Primary Objective: To investigate motor developmental outcome at school age of children with CHD compared to typically developing peers.Secondary Objective(s): - To investigate neurodevelopmental outcom eat school age on several developmental…
ID
Source
Brief title
Condition
- Congenital cardiac disorders
- Cardiac and vascular disorders congenital
Synonym
Research involving
Sponsors and support
Intervention
Outcome measures
Primary outcome
The study parameters are the scores obtained through the assessments and
questionnaires to assess developmental disorders. The scores of the MABC-2 are
dichotomized as *at risk of DCD* (<=16th percentile) or not. As a secondary
endpoint also the raw MABC-2 scores will be used. For all other study
parameters we will calculate the total scores (except for neurological
condition). These total scores will be used as continuous variables in our
primary analyses. In some of the exploratory analyses the total scores are also
dichotomized, by using valid cut-off points as mentioned in the corresponding
manuals as well.
Main study parameter/endpoint:
The Movement Assessment Battery for Children - second edition (MABC-2;
Henderson & Sugden, 2007): a valid, reliable, and easily administered motor
assessment for children aged 3 to 16 years which is developed to assess DCD. It
measures competences in three areas of motor performance; manual dexterity,
aiming and catching and balance. The MABC-2 is a composite of two complementary
assessments; a checklist and a performance test. In the current study only the
performance test is used. The dichotomized total score (composite of the three
areas) is the primary outcome parameter in our analysis. A score <=16th
percentile means *at risk of DCD*, as mentioned in the corresponding manuals.
Secondary outcome
Other primary study parameters are:
Tests:
- Minor Neurological Dysfunction assessment (MND; Hadders-Algra, 2010):
neurological age-specific examination to detect minor neurological dysfunction.
The assessment includes traditional neurological items, such as the evaluation
of posture in various positions, muscle tone and reflexes, as well as
developmental items, i.e. the items dealing with coordination, fine
manipulative abilities and associated movements. The assessment can distinguish
between two basic forms of MND: simple MND (s-MND) and complex MND (c-MND).
- Wechsler Intelligence Scale for Children V (WISC-V-NL): IQ test for children
aged 6-16 years with sub-indexes on verbal understanding, spatial awareness,
fluid reasoning, random access memory and processing speed (Hendriks and
Ruiter, 2017).
- Personal Well-being Index: School Children (PWI-SC): an instrument which
measures personal well-being in school-aged children with 7 questions on
standard of living, personal health, achievement in life, personal
relationships, personal safety, feeling part of the community, and future
security (Cummins and Lau, 2005).
Parental questionnaires:
- Child Behavior Checklist (CBCL): Questionnaire to screen for behavioural
problems, ADHD, anxiety, ASD and depression in children aged 6 to 18 years
(Achenbach, 2006)
- Behavior Rating Inventory of Executive Function (BRIEF): Questionnaire to
screen for executive functioning in 5- to 18 year olds (Huizinga and Smidts
2020).
- Social Responsiveness Scale (SRS-2): Questionnaire to screen for social
shortcomings associated with ASD (Constantino & Gruber 2012, Roeyers et al.
2015).
- Conners* Rating Scale Revised (CRS-R): Questionnaire to screen for ADHD
(Conners 1997).
- The Developmental Coordination Disorder Questionnaire-revised (DCDQ): to
assist in the identification of DCD in children (Wilson et al., 2009, Dutch
translation; Schoemaker et al., 2006).
- General questionnaire on presence of illnesses, medication use and visual and
auditory problems, known developmental problems and history of related therapy,
current school of the children, socio-economic background, psychiatric and
developmental disorders in the parents. For the children with CHD number of
cardiac interventions/surgeries and actual medical information such as latest
ECG, ultrasound data and saturation will be collected from their electronic
patient files.
Clinical data:
For the children with congenital heart disease we will collect medical history
and current medical status, including ECG and ultrasound results. We will also
use the previously collected data on prenatal cerebral perfusion and postnatal
cerebral oxygenation in order to correlate this with the current neurological
results.
Background summary
Children with severe congenital heart disease (CHD) are at increased risk for a
different neurodevelopmental development in comparison to healthy peers. Think
of cognitive difficulties, problems with executive and adaptive functioning,
motor difficulties and language problems. In addition, CHD has been associated
with more externalizing and internalizing behavioural problems, attention
deficit hyperactivity disorder (ADHD) and autism spectrum disorder (ASD)
(Mebius et al. 2017, Huisenga et al. 2020). These developmental problems are
related to altered cerebral oxygenation and circulation, already in utero,
causing suboptimal brain development. (Sun L et al., 2015). Delayed neonatal
brain development is related to less favorable early motor outcomes (Stegeman R
et al., 2022). Early detection is key to optimizing developmental outcomes
through early intervention at young age when brain plasticity is highest (Kolb
et al. 2017). In addition, early recognition of developmental problems is
important for children with CHD and their parents for getting the appropriate
guidance and support, knowing that part of the developmental problems will
persist into adulthood (Mebius et al. 2017).
This research project consists of a follow-up project to the study by Mebius et
al. from 2020, performed at the UMCG. This study investigated the association
between prenatal cerebral perfusion, postnatal cerebral oxygenation and
neurological outcome at the age of 3 months in children with CHD. Inclusion
took place between May 2014 and August 2016, with the inclusion criteria being
the presence of a prenatally diagnosed heart condition with NICU indication.
Forty-five fetuses were eligible in this timeframe. Postnatal exclusion
criteria were prematurity <36 weeks and/or serious chromosomal, genetic or
structural defects. Based on these exclusion criteria nine neonates were
excluded. In the neonatal period, seven neonates died. This resulted in a final
study population of 29 neonates with follow-up until the age of 3 months. Tests
included prenatal doppler imaging of the middle cerebral artery and the
umbilical artery, resulting in a cerebroplacental ratio as a proxy for prenatal
cerebral perfusion. Furthermore, postnatal Near-InfraRed Spectroscopy (NIRS),
and assessment of the general movements at the age of 7 days and 3 months were
performed. In addition, the Motor Optimality Score (MOS) was used to assess the
motor repertoire in detail. Main result was that prenatal cerebral perfusion
was found to be associated with neurologic outcome at the age of three months.
In addition, neonates with both prenatal and postnatal perfusion or oxygenation
problems had the highest risk of having a deviant neurological condition.
To the best of our knowledge, no research has been done yet on the association
between prenatal cerebral perfusion, postnatal cerebral oxygenation and
neurodevelopmental outcome at school age in children with severe CHD. The UMCG
CHD study group provides an excellent opportunity to investigate this. The
current study therefore consists of a follow-up study in which the CHD-group
will be re-invited for neurodevelopmental assessments to associate previous
cerebral perfusion and oxygenation with cognitive development, neurological
condition, motor development, adaptive and psychosocial functioning and signs
of ASD and ADHD. An age and sex matched control group of typically developing
children will be added for comparison.
We think the results of this study will add to the quality of care for children
with CHD and their parents and that it will provide more insight in the
pathophysiological background of brain damage and developmental problems in
this specific group of patients.
Study objective
Primary Objective:
To investigate motor developmental outcome at school age of children with CHD
compared to typically developing peers.
Secondary Objective(s):
- To investigate neurodevelopmental outcom eat school age on several
developmental domains other than motor development of children with CHD
compared to typically developing peers.
- To investigate associations between prenatal cerebral perfusion, postnatal
cerebral oxygenation and neurodevelopmental outcome at school age of children
with CHD.
Study design
Study design
This study is a longitudinal cohort study and is a follow-up study of the
previous project of Mebius et al. (Mebius et al. 2020) in which a group of 29
children with CHD was assessed pre- and postnatally until the age of 3 months.
For the current project these participants will be re-invited via mail and a
control group of healthy, age- and sex-matched typically developing children
will be recruited via social media and/or local primary schools.
Both groups will perform multiple assessments to investigate development, while
their parents fill in additional questionnaires. Tests will be performed at the
UMCG and if possible combined with regular check-ups at the cardiology
department. The test procedure will take maximum 3 hours, including a small
break in between. Children at this age, with or without developmental problems,
have shown to be able to endure such test durations, so we are confident this
will be achievable. Tests will be carried out by well-trained medical students
and recorded on video, allowing further supervision. Parents will be in the
same room as their child during the whole assessment and will be asked to fill
in a set of questionnaires on an Ipad.
Study duration
We aim to finish the study within 1.5 years, using the following time schedule.
The study will start in November 2024 and will be finished in March 2026.
Setting
The assessments will take place at the child-lab of the department of
Developmental Neurology in the University Medical Center Groningen. For the CHD
group the appointment will, if possible, be combined with the regular follow-up
visit at the paediatric cardiology department. Travel costs and parking costs
of the parents will be re-imbursed
Study burden and risks
In our opinion the burden of this study, namely a 3 hour visit to our hospital
with playful tests for the children and questionnaires for the parents, is very
much acceptable for the benefits of learning more about neurodevelopmental
problems in children with congenital heart disease with which we hope to be
able to provide better guidance.
Hanzeplein 1
Groningen 9713GZ
NL
Hanzeplein 1
Groningen 9713GZ
NL
Listed location countries
Age
Inclusion criteria
Inclusion patient group: all children who participated in the first study
Control: age- and sex-matched healthy and typically developing children who
attend regular school.
Exclusion criteria
Control group: congenital abnormalities, other medical conditions, special
education, ASD/ADHD/neurodevelopmental disorder
Design
Recruitment
Followed up by the following (possibly more current) registration
No registrations found.
Other (possibly less up-to-date) registrations in this register
No registrations found.
In other registers
| Register | ID |
|---|---|
| CCMO | NL87417.042.24 |