Decisional Capacity and Informed Consent in Fragile X Syndrome

脆性 X 综合征的决策能力和知情同意

基本信息

批准号：
8437309
负责人：
Donald B Bailey
金额：
$ 62.7万
依托单位：
RESEARCH TRIANGLE INSTITUTE
依托单位国家：
美国
项目类别：
财政年份：
2012
资助国家：
美国
起止时间：
2012-09-26 至 2017-06-30
项目状态：
已结题

来源：
https://reporter.nih.gov/project-details/8437309
关键词：
Adult Adverse effects Affect Behavior Blood Caregivers Clinical Treatment Clinical Trials Cognitive Consent Data Daughter Decision Aid Decision Making Developmental Disabilities Developmental Process Educational Assessment Ethics Family Female Female Adolescents Fragile X Syndrome Generations Gold Health Communication Heart Rate Human Impairment Individual Individual Differences Informed Consent Inherited Institutional Review Boards Intellectual functioning disability Intervention Learning Life Life Cycle Stages Literature Measures Molecular Nature Neurocognitive Parental Consent Parents Participant Perception Pharmacologic Substance Process Program Development Randomized Controlled Trials Reading Research Research Personnel Saliva Son Specialist Surveys Tablets Technology Testing Videotape base comparative efficacy consent capacity design digital evidence based guidelines experience male member mild neurocognitive impairment neuroimaging neuropsychological novel psychologic research study skills standard measure

项目摘要

DESCRIPTION (provided by applicant): Fragile X syndrome (FXS) is the most common inherited form of intellectual disability. The typical male has an IQ of 50 to 55. Some are severely affected (low IQ, nonverbal), while others are mildly affected (mild intellectual disabiliy, can read, very sociable). Females are generally less affected than males but have an even broader range in function. A few females have severe intellectual and adaptive impairments, but most have a normal or borderline intellectual ability; about one-third have mild cognitive impairment. This wide range in cognitive ability almost certainly leads to variable ability to make choices and live independently. This application focuses on a particular form of choice making-decisional capacity-the ability to make a meaningful decision about whether to participate in research. Individuals with FXS have served as participants in research studies for more than 30 years. For the most part, this research has been relatively noninvasive, limited to parent surveys and neuropsychological or psycho-educational assessments. Some studies have drawn blood, collected saliva, measured heart rate, or used neuroimaging, but until recently there has been a paucity of treatment research. This scenario has changed dramatically in the last 3 years. Advances in understanding the molecular basis of FXS have led to a new generation of targeted treatment approaches, and clinical trials are underway using a variety of compounds. The possibility of side effects and the potential for significant changes in behavior and learning abilty elevate to a new level the importance of obtaining meaningful consent. Researchers and IRB members need data to help guide decisions about involving individuals with FXS in the consent process. Only a few studies have investigated decisional capacity of individuals with intellectual and developmental disabilities, and none have studied individuals with FXS. Virtually nothing is known about the extent to which individuals with FXS are involved in decisions about research participation, and no studies have investigated decisional capacity in FXS. This project will assess the range of decisional capacity in FXS, identify factors associated with variability in decisional capacity, and develop and evaluate a tablet-based decision aid to assist in obtaining informed consent. The study team includes experts in FXS, human research protection professionals, specialists in developing materials for informed decision making, a leading researcher in FXS clinical trials, and researchers who have developed and evaluated gold-standard measures for characterizing decisional capacity. The study will provide important information about the nature of decisional capacity in FXS and will provide guidance for researchers, IRB members, and pharmaceutical companies seeking to include individuals with FXS as study participants. PUBLIC HEALTH RELEVANCE: This project will be the first to study the ability of individuals with fragile X syndrome to provide informed consent to participate in research. Given the advent of clinical trials and treatment possibilities for these individuals, understanding their decisiona capacity will be important for researchers, clinicians, IRB members, and pharmaceutical companies. This project will assess the range of decisional capacity in FXS, identify factors associated with variability in decisional capacity, and develop and evaluate a tablet-based decision aid to assist in obtaining informed consent.

描述（由申请人提供）：脆弱的X综合征（FXS）是智障最常见的形式。典型的男性的智商为50至55。有些人受到严重影响（低智商，非语言），而另一些则受到轻度影响（轻度的智力上的不稳定，可以阅读，非常社交）。女性通常比男性受到的影响较小，但功能范围更大。一些女性患有严重的智力和适应性障碍，但大多数具有正常或边缘性的智力能力。大约三分之一的认知障碍。认知能力的广泛范围几乎可以肯定会导致可变能力选择和独立生活。该申请专注于一种选择的特定形式，使得确定能力 - 对是否参与研究做出有意义的决定的能力。 FXS的个人已担任研究参与者已有30多年了。在大多数情况下，这项研究相对无创，仅限于父母调查以及神经心理学或心理教育评估。一些研究已吸收血液，收集唾液，测量心率或使用神经影像学，但直到最近才有很少的治疗研究。在过去的三年中，这种情况发生了巨大变化。了解FX的分子基础的进步导致了新一代的目标治疗方法，并且使用多种化合物正在进行临床试验。副作用的可能性以及行为和学习的重大变化的可能性提高到新的水平，即获得有意义的同意的重要性。研究人员和IRB成员需要数据来帮助指导有关让FXS参与同意过程的决策。只有少数研究调查了智力和发育障碍的人的决策能力，没有人研究过FXS的人。实际上，对于FXS的个人参与研究参与的决策的程度几乎一无所知，并且没有研究研究FXS的决策能力。该项目将评估FXS中的决策能力的范围，确定与决策能力变异性相关的因素，并开发和评估基于平板电脑的决策援助，以帮助获得知情同意。该研究团队包括FXS，人类研究保护专业人员，开发知情决策材料的专家，FXS临床试验的主要研究人员，以及制定和评估金标准措施以表征决策能力的研究人员。该研究将提供有关FXS决策能力的性质的重要信息，并将为寻求包括以FXS作为研究参与者的个人的研究人员，IRB成员和制药公司提供指导。公共卫生相关性：该项目将是第一个研究脆弱X综合症的人提供知情同意以参与研究的能力的项目。考虑到这些人的临床试验和治疗可能性的出现，了解其决策能力对于研究人员，临床医生，IRB成员和制药公司至关重要。该项目将评估FXS中的决策能力的范围，确定与决策能力变异性相关的因素，并开发和评估基于平板电脑的决策援助，以帮助获得知情同意。