Variation in follow-up for children born very preterm in Europe.

BACKGROUND: Children born very preterm (<32 weeks of gestation) face high risks of neurodevelopmental and health difficulties compared with children born at term. Follow-up after discharge from the neonatal intensive care unit is essential to ensure early detection and intervention, but data on policy approaches are sparse. METHODS: We investigated the characteristics of follow-up policy and programmes in 11 European countries from 2011 to 2022 using healthcare informant questionnaires and the published/grey literature. We further explored how one aspect of follow-up, its recommended duration, may be reflected in the percent of parents reporting that their children are receiving follow-up services at 5 years of age in these countries using data from an area-based cohort of very preterm births in 2011/12 (N = 3635). RESULTS: Between 2011/12 and 22, the number of countries with follow-up policies or programmes increased from 6 to 11. The policies and programmes were heterogeneous in eligibility criteria, duration and content. In countries that recommended longer follow-up, parent-reported follow-up rates at 5 years of age were higher, especially among the highest risk children, born <28 weeks' gestation or with birthweight <1000 g: between 42.1% and 70.1%, vs. <20% in most countries without recommendations. CONCLUSIONS: Large variations exist in follow-up policies and programmes for children born very preterm in Europe; differences in recommended duration translate into cross-country disparities in reported follow-up at 5 years of age.

The early educational environment at five years of age in a European cohort of children born very preterm: challenges and opportunities for research.

BACKGROUND: Early childhood education offers opportunities for stimulation in multiple developmental domains and its positive impact on long-term outcomes and wellbeing for children is well documented. Few studies have explored early education in children born very preterm (VPT; <32 weeks of gestation) who are at higher risk of neurodevelopmental disorders and poor educational outcomes than their term-born peers. The purpose of the study is to describe and compare the educational environment of children born VPT in European countries at 5 years of age according to the degree of perinatal risk. METHODS: Data originated from the population-based Screening to Improve Health In very Preterm infants (SHIPS) cohort of children born VPT in 2011/2012 in 19 regions from 11 European countries. Perinatal data were collected from medical records and the 5-year follow-up was conducted using parental questionnaires. Outcomes at 5 years were participation in early education (any, type, intensity of participation) and receipt of special educational support, which were harmonized across countries. RESULTS: Out of 6,759 eligible children, 3,687 (54.6%) were followed up at 5 years (mean gestational age 29.3 weeks). At 5 years, almost all children (98.6%) were in an educational program, but type (preschool/primary), attendance (full-time/part-time) and use and type of school support/services differed by country. In some countries, children with high perinatal risk were more likely to be in full-time education than those with low risk (e.g. Estonia: 97.9% vs. 87.1%), while the inverse pattern was observed elsewhere (e.g. Poland: 78.5% vs. 92.8%). Overall, 22.8% of children received special educational support (country range: 12.4-34.4%) with more support received by children with higher perinatal risk. Large variations between countries remained after adjustment for socio-demographic characteristics. CONCLUSIONS: There are marked variations in approaches to early education for children born VPT in Europe, raising opportunities to explore its impact on their neurodevelopment and well-being.

Developmental motor problems and health-related quality of life in 5-year-old children born extremely preterm: A European cohort study.

AIM: To measure the association between cerebral palsy (CP) and non-CP-related movement difficulties and health-related quality of life (HRQoL) among 5-year-old children born extremely preterm (<28 weeks gestational age). METHOD: We included 5-year-old children from a multi-country, population-based cohort of children born extremely preterm in 2011 to 2012 in 11 European countries (n = 1021). Children without CP were classified using the Movement Assessment Battery for Children, Second Edition as having significant movement difficulties (≤5th centile of standardized norms) or being at risk of movement difficulties (6th-15th centile). Parents reported on a clinical CP diagnosis and HRQoL using the Pediatric Quality of Life Inventory. Associations were assessed using linear and quantile regressions. RESULTS: Compared to children without movement difficulties, children at risk of movement difficulties, with significant movement difficulties, and CP had lower adjusted HRQoL total scores (ß [95% confidence interval] = -5.0 [-7.7 to -2.3], -9.1 [-12.0 to -6.1], and - 26.1 [-31.0 to -21.2]). Quantile regression analyses showed similar decreases in HRQoL for all children with CP, whereas for children with non-CP-related movement difficulties, reductions in HRQoL were more pronounced at lower centiles. INTERPRETATION: CP and non-CP-related movement difficulties were associated with lower HRQoL, even for children with less severe difficulties. Heterogeneous associations for non-CP-related movement difficulties raise questions for research about mitigating and protective factors.

Prediction of movement difficulties at 5 years from parent report at 2 years in children born extremely preterm.

AIM: To assess the predictive validity of parent-reported gross motor impairment (GMI) at age 2 years to detect significant movement difficulties at age 5 years in children born extremely preterm. METHOD: Data were from 556 children (270 males, 286 females) born at less than 28 weeks' gestation in 2011 to 2012 in 10 European countries. Parent report of moderate/severe GMI was defined as walking unsteadily or unable to walk unassisted at 2 years corrected age. Examiners assessed significant movement difficulties (score ≤ 5th centile on the Movement Assessment Battery for Children, Second Edition) and diagnoses of cerebral palsy (CP) were collected by parent report at 5 years chronological age. RESULTS: At 2 years, 66 (11.9%) children had moderate/severe GMI. At 5 years, 212 (38.1%) had significant movement difficulties. Parent reports of GMI at age 2 years accurately classified CP at age 5 years in 91.0% to 93.2% of children. Classification of moderate/severe GMI at age 2 years had high specificity (96.2%; 95% confidence interval 93.6-98.0) and positive predictive value (80.3%; 68.7-89.1) for significant movement difficulties at age 5 years. However, 74.5% of children with significant movement difficulties at 5 years were not identified with moderate/severe GMI at age 2 years, resulting in low sensitivity (25.1%; 19.4-31.5). INTERPRETATION: This questionnaire may be used to identify children born extremely preterm who at age 2 years have a diagnosis of CP or movement difficulties that are likely to have a significant impact on their functional outcomes at age 5 years.

Neurodevelopment and behavior after transcatheter versus surgical closure of secundum type atrial septal defect.

OBJECTIVE: To assess the neuropsychological and behavioral profiles of school-aged children treated for atrial septal defect, secundum type (ASD-II) with open-heart surgery or catheterization. STUDY DESIGN: Patients (n = 48; mean age, 9 years, 3 months) and a matched healthy group (mean age, 9 years, 2 months) were evaluated with a shortened intelligence scale (Wechsler Intelligence Scale for Children, third edition, Dutch version) and a developmental neuropsychological test battery (Developmental Neuropsychological Assessment, second edition, Dutch version). Parents completed behavioral checklists (Achenbach Child Behavior Checklist for Children aged 6-18). Hospitalization variables were retrieved from medical files for studying associations with long-term neurodevelopment. RESULTS: Compared with the healthy matched controls, patients treated for ASD-II had significantly lower scores on subtasks underlying such Developmental Neuropsychological Assessment, second edition, Dutch version domains as Attention and Executive Functioning, Language, Working Memory, Sensorimotor Functioning, Social Cognition, and Visuospatial Information Processing. Only subtle differences, mainly in Visuospatial Information Processing, were found between the surgical repair and transcatheter repair groups. Socioeconomic status, longer hospital stay, and larger defect size were associated with neurocognitive outcome measures. Parents of patients reported more thought problems, posttraumatic stress problems, and lower school performance compared with parents of healthy peers. CONCLUSION: After treatment for ASD-II, children display a range of neuropsychologic difficulties that may increase their risk for learning problems and academic underachievement. Differences related to treatment were not found. Our results suggest that neurodevelopmental and behavioral follow-up at school age is warranted in this group.

Neurobehavioural functioning in school-aged children with a corrected septal heart defect.

OBJECTIVE: This cross-sectional study assesses neurobehavioural consequences after surgical treatment for acyanotic congenital heart defect at the age of 5 to 12 years. METHODS AND RESULTS: Fifteen school-aged children who underwent surgical intervention for a septal heart defect were examined with the short form Wechsler Intelligence Scale for Children-third Edition, Dutch Version (WISC-III-NL), and a neuropsychological assessment with the Nepsy. Performances were compared to a matched healthy control group. Parents completed the Child Behaviour Checklist and were interviewed to rate the child's cognitive and emotional functioning. Children older than eight years filled out a self-perception questionnaire. Analyses revealed significant differences between the study groups on several cognitive and behavioural domains. Estimated intelligence scores were in the average range. Domains of attention and fine motor skills, and the subtest level of language abilities, elicited significant group differences, with less favourable outcomes for the patient group. Parents of patients reported more withdrawn behaviour, social difficulties, thought problems, attentional shortcomings and lower competence for activities. These parents also indicated poor problem solving skills in everyday activities. CONCLUSIONS: Surgical patients displayed subtle neuropsychological difficulties concerning language and fine motor skills. Behavioural difficulties were common. Future research should address risk factors for adverse neurobehavioural manifestations in this patient group.

Maternal Wellbeing Five Years after a Very Preterm Delivery: Prevalence and Influencing Factors in a European Cohort.

(1) Background: Mothers of very preterm (VPT) infants may experience psychological symptoms compromising long-term emotional wellbeing. This study describes the emotional wellbeing of mothers of five-year-old children born VPT. We assess the association between sociodemographic, perinatal and neonatal characteristics, and the child's health and development at five years old and maternal emotional wellbeing. (2) Methods: Data are from the prospective European "Effective Perinatal Intensive Care in Europe" (EPICE) and subsequent "Screening for Health In very Preterm infantS in Europe" (SHIPS) projects including births <32 weeks' gestational age in 11 countries in 2011/12. Data were abstracted from obstetric and neonatal records. At five years old, 2605 mothers answered a parental questionnaire including the Mental Health Inventory-5 (MHI-5). Associations between sociodemographic and health characteristics and the mother's MHI-5 score were investigated using multilevel multivariate linear regression analysis with the country modelled as a random effect and inverse probability weighting to correct for attrition bias. (3) Results: The mean MHI-5 score was 71.3 (SD 16.7) out of 100 (highest emotional wellbeing) with a variation among countries from 63.5 (SD 16.8; Poland) to 82.3 (SD 15.8; the Netherlands). MHI-5 scores were significantly lower for mothers whose child had a severe health problem, developmental, or speech delay, for multiparous and single mothers, and when at least one of the parents was unemployed. (4) Conclusions: The emotional wellbeing of mothers of VPT infants differs between European countries. Identifying sociodemographic characteristics and child's health and developmental conditions that affect maternal emotional wellbeing may help to identify groups of mothers who need special assistance to cope with consequences of the delivery of a VPT child.

Movement Difficulties at Age Five Among Extremely Preterm Infants.

BACKGROUND AND OBJECTIVES: Children born extremely preterm (EPT), <28 weeks' gestational age, face higher risks of movement difficulties than their term-born peers. Studies report varying prevalence estimates and prognostic factors identifying children who could benefit from early intervention are inconsistent. This study investigated the prevalence of movement difficulties in children born EPT and associated risk factors. METHODS: Data come from a population-based EPT birth cohort in 2011 and 2012 in 11 European countries. Children without cerebral palsy were assessed at 5 years of age (N = 772) with the Movement Assessment Battery for Children-Second Edition, which classifies movement difficulties as none (>15th percentile), at risk (6th-15th percentile) and significant (≤5th percentile). Associations with sociodemographic, perinatal, and neonatal characteristics collected from obstetric and neonatal medical records and parental questionnaires were estimated using multinomial logistic regression. RESULTS: We found 23.2% (n = 179) of children were at risk for movement difficulties and 31.7% (n = 244) had significant movement difficulties. Lower gestational age, severe brain lesions, and receipt of postnatal corticosteroids were associated with significant movement difficulties, whereas male sex and bronchopulmonary dysplasia were associated with being at risk and having significant movement difficulties. Children with younger, primiparous, less educated, and non-European-born mothers were more likely to have significant movement difficulties. Differences in prevalence between countries remained after population case-mix adjustments. CONCLUSIONS: This study confirms a high prevalence of movement difficulties among EPT children without cerebral palsy, which are associated with perinatal and neonatal risk factors as well as sociodemographic characteristics and country.

Increased interaction and procedural flexibility favoured participation: Study across European cohorts of preterm born individuals.

OBJECTIVE: To understand participation and attrition phenomena variability in European cohorts of individuals born preterm through in-depth exploration of the interplay of situational elements involved. METHODS: Multi-situated qualitative design, using focus groups, semi-structured interviews and collaborative visual methodology with a purposive sample of adults born preterm, parents and professionals (n = 124) from eight cohorts in seven European countries. RESULTS: Most cohort participants were motivated by altruism/solidarity and gratitude/sense of duty to reciprocate (only absent in adults aged 19 - 21), followed by expectation of direct benefit to one's health and knowledge amongst participating adults. Common deterrents were perceived failure in reciprocity as in insufficient/inadequate interaction and information sharing, and postal questionnaires. Combining multipurpose, flexible strategies for contact and assessment, reminders, face-to-face and shorter periodicity and not simply adding retention strategies or financial incentives favoured participation. Professionals' main challenges entailed resources, funding and, European societal changes related to communication and geopolitical environment. CONCLUSION: Retention would benefit from tailoring inclusive strategies throughout the cohorts' life cycle and consistent promotion of reciprocal altruistic research goals. Investing in regular interaction, flexibility in procedures, participant involvement and return of results can help mitigate attrition as well as considering mothers as main facilitators to participating children and impaired adults.

Maternal education and cognitive development in 15 European very-preterm birth cohorts from the RECAP Preterm platform.

BACKGROUND: Studies are sparse and inconclusive about the association between maternal education and cognitive development among children born very preterm (VPT). Although this association is well established in the general population, questions remain about its magnitude among children born VPT whose risks of medical and developmental complications are high. We investigated the association of maternal education with cognitive outcomes in European VPT birth cohorts. METHODS: We used harmonized aggregated data from 15 population-based cohorts of children born at <32 weeks of gestational age (GA) or <1500 g from 1985 to 2013 in 13 countries with information on maternal education and assessments of general development at 2-3 years and/or intelligence quotients between 4 and 15 years. Term-born controls (≥37 weeks of GA) were available in eight cohorts. Maternal education was classified as: low (primary/lower secondary); medium (upper secondary/short tertiary); high (bachelor's/higher). Pooled standardized mean differences (SMDs) in cognitive scores were estimated (reference: high educational level) for children assessed at ages 2-3, 4-7 and 8-15 years. RESULTS: The study included 10 145 VPT children from 12 cohorts at 2-3 years, 8829 from 12 cohorts at 4-7 years and 1865 children from 6 cohorts at 8-15 years. Children whose mothers had low, compared with high, educational attainment scored lower on cognitive measures [pooled unadjusted SMDs: 2-3 years = -0.32 (95% confidence intervals: -0.43 to -0.21); 4-7 years = -0.57 (-0.67; -0.47); 8-15 years = -0.54 (-0.72; -0.37)]. Analyses by GA subgroups (<27 vs ≥27 weeks) in children without severe neonatal morbidity and term controls yielded similar results. CONCLUSIONS: Across diverse settings and regardless of the degree of prematurity, low maternal education was associated with lower cognition.

The impact of choice of norms on classification of motor impairment for children born very preterm.

BACKGROUND: The Movement Assessment Battery for Children-Second Edition (Movement ABC-2) is widely used to assess children's motor function, yet there is a lack of normative data for many countries. AIMS: To assess the extent to which the application of different population reference norms for the Movement ABC-2 affects the classification and prevalence of motor impairment. DESIGN: Data were obtained from two Portuguese regions participating in the Screening to Improve Health in Very Preterm Infants in Europe (SHIPS) Study, which was a five year follow-up of a cohort of children born at <32 weeks' gestation in 2011-2012 in 19 regions in 11 European countries. Perinatal data were extracted from medical records and the Movement ABC-2 was administered at five years of age. SUBJECTS: Of 542 Portuguese children eligible for the five-year follow-up, 413 (76.2%) were evaluated. OUTCOME MEASURES: Movement ABC-2 raw scores were converted to standardized scores using norms from four countries with national standardisations (UK, Netherlands/Belgium, France and Italy). RESULTS: The prevalence of significant movement difficulties (total score ≤ 5th percentile) was 28.5% using Dutch/Flemish norms, 23.3% using French norms, 16.5% using UK norms and 11.4% using Italian norms; 10.8% and 68.3% of the children were consistently classified as having significant movement difficulties and as not having significant movement difficulties, respectively, according to any norms. However, for 20.9% of children there was a disagreement in motor function status using different norms. CONCLUSION: The use of different test norms has a large impact on the proportion of children classified with significant movement difficulties, with implications for clinical referrals, healthcare costs and research. Our results underscore the importance of using appropriately validated tests with sound psychometric properties, and raise questions about the large differences in norms for the Movement ABC-2 in European countries.

Neurocognitive development and behaviour in school-aged children after surgery for univentricular or biventricular congenital heart disease.

OBJECTIVES: To assess the long-term neuropsychological and behavioural profile of school-aged children who were treated for univentricular heart (UVH) conditions or biventricular heart defect (BiVH) in infancy in a cross-sectional study design. METHODS: Sixty-three patients, 17 UVH (13 males, 4 females) and 46 BiVH (19 males, 27 females), were assessed at a mean age of 9.1 years (2.2 years) with an intelligence and neuropsychological test battery. Results were compared between subgroups (UVH, BiVH and a healthy control group). Associations between cognitive outcome, medical and socio-demographic factors were explored. Parents completed the Child Behavior Checklist (CBCL). RESULTS: Mean intelligence and neuropsychological scores were found within normal ranges for all diagnostic groups. Significant differences between UVH patients and controls emerged on auditory sustained and alternating attention, fine motor skills, visuospatial information processing, and to a lesser extent, memory performance. Parents of UVH patients reported more externalizing problems and school problems. Patient groups did not differ on neuropsychological outcome measures, nor on behavioural problems as rated by parents. CONCLUSIONS: After Fontan completion, patients at school age display intelligence scores within normal population-based ranges. However, they were found at risk for subtle shortcomings in attention, fine motor skills, visuospatial information processing and externalizing behaviour problems. Considerations pertaining to neurobehavioural outcome in school-aged children are discussed.

Neurodevelopmental outcome after surgery for acyanotic congenital heart disease.

BACKGROUND: Inconsistent results on neuropsychological outcome in patients treated for acyanotic congenital heart disease (aCHD) questioned the clinical relevance of possible neurobehavioral sequelae in this group. This study was designed to objectify the neuropsychological profile and evaluate associations with medical data. METHODS: Patients with a corrected atrial or ventricular septal defect, ASD-II or VSD, (n=46; mean age 9 years, 2 months) and a matched control group were submitted to an intelligence test (Wechsler Intelligence Scale for Children, third edition, Dutch version) and evaluated with a neuropsychological test battery (Developmental Neuropsychological Assessment, second edition, Dutch version). Hospitalization variables were retrieved to evaluate associations with cognitive outcome. Parents completed a behavioral checklist (Achenbach Child Behavior Checklist for Children aged 6-18). RESULTS: ASD-II patients showed lower scores in domains of visuospatial processing, language, attention, and social perception. VSD patients displayed subtle problems in attention and visuospatial information processing. Only few perioperative medical factors, but also socioeconomic variables were associated with cognitive outcomes. Parents of ASD-II patients reported more school problems when compared to controls. CONCLUSIONS: After treatment for aCHD, subtle cognitive difficulties can present in domains of visuospatial information processing, language, attention, and social perception. These shortcomings might hamper school performances, as is suggested by lower school competence ratings. Ongoing follow-up and cognitive screening is warranted to promote developmental progress, in which both parents and clinicians share responsibility.

Individual differences in degree of handedness and somesthetic asymmetry predict individual differences in left-right confusion.

Confusion or frustration connected with daily demands involving left-right discrimination is a common observation even in neurologically intact adults. We aimed to test the hypothesis that the degree of left-right confusion is associated with bodily asymmetry. Sixty-two female volunteers performed a left-right decision task that required fast responses to visually presented directional words (left, right, up, down) or pictograms (<--, -->, upward arrow, downward arrow). Participants also performed several tests that measured asymmetry of handedness, grip strength, and tactile sensitivity, and completed self-reports on left-right confusion and perceived bodily asymmetry. Results showed significant correlations between left-right confusion and the degree of handedness and asymmetry in tactile sensitivity. These results suggest that individuals who reveal a stronger internal bias between both sides of the body show less left-right confusion than people with less salient bodily asymmetry.