Radiographic severity is associated with worse executive function in metopic craniosynostosis.

IF 1.3 4区医学 Q4 CLINICAL NEUROLOGY Child's Nervous System Pub Date : 2024-12-01 Epub Date: 2024-06-21 DOI:10.1007/s00381-024-06493-7

Mariana N Almeida, David P Alper, Carlos Barrero, Neil Parikh, Sacha C Hauc, Jake Moscarelli, Michael Golinko, John Persing, Jordan Swanson, Michael Alperovich

{"title":"Radiographic severity is associated with worse executive function in metopic craniosynostosis.","authors":"Mariana N Almeida, David P Alper, Carlos Barrero, Neil Parikh, Sacha C Hauc, Jake Moscarelli, Michael Golinko, John Persing, Jordan Swanson, Michael Alperovich","doi":"10.1007/s00381-024-06493-7","DOIUrl":null,"url":null,"abstract":"Background: Children with metopic synostosis have been found to have more neurocognitive and behavioral difficulties. The variables that may affect future neurodevelopmental outcomes, including presenting morphologic severity, have not been fully studied. In the largest study to date, we aimed to assess what portends worse neurocognitive and behavioral outcomes at school age.Methods: Children 6-18 years old with surgically corrected metopic nonsyndromic craniosynostosis underwent neurocognitive testing. Parents completed behavior rating surveys about their child: Conners-3 (ADHD), Social Responsiveness Scale-2 (autism spectrum disorder), Behavior Rating Inventory of Executive Function-2 (BRIEF-2: executive function), and Child's Behavior Checklist (overall behavior). The endocranial bifrontal angle (EBA), adjusted EBA (aEBA), frontal angle (FA), and AI-derived metopic severity score (MSS) were determined on pre-operative CT images. Multivariate linear regressions were used to evaluate the association of age at surgery and severity.Results: There were 87 children who underwent neurocognitive testing (average age 10.9 ± 3.3 years) of whom 67 also completed behavioral assessments. Greater phenotypical severity of metopic synostosis (lower FA, aEBA, and EBA) was associated with worse scores on the subscales of the BRIEF-2 (executive function) and executive subscale of the Conners-3. Increasing age at surgery was associated with worse executive function subscale scores of the Conners-3 when controlling for each severity measurement and sociodemographic risk.Conclusion: Children with greater phenotypic severity of metopic synostosis have worse executive function at school age. The majority of children with metopic synostosis have signs of ADHD. Later surgeries (greater than 12 months) may impact executive functioning, regardless of the degree of severity. Future research should aim at identifying the direct structural changes to the brain.","PeriodicalId":9970,"journal":{"name":"Child's Nervous System","volume":" ","pages":"3971-3982"},"PeriodicalIF":1.3000,"publicationDate":"2024-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Child's Nervous System","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1007/s00381-024-06493-7","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2024/6/21 0:00:00","PubModel":"Epub","JCR":"Q4","JCRName":"CLINICAL NEUROLOGY","Score":null,"Total":0}

引用次数: 0

Abstract

Background: Children with metopic synostosis have been found to have more neurocognitive and behavioral difficulties. The variables that may affect future neurodevelopmental outcomes, including presenting morphologic severity, have not been fully studied. In the largest study to date, we aimed to assess what portends worse neurocognitive and behavioral outcomes at school age.

Methods: Children 6-18 years old with surgically corrected metopic nonsyndromic craniosynostosis underwent neurocognitive testing. Parents completed behavior rating surveys about their child: Conners-3 (ADHD), Social Responsiveness Scale-2 (autism spectrum disorder), Behavior Rating Inventory of Executive Function-2 (BRIEF-2: executive function), and Child's Behavior Checklist (overall behavior). The endocranial bifrontal angle (EBA), adjusted EBA (aEBA), frontal angle (FA), and AI-derived metopic severity score (MSS) were determined on pre-operative CT images. Multivariate linear regressions were used to evaluate the association of age at surgery and severity.

Results: There were 87 children who underwent neurocognitive testing (average age 10.9 ± 3.3 years) of whom 67 also completed behavioral assessments. Greater phenotypical severity of metopic synostosis (lower FA, aEBA, and EBA) was associated with worse scores on the subscales of the BRIEF-2 (executive function) and executive subscale of the Conners-3. Increasing age at surgery was associated with worse executive function subscale scores of the Conners-3 when controlling for each severity measurement and sociodemographic risk.

Conclusion: Children with greater phenotypic severity of metopic synostosis have worse executive function at school age. The majority of children with metopic synostosis have signs of ADHD. Later surgeries (greater than 12 months) may impact executive functioning, regardless of the degree of severity. Future research should aim at identifying the direct structural changes to the brain.

Abstract Image

查看原文

微信好友朋友圈 QQ好友复制链接

本刊更多论文

放射学严重程度与偏位颅畸形患者的执行功能恶化有关。

背景：研究发现，患有偏侧突眼的儿童有更多的神经认知和行为障碍。目前尚未对可能影响未来神经发育结果的变量（包括目前的形态学严重程度）进行全面研究。在迄今为止最大规模的研究中，我们旨在评估哪些因素会导致学龄期的神经认知和行为结果更差：方法：6-18 岁经手术矫正的异位非综合征颅脑发育不良儿童接受神经认知测试。家长填写有关孩子的行为评分调查表：康纳斯-3（多动症）、社会反应量表-2（自闭症谱系障碍）、执行功能行为评级量表-2（BRIEF-2：执行功能）和儿童行为检查表（整体行为）。根据术前 CT 图像确定颅内双额角 (EBA)、调整后 EBA (aEBA)、额角 (FA) 和源自 AI 的偏侧严重性评分 (MSS)。采用多变量线性回归评估手术年龄与严重程度的关系：共有 87 名儿童接受了神经认知测试（平均年龄为 10.9 ± 3.3 岁），其中 67 名儿童还完成了行为评估。偏侧突触症的表型严重程度越高（FA、aEBA和EBA越低），BRIEF-2（执行功能）分量表和Conners-3执行分量表的得分越低。在控制各项严重程度测量和社会人口风险的情况下，手术年龄的增加与Conners-3执行功能分量表得分的降低有关：结论：表型严重度较高的异位突触症儿童在学龄期的执行功能较差。大多数患有异位突触症的儿童都有多动症的症状。无论严重程度如何，后期手术（超过 12 个月）都可能影响执行功能。未来的研究应着眼于确定大脑结构的直接变化。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

求助全文

约1分钟内获得全文去求助

来源期刊

Child's Nervous System 医学-临床神经学

CiteScore

3.00

自引率

7.10%

发文量

322

审稿时长

3 months

期刊介绍： The journal has been expanded to encompass all aspects of pediatric neurosciences concerning the developmental and acquired abnormalities of the nervous system and its coverings, functional disorders, epilepsy, spasticity, basic and clinical neuro-oncology, rehabilitation and trauma. Global pediatric neurosurgery is an additional field of interest that will be considered for publication in the journal.