Journal of neurosurgery. Pediatrics最新文献

{"title":"Pediatric CSF diversion procedures for treatment of hydrocephalus during the COVID-19 pandemic.","authors":"Paige Lundy, Ariana Barkley, A K M Fazlur Rahman, Anastasia Arynchyna-Smith, Jessica Thrower, Addison Stewart, Klaudia Dziugan, Sandi Lam, Koko Hall, Jason Hauptman, Keshari Shrestha, Susan Staulcup, Todd C Hankinson, Benjamin Best, Irene Kim, Joan Yea, Carly Weber-Levine, Eric M Jackson, Christine Park, Daniel Sexton, Eric M Thompson, Anna L Slingerland, Joanna Papadakis, Katie Pricola Fehnel, Sarah Wisor-Martinez, David F Bauer, S Hassan A Akbari, Brandon G Rocque","doi":"10.3171/2024.7.PEDS24249","DOIUrl":"https://doi.org/10.3171/2024.7.PEDS24249","url":null,"abstract":"<p><strong>Objective: </strong>Because there is not a link between COVID-19 and pediatric hydrocephalus, the COVID-19 pandemic should not have altered the incidence of pediatric hydrocephalus or the rate of CSF diversion procedures or shunt failure. Therefore, hydrocephalus-related surgical volume should have remained constant. The goal of this study was to evaluate the rates of hydrocephalus surgeries in the COVID-19 era compared with the baseline pre-COVID-19 era.</p><p><strong>Methods: </strong>Ten institutions collected information about all hydrocephalus-related surgeries performed between March 2018 and February 2022. The period after March 1, 2020, was considered the COVID-19 era; the period prior to this date was considered the baseline pre-COVID era. Four COVID surge periods were defined using the New York Times COVID-19 database. Total case volumes were compared between the COVID era and baseline, both overall and for each surge period. Sex, race, ethnicity, insurance status, Area Deprivation Index (ADI), and rural-urban commuter area were collected for each surgery. Proportions of patients were then compared overall and for each surge based on these variables.</p><p><strong>Results: </strong>Of 8056 procedures, 54% were in male patients (n = 4375), 65% in White patients (n = 5247), 18% in Hispanic patients (n = 1423), and 54% in patients with public insurance (n = 4371). There was no change in the number of surgeries per site per month in the COVID era compared with baseline (16.7 vs 17.9, p = 0.113). However, there was a significant decrease in the first surge period (April 2020; 11.5 vs 17.7, p = 0.034). Male sex (p < 0.0039) and Black race (p < 0.001) were found to be associated with a significantly higher proportion of hydrocephalus procedures during the COVID-19 era. Some surge periods showed different proportions of privately insured patient and ADI levels. However, these relationships were inconsistent between surges.</p><p><strong>Conclusions: </strong>Overall average monthly case numbers were not significantly different between the pre-COVID and COVID eras. There was a significant decrease in hydrocephalus surgery during the first COVID surge. More hydrocephalus surgeries were performed in children of male sex and Black race proportionally during the COVID period overall, but not during individual surges.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":null,"pages":null},"PeriodicalIF":2.1,"publicationDate":"2024-10-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142406460","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}

{"title":"The impact of spheno-occipital synchondrosis exposure via extended endoscopic endonasal surgery on midface growth in pediatric patients.","authors":"Manish Beniwal, Hiroki Morisako, Tsuyoshi Sasaki, Masaki Ikegami, Atsufumi Nagahama, Yuta Tanoue, Hiroaki Sakamoto, Takeo Goto","doi":"10.3171/2024.7.PEDS24174","DOIUrl":"https://doi.org/10.3171/2024.7.PEDS24174","url":null,"abstract":"&lt;p&gt;&lt;strong&gt;Objective: &lt;/strong&gt;Pediatric extended endonasal procedures pose significant surgical challenges. Lesions from the suprasellar region to the lower clivus necessitate extensive exposure. This study examined whether drilling the spheno-occipital synchondrosis (SOS) to remove the posterior clinoid process and dorsum sellae (DS) for greater exposure affects pediatric midfacial growth.&lt;/p&gt;&lt;p&gt;&lt;strong&gt;Methods: &lt;/strong&gt;From 2014 to 2020, the authors performed endoscopic endonasal surgery (EES) in 14 patients aged 12 years or younger. The lesions consisted of 11 cases of craniopharyngioma, 1 pituitary neuroendocrine tumor, 1 Rathke's cleft cyst, and 1 Langerhans cell histiocytosis. In 8 of the 14 cases, an extended EES procedure was used by exposing the SOS to remove the posterior clinoid process and DS. Measurement of the central face was based on head MRI before and after surgery. Measuring points were the sellae-nasion (SN) plane, the foremost points of the anterior maxilla (point A), and the maximum concavity point of the mandibular symphysis (point B). The authors measured and evaluated the SNA angle (angle created by the SN plane and the NA [a line connecting point A and the nasion] plane), SNB angle (angle created by the SN plane and the NB [a line connecting point B and the nasion] plane), and the ANB angle (angle created by the NA plane and the NB plane). In addition, a comparison was made with 6 pediatric cases in which transcranial surgery was performed for craniopharyngiomas.&lt;/p&gt;&lt;p&gt;&lt;strong&gt;Results: &lt;/strong&gt;In the extended EES group, the average preoperative age was 7 years, and the average postoperative age was 12 years. Mean preoperative angles in this group were 84° (SNA), 80.9° (SNB), and 3.1° (ANB); mean postoperative angles were 83.5° (SNA), 83.9° (SNB), and -0.4° (ANB). In the standard EES group, the average preoperative age was 9 years, and the average postoperative age was 14.5 years. Average preoperative angles in the standard EES group were 83° (SNA), 80.3° (SNB), and 2.7° (ANB); average postoperative angles were 82.7° (SNA), 81° (SNB), and 1.6° (ANB). In the transcranial surgery group, the average preoperative age was 4.5 years, and the average postoperative age was 9.8 years. Mean preoperative angles were 83.8° (SNA), 80.3° (SNB), and 3° (ANB); mean postoperative angles were 83.8° (SNA), 82.6° (SNB), and 1.2° (ANB). The only significant difference between groups was the postoperative ANB angle, which was negative in the extended EES group compared to the standard EES group, indicating the maxilla was positioned posteriorly compared to the mandible.&lt;/p&gt;&lt;p&gt;&lt;strong&gt;Conclusions: &lt;/strong&gt;The measurement values of the EES groups and the transcranial surgery group exhibited minimal differences, except for a significant decrease in the postoperative ANB angle in the extended EES group compared with the standard EES group. These results show that extended EES may impact midface growth. Further research is required to understan","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":null,"pages":null},"PeriodicalIF":2.1,"publicationDate":"2024-10-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142406461","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}

{"title":"Three-dimensional imaging in craniofacial surgery: utilization of a novel 3D mobile application to evaluate the surgical outcomes of a skull recontouring procedure for cephalohematoma.","authors":"Geena Jung, Emery Buckner-Wolfson, Hailey Reisert, Margaret Keymakh, Timothy Kim, Ryan Fatemi, Andres Pasuizaca, Pushti Shah, Joshua Cohen, Seyed Ahmad Naseri Alavi, Genesis Liriano, Andrew Kobets","doi":"10.3171/2024.7.PEDS24271","DOIUrl":"https://doi.org/10.3171/2024.7.PEDS24271","url":null,"abstract":"<p><strong>Objective: </strong>Cranial abnormalities are common birth defects that frequently alter skull shape and appearance. Despite their prevalence, objective quantification of defect severity pre- and posttreatment is limited. The authors evaluated the ability of MirrorMe3D, a novel 3D mobile iPhone application, to measure changes in the contour of the skull for patients undergoing contouring of a calcified cephalohematoma.</p><p><strong>Methods: </strong>The heads of two 20-month-old patients with disfiguring right parietal cephalohematomas undergoing a skull recontouring operation were scanned pre- and postsurgery. Four scans of the cranial abnormality were taken throughout the procedure and intraoperative 3D models were generated. Models of the head were overlapped pre- and postsurgery and compared using a depth analyzer built into MirrorMe3D.</p><p><strong>Results: </strong>Depth analysis revealed 6.0-mm and 9.9-mm differences for patients 1 and 2, respectively. Volume analysis revealed 33-cm3 and 85-cm3 differences for patients 1 and 2, respectively. Currently, no standard for quantitative measurement of the surgical outcomes of a skull reconstruction procedure exists.</p><p><strong>Conclusions: </strong>MirrorMe3D provides an efficient method for monitoring patients with simple topographic scans that create accurate models of the head. The authors show the app's ability to capture the severity of a calcified cephalohematoma and quantify the changes in the contour of the skull before and after surgery.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":null,"pages":null},"PeriodicalIF":2.1,"publicationDate":"2024-10-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142375505","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}

{"title":"Letter to the Editor. Sleep apnea and Chiari malformation type I.","authors":"Atul Goel","doi":"10.3171/2024.8.PEDS24422","DOIUrl":"https://doi.org/10.3171/2024.8.PEDS24422","url":null,"abstract":"","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":null,"pages":null},"PeriodicalIF":2.1,"publicationDate":"2024-10-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142375502","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}

{"title":"Stereotactic radiosurgery for arteriovenous malformations in pediatric patients: an updated systematic review and meta-analysis.","authors":"Salem M Tos, Georgios Mantziaris, Ahmed Shaaban, Neil Dayawansa, Ahmed Sallam Motawei, Jason P Sheehan","doi":"10.3171/2024.7.PEDS24230","DOIUrl":"https://doi.org/10.3171/2024.7.PEDS24230","url":null,"abstract":"<p><strong>Objective: </strong>Intracranial arteriovenous malformations are the most common cause of intracranial hemorrhages in pediatric patients. Stereotactic radiosurgery (SRS) has been used extensively to treat these lesions. The authors conducted a systematic review and meta-analysis to report treatment outcomes and long-term complications.</p><p><strong>Methods: </strong>This study follows the PRISMA and MOOSE guidelines, with the search spanning electronic databases up to February 6, 2024. The outcome measures included obliteration rate, hemorrhage in the latency period, symptomatic radiation-induced changes (RICs), cyst formation, and radiation-induced tumorigenesis.</p><p><strong>Results: </strong>A total of 1493 patients across 24 studies were included. The pooled complete obliteration after single-fraction SRS was 64.7% (95% CI 58%-69%). The pooled post-SRS hemorrhage rate at the final follow-up was 6.2% (95% CI 5%-8%). The overall incidence rate of RIC was 31.3% (267/854 patients), and the incidence of symptomatic RIC was 8.8% (114/1289 patients). For permanent symptomatic RIC, the pooled incidence was 4.8% (62/1283 patients). At final follow-up, 17 cases of radiation-induced necrosis were documented among 654 patients (2.6%). Similarly, cyst formation was reported in 1.3% of cases (17/1265 patients) and radiation-induced tumors occurred in 0.15% of cases (2/1342 patients).</p><p><strong>Conclusions: </strong>SRS can be considered an effective intervention for appropriately selected pediatric patients with arteriovenous malformations. Long-term complication rates appear to be low but additional longitudinal studies are required to better define the long-term outcomes.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":null,"pages":null},"PeriodicalIF":2.1,"publicationDate":"2024-10-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142375504","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}

{"title":"Predictors of postsurgical retethering in pediatric fatty or tight filum terminale: a systematic review and meta-analysis of 1167 patients.","authors":"Parker D Smith, Mina Guirguis, Michael Farid, Kwadwo Darko, Shubhang Bhalla, Jason Wang, Umaru Barrie, Brett Whittemore","doi":"10.3171/2024.9.PEDS24277","DOIUrl":"https://doi.org/10.3171/2024.9.PEDS24277","url":null,"abstract":"<p><strong>Objective: </strong>The objective of this study was to characterize pediatric patients with tight filum terminale (TFT) or fatty filum terminale (FFT) who experienced retethering after transection of the filum, and to determine the risk factors for retethering.</p><p><strong>Methods: </strong>A systematic review was conducted on May 31, 2023, using PubMed, Google Scholar, SCOPUS, and Web of Science databases according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines to identify studies detailing retethering following transection for TFT or FFT. Studies were then screened using the Newcastle-Ottawa Scale for risk of bias assessment.</p><p><strong>Results: </strong>Eleven articles met the inclusion criteria with an overall cohort of 1167 patients evaluated for retethering following transection for TFT or FFT. The combined retethering rate across all retrospective studies was 3.4% (95% CI 1.6%-5.2%). A random-effects model was used to estimate the prevalence of presenting symptoms, with the most common being lower-extremity weakness in the overall cohort (54.5%, 95% CI 32.6%-76.4%) and bowel or bladder dysfunction in the retethered cohort (57.9%, 95% CI 41.1%-74.8%). Patients who experienced retethering had a similar estimated prevalence of low-lying conus (71.1%, 95% CI 45.1%-97.1%) compared with the overall cohort (51.1%, 95% CI 39.3%-63.0%). The most common complication following surgery for the overall cohort was a wound complication (2.7%, 95% CI 0.8%-4.6%). Postoperative CSF leakage (OR 13.8, 95% CI 3.9-49.4) was a strong predictor of retethering. Sensory changes at initial presentation were also found to be a predictor of retethering (OR 2.9, 95% CI 1.3-6.5). Low-lying conus was not predictive of retethering.</p><p><strong>Conclusions: </strong>Preoperative sensory changes and postsurgical CSF leakage were associated with an increased retethering rate following transection of the filum in cases of tethered cord syndrome secondary to fatty filum terminale.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":null,"pages":null},"PeriodicalIF":2.1,"publicationDate":"2024-10-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142375503","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}

{"title":"Association between social determinants of health and select neurosurgical procedures in the National Spina Bifida Patient Registry.","authors":"Maria A Punchak, Stephen Miranda, Canada Montgomery, Ena Agbodza, Daksh Chauhan, Amy Houtrow, Kathryn Smith, Andrew B Foy, Jeffrey R Leonard, Heidi Castillo, Jonathan Castillo, Rhonda G Cady, Robin M Bowman, Kurt A Freeman, Brandon G Rocque, Tracy M Flanders, Gregory G Heuer","doi":"10.3171/2024.7.PEDS24159","DOIUrl":"https://doi.org/10.3171/2024.7.PEDS24159","url":null,"abstract":"<p><strong>Objective: </strong>Myelomeningocele (MMC) is a lifelong condition requiring complex multidisciplinary management. Using the National Spina Bifida Patient Registry (NSBPR), the authors tested the association between sociodemographic variables and odds of undergoing neurosurgical procedures.</p><p><strong>Methods: </strong>The authors extracted sociodemographic, clinical, and neurosurgical procedure data on participants with MMC aged ≥ 1 year who visited an NSBPR clinic between 2009 and 2020. The zip code of the participant's residence at the time of the last spina bifida clinic visit was linked to the Distressed Communities Index (DCI) tier. Multivariate models were built to identify factors associated with undergoing CSF diversion, shunt revision, tethered cord release (TCR), and Chiari decompression.</p><p><strong>Results: </strong>There were 7924 participants with a median visit age of 13 years (IQR 7-20 years); 49.1% were male, 30.2% were non-Hispanic Black or Hispanic, 54.5% had public/supplemental insurance, and 16.9% were from distressed communities. CSF diversion, shunt revision, TCR, and Chiari decompression were performed in 81.8%, 47.7%, 22.9%, and 8.7% of participants, respectively. In multivariate analyses controlling for age, sex, insurance, DCI tier, lesion level, and surgical closure timing, Hispanic individuals were less likely than their non-Hispanic White counterparts to undergo shunt revision (p = 0.013). Non-Hispanic Black and Hispanic individuals were less likely to undergo TCR (p < 0.001 each) or Chiari decompression (p < 0.001 each). Compared with privately insured individuals, publicly insured individuals were more likely to undergo CSF diversion (p = 0.031). Those in distressed communities had increased odds of undergoing CSF diversion (p = 0.004) than those in prosperous communities.</p><p><strong>Conclusions: </strong>Among individuals with MMC participating in the NSBPR, there were differences in receiving neurosurgical procedures by race/ethnicity, insurance type, and DCI tier. Additional prospective studies are necessary to elucidate the reasons for these variations and their impact on long-term outcomes for this patient population in order to created targeted interventions.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":null,"pages":null},"PeriodicalIF":2.1,"publicationDate":"2024-10-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142375501","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}

{"title":"Unruptured intracranial aneurysms in pediatric sickle cell disease: clinical and MR imaging follow-up of 296 patients.","authors":"Deepak Khatri, Richard Zampolin, Adisson Fortunel, Seon-Kyu Lee","doi":"10.3171/2024.7.PEDS24231","DOIUrl":"https://doi.org/10.3171/2024.7.PEDS24231","url":null,"abstract":"<p><strong>Objective: </strong>Currently available data regarding unruptured intracranial aneurysms (UIAs) in sickle cell disease (SCD) are largely based on adult patients; this has made it challenging to reach a consensus on treatment decisions and follow-up strategies for pediatric SCD patients with UIAs.</p><p><strong>Methods: </strong>A retrospective review of 296 SCD patients (aged 18 years or younger) who underwent MRA from January 2008 to September 2022 was performed. The vessel of origin and characteristics of the UIAs, including maximum diameter, laterality, and number, were evaluated. Demographic data, as well as history of vaso-occlusive crisis and moyamoya syndrome, were recorded. Interval change in size or morphology, as well as a new aneurysm development, were reviewed on follow-up MRA.</p><p><strong>Results: </strong>Forty-nine aneurysms were identified in 32/296 (10.81%) patients, and they all had saccular morphology. In total, 30/49 (61.22%) aneurysms were in the anterior circulation. The ophthalmic segment of the internal carotid artery and the posterior cerebral artery were the most common locations (n = 12 [24.49%] each). Multiple aneurysms were seen in 11/32 patients (34.37%). Most of the African American patients had multiple aneurysms (n = 25/28 [89.3%]). Most aneurysms (n = 43 [87.75%]) were 3 mm or less in diameter. No significant difference in aneurysm size was noticed between aneurysms located in anterior or posterior circulation (p = 0.22). Similarly, age, sex, ethnicity, genotype, or aneurysm multiplicity were not associated with size. Follow-up MRA was available for 24/32 patients (75%). All except 2 aneurysms (n = 2/38 [5.3%]) were stable in size and morphology on follow-up. Interval progression in aneurysm size was noticed in 2 patients with multiple intracranial aneurysms (n = 2/11 [18.2%]).</p><p><strong>Conclusions: </strong>In pediatric SCD patients, the incidence of UIAs was higher than previously reported. Those aneurysms demonstrated a tendency for multiplicity, an atypical anatomical distribution compared with that of adult brain aneurysms, bilateral involvement, and higher prevalence in African Americans. Although most of the aneurysms were stable, some showed interval growth in size.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":null,"pages":null},"PeriodicalIF":2.1,"publicationDate":"2024-09-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142348496","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}

{"title":"Letter to the Editor. Pediatric traumatic venous sinus thrombosis: anticoagulation dilemma.","authors":"Fardad T Afshari","doi":"10.3171/2024.7.PEDS24353","DOIUrl":"https://doi.org/10.3171/2024.7.PEDS24353","url":null,"abstract":"","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":null,"pages":null},"PeriodicalIF":2.1,"publicationDate":"2024-09-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142348495","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}

{"title":"Does pre-irradiation gross tumor volume predict the risk of progression after radiation therapy in pediatric patients with adamantinomatous craniopharyngioma?","authors":"Tamar Brooks, Dexiang Gao, Kathleen Dorris, Karlie Boone, David M Mirsky, Susan Staulcup, Eric Prince, Marina Moskalenko, Elizabeth Ignowski, Narine Wandrey, Kareem Fakhoury, Todd C Hankinson, Sarah A Milgrom","doi":"10.3171/2024.7.PEDS2429","DOIUrl":"https://doi.org/10.3171/2024.7.PEDS2429","url":null,"abstract":"<p><strong>Objective: </strong>In a cohort of patients who were treated with resection and adjuvant radiotherapy (RT) for adamantinomatous craniopharyngioma (ACP), the authors aimed to determine whether gross tumor volume (GTV) at the initiation of RT was associated with the risk of progressive disease (PD) following treatment.</p><p><strong>Methods: </strong>Pediatric and adolescent patients who received surgery and RT for ACP at a single institution from 1998 to 2021 were identified. Univariable Cox regression analyses (UVAs) were performed to assess the association between pre-RT GTV and PD after RT. Multivariable analyses (MVAs) were used to control for potential confounders. Two different endpoints were used to define PD. The first definition was based on radiographic tumor growth, with or without progression of clinical symptoms. The second definition was the requirement for an additional tumor-directed intervention following the completion of RT.</p><p><strong>Results: </strong>Forty-eight patients were eligible for inclusion. The median age at diagnosis was 7.9 years (range 2.1-17.4 years). All patients were treated with surgery and RT with a median dose of 52.2 Gy (range 45-55.8 Gy) and median GTV of 9.86 cm3 (range 0.7-117.7 cm3). After a median follow-up of 66.4 months, 8 patients experienced PD based on both definitions. The 5-year event-free survival rate was 85.4% (95% CI 74.1%-98.3%). On both UVA and MVA, GTV was significantly associated with an increased likelihood of PD (UVA: HR 1.02, 95% CI 1.00-1.04, p = 0.02; MVA: HR 1.10, 95% CI 1.02-1.19, p = 0.01). However, after exclusion of a single outlier with a GTV of 117.7 cm3 prior to RT (remainder of the cohort: range 0.7-37.3 cm3), a second analysis identified no significant association between GTV and PD (UVA: HR 1.03, 95% CI 0.96-1.10, p = 0.4; MVA: HR 1.06, 95% CI 0.96-1.17, p = 0.24).</p><p><strong>Conclusions: </strong>The authors conclude that for most children and adolescents with ACP, the GTV at the initiation of RT is not associated with the risk of PD. This finding may influence surgical practice, because it suggests that aggressive tumor debulking for the purpose of improving the efficacy of RT may not be necessary. In the case of giant tumors, however, novel strategies may be needed for tumor control.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":null,"pages":null},"PeriodicalIF":2.1,"publicationDate":"2024-09-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142348493","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}