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Predictors and timing of hydrocephalus treatment in patients undergoing prenatal versus postnatal surgery for myelomeningocele

Sasidhar Karuparti, Ashley Dunbar, Kaamya Varagur, Kavya Sudanagunta, Mark Mingo, Katherine H. Bligard, Anthony Odibo, Jesse Vrecenak, Sean McEvoy, David Limbrick Jr., Lindsay Peglar Marsala, Jagruti Anadkat, Ali Mian, and Jennifer M. Strahle

OBJECTIVE

Although hydrocephalus rates have decreased with intrauterine surgery for myelomeningocele (MMC), 40%–85% of children with MMC still go on to develop hydrocephalus. Prenatal ventricle size is known to be associated with later development of hydrocephalus; however, it is not known how prediction measures or timing of hydrocephalus treatment differ between pre- and postnatal surgery for MMC. The goal of this study was to determine anatomical, clinical, and radiological characteristics that are associated with the need for and timing of hydrocephalus treatment in patients with MMC.

METHODS

The authors retrospectively identified patients from Barnes Jewish Hospital or St. Louis Children’s Hospital between 2016 and 2021 who were diagnosed with MMC prenatally and underwent either pre- or postnatal repair. Imaging, clinical, and demographic data were examined longitudinally between treatment groups and hydrocephalus outcomes.

RESULTS

Fifty-eight patients were included (27 females, 46.6%), with a mean gestational age at birth of 36.8 weeks. Twenty-three patients (39.7%) underwent prenatal surgery. For the overall cohort, the ventricle size at prenatal ultrasound (HR 1.175, 95% CI 1.071–1.290), frontal-occipital horn ratio (FOHR) at birth > 0.50 (HR 3.603, 95% CI 1.488–8.720), and mean rate of change in head circumference (HC) in the first 90 days after birth (> 0.10 cm/day: HR 12.973, 95% CI 4.262–39.486) were identified as predictors of hydrocephalus treatment. The factors associated with hydrocephalus in the prenatal cohort were FOHR at birth > 0.50 (HR 27.828, 95% CI 2.980–259.846) and the rate of change in HC (> 0.10 cm/day: HR 39.414, 95% CI 2.035–763.262). The factors associated with hydrocephalus in the postnatal cohort were prenatal ventricle size (HR 1.126, 95% CI 1.017–1.246) and the mean rate of change in HC (> 0.10 cm/day: HR 24.202, 95% CI 5.119–114.431). FOHR (r = −0.499, p = 0.008) and birth HC (−0.409, p = 0.028) were correlated with time to hydrocephalus across both cohorts. For patients who underwent treatment for hydrocephalus, those in the prenatal surgery group were significantly more likely to develop hydrocephalus after 3 months than those treated with postnatal surgery, although the overall rate of hydrocephalus was significantly higher in the postnatal surgery group (p = 0.018).

CONCLUSIONS

Clinical and imaging factors associated with hydrocephalus treatment differ between those receiving pre- versus postnatal MMC repair, and while the overall rate of hydrocephalus is lower, those undergoing prenatal repair are more likely to develop hydrocephalus after 3 months of age. This has implications for clinical follow-up timing for patients treated prenatally, who may live at a distance from the treatment site.

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Risk factors for postoperative ventriculoperitoneal shunt requirement in pediatric patients with brain tumors invading or adjacent to CSF circulation pathways

Zesheng Ying, Wei Yang, Nijia Zhang, Hailang Sun, Di Zhang, Baojin Shang, Jiashu Chen, and Ming Ge

OBJECTIVE

Hydrocephalus is a common comorbidity of brain tumors in children that may persist following brain tumor resection. This study aimed to explore perioperative risk factors associated with postoperative ventriculoperitoneal shunt (VPS) placement for tumors located at or adjacent to the CSF circulation pathway.

METHODS

Patients aged 0–18 years with tumors invading or adjacent to the CSF circulation pathways who underwent brain tumor resection between October 2015 and September 2021 were included in this study. The outcome metric was whether patients underwent VPS placement within 6 months of tumor resection. Patients were followed up every 3–6 months after surgery. Demographic and perioperative imaging characteristics, clinical variables, and long-term treatments, including radiotherapy or chemotherapy, were included in the analysis.

RESULTS

Two hundred sixty-five children were included in this study. Of these patients, 38 (14.34%) underwent VPS placement within 6 months of tumor resection. One hundred thirty-two patients (49.81%) presented with preoperative hydrocephalus. Results from the multivariate analysis showed that medulloblastoma (OR 4.15, 95% CI 1.74–9.91, p = 0.001), lateral/third ventricle tumors (OR 4.07, 95% CI 1.33–12.30, p = 0.014), postoperative intraventricular hematoma (OR 3.36, 95% CI 1.53–7.38, p = 0.003), and presence of subdural hygroma in the nonoperated area within 48 hours after tumor resection (OR 2.78, 95% CI 1.15–6.74, p = 0.024) were independent risk factors for postoperative VPS placement.

CONCLUSIONS

Postoperative lateral/third ventricle hematoma and subdural hygroma in the nonoperated area, anatomical location, and tumor histology may be potential risk factors for a postoperative VPS after brain tumor resection.

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47th Annual Meeting The American Society of Pediatric Neurosurgeons

Jointly provided by AANS

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Auditory brainstem implantation: surgical experience and audiometric outcomes in the pediatric population

Yosef M. Dastagirzada, Alexander Eremiev, Jeffrey H. Wisoff, Emily Kay-Rivest, William H. Shapiro, Ansley Unterberger, Susan B. Waltzman, J. Thomas Roland, John G. Golfinos, and David H. Harter

OBJECTIVE

Pediatric data regarding treatment via an auditory brainstem implant (ABI) remains sparse. The authors aimed to describe their experience at their institution and to delineate associated demographic data, audiometric outcomes, and surgical parameters.

METHODS

An IRB-approved, retrospective chart review was conducted among the authors’ pediatric patients who had undergone auditory brainstem implantation between 2012 and 2021. Demographic information including sex, age, race, coexisting syndrome(s), history of cochlear implant placement, average duration of implant use, and follow-up outcomes were collected. Surgical parameters collected included approach, intraoperative findings, number of electrodes activated, and complications.

RESULTS

A total of 19 pediatric patients had an ABI placed at the authors’ institution, with a mean age at surgery of 4.7 years (range 1.5–17.8 years). A total of 17 patients (89.5%) had bilateral cochlear nerve aplasia/dysplasia, 1 (5.3%) had unilateral cochlear nerve aplasia/dysplasia, and 1 (5.3%) had a hypoplastic cochlea with ossification. A total of 11 patients (57.9%) had a history of cochlear implants that were ineffective and required removal. The mean length of implant use was 5.31 years (0.25–10 years). Two patients (10.5%) experienced CSF-related complications requiring further surgical intervention. The most recent audiometric outcomes demonstrated that 15 patients (78.9%) showed improvement in their hearing ability: 5 with sound/speech awareness, 5 able to discriminate among speech and environmental sounds, and 5 able to understand common phrases/conversation without lip reading. Nine patients (47.4%) are in a school for the deaf and 7 (36.8%) are in a mainstream school with support.

CONCLUSIONS

The authors’ surgical experience with a multidisciplinary team demonstrates that the retrosigmoid approach for ABI placement in children with inner ear pathologies and severe sensorineural hearing loss is a safe and effective treatment modality. Audiometric outcome data showed that nearly 79% of these patients had an improvement in their environmental and speech awareness. Further multicenter collaborations are necessary to improve these outcomes and potentially standardize/enhance electrode placement.

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Editorial. Occult dysraphism, spinopelvic angles, and machine learning: applying new tools to complex unsolved problems in pediatric neurosurgery

Robert J. Bollo and Sandi K. Lam

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Pediatric thoracic outlet syndrome: a systematic review and meta-analysis

Anthony Price, Nathan Fredricks, Nina Truong, and Robert Y. North

OBJECTIVE

Thoracic outlet syndrome (TOS) is a complex disorder affecting the neurovascular structures of the upper extremity as they traverse from the neck and thorax to the upper extremity. This systematic review and meta-analysis focuses on pediatric TOS, offering insights into its clinical presentation, etiology, treatment modalities, and outcomes in contrast to those reported in adult TOS.

METHODS

A comprehensive search for pediatric TOS in the PubMed database using PRISMA guidelines identified 6 relevant studies published between 2008 and 2022. In total, 227 pediatric TOS cases in 216 patients were analyzed. Data categories explored for TOS in pediatric patients included study design, number of patients included, mean age and sex of patients, TOS type, laterality, bony abnormalities, time to surgery, symptoms, treatment modalities, initial surgical technique, surgical complications, percent lost to follow-up, mean follow-up period, and treatment outcome.

RESULTS

The results from the 6 studies of 216 patients show a distinct pattern in pediatric TOS, with a 1.84:1 female-to-male ratio, a mean age of 15.49 years, and a lower prevalence of neurogenic TOS (75%, 95% CI 0.41–0.93; I2 = 86%, p < 0.01) compared with the prevailing literature on adults (87.5%–99%). Venous and arterial TOS accounted for a higher proportion of cases in pediatric patients than in adults, challenging the traditional adult-oriented perspective. Right-sided presentations were more common, reflecting right-arm dominance in most individuals. Additionally, bony abnormalities were more common in adults (30%) than in children (10.65%). Treatments involved mixed methods, predominantly using combinations of muscle resection (95.26%), neurolysis (78.02%), and bone resection (72.41%). Patients had high rates of symptom improvement (89%, 95% CI 0.67–0.97; I2 = 85%, p < 0.01) following surgery, with improvement of symptoms ranging from slight to complete relief. Complications were infrequent (5.66%), and most patients reported positive outcomes. The limitations of this analysis include subjective diagnostic and reporting criteria for TOS given its broad range of presentations.

CONCLUSIONS

This systematic review and meta-analysis brings to light the distinctive characteristics of pediatric TOS and underscores the importance of recognizing these differences to ensure accurate diagnosis and effective treatment in this patient population. Further research is needed to understand the predictive value of conservative treatments, especially in pediatric TOS cases.

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Use of a machine learning algorithm with a focus on spinopelvic parameters to predict development of symptomatic tethered cord after initial untethering surgery

Maria A. Punchak, Kamila M. Bond, Connor A. Wathen, Madison L. Hollawell, Chao Zhao, Christina Sarris, Tracy M. Flanders, Peter J. Madsen, Alexander M. Tucker, and Gregory G. Heuer

OBJECTIVE

Among patients with a history of prior lipomyelomeningocele repair, an association between increased lumbosacral angle (LSA) and cord retethering has been described. The authors sought to build a predictive algorithm to determine which complex tethered cord patients will develop the symptoms of spinal cord retethering after initial surgical repair with a focus on spinopelvic parameters.

METHODS

An electronic medical record database was reviewed to identify patients with complex tethered cord (e.g., lipomyelomeningocele, lipomyeloschisis, myelocystocele) who underwent detethering before 12 months of age between January 1, 2008, and June 30, 2022. Descriptive statistics were used to characterize the patient population. The Caret package in R was used to develop a machine learning model that predicted symptom development by using spinopelvic parameters.

RESULTS

A total of 72 patients were identified (28/72 [38.9%] were male). The most commonly observed dysraphism was lipomyelomeningocele (41/72 [56.9%]). The mean ± SD age at index MRI was 2.1 ± 2.2 months, at which time 87.5% of patients (63/72) were asymptomatic. The mean ± SD lumbar lordosis at the time of index MRI was 23.8° ± 11.1°, LSA was 36.5° ± 12.3°, sacral inclination was 30.4° ± 11.3°, and sacral slope was 23.0° ± 10.5°. Overall, 39.6% (25/63) of previously asymptomatic patients developed new symptoms during the mean ± SD follow-up period of 44.9 ± 47.2 months. In the recursive partitioning model, patients whose LSA increased at a rate ≥ 5.84°/year remained asymptomatic, whereas those with slower rates of LSA change experienced neurological decline (sensitivity 77.5%, specificity 84.9%, positive predictive value 88.9%, and negative predictive value 70.9%).

CONCLUSIONS

This is the first study to build a machine learning algorithm to predict symptom development of spinal cord retethering after initial surgical repair. The authors found that, after initial surgery, patients who demonstrate a slower rate of LSA change per year may be at risk of developing neurological symptoms.

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Both ends of the scalpel: a child’s journey from "Nemo fin" to neurosurgeon

Anthony Price and Robert North

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Multicenter comparison of Chiari malformation type I presentation in children versus adults

Armin Mortazavi, Neil D. Almeida, Katherine Hofmann, Laurence Davidson, Juliana Rotter, Tiffany N. Phan, Deki Tsering, Christina Maxwell, Jehshua Karunakaran, Erol Veznedaroglu, Anthony J. Caputy, John D. Heiss, Faheem A. Sandhu, John S. Myseros, Chima Oluigbo, Suresh N. Magge, Donald C. Shields, Michael K. Rosner, Grégoire P. Chatain, and Robert F. Keating

OBJECTIVE

Treatment for Chiari malformation type I (CM-I) often includes surgical intervention in both pediatric and adult patients. The authors sought to investigate fundamental differences between these populations by analyzing data from pediatric and adult patients who required CM-I decompression.

METHODS

To better understand the presentation and surgical outcomes of both groups of patients, retrospective data from 170 adults and 153 pediatric patients (2000–2019) at six institutions were analyzed.

RESULTS

The adult CM-I patient population requiring surgical intervention had a greater proportion of female patients than the pediatric population (p < 0.0001). Radiographic findings at initial clinical presentation showed a significantly greater incidence of syringomyelia (p < 0.0001) and scoliosis (p < 0.0001) in pediatric patients compared with adult patients with CM-I. However, presenting signs and symptoms such as headaches (p < 0.0001), ocular findings (p = 0.0147), and bulbar symptoms (p = 0.0057) were more common in the adult group. After suboccipital decompression procedures, 94.4% of pediatric patients reported symptomatic relief compared with 75% of adults with CM-I (p < 0.0001).

CONCLUSIONS

Here, the authors present the first retrospective evaluation comparing adult and pediatric patients who underwent CM-I decompression. Their analysis reveals that pediatric and adult patients significantly differ in terms of demographics, radiographic findings, presentation of symptoms, surgical indications, and outcomes. These findings may indicate different clinical conditions or a distinct progression of the natural history of this complex disease process within each population, which will require prospective studies to better elucidate.

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Near-infrared spectroscopy monitoring in severe pediatric abusive head trauma

Shih-Shan Lang, Nankee K. Kumar, Raphia Rahman, Alexander Tucker, Tracy M. Flanders, Gregory G. Heuer, Phillip B. Storm, Chao Zhao, and Jimmy W. Huh

OBJECTIVE

Abusive head trauma (AHT) is one of the most devastating forms of pediatric traumatic brain injury (TBI). It commonly presents with seizures, which may contribute to poor neurological outcome following trauma. Noninvasive near-infrared spectroscopy (NIRS) neuromonitoring may provide information on cerebral oxygenation and perfusion. In this study, the authors evaluated whether NIRS regional cerebral oxygen saturation (rSO2) values were associated with seizure activity confirmed by electroencephalography (EEG) and whether NIRS neuromonitoring could aid in seizure detection in patients with severe AHT.

METHODS

The authors retrospectively analyzed pediatric patients aged ≤ 18 years who were admitted to a quaternary urban pediatric hospital from 2016 to 2022 with severe AHT, who received NIRS and EEG monitoring during their hospital course. They evaluated clinical presentation and hospital course, including imaging findings, EEG findings, and NIRS rSO2 values.

RESULTS

Nineteen patients with severe AHT were monitored with both EEG and NIRS. The median age was 3.4 months, and 14 patients experienced seizures confirmed by EEG. On average, rSO2 values before, during, and after seizure did not differ significantly. However, within individual patients, bilateral regional NIRS rSO2 (bilateral forehead region) was seen to rise in the hour preceding seizure activity and during periods of frequent seizure activity, confirmed by EEG in the bilateral frontal-midline brain regions.

CONCLUSIONS

To the best of the authors’ knowledge, this is the largest study to analyze NIRS and seizures confirmed by EEG in the severe AHT population. The relationship between NIRS values and seizures in this series of pediatric patients with severe AHT suggests that, overall, regional NIRS cannot predict early seizures. However, increased cerebral oxygenation preceding seizure activity and during seizure activity may be detected by regional NIRS in certain patients with local seizure activity. Future studies with larger sample sizes may help elucidate the relationship between seizures and cerebral oxygenation in different regions in severe pediatric AHT.