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Demographic and social determinants of the interval from symptom onset to diagnosis of pediatric brain tumors: a study of the Connecticut Tumor Registry.
IF 2.1 3区 医学
Journal of neurosurgery. Pediatrics Pub Date : 2025-03-28 DOI: 10.3171/2024.12.PEDS24394
William A Lambert, Marcus Alexander, Jackson Ayers, Amirul Anuar, Katrina R Swett, Lou Gonsalves, Evan Cantor, Jonathan E Martin, Markus J Bookland, David S Hersh
{"title":"Demographic and social determinants of the interval from symptom onset to diagnosis of pediatric brain tumors: a study of the Connecticut Tumor Registry.","authors":"William A Lambert, Marcus Alexander, Jackson Ayers, Amirul Anuar, Katrina R Swett, Lou Gonsalves, Evan Cantor, Jonathan E Martin, Markus J Bookland, David S Hersh","doi":"10.3171/2024.12.PEDS24394","DOIUrl":"https://doi.org/10.3171/2024.12.PEDS24394","url":null,"abstract":"<p><strong>Objective: </strong>Pediatric patients frequently experience long intervals between the initial onset of symptoms and the diagnosis of a brain tumor. Understanding the determinants of the prediagnostic symptomatic interval (PSI) has the potential to facilitate earlier initiation of treatment. Here, the authors studied the impact of demographic factors and social determinants of health on the PSI among pediatric brain tumor patients.</p><p><strong>Methods: </strong>All pediatric patients with brain tumors included in the Connecticut Department of Public Health's Tumor Registry from 2004 to 2018 were reviewed. Demographic, clinical, and outcomes data were collected. The primary outcome was the log transformation of the PSI (ln(PSI)), which was modeled as a continuous variable. Statistical analyses included multiple linear regression models with stepwise variable selection. The Akaike Information Criterion (AIC) was used to indicate model fit, and bootstrapping was performed to validate the findings.</p><p><strong>Results: </strong>Overall, 153 patients met the inclusion criteria with a median (IQR) PSI of 30 (14-60) days. Of the tumors, 91 (59.5%) were classified as malignant and 62 (40.5%) as benign. The multivariable linear regression analysis of ln(PSI) demonstrated that longer PSI was positively associated with older age (β = 0.06, p = 0.004) and was negatively associated with Black or African American race (β = -0.64, p = 0.023), the presence of hydrocephalus on presentation (β = -0.79, p = 0.011), fatigue (β = -0.61, p = 0.047) or hemiparesis (β = -0.82, p = 0.041) as presenting symptoms, and malignant tumor behavior (β = -0.45, p = 0.042). The multiple linear regression model was statistically significant overall (F(7,145) = 5.42, p < 0.001), explaining approximately 21% of the variance ln(PSI), and bootstrapping with 1000 resamples confirmed the reliability of the regression analysis.</p><p><strong>Conclusions: </strong>Demographic factors, patterns of presentation, and tumor biology play a role in the interval between symptom onset and the diagnosis of pediatric brain tumors, with potential implications for quality of care and survival. Although this study did not identify a significant association between nondemographic social determinants of health and the PSI, additional studies are needed with more granular measures of economic stability, healthcare access and quality, education access and quality, neighborhood and built environment, and social and community context. Future efforts should target patient populations at increased risk of a prolonged PSI.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":" ","pages":"1-9"},"PeriodicalIF":2.1,"publicationDate":"2025-03-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143735934","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Overlap of genetic factors among craniosynostosis and autism spectrum disorder: the presence of autistic cases without craniosynostosis in carriers of shared variants. 颅骨发育不良与自闭症谱系障碍之间遗传因素的重叠:在共享变异体的携带者中存在没有颅骨发育不良的自闭症病例。
IF 2.1 3区 医学
Journal of neurosurgery. Pediatrics Pub Date : 2025-03-28 DOI: 10.3171/2024.12.PEDS24296
Shinji Ijichi, Naomi Ijichi, Mayuko Sakuma, Daisuke Yokoyama, Tsuneari Hayashi, Tomoya Hirota, Bryan H King
{"title":"Overlap of genetic factors among craniosynostosis and autism spectrum disorder: the presence of autistic cases without craniosynostosis in carriers of shared variants.","authors":"Shinji Ijichi, Naomi Ijichi, Mayuko Sakuma, Daisuke Yokoyama, Tsuneari Hayashi, Tomoya Hirota, Bryan H King","doi":"10.3171/2024.12.PEDS24296","DOIUrl":"https://doi.org/10.3171/2024.12.PEDS24296","url":null,"abstract":"<p><strong>Objective: </strong>A significant overlap of the risk genes respectively listed for craniosynostosis and autism was recently demonstrated in each case series. The purposes of this article were to review the clinical manifestations systemically in previously reported individual cases with the shared variants and to confirm the pleiotropic associations between genotype and phenotype.</p><p><strong>Methods: </strong>The risk genes or loci were retrieved from the PubMed database (last searched March 2024) as genetic factors for which genotype-phenotype associations were reported in at least 2 different individuals. Clinical details were reviewed in cases with shared genetic factors between craniosynostosis and autism spectrum disorder. In the genotype-phenotype association of syndromic craniosynostosis genes, both syndromic involvement of autistic traits and nonsyndromic autism cases with nonsyndromic variants in the flanking regions of the syndromic mutations were included.</p><p><strong>Results: </strong>Nonsyndromic craniosynostosis and autism are both underpinned by complex polygenic networks that include multiple risk genes or loci, and a considerable part of the networks might overlap. Although nonsyndromic mild trigonocephaly and autism might be closely associated in carriers of the shared variants, the presence of cases with autism without mild trigonocephaly was confirmed in a PJA1 variant case. As one of the characteristics of epistasis, ethnic specificity has been demonstrated in the interactions between SMAD6 and BMP2 variants. The presence of autism cases without craniosynostosis on a shared genetic basis was also confirmed in syndromic craniosynostosis risk genes and loci, suggesting that the causal relationship from craniosynostosis to autism is unnecessary for the development of autism spectrum disorder.</p><p><strong>Conclusions: </strong>Regarding the 3 possibilities of the association between craniosynostosis and neurodevelopmental comorbidities proposed by Kapp-Simon's group, only the independent pathway theory can explain the genotype-phenotype findings. There is no causal relationship between craniosynostosis and autism spectrum disorder in the independent pathway theory. Because the mechanical hindrance of brain expansion from poor compliance of the affected cranial vault is less likely in mild craniosynostosis cases, surgical indication of cranioplasty for mild trigonocephaly should never be confused with that of overt craniosynostosis. If there is no cosmetic indication for the metopic ridge in mild trigonocephaly cases with developmental comorbidities, there is no reason for surgical intervention.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":" ","pages":"1-10"},"PeriodicalIF":2.1,"publicationDate":"2025-03-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143735948","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Healthcare resource utilization for the management of neonatal head shape deformities: a propensity-matched analysis of AI-assisted and conventional approaches.
IF 2.1 3区 医学
Journal of neurosurgery. Pediatrics Pub Date : 2025-03-28 DOI: 10.3171/2024.12.PEDS24429
Jimin Shin, Gabrielle Caron, Petronella Stoltz, Jonathan E Martin, David S Hersh, Markus J Bookland
{"title":"Healthcare resource utilization for the management of neonatal head shape deformities: a propensity-matched analysis of AI-assisted and conventional approaches.","authors":"Jimin Shin, Gabrielle Caron, Petronella Stoltz, Jonathan E Martin, David S Hersh, Markus J Bookland","doi":"10.3171/2024.12.PEDS24429","DOIUrl":"https://doi.org/10.3171/2024.12.PEDS24429","url":null,"abstract":"<p><strong>Objective: </strong>Overuse of radiography studies and underuse of conservative therapies for cranial deformities in neonates is a known inefficiency in pediatric craniofacial healthcare. This study sought to establish whether the introduction of artificial intelligence (AI)-generated craniometrics and craniometric interpretations into craniofacial clinical workflow improved resource utilization patterns in the initial evaluation and management of neonatal cranial deformities.</p><p><strong>Methods: </strong>A retrospective chart review of pediatric patients referred for head shape concerns between January 2019 and June 2023 was conducted. Patient demographics, final encounter diagnosis, review of an AI analysis, and provider orders were documented. Patients were divided based on whether an AI cranial deformity analysis was documented as reviewed during the index evaluation, then both groups were propensity matched. Rates of index-encounter radiology studies, physical therapy (PT), orthotic therapy, and craniofacial specialist follow-up evaluations were compared using logistic regression and ANOVA analyses.</p><p><strong>Results: </strong>One thousand patient charts were reviewed (663 conventional encounters, 337 AI-assisted encounters). One-to-one propensity matching was performed between these groups. AI models were significantly more likely to be reviewed during telemedicine encounters and advanced practice provider (APP) visits (54.8% telemedicine vs 11.4% in-person, p < 0.0001; 12.3% physician vs 44.4% APP, p < 0.0001). All AI diagnoses of craniosynostosis versus benign deformities were congruent with final diagnoses. AI model review was associated with a significant increase in the use of orthotic therapies for neonatal cranial deformities (31.5% vs 38.6%, p = 0.0132) but not PT or specialist follow-up evaluations. Radiology ordering rates did not correlate with AI-interpreted data review.</p><p><strong>Conclusions: </strong>As neurosurgeons and pediatricians continue to work to limit neonatal radiation exposure and contain healthcare costs, AI-assisted clinical care could be a cheap and easily scalable diagnostic adjunct for reducing reliance on radiography and encouraging adherence to established clinical guidelines. In practice, however, providers appear to default to preexisting diagnostic biases and underweight AI-generated data and interpretations, ultimately negating any potential advantages offered by AI. AI engineers and specialty leadership should prioritize provider education and user interface optimization to improve future adoption of validated AI diagnostic tools.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":" ","pages":"1-9"},"PeriodicalIF":2.1,"publicationDate":"2025-03-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143735937","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Lower prefrontal blood flow associated with intraindividual weakness in successive processing: a neurocognitive study of pediatric moyamoya disease.
IF 2.1 3区 医学
Journal of neurosurgery. Pediatrics Pub Date : 2025-03-28 DOI: 10.3171/2024.11.PEDS24495
Yusuke Kusano, Takeshi Funaki, Keita Ueda, Tsukasa Ueno, Kanade Tanaka, Noyuri Nishida, Ami Tabata, Yasutaka Fushimi, Katsuhiko Mitsumoto, Takayuki Kikuchi, Ryosuke Ikeguchi, Nan Liang, Hiroyuki Inadomi, Susumu Miyamoto, Yoshiki Arakawa
{"title":"Lower prefrontal blood flow associated with intraindividual weakness in successive processing: a neurocognitive study of pediatric moyamoya disease.","authors":"Yusuke Kusano, Takeshi Funaki, Keita Ueda, Tsukasa Ueno, Kanade Tanaka, Noyuri Nishida, Ami Tabata, Yasutaka Fushimi, Katsuhiko Mitsumoto, Takayuki Kikuchi, Ryosuke Ikeguchi, Nan Liang, Hiroyuki Inadomi, Susumu Miyamoto, Yoshiki Arakawa","doi":"10.3171/2024.11.PEDS24495","DOIUrl":"https://doi.org/10.3171/2024.11.PEDS24495","url":null,"abstract":"<p><strong>Objective: </strong>Selective intraindividual weakness in successive processing, which is a unique verbal working memory scale included in the Das-Naglieri Cognitive Assessment System (CAS), is considered one of the intrinsic neurocognitive characteristics in pediatric moyamoya disease (MMD). The aim of the study was to elucidate the association between cerebral blood flow (CBF) and weakness in successive processing, and to identify regions related to the weakness.</p><p><strong>Methods: </strong>The present cross-sectional study included children who had been diagnosed with MMD and were assessed using neuropsychological tests before surgery between June 2016 and December 2023. According to the CAS manual, intraindividual difference was calculated by subtracting the mean of the 4 standard scores from each standard score. Each patient was classified as either manifesting \"intraindividual weakness in successive processing\" (intraindividual difference of successive processing < 0) or not (intraindividual difference of successive processing ≥ 0), and CBF acquired with resting-state SPECT was compared between groups. Three-dimensional stereotactic surface projection (3D-SSP) was also used for topographical comparison of CBF.</p><p><strong>Results: </strong>Of 51 children (mean age ± SD at admission 8.0 ± 2.6 years) who underwent preoperative neuropsychological tests, 43 were included in the CBF analysis. Both standard scores and intraindividual difference of the CAS significantly varied across 4 domains (p = 0.006 and p < 0.001, respectively), and those of successive processing were the lowest. Of the children analyzed, 35 (68.7%) were classified as having intraindividual weakness in successive processing. Multiple logistic regression analysis revealed the severest ischemic grade was significantly associated with intraindividual weakness in successive processing (OR 5.49 [95% CI 1.12-27.06]). Three-dimensional SSP analysis demonstrated a significant CBF decrease in the left dorsolateral and medial prefrontal cortexes in the children showing intraindividual weakness in successive processing compared with those who did not.</p><p><strong>Conclusions: </strong>Intraindividual weakness in successive processing typical of MMD might be associated with reduced CBF in the dorsolateral and medial prefrontal cortex, predominantly in the left hemisphere. Further studies in this area could contribute to the improvement of long-term social outcomes for patients with MMD.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":" ","pages":"1-10"},"PeriodicalIF":2.1,"publicationDate":"2025-03-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143735945","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
The risks of omitting routine postoperative imaging after insertion of new ventricular shunts in children: a 10-year retrospective cohort study.
IF 2.1 3区 医学
Journal of neurosurgery. Pediatrics Pub Date : 2025-03-28 DOI: 10.3171/2024.12.PEDS24496
Michael J Stuart, Annabelle M Harbison, Timothy Ruder, Joshua Hackney, Norman Ma, Robert A J Campbell, Amelia J Jardim, Liam G Coulthard
{"title":"The risks of omitting routine postoperative imaging after insertion of new ventricular shunts in children: a 10-year retrospective cohort study.","authors":"Michael J Stuart, Annabelle M Harbison, Timothy Ruder, Joshua Hackney, Norman Ma, Robert A J Campbell, Amelia J Jardim, Liam G Coulthard","doi":"10.3171/2024.12.PEDS24496","DOIUrl":"https://doi.org/10.3171/2024.12.PEDS24496","url":null,"abstract":"<p><strong>Objective: </strong>Ventriculoperitoneal shunt insertion is the primary treatment for pediatric hydrocephalus and is one of the most common procedures performed in pediatric neurosurgery. However, the value of routine postoperative imaging remains controversial. Although some evidence suggests that routine postoperative imaging provides a low yield after shunt revision, its role following initial shunt insertion in children has not been reported. Additionally, the role of routine postoperative imaging as a baseline study in altering the rate of longer-term shunt revision rates remains unexplored.</p><p><strong>Methods: </strong>A retrospective review was conducted using a prospectively maintained surgical database, encompassing 10 years of consecutive cases from a quaternary pediatric neurosurgical center serving a population of 5.3 million. Statewide electronic medical records including imaging and mortality data were reviewed. Only cases involving the insertion of a new ventricular shunt system were included. Based on longstanding physician preferences, approximately half the neurosurgeons in the department did not routinely perform postoperative imaging during the study period. The primary outcomes were shunt-related mortality during the study period, shunt revision or mortality within 7 days of surgery, or hospital discharge, with time to first revision as a secondary outcome.</p><p><strong>Results: </strong>During the study period 1485 shunt procedures were performed, of which 427 involved the placement of a new ventriculoperitoneal shunt system. The mean age of the patients was 5.2 years (range premature to 18 years). A total of 206/427 (48%) underwent routine postoperative imaging with MR or CT, and 153/427 (36%) underwent routine postoperative shunt series radiographs. During the study period, 2 abdominal radiographs identified preperitoneal placement; however, in both cases a symptomatic subcutaneous fluid collection was present (2/427, 0.5%). Postoperative CT or MR studies identified 1 case of extraventricular catheter placement and 5 cases of a kink adjacent to the valve (6/427, 1.4%), of which 4 cases were detected on routine imaging (4/427, 0.9%). No significant differences were detected between patients with or without imaging in 7-day postoperative (14 [7%] vs 8 [4%], p = 0.25) or 7-day postdischarge (2 [1%] vs 1 [< 1%], p = 0.96) rates of noninfective shunt revision, shunt-related mortality (4 [2%] vs 5 [2%], respectively, p = 0.8) or Kaplan-Meier long-term shunt survival curves (all p > 0.05).</p><p><strong>Conclusions: </strong>Routine postoperative imaging after pediatric shunt insertion rarely identifies clinically significant issues, and consideration could be given to its omission.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":" ","pages":"1-8"},"PeriodicalIF":2.1,"publicationDate":"2025-03-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143735951","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Does interfacility transfer affect outcomes in myelomeningocele repair? A National Surgical Quality Improvement Program Pediatric analysis.
IF 2.1 3区 医学
Journal of neurosurgery. Pediatrics Pub Date : 2025-03-28 DOI: 10.3171/2025.1.PEDS24400
Sara C Chaker, Andrew J James, Lauren E Sullivan, Mariam Saad, Michael S Golinko, Christopher M Bonfield, J Michael Newton, Stephane A Braun, Kelly A Bennett, John C Wellons, Matthew E Pontell
{"title":"Does interfacility transfer affect outcomes in myelomeningocele repair? A National Surgical Quality Improvement Program Pediatric analysis.","authors":"Sara C Chaker, Andrew J James, Lauren E Sullivan, Mariam Saad, Michael S Golinko, Christopher M Bonfield, J Michael Newton, Stephane A Braun, Kelly A Bennett, John C Wellons, Matthew E Pontell","doi":"10.3171/2025.1.PEDS24400","DOIUrl":"https://doi.org/10.3171/2025.1.PEDS24400","url":null,"abstract":"<p><strong>Objective: </strong>Myelomeningocele (MMC) remains one of the most common birth deformities, occurring in every 0.5 to 1 in 1000 pregnancies in the United States. Neonates born with MMC may require transfer to specialized institutions for surgical repair. This study aimed to investigate the impact of interfacility transfer on neonates undergoing MMC repair.</p><p><strong>Methods: </strong>All MMC repair cases from 2015 to 2021 were extracted from the National Surgical Quality Improvement Program Pediatric database. Transferred and nontransferred cohorts were compared for associations between transfer status and postoperative complications. Stepwise regression was completed to identify predictors of adverse outcomes.</p><p><strong>Results: </strong>From 2015 to 2021, 1672 MMC patients were identified, 753 of whom were transferred from an outside facility. Transferred patients were significantly more likely to be born vaginally (27.2% vs 18.9%, p = 0.005), premature (20.6% vs 15.3%, p = 0.033), and with low birth weight (16.7% vs 12.6%, p < 0.001). A greater number of transferred patients were classified as American Society of Anesthesiologists class III or higher (78.6% vs 73.2%, p = 0.009). Transferred patients were more frequently classified as an emergency (30.0% vs 19.2%, p < 0.001) or urgent (37.3% vs 24.9%, p < 0.001) case. Postoperatively, transferred patients were significantly more likely to experience cardiac arrest (0.9% vs 0.2%, p = 0.026), require supplemental oxygen at discharge (9.6% vs 4.8%, p < 0.001), have higher 30-day all-cause mortality (1.7% vs 0.5%, p = 0.020), and die more than 1 week after surgery (13 vs 3 deaths, p = 0.016).</p><p><strong>Conclusions: </strong>Patients transferred in the neonatal period may experience worse postoperative outcomes after MMC repair. These data support a recommendation for patients prenatally diagnosed with MMC to be delivered at centers equipped for pre- and postnatal management, when possible.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":" ","pages":"1-8"},"PeriodicalIF":2.1,"publicationDate":"2025-03-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143735936","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Long-term outcomes of intraventricular baclofen therapy for medically refractory generalized secondary dystonia.
IF 2.1 3区 医学
Journal of neurosurgery. Pediatrics Pub Date : 2025-03-21 DOI: 10.3171/2024.12.PEDS24418
Joyce Koueik, Tenzing Lhamo, Emily Meyer, Susan Hubanks, Brandon G Rocque, A Leland Albright
{"title":"Long-term outcomes of intraventricular baclofen therapy for medically refractory generalized secondary dystonia.","authors":"Joyce Koueik, Tenzing Lhamo, Emily Meyer, Susan Hubanks, Brandon G Rocque, A Leland Albright","doi":"10.3171/2024.12.PEDS24418","DOIUrl":"https://doi.org/10.3171/2024.12.PEDS24418","url":null,"abstract":"<p><strong>Objective: </strong>Intrathecal baclofen (ITB) is commonly used to treat secondary generalized dystonia. Intraventricular baclofen (IVB) has been shown to be a safe alternative treatment with low complications. The objective of this study was to report the long-term effects of IVB.</p><p><strong>Methods: </strong>This retrospective analysis included patients who underwent IVB therapy from April 2005 to June 2024. The decision to use IVB and the surgical technique have been previously described. Data collection included sex, race, etiology of dystonia, Gross Motor Functional Classification System scores, Barry-Albright Dystonia Scale (BADS) scores, Ashworth Scale scores, medical and surgical management of dystonia, follow-up duration, and complications. Patients whose IVB pump was removed within 1 year and those with less than 1 year of follow-up were excluded.</p><p><strong>Results: </strong>Thirty-six patients with IVB were identified, and 27 patients (median age 12.9 years) were ultimately included. The most common cause of secondary dystonia was cerebral palsy in 21 patients (78%), followed by metabolic and neurodegenerative disorders in 4 (14%), infection in 1 (4%), and severe traumatic brain injury (TBI) in 1 (4%). The follow-up duration ranged from 1.2 to 16.7 years, with a median of 7.7 years and mean of 8.4 years. The baclofen dosage was twice as high in patients with metabolic and neurodegenerative disorders compared with those with cerebral palsy, TBI, and infection, with median dosages of 1455 μg/day and 725 μg/day, respectively. Both posttreatment BADS and Ashworth Scale scores showed statistically significant improvement. Complications included infection that necessitated pump removal in 1 patient (4%), wound dehiscence that was treated with wound revision and antibiotics in 1 patient (4%), and hydrocephalus that required CSF diversion in 4 patients (15%). In 2 patients, IVB therapy was ineffective at controlling dystonia and, thus, was discontinued after 1.3 and 2.7 years. One patient asked for the pump to be removed after 1.2 years due to \"does not like the physical pump itself.\" Four patients (15%) developed hydrocephalus, which required CSF diversion via a ventriculoperitoneal shunt in 3 patients and a lumboperitoneal shunt in 1 patient, with no further shunt or IVB complications.</p><p><strong>Conclusions: </strong>IVB is well tolerated in patients with generalized secondary dystonia refractory to conventional medical therapy, ITB, and deep brain stimulation. Positive long-term outcomes were reported in this cohort, with significant improvement in dystonia and overall complication rates similar to those reported with ITB.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":" ","pages":"1-6"},"PeriodicalIF":2.1,"publicationDate":"2025-03-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143673960","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Long-term parental satisfaction and cosmetic, ophthalmological, and cognitive outcomes after sagittal strip craniectomy with barrel stave osteotomies and occipital release.
IF 2.1 3区 医学
Journal of neurosurgery. Pediatrics Pub Date : 2025-03-21 DOI: 10.3171/2024.11.PEDS24263
Serife Maide Pinar, Tim Jonas Hallenberger, Maria Licci, Benito K Benitez, Anja Palmowski-Wolfe, Françoise Roulez, Valérie Oesch, Raphael Guzman, Jehuda Soleman
{"title":"Long-term parental satisfaction and cosmetic, ophthalmological, and cognitive outcomes after sagittal strip craniectomy with barrel stave osteotomies and occipital release.","authors":"Serife Maide Pinar, Tim Jonas Hallenberger, Maria Licci, Benito K Benitez, Anja Palmowski-Wolfe, Françoise Roulez, Valérie Oesch, Raphael Guzman, Jehuda Soleman","doi":"10.3171/2024.11.PEDS24263","DOIUrl":"https://doi.org/10.3171/2024.11.PEDS24263","url":null,"abstract":"<p><strong>Objective: </strong>Craniosynostosis is ideally treated within the 1st year of life to correct the physical deformity, achieve optimal aesthetic head shape, and facilitate normal neurocognitive development. While objective aesthetic assessments have been widely studied, limited reports on long-term parent-reported outcomes (PROs) exist. The authors herein report long-term follow-up after open midline strip craniectomy with barrel stave osteotomies (BSOs) and occipital release of nonsyndromic sagittal craniosynostosis (SC), including parental satisfaction; cosmetic, ophthalmological, and neurocognitive outcomes; and health-related quality of life (HRQOL).</p><p><strong>Methods: </strong>Patients younger than 12 months of age who underwent surgical reconstruction of isolated SC between March 2014 and March 2019 with a minimum follow-up period of 3 years were included in this study. The primary outcome was parental satisfaction assessed by PROs. Secondary outcomes were neurocognitive development using the Vineland Adaptive Behavior Scale (VABS), ophthalmological and cosmetic outcomes based on clinical examination, and HRQOL and 3D imaging of the patients.</p><p><strong>Results: </strong>Included were 25 patients (16% female) with a median age of 5.91 (IQR 4.97-6.55) months. Parental satisfaction was excellent, with 96% reporting satisfaction with the overall surgical outcome. Cosmetic outcomes assessed by the treating surgeons were considered good in 94.7% of the cases, while the cephalic index was within the mesocephalic range in 78.22% of the cases. The aesthetic perception of parents and surgeons was concordant in 89.5% of cases. Children of all age groups showed an age-appropriate adaptive level on the VABS (mean score 108.21 ± 9.60). The median HRQOL score was 87.5 (IQR 75-91.66), which was above the normal range. Ophthalmological examinations were unremarkable in all patients.</p><p><strong>Conclusions: </strong>These results suggest that open midline strip craniectomy with BSO and occipital release for isolated SC yields high levels of satisfaction among both parents and clinicians, along with HRQOL surpassing norms and no reported ophthalmological or developmental disability.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":" ","pages":"1-10"},"PeriodicalIF":2.1,"publicationDate":"2025-03-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143673963","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Racial disparities in hydrocephalus mortality and shunt revision: a study from the Hydrocephalus Clinical Research Network.
IF 2.1 3区 医学
Journal of neurosurgery. Pediatrics Pub Date : 2025-03-21 DOI: 10.3171/2024.12.PEDS24371
Brandon G Rocque, Hailey Jensen, Ron W Reeder, Curtis J Rozzelle, Abhaya V Kulkarni, Ian F Pollack, Michael M McDowell, Robert P Naftel, Eric M Jackson, William E Whitehead, Jonathan A Pindrik, Albert M Isaacs, Jennifer M Strahle, Patrick J McDonald, Mandeep S Tamber, Todd C Hankinson, Samuel R Browd, Jason S Hauptman, Mark D Krieger, Jason Chu, Jay Riva-Cambrin, David D Limbrick, Richard Holubkov, John R W Kestle, John C Wellons
{"title":"Racial disparities in hydrocephalus mortality and shunt revision: a study from the Hydrocephalus Clinical Research Network.","authors":"Brandon G Rocque, Hailey Jensen, Ron W Reeder, Curtis J Rozzelle, Abhaya V Kulkarni, Ian F Pollack, Michael M McDowell, Robert P Naftel, Eric M Jackson, William E Whitehead, Jonathan A Pindrik, Albert M Isaacs, Jennifer M Strahle, Patrick J McDonald, Mandeep S Tamber, Todd C Hankinson, Samuel R Browd, Jason S Hauptman, Mark D Krieger, Jason Chu, Jay Riva-Cambrin, David D Limbrick, Richard Holubkov, John R W Kestle, John C Wellons","doi":"10.3171/2024.12.PEDS24371","DOIUrl":"https://doi.org/10.3171/2024.12.PEDS24371","url":null,"abstract":"<p><strong>Objective: </strong>Several studies of administrative data have noted higher mortality rates for Black/African American children with shunted hydrocephalus. A longitudinal study of children with hydrocephalus secondary to myelomeningocele showed lower lifetime rates of shunt revision in minority children compared to White children, indicating a possible disparity in hydrocephalus treatment. The goal of this study is to identify racial and ethnic disparities in mortality or in shunt revision rates by using the Hydrocephalus Clinical Research Network (HCRN) hydrocephalus registry sample.</p><p><strong>Methods: </strong>The HCRN registry was queried for patients with shunted hydrocephalus for whom data on all lifetime hydrocephalus procedures were available. Patients with a primary shunt placement prior to 2023 were included, with follow-up extending through March 19, 2024. A Cox proportional hazards model was created to determine the effect of race and ethnicity on mortality while controlling for age at initial shunt placement, sex, hydrocephalus etiology, gestational age at birth, and presence of complex chronic conditions. Similarly, a proportional means model was used to evaluate association with lifetime number of shunt revision surgeries. The authors' hypothesis was that when controlling for other variables, minority children would have higher mortality and fewer shunt revision surgeries than White children.</p><p><strong>Results: </strong>A total of 5656 children were included in the analysis of mortality. There were 579 deaths. Race and ethnicity were independently associated with mortality, with Black (HR 1.32, 95% CI 1.05-1.65), other non-White (HR 1.39, 95% CI 1.03-1.86), and Hispanic (HR 1.50, 95% CI 1.22-1.84) children having a higher mortality rate than White children. In the analysis of 4081 children with shunts, Hispanic ethnicity was also independently associated with fewer total shunt revisions (HR 0.84, 95% CI 0.72-0.98).</p><p><strong>Conclusions: </strong>In children with hydrocephalus, when controlling for other factors, there is a higher mortality rate among Hispanic, Black, and other non-White children, and fewer shunt revisions among Hispanic children. These findings highlight important potential disparities in hydrocephalus treatment.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":" ","pages":"1-9"},"PeriodicalIF":2.1,"publicationDate":"2025-03-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143673872","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Efficacy of selective dorsal rhizotomy in the treatment of spasticity in children with cerebral palsy: a systematic review and meta-analysis.
IF 2.1 3区 医学
Journal of neurosurgery. Pediatrics Pub Date : 2025-03-21 DOI: 10.3171/2024.11.PEDS24398
Iris Otero-Luis, Arturo Martinez-Rodrigo, Iván Cavero-Redondo, Nerea Moreno-Herráiz, Felipe Araya-Quintanilla, Carla Geovanna Lever-Megina, Alicia Saz-Lara
{"title":"Efficacy of selective dorsal rhizotomy in the treatment of spasticity in children with cerebral palsy: a systematic review and meta-analysis.","authors":"Iris Otero-Luis, Arturo Martinez-Rodrigo, Iván Cavero-Redondo, Nerea Moreno-Herráiz, Felipe Araya-Quintanilla, Carla Geovanna Lever-Megina, Alicia Saz-Lara","doi":"10.3171/2024.11.PEDS24398","DOIUrl":"https://doi.org/10.3171/2024.11.PEDS24398","url":null,"abstract":"<p><strong>Objective: </strong>Spasticity is one of the most prevalent neurological disorders, affecting > 90% of patients with cerebral palsy (CP). Selective dorsal rhizotomy (SDR) is a permanent neurosurgical procedure indicated for the treatment of spasticity. This study aimed to analyze the efficacy of SDR in managing spasticity in children with CP and as well as the efficacy of this therapy over time after the intervention using different scales.</p><p><strong>Methods: </strong>A comprehensive systematic search was conducted across several databases, spanning from inception to November 5, 2023. Random-effects meta-analyses were used to calculate pooled mean differences and their corresponding confidence intervals to assess the efficacy of SDR using different scales.</p><p><strong>Results: </strong>All the included studies involved before-after single-arm studies, with the exception of four randomized clinical trials and one quasi-experimental study. The effect of SDR on spasticity, measured with the modified Ashworth scale (MAS) and the Gross Motor Function Measure (GMFM), was significantly improved in children with lower limb spasticity. The efficacy of the treatment was significant up to 12 months posttreatment.</p><p><strong>Conclusions: </strong>SDR has been demonstrated to be effective in treating children with CP. This efficacy is evidenced by notable improvements, as measured by both the MAS and GMFM. These improvements suggested enhanced mobility and overall quality of life.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":" ","pages":"1-10"},"PeriodicalIF":2.1,"publicationDate":"2025-03-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143673950","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
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