Adaptive behaviour, executive function and employment in adults with Down syndrome

Tomaszewski, Brianne; Fidler, Deborah J.; Talapatra, Devadrita; Riley, Karen

doi:10.1111/jir.12450

Cited by 58 publications

(43 citation statements)

References 51 publications

(135 reference statements)

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“…It is important to note that the previous studies involved samples of children with ASD and TD. As stated before, research (including this study) has consistently showed that social skills are a relative strength in people with DS, compared to other adaptive and intellectual skills 32,33 , while EF has been reported as a difficulty in people with DS 16,27,64 . Based on this, our hypothesis is that people with DS might tap other abilities to develop their social skills, including understanding and successfully resolving social situations.…”

Section: Discussionsupporting

confidence: 76%

Different abilities needed at home and school: The relation between executive function and adaptive behaviour in adolescents with Down syndrome

Sabat

Arango

Tassé

et al. 2020

Sci Rep

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Studies have shown that executive function abilities are related and have predictive power over adaptive behaviour in both typical and atypical populations. This study examined the relationship between executive functioning and adaptive behaviour in adolescents with Down syndrome, as it has not been studied before in this population. We propose and test a model of how each core EF (i.e., working memory, inhibition, and flexibility) contributes to each domain of AB (i.e., conceptual, social, and practical). We found that parent reported Conceptual skills were related to working memory, while teacher reported Conceptual and Practical skills were related to inhibition and flexibility. We hypothesise that these findings are related to the different requirements and expectations of the home and school environments: the more predictable home environment requires the adolescent to rely on working memory for his everyday activities, while the changing and challenging school environment requires the inhibition common behaviours and to flexibly change actions to be successful. Down syndrome (DS) is the most common chromosomal disorder, with a total mean prevalence (live, stillbirths and termination of pregnancies) of about 18.2 in 10,000 births 1. In most cases, DS is caused by an extra copy of all or part of chromosome 21 2 , and it is considered one of the leading genetic causes of intellectual disability (ID) 3 , as cognitive functioning and adaptive behaviour (AB) seem to be somehow affected by the condition 2,4. Regarding cognitive functioning, executive function (EF) appears as a set of skills that have consistently been shown to be different in people with DS when compared to typically developing (TD) children and adults 4-6. EF is a set of top-down skills used in the conscious control of attention, thoughts, and actions, when relying on our automatic processes would be unwise, insufficient, or impossible 7,8. The three core EFs include working memory (i.e., the ability to hold information in mind and mentally work with it 9), inhibition (i.e., the capacity to override automatic or impulsive responses, thoughts, or emotions, in order to act according to one's goals and/or what is appropriate for the situation 7,10), and cognitive flexibility (i.e., the ability that allows for change in perspectives, means to reach a goal, or the goal itself, in order to optimize resources and/or more effective use of the feedback from the environment 7,10). These abilities form the foundation from which higher order cognitive processes, such as reasoning, problem solving, and planning are built. This multidimensional approach to EF has been supported by studies in children 11,12 and adults 13. Studies that have explored EF in DS have reported that children and adolescents with DS have overall difficulties in EF, but with an age dependent profile of strengths and weaknesses across the different dimensions. Two studies with preschool children with DS 6,14 reported greater difficulties in working memory, planning, and inhibition, compa...

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Section: Discussionsupporting

confidence: 76%

Different abilities needed at home and school: The relation between executive function and adaptive behaviour in adolescents with Down syndrome

Sabat

Arango

Tassé

et al. 2020

Sci Rep

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“…[169][170][171][172][173][174] DS is characterized by learning, memory, and language abnormalities that result in mild to moderate intellectual disability. [175][176][177][178][179][180][181][182] Additional neurological hallmarks of DS include motor dysfunction and hypotonia 178,[183][184][185] and predisposition to AD pathology. 178,[186][187][188][189] In recent years, research in DS has been increasingly focused on the degeneration and dementia hallmarks of AD in DS.…”

Section: Modeling Neurological Symptoms and Behavioral Deficitsmentioning

confidence: 99%

Human Models Are Needed for Studying Human Neurodevelopmental Disorders

Zhao

Bhattacharyya

2018

The American Journal of Human Genetics

129

103

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The analysis of animal models of neurological disease has been instrumental in furthering our understanding of neurodevelopment and brain diseases. However, animal models are limited in revealing some of the most fundamental aspects of development, genetics, pathology, and disease mechanisms that are unique to humans. These shortcomings are exaggerated in disorders that affect the brain, where the most significant differences between humans and animal models exist, and could underscore failures in targeted therapeutic interventions in affected individuals. Human pluripotent stem cells have emerged as a much-needed model system for investigating human-specific biology and disease mechanisms. However, questions remain regarding whether these cell-culture-based models are sufficient or even necessary. In this review, we summarize human-specific features of neurodevelopment and the most common neurodevelopmental disorders, present discrepancies between animal models and human diseases, demonstrate how human stem cell models can provide meaningful information, and discuss the challenges that exist in our pursuit to understand distinctively human aspects of neurodevelopment and brain disease. This information argues for a more thoughtful approach to disease modeling through consideration of the valuable features and limitations of each model system, be they human or animal, to mimic disease characteristics.

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“…Ages and scores given as mean (SD; range). NR indicates “not reported”StudyIQ testScore type(s) providedParticipantsParticipant agesRaw scoresStandardised scoresFloor effectsTests not specifically designed for children and adolescents (most recent first) Lao et al, [26]PPVT-IVStandardised score and age-equivalent score5237.3 (6.6; 30–50)NR56.6 (17.2) standardised score; 8.19 (3.44) age-equivalent scoreNR Hartley et al [14]*PPVT-IVAge-equivalent score5837.6 (6.8; ≥ 30)NR8.10 (3.34)NR Tomaszewski et al [15]Stanford Binet 5th EdFull IQ score3125.9 (5.92)NR46.6 (9.1)NR Sinai et al [11]KBIT-2Raw scores for verbal and non-verbal subscales30 no dementia50.9 (4.83)Total, 23.17 (19.50; 3–63);Verbal, 16.37 (13.33; 1–47);Non-verbal, 6.8 (6.92; 0–15)NRVerbal, 0%;Non-verbal, 16.7%19 diagnosed or possible dementia55.6 (6.77)Total, 9.74 (11.06; 1–49);Verbal, 6.53 (7.16; 0–34);Non-verbal, 3.21 (4.16; 0–20)NRVerbal, 5.3%;Non-verbal, 21.1% Startin et al [16]KBIT-2Raw scores for verbal and non-verbal subscales; full IQ scores floor effects only130 aged 36+ years without dementia47.77 (7.01; 36–71)Verbal, 30.55 (17.47; 2–80);Non-verbal, 12.55 (6.57; 0–32);NRVerbal raw, 0%;Verbal IQ, 66.7%;Non-verbal raw, 6.7%;Non-verbal IQ, 39.4%51 aged 36+ years with dementia54.20 (6.95; 38–67)Verbal, 18.68 (13.77; 1–51);Non-verbal, 8.29 (6.45; 0–19)NRVerbal raw, 0%;Verbal IQ, 84.0%;Non-verbal raw, 16.7%;Non-verbal IQ, 62.5%124 aged 16–35 years25.24 (5.53; 16–35)Verbal, 35.03 (16.77; 2–82);Non-verbal, 14.98 (6.9; 0–32)NRVerbal raw, 0%;Verbal IQ, 50.8%;Non-verbal raw, 4.1%;Non-verbal IQ, 33.9% de Sola et al [17]KBIT (Spanish version)Full IQ score; combined verbal and non-verbal standardised KBIT score8623.3 (4.3; 16–34)NRFull IQ median, 41;Standardised KBIT score, 105 (17.8; 80–180)41.9% Ghezzo et al [8]WAIS-RFull IQ score; verbal IQ score; performance IQ score36 adults with DS (of a larger sam...…”

Section: Iq and General Ability Tests In People With Dsmentioning

confidence: 99%

Assessing general cognitive and adaptive abilities in adults with Down syndrome: a systematic review

Hamburg

Lowe

Startin

et al. 2019

J Neurodevelop Disord

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Background Measures of general cognitive and adaptive ability in adults with Down syndrome (DS) used by previous studies vary substantially. This review summarises the different ability measures used previously, focusing on tests of intelligence quotient (IQ) and adaptive behaviour (AB), and where possible examines floor effects and differences between DS subpopulations. We aimed to use information regarding existing measures to provide recommendations for individual researchers and the DS research community. Results Nineteen studies reporting IQ test data met inclusion for this review, with 17 different IQ tests used. Twelve of these IQ tests were used in only one study while five were used in two different studies. Eleven studies reporting AB test data met inclusion for this review, with seven different AB tests used. The only AB scales to be used by more than one study were the Vineland Adaptive Behaviour Scale (VABS; used by three studies) and the Vineland Adaptive Behavior Scale 2nd Edition (VABS-II; used by two studies). A variety of additional factors were identified which make comparison of test scores between studies problematic, including different score types provided between studies (e.g. raw scores compared to age-equivalent scores) and different participant inclusion criteria (e.g. whether individuals with cognitive decline were excluded). Floor effects were common for IQ tests (particularly for standardised test scores). Data exists to suggest that floor effects may be minimised by the use of raw test scores rather than standardised test scores. Raw scores may, therefore, be particularly useful in longitudinal studies to track change in cognitive ability over time. Conclusions Studies assessing general ability in adults with DS are likely to benefit from the use of both IQ and AB scales. The DS research community may benefit from the development of reporting standards for IQ and AB data, and from the sharing of raw study data enabling further in-depth investigation of issues highlighted by this review.

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Adaptive behaviour, executive function and employment in adults with Down syndrome

Cited by 58 publications

References 51 publications

Different abilities needed at home and school: The relation between executive function and adaptive behaviour in adolescents with Down syndrome

Different abilities needed at home and school: The relation between executive function and adaptive behaviour in adolescents with Down syndrome

Human Models Are Needed for Studying Human Neurodevelopmental Disorders

Assessing general cognitive and adaptive abilities in adults with Down syndrome: a systematic review

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