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.

The Knowledge Translation of Early Cerebral Palsy (KiTE CP) Study: Implementing Screening Among a High-Risk Prospective Cohort of Australian Infants.

OBJECTIVE: To describe the implementation of the international guidelines for the early diagnosis of cerebral palsy (CP) and engagement in the screening process in an Australian cohort of infants with neonatal risk factors for CP. STUDY DESIGN: Prospective cohort study of infants with neonatal risk factors recruited at <6 months corrected age from 11 sites in the states of Victoria, New South Wales, and Queensland, Australia. First, we implemented a multimodal knowledge translation strategy including barrier identification, technology integration, and special interest groups. Screening was implemented as follows: infants with clinical indications for neuroimaging underwent magnetic resonance imaging and/or cranial ultrasound. The Prechtl General Movements Assessment (GMA) was recorded clinically or using an app (Baby Moves). Infants with absent or abnormal fidgety movements on GMA videos were offered further assessment using the Hammersmith Infant Neurological Examination (HINE). Infants with atypical findings on 2/3 assessments met criteria for high risk of CP. RESULTS: Of the 597 infants (56% male) recruited, 95% (n = 565) received neuroimaging, 90% (n = 537) had scorable GMA videos (2% unscorable/8% no video), and 25% (n = 149) HINE. Overall, 19% of the cohort (n = 114/597) met criteria for high risk of CP, 57% (340/597) had at least 2 normal assessments (of neuroimaging, GMA or HINE), and 24% (n = 143/597) had insufficient assessments. CONCLUSIONS: Early CP screening was implemented across participating sites using a multimodal knowledge translation strategy. Although the COVID-19 pandemic affected recruitment rates, there was high engagement in the screening process. Reasons for engagement in early screening from parents and clinicians warrant further contextualization and investigation.

Use of the Motor Optimality Score-Revised (MOS-R) to predict neurodevelopmental outcomes in infants with congenital anomalies.

AIMS: To describe the Motor Optimality Score-Revised (MOS-R) in infants with congenital anomalies requiring major surgery in the neonatal period; and to determine the predictive validity of the MOS-R, including specific movement and postural patterns, for neurodevelopmental outcomes at 3 years of age. METHOD: A retrospective cohort study of 201 infants born with congenital anomalies requiring surgery in the neonatal period (mean gestational age 38.2 weeks, SD 2.2). MOS-R completed using the pre-recorded General Movements Assessment (GMA) videos taken at 12 to 14 weeks post-term age (mean 12.45, SD 1.54). Developmental outcomes were assessed at 3 years of age (38.13 months, SD 1.76) using the Bayley Scales of Infant and Toddler Development (3rd ed). RESULT: The mean score for the MOS-R was 21.85 (SD 5.16), with scores ranging from 6 to 28. Fifty-six infants (27.9 %) scored within the optimal range (25-28) with only 12 % demonstrating a normal movement character. A MOS-R total score of <21 was identified as the best performing cut-off to predict a mild, moderate or severe delay or CP diagnosis with sensitivity 0.39 (95 % CI: 0.25, 0.54) and specificity 0.86 (95 % CI: 0.80, 0.91), and an area under the ROC curve of 0.63. Outcome at 3 years was significantly associated with the MOS-R total (p < 0.01) and the subscales for observed movement patterns (p < 0.01) and age adequate repertoire (p = 0.02). CONCLUSION: The MOS-R may be an effective tool to use in addition to existing assessments to identify infants who are at risk of adverse developmental outcomes. Our study found that a MOS-R of <21 identified infants who would benefit from referral to early intervention.

The General Movements Motor Optimality Score in High-Risk Infants: A Systematic Scoping Review.

PURPOSE: The aim of this systematic scoping review was to explore the use of the motor optimality score in the fidgety movement period in clinical practice, and to investigate evidence for the motor optimality score in predicting neurodevelopmental outcomes. SUMMARY OF KEY POINTS: Thirty-seven studies, with 3662 infants, were included. Studies were conceptualized and charted into 4 categories based on the motor optimality score: prediction, outcome measure, descriptive, or psychometric properties. The most represented populations were preterm or low-birth-weight infants (16 studies), infants with cerebral palsy or neurological concerns (5 studies), and healthy or term-born infants (4 studies). CONCLUSION: The motor optimality score has the potential to add value to existing tools used to predict risk of adverse neurodevelopmental outcomes. Further research is needed regarding the reliability and validity of the motor optimality score to support increased use of this tool in clinical practice. What this adds to the evidence : The motor optimality score has potential to improve the prediction of adverse neurodevelopmental outcomes. Further research on validity and reliability of the motor optimality score is needed; however, a revised version, the motor optimality score-R (with accompanying manual) will likely contribute to more consistency in the reporting of the motor optimality score in future.

Neurodevelopmental outcomes in neonatal extracorporeal membrane oxygenation survivors: An institutional perspective.

AIM: To describe the neurodevelopmental outcomes following extracorporeal membrane oxygenation (ECMO) in early infancy. METHODS: Thirty-seven patients who had survived following ECMO support from 2008 to 2018 had their neurodevelopmental outcomes assessed and analysed using the Bayley Scales of Infant and Toddler Development. Developmental outcome was defined as impairment in any of the developmental domains of motor function, cognition and language with mild impairment being 1-2 standard deviations below the test mean, moderate being 2-3 standard deviations below and severe being greater than 3 standard deviations below. RESULTS: Of these 37 patients, the median age at admission to Paediatric Intensive Care Unit was 0.4 months (interquartile range 2.8 months) with all of the study patients having an underlying diagnosis of congenital cardiac disease and 37/40 (92.5%) ECMO runs occurring in the immediate post-operative period. Of the 29 patients who had had follow-up at 12 months of age or older, 3 (10.3%) had severe impairment, 4 (13.8%) had moderate impairment, 12 (41.3%) had mild impairment and 10 (34.5%) had no impairment. Gross motor function was most significantly impacted in 18/29 (62.1%) patients, of which 7/18 (38.9%) had severe impairment. This was followed by impairment of receptive language in 8/29 (27.6%) patients and expressive language in 6/29 (20.1%) patients. CONCLUSIONS: One in four infants undergoing ECMO treatment in early infancy has moderate to severe neurodevelopmental impairment. Gross motor and language are the most affected developmental domains.

The Evolution of an Interdisciplinary Developmental Round in a Surgical Neonatal Intensive Care Unit.

BACKGROUND: Developmentally supportive environments are known to improve medical outcomes for hospitalized neonates and are considered the overarching philosophy for practice in the neonatal setting. Developmental rounds are a strategy incorporated by multidisciplinary teams to support development within and beyond the neonatal unit. Typically, they consist of bedside consultations and individualized developmentally supportive recommendations for families and clinicians. Globally, the use of developmental rounds has been described since the early 1990s. They are viewed as a measure to counter some of the barriers to developmental care implementation while buffering against the effect of an intensive care admission. To date, their use in the surgical neonatal intensive care unit (sNICU) has been minimally reported in literature. PURPOSE: This article describes the focus and work of a developmental round team and strategy in the sNICU. METHOD: A retrospective audit of developmental round key performance criteria undertaken over a 4-year period (2015-2018). FINDINGS/RESULTS: More than 300 developmental consults and 2000 individualized developmental recommendations occurred annually. Parental presence during the developmental round increased by 10%, from 48% to 58%, during the audit period. IMPLICATIONS FOR PRACTICE/IMPLICATIONS FOR RESEARCH: Literature has supported the use of developmental round interventions; however, minimal data have been reported to date. This article provides retrospective audit data of a developmental round intervention in the sNICU with a focus on data over 4 years to highlight key areas, including the structure and process, recommended educational standards for team members, and parental engagement, as key markers for developmental round efficacy. Future research should focus on the link between the developmental round intervention and long-term neonatal outcomes.

Sensitivity and specificity of general movements assessment for detecting cerebral palsy in an Australian context: 2-year outcomes.

AIM: We previously reported sensitivity and specificity levels of the general movements assessment (GMA) to detect cerebral palsy (CP) at 1 year within a clinical setting and with the assistance of the New South Wales (NSW) Rater Network. The aim of this study was to determine whether similarly high levels of validity could be maintained in the same group at 2 years. METHODS: A prospective longitudinal cross-sectional study was conducted. GMA was blind-rated from conventional video by two independent certified raters, blinded to medical history. A third rater resolved disagreements. High-risk population screening for CP using the GMA during the fidgety period (12-20 weeks) was carried out in four neonatal intensive care units and one CP service over a 30-month period. Participants were 259 high-risk infants for the initial study. Multidisciplinary follow-up at 2-3 years included Bayley Scales of Infant Development and confirmed diagnosis of CP. Sensitivity and specificity values were calculated with true positives defined as a confirmed diagnosis of CP. RESULTS: At 2-3 years, 184 (71%) completed the follow-up assessment. GMA was normal for 134 (73%, low risk for CP), absent fidgety for 48 (26%, high risk for CP) and abnormal fidgety for 2 (1%, high risk for abnormal neurological disorder). Sensitivity for detecting CP was 97.6% (40/41) and specificity 95.7% (133/139). Sensitivity for detecting any abnormal outcome with absent/abnormal fidgety general movements (GMs) was 57.9% (44/76) and specificity 94.4% (101/107). CONCLUSION: Excellent levels of sensitivity and specificity of the GMA for detecting CP in the clinical setting were maintained at 2 years and were similar to our previously reported findings.

Use of the General Movements Assessment for the Early Detection of Cerebral Palsy in Infants with Congenital Anomalies Requiring Surgery.

The general movements (GMs) assessment is recognised as one of the most important tools in the early detection of cerebral palsy (CP). However, there remains a paucity of data on its application to infants with congenital anomalies requiring surgery. This was a prospective study of 202 infants (mean gestation 38 weeks, SD 2.2) who had undergone major surgery for congenital anomalies in the neonatal period. Infants were assessed at three months of age (mean 12 weeks, SD 1.6) and GMs videos were independently rated by three clinicians, two blinded to clinical details. Developmental follow-up was at three years of age. Of the twenty-five infants (9%) rated as having an absence of fidgety movements, 22 were seen at 3 years, and 17 had an abnormal outcome: 11 with CP, and 6 with a developmental disability. Infants with absent fidgety movements were 21.5 (95% CI 7.3-63.8) times more likely to have an abnormal outcome including CP. None of the infants with normal fidgety movements had a diagnosis of CP and 86% were assessed to be developing normally. The GMs assessment has predictive value for cerebral palsy and neurodevelopment for infants with congenital anomalies, and should be incorporated into routine follow-up to facilitate early referral.

Prediction of neurodevelopment at one year of age using the General Movements assessment in the neonatal surgical population.

BACKGROUND: Recent evidence indicates neonatal surgery is associated with an increased risk of neurodevelopmental disability, including cerebral palsy (CP). Despite evidence for prediction of CP there is limited information on use of the General Movements Assessment (GMA) with this population. AIM: To investigate the utility of the GMA for prediction of neurodevelopment in an infant surgical population. STUDY DESIGN: Prospective cohort study Subjects: 278 infants following cardiac surgery (n = 149), non-cardiac surgery (n = 123) or both surgeries (n = 6). OUTCOME MEASURES: GMA at three months of age (mean 12 weeks, SD 1.6) rated by three assessors, two blinded to clinical details. Follow-up at one year of age (mean 372 days, SD 13) using Bayley Scales of Infant and Toddler Development III (BSID-III), clinical and neurological examination. RESULTS: At one year, none of the 248 (89%) infants with normal fidgety movements had a diagnosis of CP, however a large proportion (n = 118, 48%) demonstrated delayed development. Infants who had absent fidgety movements (n = 25, 9%) showed a significant difference on all subtests of the BSID-III (p > 0.05). For prediction of CP there was 100% sensitivity and 96% specificity. CONCLUSIONS: The GMA is a valid complementary assessment tool to enhance prediction of neurodevelopment, specifically CP, following early neonatal surgery and should be incorporated into routine follow-up for this population.

General movement trajectories and neurodevelopment at 3months of age following neonatal surgery.

BACKGROUND: Neonates who undergo major surgery are at risk of neurodevelopmental disability. The General Movements Assessment (GMA) is a valid and reliable method to predict neurodevelopment, however, there are minimal data on the applicability among infants post-surgery. AIM: To describe GMs trajectories following neonatal surgery. STUDY DESIGN: Prospective cohort study. SUBJECTS: 217 infants following major cardiac and non-cardiac neonatal surgery. OUTCOME MEASURES: Infants were assessed following surgery at term age (mean 40weeks, SD 2.3), and at 3months of age (mean 12weeks, SD 1.6) using the GMA and the Bayley Scales of Infant and Toddler Development III. GMA videos were independently scored by three advanced trained assessors, two blinded to infant details. RESULTS: The most common result in the writhing period was 'poor repertoire' (n=117, 54%), however, 99 (84%) of these infants had normal fidgety movements. For infants with normal writhing (n=75, 34%), only four had absent fidgety movements. Cramped synchronised movements were seen in 10 infants, and three of these were rated as absent fidgety. There was no significant difference between the surgical groups. In total, 24 infants (11%) had absent fidgety movements and lower scores on average in all subtests of the BSID-III than those with normal fidgety movements. CONCLUSIONS: This is the first report describing GMs trajectories in infants who have undergone neonatal surgery. Similar to other high risk infant populations, this group showed a high proportion of poor repertoire writhing movements, however, most infants demonstrated normal fidgety movements and development at 3months of age.

Inter-observer agreement of the General Movements Assessment with infants following surgery.

BACKGROUND: The General Movements Assessment (GMA) is a validated and reliable method of identifying infants at risk of adverse neurodevelopmental outcomes, however there is minimal data available on the use of the GMA with infants following surgery. AIMS: The aim of this study was to investigate the inter-observer agreement for the GMA with this infant population. STUDY DESIGN: Reliability and agreement study. SUBJECTS: This was a prospective cohort study of 190 infants (male n=112) born at term (mean 38weeks, SD 2weeks). OUTCOME MEASURES: A GMA was conducted in the Neonatal Intensive Care Unit (NICU) following either cardiac surgery (n=92), non-cardiac surgery (n=93) or both types of surgery (n=5), and then again at three months of age. All videos were independently assessed by three advanced trained clinicians. Agreement and reliability statistics were calculated between each pair. RESULTS: We found moderate to substantial levels of agreement in the writhing period (66-77%, AC1=0.53-0.69). For fidgety general movements, agreement was classified as almost perfect, ranging from 86 to 89% (AC1=0.84-0.88). CONCLUSIONS: The GMA has high levels of inter-observer agreement when used with infants who have undergone surgery in the neonatal period, making it a valid, complementary assessment tool. Research is now underway to determine the ability of the GMA to predict neurodevelopmental outcomes in this population.

Sensitivity and specificity of General Movements Assessment for diagnostic accuracy of detecting cerebral palsy early in an Australian context.

AIM: The aim of this study was to calculate the sensitivity and specificity of the General Movements Assessment (GMA) for estimating diagnostic accuracy in detecting cerebral palsy (CP) in an Australian context by a newly established NSW rater network. METHODS: A prospective longitudinal cross-sectional study was conducted. The GMA was blind-rated from conventional video by two independent certified raters, blinded to medical history. A third rater resolved disagreements. High-risk population screening for CP using the GMA during the fidgety period (12-20 weeks) was carried out in four neonatal intensive care units and one CP service over a 30-month period (2012-2013). Participants were 259 high-risk infants. Sensitivity and specificity values were calculated with true positives defined as a confirmed diagnosis of CP from a medical doctor. RESULTS: Of the 259 infants assessed, 1-year follow-up data were available for 187. Of these, n = 48 had absent fidgety (high risk for CP), n = 138 had normal fidgety (low risk for CP), and n = 1 had abnormal fidgety (high risk for a neurological disorder). Of the 48 with absent fidgety movements, 39 had received a diagnosis of CP by 18 months and another 6 had an abnormal outcome. Of the n = 138 normal fidgety cases, n = 99 cases had a normal outcome, n = 38 had an abnormal outcome but not CP, and n = 1 had CP. For detecting CP, we had a sensitivity of 98% and specificity of 94%. CONCLUSION: GMA was feasible in an Australian context and accurately identified CP with a sensitivity and specificity comparable with European standards and published neuroimaging data.

General Movements Assessment of infants in the neonatal intensive care unit following surgery.

AIM: There is an identified gap in the literature regarding the use of the General Movements (GMs) Assessment to identify risk in infants post-surgery. This paper aims to examine the GMs for infants who have undergone surgery to identify common risk profiles in the writhing age. METHODS: This was a prospective cohort study of 170 infants (99 male, 58%) admitted to a neonatal surgical unit from 2012 until June 2014. Infants were born at a mean age of 38 weeks (standard deviation (SD) 2.3), and writhing GMs were conducted at term (40 weeks, SD 2.6) following either cardiac (n = 84, 49%) or non-cardiac surgery (n = 86, 51%). GMs were categorised by three trained assessors. RESULTS: Normal writhing was recorded in 64 infants (38%). The most common profile was poor repertoire (n = 80, 47%), with 14 (8%) cramped synchronised. There was no difference in scores between infants who had undergone cardiac versus non-cardiac surgery (P = 0.45). CONCLUSION: This is the first report on the GMs assessment in an infant surgical population. Infants who have undergone surgery most commonly display poor repertoire writhing movements. Research is needed to determine the ability of the GMs to predict neurodevelopmental outcomes in this population.