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结节性硬化症患儿认知预测临床因素的相互依赖性。

Interdependence of clinical factors predicting cognition in children with tuberous sclerosis complex.

作者信息

Overwater I E, Verhaar B J H, Lingsma H F, Bindels-de Heus G C B, van den Ouweland A M W, Nellist M, Ten Hoopen L W, Elgersma Y, Moll H A, de Wit M C Y

机构信息

Department of Neurology, Erasmus University Medical Centre, PO Box 2060, 3000 CB, Rotterdam, The Netherlands.

ENCORE Expertise Center for Neurodevelopmental Disorders, Erasmus University Medical Center, PO Box 2060, 3000 CB, Rotterdam, The Netherlands.

出版信息

J Neurol. 2017 Jan;264(1):161-167. doi: 10.1007/s00415-016-8335-5. Epub 2016 Nov 22.

Abstract

Cognitive development in patients with tuberous sclerosis complex is highly variable. Predictors in the infant years would be valuable to counsel parents and to support development. The aim of this study was to confirm factors that have been reported to be independently correlated with cognitive development. 102 patients included in this study were treated at the ENCORE-TSC expertise center of the Erasmus Medical Center-Sophia Children's Hospital. Data from the first 24 months of life were used, including details on epilepsy, motor development and mutation status. Outcome was defined as cognitive development (intellectual equivalent, IE) as measured using tests appropriate to the patients age and cognitive abilities (median age at testing 8.2 years, IQR 4.7-12.0). Univariable and multivariable regression analyses were used. In a univariable analysis, predictors of lower IE were: the presence of infantile spasms (β = -18.3, p = 0.000), a larger number of antiepileptic drugs used (β = -6.3, p = 0.000), vigabatrin not used as first drug (β = -14.6, p = 0.020), corticosteroid treatment (β = -33.2, p = 0.005), and a later age at which the child could walk independently (β = -2.1, p = 0.000). An older age at seizure onset predicted higher IE (β = 1.7, p = 0.000). In a multivariable analysis, only age at seizure onset was significantly correlated to IE (β = 1.2, p = 0.005), contributing to 28% of the variation in IE. In our cohort, age at seizure onset was the only variable that independently predicted IE. Factors predicting cognitive development could aid parents and physicians in finding the appropriate support and schooling for these patients.

摘要

结节性硬化症患者的认知发展差异很大。婴儿期的预测因素对于为家长提供咨询和支持其发展很有价值。本研究的目的是确认据报道与认知发展独立相关的因素。本研究纳入的102例患者在伊拉斯姆斯医学中心 - 索菲亚儿童医院的ENCORE - TSC专业中心接受治疗。使用了生命最初24个月的数据,包括癫痫、运动发育和突变状态的详细信息。结果定义为使用适合患者年龄和认知能力的测试所测量的认知发展(智力当量,IE)(测试时的中位年龄为8.2岁,四分位距为4.7 - 12.0)。采用单变量和多变量回归分析。在单变量分析中,IE较低的预测因素为:婴儿痉挛的存在(β = -18.3,p = 0.000)、使用的抗癫痫药物数量较多(β = -6.3,p = 0.000)、未将氨己烯酸作为首选药物(β = -14.6,p = 0.020)、皮质类固醇治疗(β = -33.2,p = 0.005)以及儿童能够独立行走的年龄较晚(β = -2.1,p = 0.000)。癫痫发作起始年龄较大预测IE较高(β = 1.7,p = 0.000)。在多变量分析中,只有癫痫发作起始年龄与IE显著相关(β = 1.2,p = 0.005),占IE变异的28%。在我们的队列中,癫痫发作起始年龄是唯一独立预测IE的变量。预测认知发展的因素可以帮助家长和医生为这些患者找到合适的支持和教育方式。

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