Longitudinal Effects of Spina Bifida on Learning

描述（由申请人提供）：脊柱裂髓脊膜膨出（SB）是一种神经管缺陷，与脊柱和大脑畸形有关，与运动、非言语认知、注意力、语言和适应性行为问题有关。虽然这些缺陷在学龄儿童中有很好的记录，但由于很少有研究检查儿童早期SB的认知发展，因此对其发展起源没有很好的了解。对学龄儿童的研究表明，年龄较大的SB儿童在时间、注意定向和运动控制/学习方面存在核心缺陷，在视觉感知、注意调节和涉及信息/偶然性学习（II/CL）整合的技能（基于规则的问题解决、语境语言和行为调节）方面存在功能缺陷。这些赤字跨越了结果领域。在目前对SB婴儿的纵向研究中，研究人员检查了6-36个月的核心缺陷和6-36个月的功能缺陷（包括II/CL技能）的发展情况，以确定其起源以及与SB后来发现的问题的可能联系。目前的纵向研究结果表明，SB婴儿在时间、运动质量和组织以及注意定向方面确实表现出核心缺陷。这些核心缺陷早在7个月大时就很明显，持续到童年早期，并且似乎与3岁时新出现的功能缺陷的增长有关，这表明它们作为后来SB问题的基础的重要性。为了将核心和功能缺陷与学龄期类似技能的评估联系起来，并与传统的结果评估联系起来，拟议的研究将对参与我们当前研究的85名SB儿童和73名正常发育儿童的纵向核心和功能缺陷进行检查。在拟议的扩展中，我们将看到这些儿童在7.5岁、8.5岁和9.5岁时检查核心和功能缺陷的增长，评估包括智力、语言、学术、适应行为和社会功能在内的传统结果。我们期望儿童在核心和功能性任务上的早期表现将预测他们在早期学龄阶段类似技能的发展，并解释传统结果的差异。然而，我们预测，随着时间的推移，技能之间的关系将被中枢神经系统（脊柱和大脑）异常的性质和严重程度以及重要的环境变量（包括同期和以前的母亲互动方式）所缓和。特别是，照顾者维持儿童注意力的能力，对儿童的信号作出偶然和热情的反应的能力，以及提供认知丰富的语言输入的能力，可能会缓和早期核心缺陷导致功能缺陷的程度，并预测这些技能的后期能力。在9.5岁时记录的SB儿童特定中枢神经系统异常与发育轨迹的关系以及与这组技能的并发关系，应该揭示更年轻SB儿童脑-行为关系的重要信息。本研究是为数不多的针对学龄前儿童的研究之一，也是唯一一项系统、全面的进入学龄期的随访研究。这项研究的完成将与学龄儿童患有SB的重要研究机构建立联系。

DESCRIPTION (provided by applicant): Spina bifida myelomeningocele (SB), a neural tube defect associated with deformities of the spine and brain, is associated with motor, non-verbal cognitive, attention, language, and adaptive behavior problems. While these deficiencies are well documented in school-age children, their developmental origins are not well understood since few studies have examined cognitive development for children with SB in early childhood. Research with school- age children has demonstrated that older children with SB have core deficits involving timing, attention orientation, and motor control/learning, and functional deficits in visual perception, attention regulation, and skills involving the integration of information/contingency learning (II/CL): rule-based problem solving, contextual language, and behavioral regulation. These deficits cut across outcome domains. In the current longitudinal study of infants with SB, the development over 6 to 36 months of core deficits and functional deficits from 6-36 months of age, including II/CL skills, have been examined in order to determine their origins and possible links to the later problems identified for SB. Findings from the current longitudinal study show that infants with SB do exhibit core deficits in timing, motor quality and organization, and attention orientation. These core deficits are apparent as early as 7 months of age, persist across early childhood, and appear to be related to growth in emerging functional deficits at 3 years, which demonstrates their importance as a foundation for later SB problems. To link core and functional deficits with school age assessments of similar skills, and to traditional assessments of outcomes, the proposed research will examine longitudinally core and functional deficits for the same 85 children with SB and 73 children normally developing controls that have participated in our current study. In the proposed extension, we will see these children at 7.5, 8.5, and 9.5 years of age to examine growth of core and functional deficits, assess traditional outcomes involving intelligence, language, academics, adaptive behavior, and social functions. We expect that the children's early performance on core and functional tasks will predict their development of similar skills during the early school-age period, and explain variations in traditional outcomes. However, we predict that relations among skills over time will be moderated by the nature and severity of CNS (spine and brain) anomalies and by important environmental variables, including concurrent and previous maternal interactive style. In particular, the caregivers' abilities to maintain the children's attention focus, respond contingently and warmly to children's signals, and provide cognitively enriching verbal input are likely to moderate the degree to which early core deficits lead to functional deficits and predict later ability in these skills. Relations between specific CNS abnormalities documented at 9.5 years for children with SB and the developmental trajectories and concurrent relations with this group of skills, should reveal important information about brain- behavior relations for younger SB children. This study is 1 of few studies of children with SB in the preschool years, and would be the only systematic, comprehensive follow- up into the school- age years. Completion of the study would allow a link with a significant body of research on school- age children with SB.