Journal of neurosurgery. Pediatrics最新文献

Objective: Brain tumors are a global problem, leading to higher cancer-related morbidity and mortality rates in children. Despite the progressive though slow advances in neuro-oncology care, research, and diagnostics in sub-Saharan Africa (SSA), the epidemiological landscape of pediatric brain tumors (PBTs) remains underestimated. This study aimed to systematically analyze the distribution of PBT types in SSA.

Methods: Ovid Medline, Global Index Medicus, African Journals Online, Google Scholar, and faculty of medicine libraries were searched for literature on PBTs in SSA published before October 29, 2022. A proportional meta-analysis was performed.

Results: Forty-nine studies, involving 2360 children, met the inclusion criteria for review; only 20 (40.82%) were included in the quantitative analysis. South Africa and Nigeria were the countries with the most abundant data. Glioma not otherwise specified (NOS) was the common PBT in the 4 SSA regions combined. However, medulloblastoma was more commonly reported in Southern SSA (p = 0.01) than in other regions. The prevalence and the overall pooled proportion of the 3 common PBTs was estimated at 46.27% and 0.41 (95% CI 0.32-0.50, 95% prediction interval [PI] 0.11-0.79), 25.34% and 0.18 (95% CI 0.14-0.21, 95% PI 0.06-0.40), and 12.67% and 0.12 (95% CI 0.09-0.15, 95% PI 0.04-0.29) for glioma NOS, medulloblastoma, and craniopharyngioma, respectively. Sample size moderated the estimated proportion of glioma NOS (p = 0.02). The highest proportion of craniopharyngiomas was in Western SSA, and medulloblastoma and glioma NOS in Central SSA.

Conclusions: These findings provide insight into the trends of PBT types and the proportion of the top 3 most common tumors across SSA. Although statistical conclusions are difficult due to the inconsistency in the data, the study identifies critical areas for policy development and collaborations that can facilitate improved outcomes in PBTs in SSA. More accurate epidemiological studies of these tumors are needed to better understand the burden of the disease and the geographic variation in their distribution, and to raise awareness in their subsequent management.

Objective: Craniovertebral junction (CVJ) abnormalities are common and well documented in mucopolysaccharidosis type I-Hurler syndrome (MPS IH), often causing severe spinal canal narrowing. However, the requirement for surgical decompression and/or fusion is uncommon. Although hematopoietic cell transplant (HCT) has been shown to prolong the lives of patients with MPS IH, its effect in halting or reversing musculoskeletal abnormalities is less clear. Unfortunately, there are currently no universal guidelines for imaging or indication for surgical interventions in these patients. The goal of this study was to track the progression of the CVJ anatomy in patients with MPS IH following HCT, and to examine radiographic features in patients who needed surgical intervention.

Methods: Patients with MPS IH treated at the University of Minnesota with allogeneic HCT between 2008 and 2020 were retrospectively reviewed. Patients who underwent CVJ surgery were identified with chart review. All MPS IH cervical scans were examined, and the odontoid retroflexion angle, clivoaxial angle (CXA), canal width, and Grabb-Oakes distance (pB-C2) were measured yearly for up to 7 years after HCT. Longitudinal models based on the measurements were made. An intraclass correlation coefficient was used to measure interrater reliability. Nine children without MPS IH were examined for control CVJ measurements.

Results: A total of 253 cervical spine MRI scans were reviewed in 54 patients with MPS IH. Only 4 (7.4%) patients in the study cohort required surgery. Three of them had posterior fossa and C1 decompression, and 1 had a C1-2 fusion. There was no statistically significant difference in the spinal parameters that were examined between surgery and nonsurgery groups. Among the measurements, canal width and CXA varied drastically in patients with different neck positions. Odontoid retroflexion angle and CXA tended to decrease with age. Canal width and pB-C2 tended to increase with age.

Conclusions: Based on the data, the authors observed an increase in canal width and pB-C2, whereas the CXA and odontoid retroflexion angle became more acute as the patients aged after HCT. The longitudinal models derived from these data mirrored the development in children without MPS IH. Spinal measurements obtained on MR images alone are not sufficient in identifying patients who require surgical intervention. Symptom monitoring and clinical examination, as well as pathological spinal cord changes on MRI, are more crucial in assessing the need for surgery than is obtaining serial imaging.

Objective: Craniosynostosis involves early closure of one or more sutures, which is known to limit normal cranium growth and interfere with normal brain development. Various surgical methods are used, ranging from minimally invasive strip craniectomy to more extensive whole-vault cranioplasty. This study aimed to evaluate neurocognitive outcomes 5 years after surgical treatment in children with craniosynostosis and to evaluate relevant clinical factors.

Methods: After exclusion of genetically confirmed syndromic craniosynostosis patients, a retrospective review was conducted on 112 nonsyndromic craniosynostosis patients who underwent surgical treatment and follow-up neurocognitive assessment. Ninety-seven patients underwent strip craniectomy with postoperative orthotic helmet therapy, and 15 received other surgical treatment: 4 with distraction osteotomy and 11 with craniofacial reconstruction. Neurocognitive assessment using the Korean Wechsler Preschool and Primary Scale of Intelligence, Fourth Edition (K-WPPSI-IV), was performed 5 years postoperatively. Clinical factors were assessed regarding neurocognitive outcomes.

Results: The mean age at surgery was significantly younger in the strip craniectomy group (strip craniectomy 4.6 months vs other surgical treatment 18.6 months, p < 0.01). Patients with 2 or more sutures involved were more likely to receive more extensive surgical treatment (16.5% in the strip craniectomy group vs 53.8% in the other group, p < 0.01). Four (3.5%) patients who showed evidence of increased intracranial pressure (ICP) also underwent more extensive surgical treatment. Multivariable linear regression revealed a significant correlation of age at neurocognitive testing (-3.18, 95% CI -5.95 to -0.40, p = 0.02), increased ICP (-34.73, 95% CI -51.04 to -18.41, p < 0.01), and the level of maternal education (6.11, 95% CI 1.01-11.20, p = 0.02) with the Full-Scale Intelligence Quotient (FSIQ). Age at surgery, involvement of 2 or more sutures, and type of operation demonstrated no correlation with FSIQ. Among the 97 patients who underwent strip craniectomy, the FSIQ ranged from mean ± SD 100.2 ± 10.2 (bicoronal) to 110.1 ± 12.7 (lambdoid), and there was no significant difference between the suture groups (p = 0.41). The 5 index scores were all within average ranges based on their age norms.

Conclusions: Age at neurocognitive assessment, increased ICP, and maternal education level showed significant correlations with the neurocognitive function of craniosynostosis patients. Although children with craniosynostosis exhibited favorable 5-year postoperative neurocognitive outcomes across various synostosis sutures, longer follow-up is needed to reveal the incidence of neurocognitive dysfunction in these patients.

Objective: Cerebrospinal fluid (CSF) shunt infections caused by gram-negative bacteria are difficult to treat given the limited treatment options and the emergence of carbapenem-resistant (CR) strains. This study aimed to evaluate the demographic and clinical characteristics of children with CSF shunt and external ventricular drain (EVD) infections caused by gram-negative bacteria, to identify the risk factors for acquiring CR CSF shunt infections, and to report on the clinical outcomes of these infections.

Methods: A retrospective cohort study was designed to evaluate pediatric patients with CSF shunt and EVD infections caused by gram-negative bacteria between January 2013 and February 2023.

Results: A total of 64 episodes in 50 patients were evaluated. There were 45 (70.3%) CSF shunt infections and 19 (29.7%) EVD infections. The median (range) ages were 1.4 years (9 months-17.5 years) for CSF shunt infection patients and 4.2 years (1 month-17 years) for EVD infection patients. The most common isolated gram-negative bacteria species in CSF shunt infections were Pseudomonas spp. (12, 26.7%), followed by Escherichia coli (11, 24.4%), Klebsiella pneumoniae (9, 20%), and Enterobacter cloacae (5, 11.1%). In EVD infections, the most common isolated gram-negative bacteria species were Acinetobacter spp. (6, 31.6%), followed by Pseudomonas spp. (4, 21.1%) and E. coli (3, 15.8%). The carbapenem resistance rate was 26.3% (n = 5) in EVD infections and 26.2% (n = 11) in CSF shunt infections. When risk factors for carbapenem resistance were evaluated for CSF shunt infections, prior carbapenem treatment and a prolonged hospital stay > 7 days were risk factors for the CR group (p = 0.032 and p = 0.042, respectively). In definitive treatment, colistin was statistically more commonly used in the CR group (p = 0.049). When outcomes were evaluated, the 30-day mortality rate (18.2% vs 0%) was higher in the CR group, without a significant difference (p = 0.064).

Conclusions: A prolonged hospital stay > 7 days and prior carbapenem exposure within 30 days were associated with CR shunt infections caused by gram-negative bacteria.

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.

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.

Objective: Posttraumatic headache (PTH) represents the most common acute and persistent symptom in children after concussion, yet there is no blood protein signature to stratify the risk of PTH after concussion to facilitate early intervention. This discovery study aimed to identify capillary blood protein markers, at emergency department (ED) presentation within 48 hours of concussion, to predict children at risk of persisting PTH at 2 weeks postinjury.

Methods: Capillary blood was collected using the Mitra Clamshell device from children aged 8-17 years who presented to the ED of the Royal Children's Hospital, Melbourne, Australia, within 48 hours of sustaining a concussion. Participants were followed up at 2 weeks postinjury to determine PTH status. PTH was defined per clinical guidelines as a new or worsened headache compared with preinjury. An untargeted proteomics analysis using data-independent acquisition (DIA) was performed. Principal component analysis and hierarchical clustering were used to reduce the dimensionality of the protein dataset.

Results: A total of 907 proteins were reproducibly identified from 82 children within 48 hours of concussion. The mean participant age was 12.78 years (SD 2.54 years, range 8-17 years); 70% of patients were male. Eighty percent met criteria for acute PTH in the ED, while one-third of participants with follow-up experienced PTH at 2 weeks postinjury (range 8-16 days). Hemoglobin subunit zeta (HBZ), cystatin B (CSTB), beta-ala-his dipeptidase (CNDP1), hemoglobin subunit gamma-1 (HBG1), and zyxin (ZYX) were weakly associated with PTH at 2 weeks postinjury based on up to a 7% increase in the PTH group despite nonsignificant Benjamini-Hochberg adjusted p values.

Conclusions: This discovery study determined that no capillary blood protein markers, measured at ED presentation within 48 hours of concussion, can predict children at risk of persisting PTH at 2 weeks postinjury. While HBZ, CSTB, CNDP1, HBG1, and ZYX were weakly associated with PTH at 2 weeks postinjury, there was no specific blood protein signature predictor of PTH in children after concussion. There is an urgent need to discover new blood biomarkers associated with PTH to facilitate risk stratification and improve clinical management of pediatric concussion.

Objective: The need for permanent CSF diversion is lower in patients who have undergone prenatal surgery for myelomeningocele (MMC) than in those who have undergone postnatal closure. Differences in brain development and head growth between treatment groups are not known, particularly for those who do not require surgical treatment for hydrocephalus. The objective of this study was to determine differences in head growth and to generate MMC-specific head circumference (HC) growth curves for patients who underwent either prenatal or postnatal surgery.

Methods: The authors retrospectively identified patients from St. Louis Children's Hospital who were treated for MMC between 2016 and 2021. HC data were obtained from birth until the most recent follow-up or hydrocephalus treatment. Nonlinear least-squares regression analysis was performed to fit the data into four models: two-term power, Gompertz, West ontogenetic, and Weibull. Subsequently, the curves were assessed for their utility in predicting hydrocephalus treatment.

Results: Sixty-one patients (29 females [47.5%], 25 [41%] underwent prenatal surgery, mean gestational age at birth 36.6 weeks) were included in the study. The Weibull model best fit the HC data (prenatal adjusted R2 = 0.95, postnatal adjusted R2 = 0.95), while the Gompertz model had the worst fit (prenatal adjusted R2 = 0.56, postnatal adjusted R2 = 0.39) across both cohorts. Prenatal MMC repair patients had significantly larger HC measurements than their postnatal repair counterparts. The 50th percentile of the Weibull curve was determined as a useful threshold for hydrocephalus treatment: children with HC measurements that crossed and remained above this threshold were significantly more likely to have hydrocephalus treatment regardless of time of MMC repair (prenatal relative risk [RR] 10.0 [95% CI 1.424-70.220], sensitivity 85.7% [95% CI 0.499-0.984], and specificity 82.4% [95% CI 0.600-0.948]; postnatal RR 4.750 [95% CI 1.341-16.822], sensitivity 90.5% [95% CI 0.728-0.980], and specificity 75.0% [95% CI 0.471-0.924]). The HC growth curves of the MMC patients treated prenatally were significantly larger than the WHO HC curves (p < 0.001).

Conclusions: The Weibull model was identified as the HC growth curve with the best fit for MMC patients and serves as a useful predictor of hydrocephalus treatment. For MMC patients with hydrocephalus, prenatal repair patients fit the model well but postnatal repair patients did not, potentially indicating different mechanisms of hydrocephalus development. Those treated prenatally had significantly larger HC measurements compared with both the general population and those treated postnatally. Further study is needed to understand the long-term cognitive outcomes and optimal management of clinically asymptomatic patients with large HC measurements who were treated prenatally for MMC.

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.