxon rank-sum test, Pearson’s chi-square test, and Fisher’s exact test as appropriate. Missing data on the primary outcome were handled via multiple imputations. Relative risks were estimated using a multivariable generalized linear model with a priori variables selected using a modified Poisson regression approach with robust error variance to estimate risk ratios.

RESULTS

The cohort includes 39 patients (24 girls and 15 boys; age range 1 month–16 years) who underwent resection of craniopharyngioma at the authors’ center between 1990 and 2020. The preoperative goal of treatment was cyst decompression (CD) in 5, subtotal resection (STR) in 13, and gross-total resection (GTR) in 21 patients. The median long-term follow-up after surgery was 8.11 years (average 8.21, range 0.4–24.33 years). Univariate analysis demonstrated a statistically significant increase in hypothalamic dysfunction in patients undergoing GTR when compared to those undergoing STR or CD at 1 month postoperatively (p = 0.006) and 6–11 months postoperatively (p = 0.010), but with this difference not persisting beyond 1 year. Multivariable analysis showed patients older than 10 years at time of surgery to be the most affected and at highest risk of developing significant hypothalamic dysfunction. There was no significant difference in pituitary or neurological function between the STR/CD and GTR groups at 12–24 months or at most recent follow-up. There was no significant difference in BMI Z-scores between the STR/CD and GTR groups at 6–12 months or at most recent follow-up.

CONCLUSIONS

Both STR and GTR of craniopharyngioma were associated with significant endocrinological sequelae after 1 year. These potential complications should be discussed with patients and their families, and postoperative protocols should include early nutritional and endocrinological interventions with endocrinologist consultation.

In Journal of Neurosurgery: Pediatrics Volume 32: Issue 5 (Nov 2023)

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Hemispherectomy Outcome Prediction Scale: a validity study

Andrew T. Hale, Dagoberto Estevez-Ordonez, Jana Badrani, Wen Sha, Anastasia Arynchyna-Smith, Monisha Goyal, Ismail Mohamed, Pongkiat Kankirawatana, Curtis J. Rozzelle, and Jeffrey P. Blount

OBJECTIVE

Hemispherectomy is highly effective for patients with medically refractory epilepsy (MRE) arising from a single hemisphere. Recently, the Hemispherectomy Outcome Prediction Scale (HOPS) was developed as a prediction tool for seizure freedom after hemispherectomy. The authors’ goal was to perform a validation study to determine the generalizability of the HOPS score.

METHODS

The authors present an observational, retrospective, 20-year, single-institution, two-surgeon experience using the lateral peri-insular hemispherectomy approach to validate the HOPS score. Variables used to derive the HOPS score included seizure onset age, semiology, PET hypometabolism, seizure substrate, and history of prior epilepsy resection. Multivariable logistic regression, multiple imputation, and Bayesian analyses were used to determine validity.

RESULTS

The authors’ cohort comprised 60 patients; 55% of patients were male and 78% were Caucasian. The median age at first hemispherectomy surgery was 72 months. At 1 year postoperatively, 80% of patients had Engel class I outcomes, analogous to most contemporary series. All patients who experienced seizure recurrence after hemispherectomy did so within the first 2 years postoperatively. Sixteen (27%) and 10 (17%) patients had contralateral MRI findings and hypometabolism on PET, respectively. Both a multivariable logistic regression model using HOPS score variables (model p = 0.2588) and a revised model that included contralateral MRI findings (model p = 0.4715) were not statistically significant in this cohort. Bayesian analysis also did not validate the HOPS score.

CONCLUSIONS

While seizure outcome prediction tools may be helpful for counseling patients about postoperative outcomes, rigorous validity and reliability testing are required. Prospective, standardized, and longitudinal evaluation of patients undergoing hemispherectomy are needed.

In Journal of Neurosurgery: Pediatrics Volume 32: Issue 4 (Oct 2023)

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Letter to the Editor. The importance of vagus nerve stimulation for young children

Nallammai Muthiah and Taylor J. Abel

In Journal of Neurosurgery: Pediatrics Volume 32: Issue 4 (Oct 2023)

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First-time tethered cord release among adults with myelomeningocele: an analysis of people in the National Spina Bifida Patient Registry

Mark S. Dias, Ming Wang, Menglu Liang, Elias B. Rizk, Robin Bowman, Michael D. Partington, Jeffrey P. Blount, Brandon G. Rocque, Betsy Hopson, Amy Lee, and William O. Walker

OBJECTIVE

The authors analyzed the National Spina Bifida Patient Registry (NSBPR), a national registry that tracks the outcomes for people with various forms of spinal dysraphism, to determine the ongoing longitudinal risk for tethered cord release (TCR) among adults with myelomeningocele who had not previously undergone TCR during childhood. The authors also sought to identify the impact of lesion level, ambulation status, and prior treatments for hydrocephalus or Chiari malformations on TCR rates.

METHODS

Adults in the registry who had not previously undergone TCR during childhood were studied. This group was compared with the remaining adults in the registry. The frequency of first-time TCR and time to TCR (using Kaplan-Meier analysis) were determined independently for males and females. Cox proportional hazards analysis identified correlations between sex, best lesion level and ambulation status prior to TCR, and previous treatments for hydrocephalus and Chiari decompression.

RESULTS

Among 967 adults in the NSBPR (422 [43.6%] males and 545 [56.4%] females) who had not undergone TCR during childhood, the authors identified 47 people (4.9%) who underwent their first TCR during adulthood. This study cohort had significantly better mean functional motor levels and ambulation compared with the remaining adult cohort (both p < 0.001). The study group included 35 females (74.5%) and 12 males (25.5%); this sex distribution was significantly different in comparison with the remaining adult cohort (p = 0.016). The Kaplan-Meier curves for first TCR for females and males were significantly different (p = 0.01, log-rank test). TCR rates were correlated with sex (males had decreased risk; OR 0.31, 95% CI 0.16–0.62, p < 0.001), prior treatment for hydrocephalus (those who underwent prior treatment had decreased risk; OR 0.21, 95% CI 0.20–0.42, p < 0.001), and prior treatment for Chiari malformation (those who underwent prior treatment had greater risk; OR 3.84, 95% CI 1.50–9.88, p = 0.005).

CONCLUSIONS

Adults with myelomeningocele who escape childhood without undergoing TCR have an ongoing, albeit decreased, risk for spinal cord tethering requiring TCR. This risk is obviously not due to spinal column growth and therefore must reflect other factors such as dynamic changes in spinal cord health over time. Among people with MMC who underwent their first TCR as adults, females seemed to be overrepresented. Similar to the authors’ prior childhood study, people who underwent previous Chiari decompression seemed to be overrepresented, whereas those who underwent previous treatment for hydrocephalus seem to be underrepresented. These novel findings deserve further study.

In Journal of Neurosurgery: Spine Volume 39: Issue 4 (Oct 2023)

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Letter to the Editor. The molecular mechanisms of folic acid fortification to prevent spina bifida

Juan Armando Mejía and Luis Garcia Rairan

In Journal of Neurosurgery: Pediatrics Volume 31: Issue 6 (Jun 2023)

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Editorial. Neurocognition in fetal myelomeningocele closure: "not shunting per se"

Jeffrey P. Blount and Brandon G. Rocque

In Journal of Neurosurgery: Pediatrics Volume 31: Issue 6 (Jun 2023)

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Understanding and identifying the needs of parent caregivers of children with hydrocephalus: a qualitative study

Katherine Barnes, Kathrin Zimmerman, Ivan Herbey, Anastasia Arynchyna-Smith, Bobby May, Caroline Arata Wessinger, Laura E. Dreer, Lieu Thompson, Nataliya V. Ivankova, Curtis J. Rozzelle, James M. Johnston, Jeffrey P. Blount, and Brandon G. Rocque

OBJECTIVE

Hydrocephalus is inherently unpredictable. Most parents whose child is diagnosed with hydrocephalus do not anticipate the diagnosis, nor can anyone predict if or when a child’s shunt will fail and require emergency surgery. Previous research has shown that children with hydrocephalus and their caregivers experience significant posttraumatic stress symptoms secondary to the diagnosis. This study aims to understand caregiver experiences and needs, identify gaps in resources/support, and determine opportunities to improve care.

METHODS

Semistructured interviews were conducted with parent caregivers of children with hydrocephalus to learn about their experiences with the hydrocephalus diagnosis, hospitalizations, surgeries, coping and support, challenges of caring for a child with hydrocephalus, and logistics for a proposed support program. De-identified interviews were audio-recorded, transcribed, and analyzed for themes.

RESULTS

Thematic saturation was reached after 17 interviews. Five major themes emerged: 1) coping with the diagnosis, 2) received support, 3) hydrocephalus management, 4) implications for intervention, and 5) psychosocial stressors for caregivers. A top priority was balanced, trustworthy information delivered with compassion and updated throughout the child’s life. Caregivers described a variety of coping strategies, but a majority reported a need for support in processing complex emotions and dealing with the uncertainty of their child’s hydrocephalus. Most agreed that having a caregiver support network, medical professionals available for referrals and questions, and referrals to support services and therapies would facilitate feeling supported and providing the best care for their children.

CONCLUSIONS

Parent caregivers are critical to the health and well-being of children with hydrocephalus, and it is essential to understand their experiences to improve care. Providing well-defined information, psychosocial support, and resources will help to equip parent caregivers to be advocates for their children and to improve both the caregiver and the child’s quality of life.

In Journal of Neurosurgery: Pediatrics Volume 31: Issue 5 (May 2023)

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Postoperative seizure freedom after vagus nerve stimulator placement in children 6 years of age and younger

Pedram Maleknia, Timothy D. McWilliams, Ariana Barkley, Dagoberto Estevez-Ordonez, Curtis Rozzelle, and Jeffrey P. Blount

OBJECTIVE

Food and Drug Administration (FDA) approval for vagus nerve stimulator (VNS) implantation is limited to patients older than 4 years of age with medically refractory partial-onset seizures. In younger children with severe generalized epilepsy, however, VNS implantation remains off-label. In this study, the authors followed up on their previously reported cohort to review the longer-term safety and efficacy of VNS placement in children younger than 6 years with generalized medically refractory epilepsy (MRE), providing the largest cohort with > 2 years of follow-up to date in this age group.

METHODS

This was a retrospective observational cohort study of patients younger than 6 years of age with generalized MRE who underwent VNS implantation at a single institution between 2010 and 2020. Inclusion criteria encompassed failure of more than two antiepileptic drugs alone or in combination, neurologist referral for vagus nerve stimulation, informed consent with knowledge of the off-label status in young children, and > 1 year of follow-up. Outcome measures included seizure reduction rate ≥ 50% and postoperative morbidity defined nominally. Statistical analysis was conducted with Stata/SE.

RESULTS

Forty-five patients were included: 11 patients younger than 4 years of age and 34 between 4 and 6 years of age. There were no intraoperative complications. Perioperative complications within 1 year occurred in 11% (n = 5) of the patients and included two wound infections, a mild cough, hyperactivity, hoarseness, and 1 patient with persistent surgical site pain.

A seizure reduction ≥ 50% was observed in 36.4% (n = 4) of the patients younger than 4 years of age at the 6-month and 1-, 2-, and 5-year follow-ups. In the 4- to 6-year-old cohort, this was observed in 32.4% (n = 11) of the patients at 6 months, 41.2% (n = 14) at 1 year, 38.2% (n = 13) at 2 years, and 41.2% (n = 14) at 5 years.

CONCLUSIONS

VNS implantation for patients younger than 4 years of age with generalized onset MRE has not been approved by the FDA. This retrospective study establishes feasibility, illustrates an acceptable safety profile in children younger than 6 years, and demonstrates efficacy comparable to that reported in older patients.

In Journal of Neurosurgery: Pediatrics Volume 31: Issue 4 (Apr 2023)

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Editorial. The elusive fibrofatty filum holy grail

Jeffrey P. Blount and Michael D. Partington

In Journal of Neurosurgery: Pediatrics Volume 31: Issue 3 (Mar 2023)

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Editorial. Myelomeningocele care: moving beyond Groningen

Jeffrey P. Blount

In Journal of Neurosurgery: Pediatrics Volume 31: Issue 1 (Jan 2023)