Exploring early life social and executive function development in infants and risk for autism: a prospective cohort study protocol of NICU graduates and infants at risk for cerebral palsy.

BACKGROUND: Delays in early social and executive function are predictive of later developmental delays and eventual neurodevelopmental diagnoses. There is limited research examining such markers in the first year of life. High-risk infant groups commonly present with a range of neurodevelopmental challenges, including social and executive function delays, and show higher rates of autism diagnoses later in life. For example, it has been estimated that up to 30% of infants diagnosed with cerebral palsy (CP) will go on to be diagnosed with autism later in life. METHODS: This article presents a protocol of a prospective longitudinal study. The primary aim of this study is to identify early life markers of delay in social and executive function in high-risk infants at the earliest point in time, and to explore how these markers may relate to the increased risk for social and executive delay, and risk of autism, later in life. High-risk infants will include Neonatal Intensive Care Unit (NICU) graduates, who are most commonly admitted for premature birth and/or cardiovascular problems. In addition, we will include infants with, or at risk for, CP. This prospective study will recruit 100 high-risk infants at the age of 3-12 months old and will track social and executive function across the first 2 years of their life, when infants are 3-7, 8-12, 18 and 24 months old. A multi-modal approach will be adopted by tracking the early development of social and executive function using behavioural, neurobiological, and caregiver-reported everyday functioning markers. Data will be analysed to assess the relationship between the early markers, measured from as early as 3-7 months of age, and the social and executive function as well as the autism outcomes measured at 24 months. DISCUSSION: This study has the potential to promote the earliest detection and intervention opportunities for social and executive function difficulties as well as risk for autism in NICU graduates and/or infants with, or at risk for, CP. The findings of this study will also expand our understanding of the early emergence of autism across a wider range of at-risk groups.

Emotion Regulation Is Associated with Anxiety, Depression and Stress in Adults with Cerebral Palsy.

Emotion regulation difficulties are associated with many neurological conditions and negatively impact daily function. Yet little is known about emotion regulation in adults with cerebral palsy (CP). Our aim was to investigate emotion regulation in adults with CP and its relationship with condition-related and/or socio-demographic factors. In a cross-sectional study of adults with CP, participants completed a survey containing the Difficulties in Emotion Regulation Scale (DERS), Depression Anxiety and Stress Scale-21 (DASS-21), and socio-demographic and condition-related questions. Descriptive statistics, chi-squared and Mann-Whitney tests were performed. Of the 42 adults with CP (x31.5 years, SD13.5) that were tested, 38 had within normal limits DERS total scores; however, a significantly higher proportion of participants experienced elevated scores (i.e., more difficulties with emotion regulation) than would be expected in the general population across five of the six DERs subdomains. Moderate-extremely severe depression and anxiety symptoms were reported by 33% and 60% of participants, respectively. The DERS total scores for participants with elevated depression, anxiety, and stress scores were significantly higher than the DERS totals score for those without elevated depression, anxiety, and stress scores. DERS and DASS-21 scores did not differ significantly by condition-related nor socio-demographic characteristics. In conclusion, emotion regulation difficulties were associated with elevated symptoms of depression and anxiety, which were overrepresented in the adults with CP participating in this study.

Proportion of Infant Neurodevelopment Trials Reporting a Null Finding: A Systematic Review.

CONTEXT: Discovering new interventions to improve neurodevelopmental outcomes is a priority; however, clinical trials are challenging and methodological issues may impact the interpretation of intervention efficacy. OBJECTIVES: Characterize the proportion of infant neurodevelopment trials reporting a null finding and identify features that may contribute to a null result. DATA SOURCES: The Cochrane library, Medline, Embase, and CINAHL databases. STUDY SELECTION: Randomized controlled trials recruiting infants aged <6 months comparing any "infant-directed" intervention against standard care, placebo, or another intervention. Neurodevelopment assessed as the primary outcome between 12 months and 10 years of age using a defined list of tools. DATA EXTRACTION: Two reviewers independently extracted data and assessed quality of included studies. RESULTS: Of n = 1283 records screened, 21 studies (from 20 reports) were included. Of 18 superiority studies, >70% reported a null finding. Features were identified that may have contributed to the high proportion of null findings, including selection and timing of the primary outcome measure, anticipated effect size, sample size and power, and statistical analysis methodology and rigor. LIMITATIONS: Publication bias against null studies means the proportion of null findings is likely underestimated. Studies assessing neurodevelopment as a secondary or within a composite outcome were excluded. CONCLUSIONS: This review identified a high proportion of infant neurodevelopmental trials that produced a null finding and detected several methodological and design considerations which may have contributed. We make several recommendations for future trials, including more sophisticated approaches to trial design, outcome assessment, and analysis.

Program evaluation of an adapted PEERS® social skills program in young adults with autism spectrum disorder and/or mild intellectual impairment and social skills difficulties.

RATIONALE, AIMS AND OBJECTIVES: Social challenges are common for young adults with autism spectrum disorder (ASD) and/or mild intellectual impairment, yet few evidence-based interventions exist to address these challenges. PEERS®, the Program for the Education and Enrichment of Relational Skills, has been shown to be effective in improving the social skills of young adults with ASD; however, it requires a significant time commitment for parents of young adults. As such, this mixed-methods study aimed to investigate the experiences of young adults, parents and PEERS® social coaches participating in an adapted PEERS® program, and to evaluate its acceptability and efficacy. METHOD: Young adults with ASD and/or mild intellectual impairment participated in a 16-week PEERS® program. Parents and PEERS® social coaches attended fewer, condensed sessions, where they learnt program content to support the young adults' social skill development at home and in the community. Focus groups were conducted post intervention. Quantitative pre-post assessment using the Social and Emotional Loneliness Scale for Adults, the Test of Young Adult Social Skills Knowledge, and Quality of Socialization Questionnaire-Young Adults was completed by young adults. The Social Responsiveness Scale Second Edition was completed by young adults and their parents. RESULT: Qualitative results revealed that, taken together, young adults, parents and PEERS® social coaches all felt that the adapted PEERS® program was 'challenging, but worth it'. The program was acceptable with a 93% attendance rate across all sessions. Whilst young adults' perceptions of their own social functioning did not change post-intervention, their knowledge of social skills content improved significantly (p < 0.05). Parent perceptions of young adults' social responsiveness also improved (p < 0.05). CONCLUSIONS: Social skill knowledge, social responsiveness, and social engagement improved significantly following the completion of the adapted PEERS® program. It was deemed acceptable and worthwhile by young adults, their parents and PEERS® social coaches.

Evaluation of a Modified Mindfulness-Based Stress Reduction Intervention for Adults with Cerebral Palsy and Anxiety and/or Emotion Regulation Difficulties-A Randomised Control Trial.

Mindfulness-Based Stress Reduction (MBSR) has not yet been evaluated for people with cerebral palsy (CP). The aims of this randomised control trial were to investigate whether a modified telehealth MBSR program could improve mindfulness and reduce depression, anxiety, and emotion regulation difficulties among adults with CP with elevated anxiety and/or emotional regulation difficulties. Participants (n = 31) with elevated anxiety and/or emotion regulation difficulties and no/mild intellectual impairment were randomised to a modified telehealth MBSR program (90 min weekly, 9 weeks) group or a wait-list group. Measurements were collected prior to (T1), after (T2), and 8 weeks post-intervention (T3). The primary outcome was the mean between-group difference in the change in Cognitive and Affective Mindfulness Scale-R (CAMS-R) scores in T1-T2. The secondary outcomes included mean within-group differences over time for the CAMS-R total scores, Depression Anxiety and Stress Scale-21 subscales, and Difficulties in Emotion Regulation Scale (DERS) total t-score. We found no statistically significant between-group difference in mean change in mindfulness scores for T1-T2 (primary outcome). Secondary outcomes: The MBSR intervention group had improved CAMS-R scores with respect to T1-T2 and T1-T3; improved mean scores for Depression and Stress subscales for T1-T2; and improved DERS t-scores for T1-T2 and T1-T3. In conclusion, this study found no significant between-group difference for the primary outcome of mindfulness. The MBSR program was successfully modified for adults with CP and was effective in improving depression, stress, and emotion regulation. ACTRN12621000960853.

Factors associated with discharge from hospital to residential aged care for younger people with neuropsychiatric disorders: an exploratory case-control study in New South Wales, Australia.

OBJECTIVES: To examine the sociodemographic and diagnostic factors associated with a discharge from hospital to residential aged care (RAC) for younger people (aged 15-64 years) with neuropsychiatric disorders. DESIGN: An exploratory case-control study using a historic cohort of people with neuropsychiatric disorders. Cases were people transferred to RAC on hospital discharge during the study period. Controls were people not transferred to RAC on discharge during the study period. SETTING: Public and private hospital admissions in New South Wales (NSW), Australia. PARTICIPANTS: People aged 15-64 years with a neuropsychiatric disorder hospitalised in NSW between July 2002 and June 2015 (n=5 16 469). OUTCOME MEASURES: The main outcome was transferred to RAC on discharge from hospital. We calculated ORs for sociodemographic and diagnostic factors to determine factors that may impact discharge to RAC. RESULTS: During the period of data capture, 4406 people were discharged from hospitals to RAC. Discharge to RAC was most strongly associated with diagnoses of progressive neurological and cognitive disorders. Acute precipitants of RAC transfer included a broad range of conditions and injuries (eg, Wernicke's encephalopathy, stroke, falls) in the context of issues such as older age, not being partnered (married or de facto), living in areas of lower socioeconomic status, functional issues and the need for palliative care. CONCLUSIONS: There are multiple intersecting and interacting pathways culminating in discharge from hospital to RAC among younger people with neuropsychiatric disorders. Improved capacity for interdisciplinary home care and alternative housing and support options for people with high support needs are required.

Are We Getting It Right? A Scoping Review of Outcomes Reported in Cell Therapy Clinical Studies for Cerebral Palsy.

Cell therapies are an emergent treatment for cerebral palsy (CP) with promising evidence demonstrating efficacy for improving gross motor function. However, families value improvements in a range of domains following intervention and the non-motor symptoms, comorbidities and complications of CP can potentially be targeted by cell therapies. We conducted a scoping review to describe all outcomes that have been reported in cell therapy studies for CP to date, and to examine what instruments were used to capture these. Through a systematic search we identified 54 studies comprising 2066 participants that were treated with a range of cell therapy interventions. We categorized the reported 53 unique outcome instruments and additional descriptive measures into 10 categories and 12 sub-categories. Movement and Posture was the most frequently reported outcome category, followed by Safety, however Quality of Life, and various prevalent comorbidities and complications of CP were infrequently reported. Notably, many outcome instruments used do not have evaluative properties and thus are not suitable for measuring change following intervention. We provide a number of recommendations to ensure that future trials generate high-quality outcome data that is aligned with the priorities of the CP community.

A validation and acceptability study of cognitive testing using switch and eye-gaze control technologies for children with motor and speech impairments: A protocol paper.

Despite the importance of knowing the cognitive capabilities of children with neurodevelopmental conditions, less than one-third of children with cerebral palsy participate in standardized assessments. Globally, approximately 50% of people with cerebral palsy have an intellectual disability and there is significant risk for domain-specific cognitive impairments for the majority of people with cerebral palsy. However, standardized cognitive assessment tools are not accessible to many children with cerebral palsy, as they require manual manipulation of objects, verbal response and/or speeded response. As such, standardised assessment may result in an underestimation of abilities for children with significant motor and/or speech impairment. The overall aim of the project is to examine and compare the psychometric properties of standardised cognitive assessment tools that have been accommodated for use with either a switch device or eye-gaze control technologies, with the specific aims to: (1) Examine the psychometric properties (measurement agreement and validity) of accommodated assessment tools by comparing the performance of typically developing children on six cognitive assessment tools administered via standardised versus accommodated (switch or eye-gaze control) administration; (2) Describe and compare the performance and user experience of children with cerebral palsy on six accommodated cognitive assessments administered via switch or eye-gaze control technologies. Secondary aims are to: (1) Describe the completion rates and time to complete assessments of participants in each group; (2) Within the group with cerebral palsy, examine the effects of condition-specific characteristics (type of cerebral palsy, functional levels, and pain) and demographics (age, socio-demographic) on participation. This protocol paper describes a two-phase validation and acceptability study that utilizes a mixed-model design. This study will collect concurrent data from 80 typically developing children and 40 children with cerebral palsy, who use switch or eye-gaze control technology as alternate access communication methods. The set of instruments will measure receptive vocabulary, fluid reasoning, sustained attention, vision perception, visuospatial working memory and executive functions. Data analyses will be conducted using SPSS v. 25 and R v 4.1.0. SPSS Sample Power 3 was used for power computation and allows for a 10% drop out rate. Quantitative descriptive statistics, measurement agreement data plotting, bivariate and multiple regressions analysis will be conducted using appropriate methods.

Accommodations to cognitive assessment for a child with dyskinetic cerebral palsy: case study.

PURPOSE: Despite the importance of having knowledge about a child's cognitive functioning, less than one-third of children with cerebral palsy (CP) are formally assessed. Consequently, the cognitive strengths of many children with severe CP may be underestimated or go overlooked. This case study aimed to test accommodations to cognitive assessment administration procedures to enable switch access. METHODS: A 9-year-old boy with dyskinetic CP tested a suite of cognitive assessments with accommodations for single switch access and measures of user experience. The cognitive assessment included: receptive vocabulary; non-verbal reasoning; sustained attention; executive functions of problem solving and shifting; and visual perception skills. RESULTS: The participant's ability to independently undertake assessment on the receptive vocabulary, non-verbal reasoning and the sustained attention measures indicates that accommodations made for single switch access were appropriate. Assessment took 1-2 h longer than expected for a typically developing child via standardised administration procedures, but was considerably faster than expected if undertaken via low-tech partner assisted scanning. Accessibility barriers continued to be present for the executive function and visual perception measures. Overall, the user experience was positive, both in regards to usability and cognitive load. CONCLUSIONS: This case study provides emerging data for usability and accessibility of accommodations to a battery of cognitive assessment tasks. Further research is needed to devise appropriate accommodations for executive function and visual perception measures and to determine whether the accommodations are accessible more generally for children with motor and/or speech impairments. Implications for rehabilitationAccommodations can be successfully made to receptive vocabulary, non-verbal reasoning and sustained attention assessment administration procedures for switch technologies.Cognitive assessment with switch accommodations takes considerably longer to complete than standardised administration estimates for a typically developing child. Assessment may need to be scheduled over more than one session.User experience, including usability and cognitive load, of accommodations was positive.

The Role of Antenatal and Postnatal Maternal Bonding in Infant Development.

OBJECTIVE: The affectional bond experienced by a mother toward her developing fetus/infant has been theorized to be a critical factor in determining infant developmental outcomes; yet there remains a paucity of research in this area, and a lack of high-quality longitudinal studies. This study aimed to examine the extent to which mother-to-infant bonding predicted infant development in a multi-wave longitudinal pregnancy cohort study (N = 1,347). METHOD: Self-reported bonding was assessed using the Maternal Antenatal Attachment Scale at each trimester, and the Maternal Postnatal Attachment Scale at 8 weeks and 12 months postpartum. Infant development was assessed using the Bayley Scales of Infant and Toddler Development, Third Edition (Bayley-III) at 12 months. RESULTS: Bonding predicted indicators of infant social-affective development, including social-emotional, behavioral, and temperamental outcomes. Effect sizes ranged from small to moderate, increasing over the perinatal period (ß = 0.11-0.27). Very small effects were also identified in the relationship between bonding and cognitive, language, and motor development (ß = 0.06-0.08). CONCLUSION: Findings suggest that a mother's perceived emotional connection with her child plays a role in predicting social-affective outcomes; prediction may not extend to other domains of infant development. Maternal bonding may therefore be a potentially modifiable predictor of infant social-affective outcomes, offering important considerations for preventive intervention.

Is the search for cerebral palsy 'cures' a reasonable and appropriate goal in the 2020s?

In the field of disability research and advocacy, the notion of 'cures' is contentious. Cerebral palsy (CP) is no exception. In this narrative review, we combine perspectives gained during community consultation undertaken for the Australian and New Zealand Cerebral Palsy Strategy, 2020 with those published in the scientific and grey literature to understand whether 'cures for CP' is a reasonable and appropriate goal. We frame these perspectives through the lens of several ethical principles central to the discussion. These include maintaining hope while also being realistic, sensitivity to sharply different viewpoints amongst people with disability and their families, and responding to community priorities, societal attitudes, and identity. Through this exploration of the literature and perspectives, we arrived at a definition of 'cures for CP' that is pluralized and focuses on functional improvement and/or symptom reduction whilst still acknowledging the potential for neural repair/regeneration strategies.

Maternal bonding, negative affect, and infant social-emotional development: A prospective cohort study.

BACKGROUND: Theoretical perspectives and empirical evidence suggest that maternal bonding and negative affect play a role in supporting infant social-emotional development (Branjerdporn et al., 2017; Kingston et al., 2012; O'Donnell et al., 2014; Van den Bergh et al., 2017). However, the complex pathways likely to exist between these constructs remain unclear, with limited research examining the temporal and potentially bi-directional associations between maternal bonding and negative affect across pregnancy and infancy. METHODS: The interrelationships between maternal bonding, negative affect, and infant social-emotional development were examined using multi-wave perinatal data from an Australian cohort study (N = 1,579). Self-reported bonding and negative affect were assessed at each trimester, and 8 weeks and 12 months postpartum. The Bayley-III social-emotional scale was administered at age 12 months. RESULTS: Results revealed strong continuities in bonding and negative affect across pregnancy and postpartum. Small associations (ß = -.10 to -.20) existed between maternal negative affect during pregnancy and poor early bonding. Higher postnatal maternal bonding predicted infant social-emotional development (ß = .17). LIMITATIONS: Limitations include a somewhat advantaged and predominantly Anglo-Saxon sample of families, and the use of self-report measures (though with strong psychometric properties). These limitations should be considered when interpreting the study findings. CONCLUSIONS: Maternal bonding and negative affect are interrelated yet unique constructs, with suggested developmental interplay between mother-to-infant bonding and infant social-affective development.

Early Life Parechovirus Infection Neurodevelopmental Outcomes at 3 Years: A Cohort Study.

OBJECTIVE: To investigate the long-term developmental and behavioral outcomes in an established cohort of children hospitalized as infants with human parechovirus (HPeV) infection and sepsis-like illness. STUDY DESIGN: The HPeV cohort was composed of children 3 years of age after HPeV infection and hospitalization in early infancy that occurred during a well-documented HPeV genotype 3 outbreak in Australia. We assessed neurodevelopmental and behavioral outcomes using the Bayley Scales of Infant and Toddler Development-III and the Child Behavior Checklist. We compared their outcomes with a subsample of healthy control infants drawn from the independently sampled Triple B Pregnancy Cohort Study. RESULTS: Fifty children, with a mean age of 41 months, were followed for 3 years after hospital admission with HPeV infection. There were 47 children whose original illness was fever without source or sepsis-like illness and 3 who had encephalitis. All children in the HPeV cohort showed age-specific development within the population normal range on the Bayley Scales of Infant and Toddler Development-III. There was no difference in developmental attainment compared with 107 healthy control infants after adjusting for measured confounders. The HPeV cohort showed higher average scores on the Child Behavior Checklist and a higher frequency of clinical range scores compared with healthy controls. CONCLUSIONS: Although HPeV sepsis-like illness did not result in neurodevelopmental delay at 3 years of age, it was associated with increased behavioral problems compared with healthy controls. The behavioral problems reached a clinical threshold in a minority of children. Results inform clinical management and planning for children after severe HPeV infection in infancy.

Correction to: Prenatal alcohol exposure and infant gross motor development: a prospective cohort study.

Following publication of the original article [1], the authors opted to revise the first paragraph of the section "Characteristics associated with maternal drinking in pregnancy". Below is the updated version.

Prenatal alcohol exposure and infant gross motor development: a prospective cohort study.

BACKGROUND: Maternal alcohol consumption in pregnancy may have adverse effects on child gross motor (GM) development. There have been few human studies on this topic, particularly ones examining low exposure. This study examined the association between prenatal alcohol exposure (PAE) and infant GM development at 12-months of age. METHODS: Participants were 1324 women recruited from antenatal clinics in Sydney and Perth, Australia. Maternal and paternal alcohol use was assessed in pregnancy via interview; offspring GM development was measured at 12-months with the Bayley Scales of Infant Development (BSID-III). RESULTS: Any alcohol use in pregnancy was common: 56.1%, of pregnant women drank early in Trimester one (0-6 weeks), however this reduced to 27.9% on average thereafter and at predominantly low levels. However, infant BSID GM scale scores were not found to differ significantly as a function of PAE in the first 6-weeks (low, moderate, binge or heavy PAE), nor with low PAE across pregnancy. CONCLUSIONS: We found no evidence to suggest that low PAE is associated with measurable impairment in infant GM development at 12-months. Further research is needed to examine potential PAE impacts on GM development in heavier exposure groups and through the childhood years when subtle GM deficits may be more detectable.

Effectiveness of paediatric occupational therapy for children with disabilities: A systematic review.

INTRODUCTION: Paediatric occupational therapy seeks to improve children's engagement and participation in life roles. A wide variety of intervention approaches exist. Our aim was to summarise the best-available intervention evidence for children with disabilities, to assist families and therapists choose effective care. METHODS: We conducted a systematic review (SR) using the Cochrane methodology, and reported findings according to PRISMA. CINAHL, Cochrane Library, MEDLINE, OTSeeker, PEDro, PsycINFO were searched. Two independent reviewers: (i) determined whether studies met inclusion: SR or randomised controlled trial (RCT); an occupational therapy intervention for children with a disability; (ii) categorised interventions based on name, core components and diagnostic population; (iii) rated quality of evidence and determined the strength of recommendation using GRADE criteria; and (iv) made recommendations using the Evidence Alert Traffic Light System. RESULTS: 129 articles met inclusion (n = 75 (58%) SRs; n = 54 (42%)) RCTs, measuring the effectiveness of 52 interventions, across 22 diagnoses, enabling analysis of 135 intervention indications. Thirty percent of the indications assessed (n = 40/135) were graded 'do it' (Green Go); 56% (75/135) 'probably do it' (Yellow Measure); 10% (n = 14/135) 'probably don't do it' (Yellow Measure); and 4% (n = 6/135) 'don't do it' (Red Stop). Green lights were: Behavioural Interventions; Bimanual; Coaching; Cognitive Cog-Fun & CAPS; CO-OP; CIMT; CIMT plus Bimanual; Context-Focused; Ditto; Early Intervention (ABA, Developmental Care); Family Centred Care; Feeding interventions; Goal Directed Training; Handwriting Task-Specific Practice; Home Programs; Joint Attention; Mental Health Interventions; occupational therapy after toxin; Kinesiotape; Pain Management; Parent Education; PECS; Positioning; Pressure Care; Social Skills Training; Treadmill Training and Weight Loss 'Mighty Moves'. CONCLUSION: Evidence supports 40 intervention indications, with the greatest number at the activities-level of the International Classification of Function. Yellow light interventions should be accompanied by a sensitive outcome measure to monitor progress and red light interventions could be discontinued because effective alternatives existed.