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非综合征性颅缝早闭患者的长期神经认知和行为结局

Long-term neurocognitive and behavioral outcomes in patients with non-syndromic craniosynostosis.

作者信息

Ihnat Jacqueline, Hu Kevin Gao, Goss Jeremy, Allam Omar, Parikh Neil, Vassallo Melanie, Mayes Linda, Persing John, Alperovich Michael

机构信息

Division of Plastic Surgery, Department of Surgery, Yale School of Medicine, 330 Cedar Street, Boardman Building, New Haven, CT, 06510, USA.

Yale Child Study Center, Yale School of Medicine, New Haven, CT, USA.

出版信息

Childs Nerv Syst. 2025 Jul 14;41(1):233. doi: 10.1007/s00381-025-06888-0.

DOI:10.1007/s00381-025-06888-0
PMID:40659876
Abstract

PURPOSE

In this study, we explore the long-term cognitive capabilities and behaviors of patients in late adolescence and early adulthood who underwent corrective surgery for craniosynostosis as infants.

METHODS

Patients 16 years of age or older who had undergone surgery as infants for any type of non-syndromic craniosynostosis were identified from the operating records of three craniofacial plastic surgeons from a single institution. Participants underwent standardized neurocognitive testing using the BEERY and WASI. A subset of patients additionally underwent behavioral testing using the CAARS-2, ASR, SRS-2, and BRIEF standardized tests.

RESULTS

Thirty-two participants underwent neurocognitive testing and 22 participants underwent behavioral testing. The mean scores for the WASI FSIQ-4, PRI, and VCI were not significantly different than the general population. The mean score for BEERY VMI was 95, BEERY VP was 97, and BEERY MC was 92, all of which were significantly lower than the population average. Twenty-three percent of participants scored high or very high on the CAARS-2 ADHD index, and of the 44 total patients contacted, 13.6% either confirmed having a diagnosis of autism or tested moderate-severe on the SRS-2 autism screener.

CONCLUSIONS

Patients have comparable neurocognition relative to the general population, with deficits in visual and motor integration. However, there is a significantly higher prevalence of ADHD and autism-related behaviors in this cohort. These findings are useful in counseling parents with infants who have been diagnosed with craniosynostosis, as well as in directing early screening and intervention for these patients to provide the resources and therapies families need.

摘要

目的

在本研究中,我们探讨了婴儿期接受颅缝早闭矫正手术的青少年晚期和成年早期患者的长期认知能力和行为。

方法

从一家机构的三位颅面整形外科医生的手术记录中识别出16岁及以上婴儿期因任何类型的非综合征性颅缝早闭接受过手术的患者。参与者使用BEERY和WASI进行标准化神经认知测试。一部分患者还使用CAARS-2、ASR、SRS-2和BRIEF标准化测试进行了行为测试。

结果

32名参与者接受了神经认知测试,22名参与者接受了行为测试。WASI FSIQ-4、PRI和VCI的平均得分与一般人群无显著差异。BEERY VMI的平均得分为95,BEERY VP为97,BEERY MC为92,所有这些均显著低于人群平均水平。23%的参与者在CAARS-2注意力缺陷多动障碍指数上得分高或非常高,在总共联系的44名患者中,13.6%要么确诊患有自闭症,要么在SRS-2自闭症筛查中测试为中度至重度。

结论

患者的神经认知与一般人群相当,但在视觉和运动整合方面存在缺陷。然而,该队列中注意力缺陷多动障碍和自闭症相关行为的患病率显著更高。这些发现有助于为被诊断患有颅缝早闭的婴儿的父母提供咨询,也有助于指导对这些患者进行早期筛查和干预,以提供家庭所需的资源和治疗。

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本文引用的文献

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Long-term neurocognitive outcomes in 204 single-suture craniosynostosis patients.204 例单骨缝颅缝早闭患者的长期神经认知结局。
Childs Nerv Syst. 2023 Jul;39(7):1921-1928. doi: 10.1007/s00381-023-05908-1. Epub 2023 Mar 6.
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Longitudinal Outpatient and School-Based Service Use among Children with Nonsyndromic Craniosynostosis.非综合征性颅缝早闭患儿的纵向门诊和基于学校的服务利用情况。
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Assessing Long-Term Neurodevelopment among Children with Non-Syndromic Single Suture Craniosynostosis.
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Functional Network Development in Sagittal Craniosynostosis Treated With Whole Vault Cranioplasty.矢状缝早闭颅骨成形术治疗后功能网络的发展。
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Nonsyndromic Craniosynostosis.非综合征性颅缝早闭
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