Trends in Congenital Heart Disease

Triedman, John K.; Newburger, Jane W.

doi:10.1161/circulationaha.116.023544

Cited by 240 publications

(175 citation statements)

References 151 publications

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“…As such, the ability to identify modifiable risk factors could be an important step towards enhancing long-term neurodevelopmental outcomes via future interventional clinical trials. 3,4 Quantitative MRS can measure multiple brain metabolites that are biomarkers not only for brain dysmaturation but also potentially for brain function, such as energy processes and neurotransmitters. Although most previous studies of MRS in patients with CHD have measured major brain metabolites (eg, NAA, choline, and lactate) via the long-echo magnetic resonance technique, 6–8,18,24–26 few studies have measured other metabolites more readily detectable with short-echo MRS, such as myo-inositol, phosphocreatine, glutamate, and GABA.…”

Section: Discussionmentioning

confidence: 99%

“…In addition, identifying modifiable risk factors that could help enhance long-term neurodevelopmental outcomes via future interventional clinical trials is a priority. 3,4 Abnormalities of cerebral maturation have been de-lineated in CHD by the use of a variety of structural and metabolic neuroimaging techniques such as brain magnetic resonance spectroscopy (MRS). 5–13 MRS measures metabolites that are not only relevant to understanding structural brain maturation but also important for delineating physiological processes such as energy metabolism.…”

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confidence: 99%

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Clinical Factors Associated with Cerebral Metabolism in Term Neonates with Congenital Heart Disease

Harbison

Votava‐Smith

Castillo

et al. 2017

The Journal of Pediatrics

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Objective To determine associations between patient and clinical factors with postnatal brain metabolism in term neonates with congenital heart disease (CHD) via the use of quantitative magnetic resonance spectroscopy. Study design Neonates with CHD were enrolled prospectively to undergo pre- and postoperative 3T brain magnetic resonance imaging. Short-echo single-voxel magnetic resonance spectroscopy of parietal white matter was used to quantify metabolites related to brain maturation (n-acetyl aspartate, choline, myo-inositol), neurotransmitters (glutamate and gamma-aminobutyric acid), energy metabolism (glutamine, citrate, glucose, and phosphocreatine), and injury/apoptosis (lactate and lipids). Multivariable regression was performed to search for associations between (1) patient-specific/prenatal/preoperative factors with concurrent brain metabolism and (2) intraoperative and postoperative factors with postoperative brain metabolism. Results A total of 83 magnetic resonance images were obtained on 55 subjects. No patient-specific, prenatal, or preoperative factors associated with concurrent metabolic brain dysmaturation or elevated lactate could be identified. Chromosome 22q11 microdeletion and age at surgery were predictive of altered concurrent white matter phosphocreatine (P < .0055). The only significant intraoperative association found was increased deep hypothermic circulatory arrest time with reduced postoperative white matter glutamate and gamma-aminobutyric acid (P < .0072). Multiple postoperative factors, including increased number of extracorporeal membrane oxygenation days (P < .0067), intensive care unit, length of stay (P < .0047), seizures in the intensive care unit (P < .0009), and home antiepileptic use (P < .0002), were associated with reduced postoperative white matter n-acetyl aspartate. Conclusion Multiple postoperative factors were found to be associated with altered brain metabolism in term infants with CHD. Interventions in the perioperative period might have the potential to improve neurodevelopmental outcomes in term CHD.

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Section: Discussionmentioning

confidence: 99%

mentioning

confidence: 99%

Clinical Factors Associated with Cerebral Metabolism in Term Neonates with Congenital Heart Disease

Harbison

Votava‐Smith

Castillo

et al. 2017

The Journal of Pediatrics

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“…Congenital heart defect (CHD) is a structural abnormality of the heart that develops before birth, affecting approximately 1% of live births and is a major source of pediatric morbidity and mortality especially within the first year of life (1–6). The past five decades have witnessed spectacular advancements in diagnostic and surgical interventions in pediatric cardiology (1, 2).…”

Section: Introductionmentioning

confidence: 99%

A Path to Implement Precision Child Health Cardiovascular Medicine

Touma

Reemtsen

Halnon

et al. 2017

Front. Cardiovasc. Med.

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Congenital heart defects (CHDs) affect approximately 1% of live births and are a major source of childhood morbidity and mortality even in countries with advanced healthcare systems. Along with phenotypic heterogeneity, the underlying etiology of CHDs is multifactorial, involving genetic, epigenetic, and/or environmental contributors. Clear dissection of the underlying mechanism is a powerful step to establish individualized therapies. However, the majority of CHDs are yet to be clearly diagnosed for the underlying genetic and environmental factors, and even less with effective therapies. Although the survival rate for CHDs is steadily improving, there is still a significant unmet need for refining diagnostic precision and establishing targeted therapies to optimize life quality and to minimize future complications. In particular, proper identification of disease associated genetic variants in humans has been challenging, and this greatly impedes our ability to delineate gene–environment interactions that contribute to the pathogenesis of CHDs. Implementing a systematic multileveled approach can establish a continuum from phenotypic characterization in the clinic to molecular dissection using combined next-generation sequencing platforms and validation studies in suitable models at the bench. Key elements necessary to advance the field are: first, proper delineation of the phenotypic spectrum of CHDs; second, defining the molecular genotype/phenotype by combining whole-exome sequencing and transcriptome analysis; third, integration of phenotypic, genotypic, and molecular datasets to identify molecular network contributing to CHDs; fourth, generation of relevant disease models and multileveled experimental investigations. In order to achieve all these goals, access to high-quality biological specimens from well-defined patient cohorts is a crucial step. Therefore, establishing a CHD BioCore is an essential infrastructure and a critical step on the path toward precision child health cardiovascular medicine.

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“…Indeed, peak oxygen uptake may be the most important prognostic parameter for survival in patients with CHD (19–21) but it is controversial whether that importance already exists in children with CHD. The idea behind is the changing landscape of older patients with CHD that will develop acquired cardiovascular disease, such as hypertension and hyperlipidemia (42, 43). From the perspective of primary prevention, it is therefore more important to shape PA behavior early in life that yield health benefits later in life.…”

Section: Discussionmentioning

confidence: 99%

Web-Based Motor Intervention to Increase Health-Related Physical Fitness in Children With Congenital Heart Disease: A Study Protocol

et al. 2018

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Objective: Exercise interventions are underutilized in children with congenital heart disease (CHD) especially when the primary outcome is not peak oxygen uptake. Most of the studies are restricted to a low sample size and proximity of the patients to the study centers. Now eHealth approaches bear a promising but also challenging opportunity to transmit such intervention programs to participants, and check progress and compliance from remote. This study will aim to improve health-related physical fitness (HRPF) with a 24 weeks web-based exercise intervention.Methods and Design: The current study is planned as a randomized control trial (RCT) with a crossover design and the aim to improve functional outcome measures. It also estimates adherence and feasibility in patients with CHD in this web-based exercise/motor intervention over 24 weeks. Primary outcome will be the improvement of HRPF. Secondary outcomes are, functional and structural arterial stiffness measures and health-related quality of life. Thus, 70 children from 10 to 18 years with CHD of moderate and complex severity will be recruited and allocated randomly 1:1 in two study arms after baseline testing for their HRPF, arterial stiffness measures and health-related quality of life. For 24 weeks, participants in the intervention arm will receive three weekly exercise video clips of 20 min each. Every video clip comprises 20 child-oriented exercises which have to be executed for 30 s followed by a recovery period of 30 s. Each session will start with 3–4 warming-up exercises, followed by 10–12 strength and flexibility exercises, and ending with 3–4 min of cool down or stretching tasks. Continuous video clips will be streamed from a web-based e-Learning platform. The participant simply has to imitate the execution and follow some short advices. After each session, a brief online survey will be conducted to assess perceived exertion and feasibility.Discussion: The study will help to determine the efficacy and applicability of a web-based exercise intervention in children with CHD in regard to functional outcome measures. In addition, it will outline the effectiveness of remote monitoring, which provides a cost effective approach to reach patients with CHD that are low in prevalence and often do not live in close proximity to their tertiary center.Trial Registration: https://ClinicalTrials.gov Identifier: NCT03488797.

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Trends in Congenital Heart Disease

Cited by 240 publications

References 151 publications

Clinical Factors Associated with Cerebral Metabolism in Term Neonates with Congenital Heart Disease

Clinical Factors Associated with Cerebral Metabolism in Term Neonates with Congenital Heart Disease

A Path to Implement Precision Child Health Cardiovascular Medicine

Web-Based Motor Intervention to Increase Health-Related Physical Fitness in Children With Congenital Heart Disease: A Study Protocol

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