Search

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., (© 2024. The Author(s).)

Background: The health-related quality of life (HRQoL) of adolescents with CP in low and middle-income countries is often poor, as is the case in Bangladesh. This exploratory study examined what factors predict the proxy-reported HRQoL of adolescents with CP in rural Bangladesh, a typical low- and middle-income country (LMIC)., Methods: Adolescents with CP (10 to 18y) were identified using the Bangladesh Cerebral Palsy Register. HRQoL was assessed using the Cerebral Palsy Quality of Life-Teens proxy-report questionnaire (CPQoL-Teens), adolescent mental health using the Strengths and Difficulty Questionnaire (SDQ) and caregiver mental health using the Depression, Anxiety and Stress Scale (DASS-21). Theoretical and statistical interests (i.e. bivariate analysis, p < 0.05) identified potential predictors which were entered into hierarchical multiple linear regression (HMLR) models in order of clinical significance; HMLR related adolescent clinical characteristics, adolescent and caregiver mental health and proxies of socioeconomic status to CPQoL-Teens dimensions., Results: One hundred fifty-four adolescents with CP (mean age 15y 1mo, SD 1y 8mo, female 31.2%) participated in this study. Twenty-four factors were identified to explore for relationship to adolescent proxy-reported HRQoL. Fifteen of the factors correlated to one or more CPQoL-Teens dimension; strongest correlation was between 'feelings about functioning' and motor impairment (r = 0.545). Nine were predictive of CPQoL-Teens dimensions; adolescent sex, school attendance, severity of motor impairment, hearing and speech impairment, mother's education, primary caregiver depression and stress, and having a sanitary latrine at home resulting in score changes of between 0.79 (95% CI 0.24 to 1.35) to 35.1 (95% CI 6.03 to 64.22)., Conclusions: Many of the factors predicting the proxy-reported HRQoL of adolescents with CP are amenable to intervention, and have the potential to improve adolescent wellbeing. Several determinants are priorities of the sustainable development goals (SDGs); these findings should inform resource prioritization to improve the wellbeing of adolescents with CP in Bangladesh and other LMICs.

Background: Hip dysplasia is common among children with cerebral palsy (CP), particularly in spastic CP. It can result in pain, reduced function and quality of life. However, the burden of hip dysplasia among children with CP in llow-and middle-income countries (LMICs) like Bangladesh is unknown. We aimed to define the burden of hip dysplasia among children with spastic CP in Bangladesh., Methods: This study includes a subset of the Bangladesh CP Register (BCPR) study cohort who were registered between January and March 2015. The BCPR is a population-based surveillance of children with CP (aged < 18 years) operating in a northern sub-district (Shahjadpur; child population ~ 226,114) of Bangladesh. Community-based key informant's method (KIM) survey conducted to identify children with CP in the surveillance area. A diagnosis of CP was made based on clinical history and examination by the study physicians and physiotherapist. Study participants had an antero-posterior (AP) X-ray of their pelvis. The degree of subluxation was assessed by calculating the migration percentage (MP)., Results: During the study period, 196 children with CP were registered, 144 had spastic CP. 40 children with spastic CP (80 hips) had pelvic X-Rays (mean age 9.4 years, range 4.0-18.0 years) and 32.5% were female. Gross Motor Function Classification System (GMFCS) showed 37.5% (n = 15) with GMFCS level I-II and 62.5% (n = 25) with GMFCS level III-V. Twenty percent (n = 8) of the children had hip subluxation (MP: 33-80%). Osteopenic changes were found in 42.5% (n = 17) children., Conclusions: To the best of our knowledge this is one of the first studies exploring hip dysplasia among children with spastic CP in Bangladesh. Our findings reflect that hip dysplasia is common among children with spastic CP. Introduction of hip surveillance programmes is imperative for prevention of secondary complications, reduced function and poor quality of life among these children.

Background: Quality of life (QoL) and health-related quality of life (HRQoL) measurement in low and middle-income countries of people with cerebral palsy (CP), the major cause of childhood physical disability, is essential to assess the impact of interventions and inform policies that best improve people's lives. The purpose of this study was to cross-culturally translate and psychometrically validate the Cerebral Palsy Quality of Life-Teens (CPQoL-Teens) self- and proxy-report questionnaires for application with adolescents with CP in Bangladesh., Method: The CPQoL-Teens questionnaires were translated to Bengali using forward and backwards cross-cultural translation protocols. The questionnaires were interviewer administered to adolescents and their primary caregivers, identified through the Bangladesh Cerebral Palsy Register. Feasibility, sensitivity, internal consistency, content, concurrent and construct validity were assessed., Results: One hundred fifty four adolescents with CP (10 to 18y; mean 15y 1mo SD 1y 8mo; 31.2% female) participated. Feasibility, sensitivity and internal consistency of both self- and proxy-report questionnaires was excellent; nil missing scores except 'school wellbeing' which was associated with non-school attendance (48.4 to 74.7%); floor and ceiling effect ≤13.6%; Cronbach's alpha 0.77 to 0.94. Instrument validity was good; confirmatory factor analysis reflected five of the seven original instrument dimensions. CPQoL-Teens correlated to Kidscreen-27 on most dimensions (r = 0.176 to 0.693, p < 0.05); minimal difference in known groups was observed by mental health status (p < 0.05) although could be accounted for by homogeneity of mental health problems in the sample., Conclusion: The CPQoL-Teens self- and proxy report questionnaires successfully translated to Bengali and showed excellent feasibility and strong psychometric properties confirming suitability to assess indicators of HRQoL among adolescents with CP in Bangladesh.

Background: Measuring the health-related quality of life (HRQoL) of adolescents, including those with cerebral palsy (CP) (the major cause of childhood physical disability worldwide) in Bangladesh is pertinent although there is a dearth of validated instruments for assessing this concept. For application in a case-control study comparing HRQoL between adolescents with CP and peers without disability in Bangladesh (a typical low- and middle-income country) we cross-culturally translated and psychometrically tested KIDSCREEN-27., Methods: KIDSCREEN-27 was translated to Bengali using forward and backwards translation protocol and interviewer administered to adolescents with CP and their age and sex matched peers without disability. Primary caregivers were included for proxy-report. Sociodeomgraphic characterists and clinical information were extracted from the Bangladesh Cerebral Palsy Register (BCPR) and adolescent mental health was assessed using the Bengali version Strenghts and Difficulties Questionnaire (SDQ). Feasibility, floor and ceiling effect, internal consistency, content and construct validity of KIDSCREEN-27 were tested., Results: Feasibility, floor and ceiling effect and internal consistency of KIDSCREEN-27 was good for both self- and proxy-report questionnaires; nil missing scores except 'school environment' (11.0% to 74.7%) which correlated to rates of non-school attendance; floor and ceiling effect ≤10.4% except 'peers and social support' 23.4%; Cronbach's alpha 0.67 to 0.91. Instrument validity was strong; factor analysis reflected original instrument dimensions within one to three factors and difference in known groups was observed by CP and adolescent mental health (p < 0.05)., Conclusion: KIDSCREEN-27 successfully translated to Bengali and both the self and proxy-report questionnaires showed good psychometric properties indicating suitability for case-control assessment of HRQoL between adolescents with CP and peers without disability in Bangladesh.

Background: Children with complex neurodevelopmental disabilities such as cerebral palsy (CP), have a higher risk of dental disease related at least in part to greater difficulties in performing and maintaining effective oral hygiene and oral care practices. However, to date, there are very few studies that have considered the impact of dental disease on the Oral Health-Related Quality of Life (OHRQoL) of children and adolescents with cerebral palsy. This study aimed to investigate the association between dental caries experience and oral health related quality of life (OHRQoL) among children and adolescents with cerebral palsy in a low-resource setting (Bangladesh)., Methods: A total of 90 children and adolescents with CP, 2-17 years old (median age 10 years; 37.8% female and 62.2% male) were randomly selected from the Bangladesh Cerebral Palsy Register (BCPR) The decayed, missing and filled teeth (dmft/DMFT) index was used to measure caries experience. Child Perceptions Questionnaire (CPQ) and Family Impact Scale (FIS) were used to assess oral health-related quality of life (OHRQoL). Binary logistic regression was used to investigate factors that may contribute to dental caries experience., Results: Dental caries were observed among 55.6% of the participants. After adjusting for age and gender, binary logistic regression analysis showed that dental caries experience was significantly associated with those who had teeth/mouth pain (rate ratio 7.3; P = 0.02), food caught between teeth (rate ratio: 6.4; P = 0.02), difficulty in eating and drinking (rate ratio 5.9; p = 0.02) and those who felt frequently upset (rate ratio: 54.7; P = 0.02)., Conclusion: In this study, we found that children and adolescents with CP in a low-resource setting had high dental caries experience and that dental caries had a negative impact on OHRQoL amongst these participants and their parents/caregivers. Health care professionals should be aware of the importance of dental health and oral hygiene in this population. These findings highlight the need for oral health promotion programs for children and adolescents with CP in these settings to reduce pain and to improve quality of life.

Background: Gastroschisis is a congenital anomaly of the fetal abdominal wall, usually to the right side of umbilical insertion. It is often detected by routine antenatal ultrasound. Significant maternal and pediatric resources are utilised in the care of women and infants with gastroschisis. Increasing rates of gastroschisis worldwide have led institutions to review local data and investigate outcomes. A collaborative project was developed to review local epidemiology and investigate antenatal and neonatal factors influencing hospital length of stay (LOS) and total parental nutrition (TPN) in infants born with gastroschisis., Methods: We performed a five-year review of infants born with gastroschisis (2011-2015) at a major Australian centre. Complex gastroschisis was defined as involvement of stenosis, atresia, ischemia, volvulus or perforation and closed or vanishing gastroschisis. We extracted data from files and databases at the two participating hospitals, a major maternal fetal medicine centre and the affiliated children's hospital., Results: There were 56 infants antenatally diagnosed with gastroschisis with no terminations, one stillbirth (2%) and one infant with 'vanishing' gastroschisis. The mean maternal age was 23.9 years (range, 15-39 years). The mean gestation at delivery was 36 weeks (range, 25-39 + 3 weeks). Of the 55 neonates who received surgical management, 62% had primary closure. The median LOS was 33 (IQR, 23-45) days and the median duration of TPN was 26 (IQR, 17-36) days. Longer days on TPN (median 35 vs 16 days, P = 0.03) was associated with antenatal finding of multiple dilated bowel loops. Postnatal diagnosis of complex gastroschisis was made in 16% of cases and was associated with both longer LOS (median 89 vs 30 days, P = 0.003) and days on TPN (median 46 vs 21 days, P = 0.009)., Conclusion: Complex gastroschisis was associated with greater days on TPN and LOS. We found no late-gestation stillbirths and a low overall rate of 1.8%, suggesting the risk for stillbirth associated with gastroschisis is lower than previously documented. This information may assist counselling families. Improved data collection worldwide may reveal causative factors and enable antenatal outcome predictors.

Background: Gastro-oesophageal reflux (GOR) is common in infants. When the condition causes pathological symptoms and/or complications it is considered gastro-oesophageal reflux disease (GORD). It appears to be increasingly diagnosed and causes great distress in the first year of infancy. In New South Wales (NSW), residential parenting services support families with early parenting difficulties. These services report a large number of babies admitted with a label of GOR/GORD. The aim of this study was to explore the maternal and infant characteristics, obstetric interventions, and reasons for clinical reporting of GOR/GORD in NSW in the first 12 months following birth (2000-2011)., Methods: A three phase, mixed method sequential design was used. Phase 1 included a linked data population based study (n = 869,188 admitted babies). Phase 2 included a random audit of 326 medical records from admissions to residential parenting centres in NSW (2013). Phase 3 included eight focus groups undertaken with 45 nurses and doctors working in residential parenting centres in NSW., Results: There were a total of 1,156,020 admissions recorded of babies in the first year following birth, with 11,513 containing a diagnostic code for GOR/GORD (1% of infants admitted to hospitals in the first 12 months following birth). Babies with GOR/GORD were also more likely to be admitted with other disorders such as feeding difficulties, sleep problems, and excessive crying. The mothers of babies admitted with a diagnostic code of GOR/GORD were more likely to be primiparous, Australian born, give birth in a private hospital and have: a psychiatric condition; a preterm or early term infant (37-or-38 weeks); a caesarean section; an admission of the baby to SCN/NICU; and a male infant. Thirty six percent of infants admitted to residential parenting centres in NSW had been given a diagnosis of GOR/GORD. Focus group data revealed two themes: "It is over diagnosed" and "A medical label is a quick fix, but what else could be going on?", Conclusions: Mothers with a mental health disorder are nearly five times as likely to have a baby admitted with GOR/GORD in the first year after birth. We propose a new way of approaching the GOR/GORD issue that considers the impact of early birth (immaturity), disturbance of the microbiome (caesarean section) and mental health (maternal anxiety in particular).

Background: Infants may be at neurodevelopmental risk from adverse events arising in the neonatal period. This study aimed to investigate the developmental outcomes and physical activity behaviours of term infants after neonatal major surgery, at age three years., Methods: This prospective study enrolled infants who underwent major surgery in their first 90 days, between August 2006 and December 2008. Developmental status was assessed using the Bayley Scales of Infant and Toddler Development, Third Edition (BSID-III). Physical activity and sedentary behaviour (i.e. small screen recreation) (SSR) were assessed using the Preschool-Age Physical Activity Questionnaire (Pre-PAQ). Activity (moving between slow to fast pace) and SSR were reported for a 3-day period., Results: One hundred and thirty five children (68 major surgery, 67 control) were assessed, using both measures, at age three years. Both groups were within the average range across all domains of the BSID-III although the surgical group was significantly below the controls for cognition (t = -3.162, p = 0.002) receptive language (t = -3.790, p < 0.001) and fine motor skills (t = -2.153, p = 0.03). Mean activity time for the surgical group was 191 mins.day(-1), and 185 mins.day-1 for controls. Mean SSR time was 77 mins.day(-1), and 83 mins.day(-1) for the respective groups. There was no significant difference between groups for either physical activity (p = 0.71) or SSR time (p = 0.49)., Conclusions: By age three, children who had major surgery in infancy are developmentally normal but have not quite caught up with their peer group in cognitive, receptive language and fine motor skill domains. Both groups met recommended 3 h of daily physical activity but exceeded 60-min SSR time recommended for preschool-age children.

Background: The causes and pathogenesis of cerebral palsy (CP) are all poorly understood, particularly in low- and middle-income countries (LMIC). There are gaps in knowledge about CP in Bangladesh, especially in the spheres of epidemiological research, intervention and service utilization. In high-income countries CP registers have made substantial contributions to our understanding of CP. In this paper, we describe a pilot study protocol to develop, implement, and evaluate a CP population register in Bangladesh (i.e., Bangladesh Cerebral Palsy Register - BCPR) to facilitate studies on prevalence, severity, aetiology, associated impairments and risk factors for CP., Methods/design: The BCPR will utilise a modified version of the Australian Cerebral Palsy Register (ACPR) on a secured web-based platform hosted by the Cerebral Palsy Alliance Research Institute, Australia. A standard BCPR record form (i.e., data collection form) has been developed in consultation with local and international experts. Using this form, the BPCR will capture information about maternal health, birth history and the nature of disability in all children with CP aged <18 years. The pilot will be conducted in the Shahjadpur sub-district of Sirajgonj district in the northern part of Bangladesh. There are 296 villages in Shahjadpur, a total population of 561,076 (child population ~ 226,114), an estimated 70,998 households and 12,117 live births per annum. Children with CP will be identified by using the community based Key Informants Method (KIM). Data from the completed BPCR record together with details of assessment by a research physician will be entered into an online data repository., Discussion: Once implemented, BCPR will be, to the best of our knowledge, the first formalised CP register from a LMIC. Establishment of the BCPR will enable estimates of prevalence; facilitate clinical surveillance and promote research to improve the care of individuals with CP in Bangladesh.

Background: The average age for the diagnosis of cerebral palsy (CP) is 19 months. Recent neuroplasticity literature suggests that intensive, task-specific intervention ought to commence as early as possible and in an enriched environment, during the critical period of neural development. Active motor interventions are effective in some populations, however the effects of active motor interventions on the motor outcomes of infants with CP have not been researched thoroughly, but pilot work is promising. The aim of this study was to determine the short- term effects of "GAME"; a new and novel goal-oriented activity-based, environmental enrichment therapy programme on the motor development of infants at high risk of CP and test study procedures for a randomized controlled trial (RCT)., Methods: Pragmatic 2-group pilot RCT to assess motor outcomes, goal attainment, parent well-being and home environment quality, after 12-weeks of GAME intervention versus standard care. GAME included: creation of movement environments to elicit motor behaviours; parent training in motor learning and task analysis; frequent practice of motor tasks using a programme that was individualised to the child, was varied and focused on self-initiated movement. Data were analyzed using multiple regression., Results: Thirteen infants were consented, randomised, treated and completed the study. At study conclusion, the GAME group (n = 6) demonstrated an advantage in Total Motor Quotient of 8.05 points on the Peabody Developmental Motor Scale-2 (PDMS-2) compared to the standard care group (n = 7) (p < .001). No significant differences existed between groups on any other measure., Conclusions: GAME appears to offer a promising and feasible new motor intervention for CP, with favourable short-term motor outcomes. A pressing need exists for an adequately powered RCT with long-term end points, to determine if GAME may advance these children's motor trajectory.

Background: Cerebral palsy is the most common physical disability of childhood and early detection is possible using evidence based assessments. Systematic reviews indicate early intervention trials rarely demonstrate efficacy for improving motor outcomes but environmental enrichment interventions appear promising. This study is built on a previous pilot study and has been designed to assess the effectiveness of a goal - oriented motor training and enrichment intervention programme, "GAME", on the motor outcomes of infants at very high risk of cerebral palsy (CP) compared with standard community based care., Methods/design: A two group, single blind randomised controlled trial (n = 30) will be conducted. Eligible infants are those diagnosed with CP or designated "at high risk of CP" on the basis of the General Movements Assessment and/or abnormal neuroimaging. A physiotherapist and occupational therapist will deliver home-based GAME intervention at least fortnightly until the infant's first birthday. The intervention aims to optimize motor function and engage parents in developmental activities aimed at enriching the home learning environment. Primary endpoint measures will be taken 16 weeks after intervention commences with the secondary endpoint at 12 months and 24 months corrected age. The primary outcome measure will be the Peabody Developmental Motor Scale second edition. Secondary outcomes measures include the Gross Motor Function Measure, Bayley Scales of Infant and Toddler Development, Affordances in the Home Environment for Motor Development - Infant Scale, and the Canadian Occupational Performance Measure. Parent well-being will be monitored using the Depression Anxiety and Stress Scale., Discussion: This paper presents the background, design and intervention protocol of a randomised trial of a goal driven, motor learning approach with customised environmental interventions and parental education for young infants at high risk of cerebral palsy., Trial Registration: This trial is registered on the Australian New Zealand Clinical Trial register: ACTRN12611000572965.