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1型神经纤维瘤病患儿幼儿期情绪和行为问题的发展

Development of emotional and behavioral problems in neurofibromatosis type 1 during young childhood.

作者信息

Rietman André B, Oostenbrink Rianne, van Noort Kimberley, Franken Marie-Christine J P, Catsman-Berrevoets Coriene E, Aarsen Femke K, Hendriksen Jos G, de Nijs Pieter F A

机构信息

Department of Pediatric Neurology, Erasmus MC-Sophia Children's Hospital and ENCORE Expertise Centre for Neurodevelopmental Disorders, Rotterdam, The Netherlands.

Department of General Pediatrics, Erasmus MC-Sophia Children's Hospital and ENCORE Expertise Centre for Neurodevelopmental Disorders, Rotterdam, The Netherlands.

出版信息

Am J Med Genet A. 2017 Sep;173(9):2373-2380. doi: 10.1002/ajmg.a.38323. Epub 2017 Jun 19.

Abstract

This retrospective longitudinal study in young children with neurofibromatosis type 1 (NF1) aimed to identify if, and how early problems in behavior, intelligence, and language development are associated with later behavioral problems. At the first assessment at preschool age, we evaluated language skills, intelligence, and emotional and behavioral problems as reported by parents. The second assessment at school-age we evaluated intelligence, and emotional and behavioral problems as reported by parents and teachers. Association of baseline assessments with secondary assessment was evaluated using multivariable linear regression analysis. Of the 61 patients (25 males, 36 females; mean age 4;5 years [SD 1;1 years]) with NF1 who had a first assessment, 38 children (21 males, 17 females; mean age 7;11 years [SD 2;1 years]) had a second assessment after a mean period of 3;5 years. Longitudinal data on behavioral problems were collected for 23 of these children. Intelligence and language development were not associated with internalizing problems. Parent-rated internalizing behavioral problems significantly increased with age in this subgroup. Baseline internalizing problems predicted later internalizing problems (adjusted R  = 0.33, p = 0.003). The presence of these problems at pre-school age may be predictive of internalizing problems at a later age.

摘要

这项针对1型神经纤维瘤病(NF1)幼儿的回顾性纵向研究旨在确定行为、智力和语言发展方面的早期问题是否以及如何与后期行为问题相关联。在学龄前的首次评估中,我们评估了父母报告的语言技能、智力以及情绪和行为问题。在学龄期的第二次评估中,我们评估了父母和教师报告的智力、情绪和行为问题。使用多变量线性回归分析评估基线评估与二次评估之间的关联。在接受首次评估的61例NF1患者(25例男性,36例女性;平均年龄4岁5个月[标准差1岁1个月])中,38名儿童(21例男性,17例女性;平均年龄7岁11个月[标准差2岁1个月])在平均3年5个月的时间后接受了第二次评估。收集了其中23名儿童关于行为问题的纵向数据。智力和语言发展与内化问题无关。在这个亚组中,父母评定的内化行为问题随年龄显著增加。基线内化问题可预测后期内化问题(调整后R=0.33,p=0.003)。这些问题在学龄前的存在可能预示着后期的内化问题。

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