Brief Title
The Boston Circulatory Arrest Study: Antecedents and Correlates of Well-Being in Adults With Congenital Heart Disease
Official Title
The Boston Circulatory Arrest Study: Antecedents and Correlates of Well-Being in Adults With Congenital Heart Disease
Brief Summary
In the landmark Boston Circulatory Arrest Study, neurologic and developmental status was measured following infant heart surgery and then prospectively at ages 1, 2.5, 4, 8, and 16 years, with findings of significant neurocognitive deficits and brain MRI abnormalities regardless of operative management. To date, no study has evaluated the neuropsychological and neuroimaging antecedents and correlates of well-being in adults with congenital heart disease, a population now >1 million and projected to grow at 5% per year. The investigators propose to study the Boston cohort at ages 24-29 years to assess the associations of adult well-being with childhood and adolescent executive function, other measures of mental health and cognitive function, adolescent brain MRI findings, and clinical variables; findings will guide the design of interventions in childhood to optimize outcomes in adults with congenital heart disease.
Detailed Description
Previously lethal, critical congenital heart disease (CHD) can now be treated effectively with surgical, catheter, and medical interventions. The resulting dramatic improvement in life expectancy has brought a major demographic shift, so that adult patients with CHD now outnumber children with CHD, even for complex conditions. Adult survivors are at increased risk of anxiety, depression, social difficulties, lower educational attainment, and underemployment. These psychosocial morbidities may be associated with deficits in executive functions (EFs) and other neurocognitive abilities that are prevalent in children and adolescents with CHD. Deficits in EFs, represented by measures of inhibitory control, working memory, cognitive flexibility, and decision-making, are highly dependent on the integrity of cortical and subcortical neural networks that continue to develop into early adulthood and can have a major adverse impact on self-regulation and management. The goal of this proposal is to bridge the gap in knowledge between known executive function deficits in childhood CHD and adult well-being. The investigators propose to accomplish our goal by studying subjects, now age 24-29 years, who were enrolled as infants in the Boston Circulatory Arrest Study and then studied with respect to neuropsychological and developmental function at ages 1, 2.5, 4, 8, and 16 years, as well as with brain MRI at 16 years. In Aim 1, the relationship of EFs to major dimensions of adult well-being will be explored. It is hypothesized that lower performance on EFs will be related to poorer overall well-being. The outcomes measured to determine well-being will be quality of life, neuropsychological function (e.g. social cognition, memory skills), mental health diagnosis and function (e.g. anxiety, depression), social relatedness, academic achievement, and adult independence (e.g. employment status, medical follow up). In Aim 2, the relationship of EFs to MRI-derived measures of brain structure, function, and connectivity will be determined. It is hypothesized that lower performance on EFs will be associated with lower global efficiency (integration) and higher modularity (segregation). The brain MRI outcomes will be measured using global white matter connectivity, regional cortical gray matter thickness, gray matter connectivity measured from interregional correlation in cortical thickness, and functional connectivity as defined by resting state functional magnetic resonance imaging. In Aim 3, longitudinal models will be used to analyze the association of adult well-being with earlier measures of EFs and other neurocognitive and mental health variables, as well as with earlier clinical variables and adolescent neuroimaging. The associations of adult well-being dimensions with childhood and adolescent EFs, other measures of mental health and cognitive function, adolescent brain MRI findings, and clinical variables will be explored. The ultimate goal is to identify early, modifiable risk factors for adult performance to guide the design of targeted treatment strategies that optimize educational achievement, employability, and quality of life in the burgeoning population of adults with CHD.
Study Type
Observational
Primary Outcome
Quality of Life health related
Secondary Outcome
Mental health - Anxiety
Condition
Transposition Great Arteries
Intervention
Brain MRI, neurocognitive and psychological testing
Study Arms / Comparison Groups
TGA Case
Description: Brain MRI, Neurocognitive and psychological testing
Publications
* Includes publications given by the data provider as well as publications identified by National Clinical Trials Identifier (NCT ID) in Medline.
Recruitment Information
Recruitment Status
Diagnostic Test
Estimated Enrollment
122
Start Date
November 9, 2017
Completion Date
January 31, 2023
Primary Completion Date
November 1, 2021
Eligibility Criteria
Study Group: Inclusion Criteria 1. Prior participation in the Boston Circulatory Arrest Study 2. Informed consent Exclusion Criteria 1. Disorders that would prevent successful completion of the planned study testing (severe developmental impairment to prevent answering surveys and participating in interviews) 2. Participants lack of reading fluency in English, which is the only language for which we have the ability to do neuropsychology testing, and for which questionnaires have been validated 3. Women who are currently pregnant will be excluded from the MRI portion of the study only Control Group: Control Inclusion Criteria 1. Age 24-30 years at the time of enrollment 2. Informed consent Control Exclusion Criteria 1. Conditions that would prevent successful completion of the planned study testing (MRI) (e.g., pacemaker, metal implants, orthodontia) 2. Congenital heart disease requiring surgical correction 3. Lack of reading fluency in English, the only language for which questionnaires have been validated 4. Because we purposefully wish to compare d-TGA patients with those in optimal neurodevelopmental health, we will use the exclusion criteria for the NIH-funded project, "MRI study of normal brain development." 5. Current pregnancy
Gender
All
Ages
24 Years - 33 Years
Accepts Healthy Volunteers
Accepts Healthy Volunteers
Contacts
Michelle Gurvitz, MD, ,
Location Countries
United States
Location Countries
United States
Administrative Informations
NCT ID
NCT03073122
Organization ID
P00023574
Secondary IDs
1R01HL135061-01
Responsible Party
Principal Investigator
Study Sponsor
Boston Children's Hospital
Collaborators
National Heart, Lung, and Blood Institute (NHLBI)
Study Sponsor
Michelle Gurvitz, MD, Principal Investigator, Boston Children's Hospital
Verification Date
March 2022