Parents' information needs during the first year at home with their very premature born child; a qualitative study.

Objective: To obtain insights into parents' information needs during the first year at home with their very preterm (VP) born infant. Methods: We conducted semi-structured interviews with parents of VP infants participating in a post-discharge responsive parenting intervention (TOP program). Online interviews were audiotaped and transcribed verbatim. Inductive thematic analysis was performed by two independent coders. Results: Ten participants were interviewed and had various and changing information needs during the developmental trajectory of their infant. Three main themes emerged; (1) Help me understand and cope, (2) Be fully responsible for my baby, and (3) Teach me to do it myself. Available and used sources, such as the Internet, did not meet their information needs. Participants preferred their available and knowledgeable healthcare professionals for reassurance, tailored information, and practical guidance. Conclusion: This study identified parents' information needs during the first year at home with their VP infant and uncovered underlying re-appearing needs to gain confidence in child-caring abilities and autonomy in decision-making about their infants' care. Innovation: This study provides valuable information for healthcare professionals and eHealth developers to support parental self-efficacy during the first year after preterm birth.

Development and evaluation of a fidelity tool in a post-discharge responsive parenting intervention program for very preterm born children.

The TOP program is a fully implemented responsive parenting intervention for very preterm born infants. Fidelity monitoring of interventions is important for preserving program adherence, impact outcomes and to make evidence-based adaptations. The aim of this study was to develop a fidelity tool for the TOP program following an iterative and co-creative process and subsequently evaluate the reliability of the tool. Three consecutive phases were carried out. Phase I: Initial development and pilot testing two methods namely self-report and video based observation. Phase II: Adaptations and refinements. Phase III: Evaluation of the psychometric properties of the tool based on 20 intervention videos rated by three experts.The interrater reliability of the adherence and competence subscales was good (ICC.81 to .84) and varied from moderate to excellent for specific items (ICC between .51 and .98). The FITT displayed a high correlation (Spearman's rho.79 to.82) between the subscales and total impression item. The co-creative and iterative process resulted in a clinical useful and reliable tool for evaluating fidelity in the TOP program. This study offers insights in the practical steps in the development of a fidelity assessment tool which can be used by other intervention developers.

Neurodevelopmental Outcomes at Two Years' Corrected Age of Very Preterm Infants after Implementation of a Post-Discharge Responsive Parenting Intervention Program (TOP Program).

OBJECTIVE: To compare neurodevelopmental outcomes at 2 years corrected age (CA) between infants born very preterm (VP) who did or did not receive a postdischarge responsive parenting intervention (Transmural developmental support for very preterm infants and their parents [TOP program]) between discharge home and 12 months' CA. STUDY DESIGN: The Systemic Hydrocortisone to Prevent Bronchopulmonary Dysplasia (SToP-BPD) study showed no differences between treatment groups in motor and cognitive development using the Dutch Bayley Scales of Infant Development and behavior using the Child Behavior Checklist at 2 years' CA. During its study period, the TOP program was gradually scaled up nationwide in the same population, providing an opportunity to evaluate the effect of this program on neurodevelopmental outcome, after adjusting for baseline differences. RESULTS: Among 262 surviving VP infants in the SToP-BPD study, 35% received the TOP program. Infants in the TOP group had a significantly lower incidence of a cognitive score <85 (20.3% vs 35.2%; adjusted absolute risk reduction: -14.1% [95% CI: -27.2 to -1.1]; P = .03), and a significantly higher mean cognitive score (96.7 ± 13.8), compared with the non-TOP group (92.0 ± 17.5; crude mean difference: 4.7 [95% CI: 0.3 to 9.2]; P = .03). No significant differences were found on motor scores. For behavior problems, a small but statistically significant effect for anxious/depressive problems was found in the TOP group (50.5 vs 51.2; P = .02). CONCLUSIONS: VP infants supported by the TOP program from discharge until 12 months' CA had better cognitive function at 2 years' CA. This study demonstrates a sustained positive effect of the TOP program in VP infants.

Factors Associated With Walking Adaptability and Its Relationship With Falling in Polio Survivors.

OBJECTIVE: To explore factors associated with walking adaptability and associations between walking adaptability and falling in polio survivors. DESIGN: Cross-sectional study. SETTING: Outpatient expert polio clinic. PARTICIPANTS: Polio survivors (N=46) who fell in the previous year and/or reported fear of falling. INTERVENTIONS: Not applicable. MAIN OUTCOME MEASURES: Walking adaptability was assessed on an interactive treadmill and operationalized as variable target-stepping and reactive obstacle avoidance performance. Further, we collected walking speed and assessed leg muscle strength, balance performance (Berg Balance Scale and Timed-Up-and-Go Test), balance confidence (Activities-specific Balance Confidence scale), ambulation level, orthosis use, fear of falling, and number of falls in the previous year. RESULTS: With walking speed included as a covariate, muscle weakness of the most affected leg and balance confidence explained 54% of the variance in variable target-stepping performance. For reactive obstacle avoidance performance, muscle weakness of the most affected leg and knee extensor strength of the least affected leg explained 32% of the variance. Only target-stepping performance was significantly related to the number of falls reported in the previous year (R2=0.277, P<.001) and mediated the relation between leg muscle weakness and balance confidence with falling. CONCLUSION: Our exploratory study suggests that leg muscle weakness and reduced balance confidence limit walking adaptability in polio survivors. Because poorer target stepping rather than obstacle avoidance performance was associated with falling, our results indicate that a limited ability to ensure safe foot placement may be a fall risk factor in this group. These findings should be confirmed in a larger sample.

Mental health monitoring in parents after very preterm birth.

AIM: To evaluate parental mental health monitoring during follow-up care for very preterm (VPT) infants, describe symptoms of anxiety and depression and risk factors for mothers and fathers at 1 and 12 months of corrected age. METHODS: Parents completed the Hospital Anxiety and Depression Scale (HADS). Psychological symptoms and risk factors were analysed within and between mothers and fathers. RESULTS: In 4 years, the monitoring reached 1260 (48%) families. Of these, 693 mothers and 340 fathers (300 couples) completed the HADS twice. At 1 month, 22% and 15% of the mothers and 10% and 9% of the fathers, respectively, reported elevated symptoms of anxiety and depression. At 12 months, these rates were significantly reduced to 14% and 9% for mothers and 5% and 4% for fathers respectively. Within couples, anxiety and depression were positively associated. At 12 months, in 20% of the couples, one or both parents reported elevated symptoms. Risk factors were length of hospital stay, migration background, educational level and employment status. CONCLUSION: The mental health of parents of VPT infants improved, but elevated symptoms were still observed in 17% of included families after one year. Acknowledging and remediating parental mental health remain essential during follow-up care.

Development and nationwide implementation of a postdischarge responsive parenting intervention program for very preterm born children: The TOP program.

A previous randomized controlled trial has suggested the effectiveness of a Dutch postdischarge responsive parenting program for very preterm (VPT) infants, indicating that nationwide implementation was justified. This paper describes the development and nationwide implementation of the intervention, known as the TOP program, which consisted of three phases. In the preparation phase (2006-2010), a theory of change and the structure of the TOP program were developed, and funding for phase two, based on a positive Business Case, was obtained. In the pilot implementation phase (2010-2014), intervention strategies were developed for a real-world setting, capacity and adoption were increased, systematic evaluations were incorporated, and sustained funding was obtained. In the full-implementation phase (2014-2019), all Dutch Healthcare Insurers reimbursed the TOP program, enabling VPT infants to participate in the program without charge. By 2018, the number of interventionists that provided the TOP program had increased from 37 to 91, and all level III hospitals and 65% of regional hospitals in the Netherlands referred VPT infants. Currently, the program reaches 70% of the Dutch target population and parental satisfaction with the TOP program is high. After a 12-year implementation period, the TOP program forms part of routine care in the Netherlands.

Un previo ensayo controlado al azar ha sugerido la eficacia de un programa holandés sobre la crianza sensible para infantes muy prematuros (VPT) posterior al momento en que se les dio de alta, indicando que la implementación a lo largo de toda la nación era justificada. Este artículo describe el desarrollo y la implementación a nivel de toda la nación de la intervención, conocida como el programa ToP, el cual consistía de tres fases. En la fase de preparación (2006-2010), se desarrollaron una teoría de cambio y la estructura del programa ToP, y se obtuvieron los fondos para la fase dos, con base en un Caso de Negocios (BC) positivo. En la fase piloto de implementación (2010-2014), se desarrollaron estrategias de intervención para un escenario del mundo real, se aumentaron la capacidad y la adopción, se incorporaron evaluaciones sistemáticas y se obtuvieron fondos para mantener el programa. En la fase de implementación completa (2014-2019), todas las Aseguradoras Holandesas del Sector Salud reembolsaron el costo del programa ToP, permitiéndoles a los infantes VPT participar en el programa sin costo alguno. Para 2018, el número de practicantes de la intervención que prestaban el servicio del programa ToP había aumentado de 37 a 91, y todos los hospitales del nivel III y 65% de los hospitales regionales en Holanda refirieron los infantes VPT al programa. Actualmente, el programa llega a 70% de la población holandesa para la cual está destinado y la satisfacción de los padres con el programa ToP es alta. Después de un período de implementación de 12 años, el programa ToP forma parte del cuidado de salud rutinario en Holanda.

Un essai contrôlé randomisé précédent a suggéré l'efficacité d'un programme hollandais de sensibilité de parentage après la sortie de l'hôpital pour les nourrissons grands prématurés (GP ici en français), indiquant qu'une mise en œuvre au niveau national était justifiée. Cet article décrit le développement et la mise en œuvre au niveau national de l'intervention, connue en tant que ToP program, qui a consisté en trois phases. Dans la phase de préparation (2006-2010), une théorie du changement et la structure du programme ToP a été développée, et le financement pour la phase deux, basée sur une Etude de Cas positive, a été sécurisé. Dans la phase pilote d'implémentation (2010-2014) des stratégies d'intervention ont été développées pour un contexte réel, la capacité et l'adoption ont été augmentées, les évaluations systématiques ont été incorporées, et un financement durable a été sécurisé. Dans la pleine phase de mise en œuvre (2014-1029), tous les Assurances Santé Hollandaises ont remboursé le programme ToP, permettant aux nourrissons GP de participer au programme sans coût. En 2018 le nombre de prestataires qui offraient le programme ToP a augmenté de 37 à 91, et tous les hôpitaux de niveau III ainsi que 65% des hôpitaux régionaux aux Pays Bas ont envoyé les nourrissons GP au programme. En ce moment le programme atteint 70% de la population cible hollandaise et la satisfaction parentale avec le programme ToP est élevée. Après une période de mise en œuvre de 12 ans le programme ToP fait partie des soins de routine aux Pays Bas.

Feasibility of a Preventive Parenting Intervention for Very Preterm Children at 18 Months Corrected Age: A Randomized Pilot Trial.

OBJECTIVE: To evaluate the feasibility and potential efficacy of an age-appropriate additional parenting intervention for very preterm born toddlers. STUDY DESIGN: In a randomized controlled pilot study, 60 of 94 eligible very preterm born children who had received a responsive parenting intervention in their first year were randomized to usual care or the additional intervention, consisting of 4-6 home visits between 18 and 22 months' corrected gestational age (CA). Parents were supported to responsively interact during increasingly complex daily activities and play. Parental satisfaction with the intervention was evaluated with a questionnaire. At baseline and 24 months CA, parents completed the Infant Toddler Social and Emotional Assessment, the Ages and Stages Questionnaire, and the Dutch Schlichting Lexilist for receptive language. At 24 months CA, motor, and cognitive development was measured by the Bayley Scales of Infant and Toddler Development, Third Edition Dutch version, and parent-child interaction was evaluated by the Emotional Availability Scales. RESULTS: Parental compliance and satisfaction with the intervention was high. Effect sizes (after correction for baseline variables) were small for internalizing and competence behavior, receptive language, and problem solving; medium for cognitive development and parent-child interaction; and large for externalizing and dysregulation behavior and motor development. CONCLUSION: After a postdischarge intervention during the first year, an additional responsive parenting support at toddler-age is feasible and associated with positive outcomes in a broad array of parental and child outcome measures. TRIAL REGISTRATION: www.toetsingonline.nl: NL40208.018.12.

Deficits in vision and visual attention associated with motor performance of very preterm/very low birth weight children.

AIM: To extend understanding of impaired motor functioning of very preterm (VP)/very low birth weight (VLBW) children by investigating its relationship with visual attention, visual and visual-motor functioning. METHODS: Motor functioning (Movement Assessment Battery for Children, MABC-2; Manual Dexterity, Aiming & Catching, and Balance component), as well as visual attention (attention network and visual search tests), vision (oculomotor, visual sensory and perceptive functioning), visual-motor integration (Beery Visual Motor Integration), and neurological status (Touwen examination) were comprehensively assessed in a sample of 106 5.5-year-old VP/VLBW children. Stepwise linear regression analyses were conducted to investigate multivariate associations between deficits in visual attention, oculomotor, visual sensory, perceptive and visual-motor integration functioning, abnormal neurological status, neonatal risk factors, and MABC-2 scores. RESULTS: Abnormal MABC-2 Total or component scores occurred in 23-36% of VP/VLBW children. Visual and visual-motor functioning accounted for 9-11% of variance in MABC-2 Total, Manual Dexterity and Balance scores. Visual perceptive deficits only were associated with Aiming & Catching. Abnormal neurological status accounted for an additional 19-30% of variance in MABC-2 Total, Manual Dexterity and Balance scores, and 5% of variance in Aiming & Catching, and neonatal risk factors for 3-6% of variance in MABC-2 Total, Manual Dexterity and Balance scores. CONCLUSION: Motor functioning is weakly associated with visual and visual-motor integration deficits and moderately associated with abnormal neurological status, indicating that motor performance reflects long term vulnerability following very preterm birth, and that visual deficits are of minor importance in understanding motor functioning of VP/VLBW children.

Rethinking preventive post-discharge intervention programmes for very preterm infants and their parents.

Post-discharge preventive intervention programmes with involvement of the parent may support the resilience and developmental outcomes of infants born very preterm. Randomized controlled trials of home-based family-centred intervention programmes in very preterm infants that aimed to improve cognitive outcome, at least at age two, were selected and updated on the basis of a recent systematic review to compare their content and effect over time to form the basis of a narrative review. Six programmes were included in this narrative review. Four of the six programmes led to improved child cognitive and/or motor development. Two programmes, which focused primarily on responsive parenting and development, demonstrated improved cognitive outcome up till 5 years after completion of the programme. The programmes that also focused on maternal anxiety remediation led to improved maternal mental well-being, along with improved child behaviour, in one study - even at 3 years after completion of the programme. The magnitude of the effects was modest. Family-centred preventive intervention programmes that aim at improvement of child development should be continued after discharge home to improve the preterm child's resilience. Programmes may be most effective when they support the evolvement of a responsive parent-infant relationship over time, as well as the parent's well-being.

Attention in 3-Year-Old Children with VLBW and Relationships with Early School Outcomes.

AIM: To compare attention skills of children with a very low birth weight (VLBW) with children with a normal birth weight (NBW) when entering primary school, and explore the association of attention skills with school career 2 years later. METHODS: Participants were 151 children with VLBW and 41 with NBW. Attention was assessed at 3 years and 8 months of corrected age (CA) and school career at 5½ years of CA. Children performed two tests, parents completed three questionnaires, and an assessor systematically observed children's attention. RESULTS: Children with VLBW had significantly lower mean scores on five of the six measures. Significantly more children with VLBW had scores in the clinical range on the Child Behavior Checklist completed by the parents (13% versus 0%) and scores representing dysfunction on assessor observations (19% versus 2%). At 5½ years of age, 36% of the children with VLBW followed special education or had grade retention. Dysfunctional attention as observed by the assessor was most strongly associated with need for learning support at 5½ years of age. CONCLUSIONS: At preschool age, children with VLBW have attention difficulties. Attentive behavior at preschool age is a predictor of school career 2 years later.

Motor impairment in very preterm-born children: links with other developmental deficits at 5 years of age.

AIM: To elucidate the relation between motor impairment and other developmental deficits in very preterm-born children without disabling cerebral palsy and term-born comparison children at 5 years of (corrected) age. METHOD: In a prospective cohort study, 165 children (81 very preterm-born and 84 term-born)were assessed with the Movement Assessment Battery for Children - 2nd edition, Touwen's neurological examination, the Wechsler Preschool and Primary Scale of Intelligence, processing speed and visuomotor coordination tasks of the Amsterdam Neuropsychological Tasks, and the Strengths and Difficulties Questionnaire. RESULTS: Motor impairment (≤15th centile) occurred in 32% of the very preterm-born children compared with 11% of their term-born peers (p=0.001). Of the very preterm-born children with motor impairment, 58% had complex minor neurological dysfunctions, 54% had low IQ, 69% had slow processing speed, 58% had visuomotor coordination problems, and 27%, 50%,and 46% had conduct, emotional, and hyperactivity problems respectively. Neurological outcome (odds ratio [OR]=41.7, 95% confidence intervals [CI] 7.5­232.5) and Full-scale IQ(OR=7.3, 95% CI 1.9­27.3) were significantly and independently associated with motor impairment. Processing speed (OR=4.6, 95% CI 1.8­11.6) and attention (OR=3.2, 95% CI1.3­7.9) were additional variables associated with impaired manual dexterity. These four developmental deficits mediated the relation between preterm birth and motor impairment. INTERPRETATION: Complex minor neurological dysfunctions, low IQ, slow processing speed,and hyperactivity/inattention should be taken into account when very preterm-born children are referred for motor impairment.

The relationship between multiple developmental difficulties in very low birth weight children at 3½ years of age and the need for learning support at 5 years of age.

This study investigated whether multiple developmental difficulties are more frequent in very low birth weight (VLBW) children than in those born full term. The association between multiple developmental difficulties assessed at 3½ years of age and educational provision for the child at 5½ years was also investigated, with 'educational provision' referring to the curriculum, school placement and the level of learning support. There were 143 VLBW children without cerebral palsy (CP) and 41 term-born peers assessed at 3½ years of age. The assessment included 6 measures of development: word comprehension, visual motor integration, visual perception, motor coordination, executive functioning and behaviour. Educational provision was determined at age 5½ years. A mildly abnormal score (score <1 standard deviation) was considered to indicate developmental difficulty. Scores from the six measures of development were analysed to determine the difficulty frequency and the presence of multiple difficulties (>1 difficulty score) in each child. This study showed that at 3½ years of age, the VLBW children had significantly more difficulty with motor coordination than their term-born peers. In addition, 27% of the VLBW children had multiple difficulties compared to 10% in the term-born group. Multiple logistic regression analyses showed that of the difficulties, impaired motor coordination was most strongly associated with the requirement for learning support two years later. Regression analyses showed that having multiple difficulties was significantly associated with the need for learning support (Odds Ratio of 3.4 (95% CI: 1.5-7.8). These results show that the presence of multiple difficulties in a VLBW child of preschool age, can impact the child's educational provision two years later.

Comparing two motor assessment tools to evaluate neurobehavioral intervention effects in infants with very low birth weight at 1 year.

BACKGROUND: Infants with very low birth weight (VLBW) are at increased risk for motor deficits, which may be reduced by early intervention programs. For detection of motor deficits and to monitor intervention, different assessment tools are available. It is important to choose tools that are sensitive to evaluate the efficacy of intervention on motor outcome. OBJECTIVE: The purpose of this study was to compare the Alberta Infant Motor Scale (AIMS) and the Psychomotor Developmental Index (PDI) of the Bayley Scales of Infant Development-Dutch Second Edition (BSID-II-NL) in their ability to evaluate effects of an early intervention, provided by pediatric physical therapists, on motor development in infants with VLBW at 12 months corrected age (CA). DESIGN: This was a secondary study in which data collected from a randomized controlled trial (RCT) were used. METHODS: At 12 months CA, 116 of 176 infants with VLBW participating in an RCT on the effect of the Infant Behavioral Assessment and Intervention Program were assessed with both the AIMS and the PDI. Intervention effects on the AIMS and PDI were compared. RESULTS: Corrected for baseline differences, significant intervention effects were found for AIMS and PDI scores. The highest effect size was for the AIMS subscale sit. A significant reduction of abnormal motor development in the intervention group was found only with the AIMS. LIMITATIONS: No Dutch norms are available for the AIMS. CONCLUSIONS: The responsiveness of the AIMS to detect intervention effects was better than that of the PDI. Therefore, caution is recommended in monitoring infants with VLBW only with the PDI, and the use of both the AIMS and the Bayley Scales of Infant Development is advised when evaluating intervention effects on motor development at 12 months CA.

Assessing independency in daily activities in very preterm children at preschool age.

This study investigates whether very low birth weight (VLBW) preschoolers experience disability in daily activities and what the risk factors for disability in daily activities are. The Dutch Pediatric Evaluation of Disability Inventory (PEDI-NL) was used to detect disability in daily activities in 143 VLBW children without cerebral palsy (CP) at 44 months of corrected age (CA). Data from the psychomotor-developmental index (PDI) and the mental developmental index (MDI) of the Bayley Scales of Infant Development II (BSID II) at 24 months CA, and data relating to perinatal and socio-economic status were available. Disability in daily activities was found in 27 (19%) VLBW children without CP. High frequencies of disability were found in 19 (13%) children on the mobility domain and in 12 (8%) children on the social functioning domain. The multiple logistic regression analyses showed that low BSID II outcomes (<2 SD) were risk factors for disability in the mobility domain, but not for disability in the social functioning domain. The predictive value of the BSID II outcomes is moderate, 46% of the VLBW children with a low PDI and 44% with a low MDI developed a disability in the mobility domain. This study showed a higher frequency of disability in daily activities in VLBW preschoolers compared to term born peers. Therefore, it is suggested to assess VLBW children's performance of daily activities before they start school.

Sustained developmental effects of the infant behavioral assessment and intervention program in very low birth weight infants at 5.5 years corrected age.

OBJECTIVE: To evaluate the effect of the Infant Behavioral Assessment and Intervention Program (IBAIP) in very low birth weight (VLBW) infants on cognitive, neuromotor, and behavioral development at 5.5 years corrected age (CA). STUDY DESIGN: In a randomized controlled trial, 86 VLBW infants received post discharge IBAIP intervention until 6 months CA, and 90 VLBW infants received standard care. At 5.5 years CA, cognitive and motor development, and visual-motor integration were assessed with the Wechsler Preschool and Primary Scale of Intelligence, third Dutch version, the Movement Assessment Battery for Children, second edition, and the Developmental Test of Visual Motor Integration. Neurologic conditions were assessed with the neurologic examination according to Touwen, and behavior with the Strengths and Difficulties Questionnaire. RESULTS: At 5.5 years CA, 69 children in the intervention and 67 children in the control group participated (response rate 77.3%). Verbal and performance IQ-scores<85 occurred significantly less often in the intervention than in the control group (17.9% vs 33.3%, P=.041, and 7.5% vs 21.2%, P=.023, respectively). However, after adjustment for differences, only the OR for performance IQ was significant: 0.24, 95% CI: 0.06-0.95. Adjusted mean scores on Wechsler Preschool and Primary Scale of Intelligence, third version subtasks block design and vocabulary, the Movement Assessment Battery for Children, second edition component aiming and catching, and the Developmental Test of Visual Motor Integration were significantly better in the intervention group. No intervention effect was found on the Strengths and Difficulties Questionnaire. CONCLUSION: The IBAIP leads, 5 years after the early neurobehavioral intervention, to improvements on performance IQ, ball skills, and visual-motor integration at 5.5 years CA.

Interrater reliability of the capacity profile in children with neurodevelopmental disabilities.

OBJECTIVE: To investigate the interrater reliability of the Capacity Profile (CAP) in children with neurodevelopmental disabilities. DESIGN: Cross-sectional study. SETTING: Six rehabilitation centers in the Netherlands. PARTICIPANTS: Children (N=70) with permanent, nonprogressive neurodevelopmental disabilities. INTERVENTIONS: Not applicable. MAIN OUTCOME MEASURES: The CAP is a method to classify additional care needs of children with nonprogressive neurodevelopmental disabilities in 5 domains of body functions: physical health, motor functions, sensory functions, mental functions, and voice/speech functions. The CAP was scored independently by 2 trained physiatrists during an outpatient visit. Interrater reliability was evaluated using an intraclass correlation coefficient (ICC). RESULTS: Interrater reliability of the CAP is as follows: physical functions, ICC=.74; motor functions, ICC=.85; sensory functions, ICC=.61; mental functions, ICC=.85; and voice/speech functions, ICC=.79. CONCLUSIONS: These findings support the interrater reliability of the CAP, when scored during a visit to the rehabilitation center.

Severe fatigue and reduced quality of life in children with hereditary motor and sensory neuropathy 1A.

Severe fatigue and low quality of life are reported by a majority of adult patients with hereditary motor and sensory neuropathy 1A. In children with hereditary motor and sensory neuropathy 1A, the prevalence and impact of fatigue have not been studied yet. In this questionnaire survey, 55 Dutch children (response rate 77%) with genetically confirmed hereditary motor and sensory neuropathy 1A participated (mean age 15 years [standard deviation 2.1]). Prevalence of severe fatigue (based on a cut-off score of the Checklist Individual Strength) was 24%, in contrast to 14% in a Dutch school-based population (P < .05). Almost all quality-of-life scores (measured with the Child Health Questionnaire-Child Form 87) were significantly worse than population norms (P < .05). Fatigue severity was associated significantly (P < .01) with all quality-of-life scores (-0.4 < r < -0.7). In conclusion, severe fatigue and diminished quality of life are more frequent among children with hereditary motor and sensory neuropathy 1A compared to healthy peers. The strong association between fatigue severity and quality of life suggests a negative impact of fatigue on quality of life in these children.

The infant behavioral assessment and intervention program in very low birth weight infants; outcome on executive functioning, behaviour and cognition at preschool age.

BACKGROUND: The Infant Behavioral Assessment and Intervention Program (IBAIP©) improved motor function at 24 months, and mental and behavioural development in high risk subgroups of very low birth weight (VLBW) infants. AIM: To determine IBAIP's effects on executive functioning, behaviour and cognition at preschool age. STUDY DESIGN: Follow-up of a randomised controlled trial (RCT). SUBJECTS: At 44 months corrected age, all 176 VLBW infants were invited for follow-up. Forty-one term born children were assessed for comparison. OUTCOME MEASURES: Visual Attention Task (VAT), Gift delay, Peabody Picture Vocabulary Test III-NL (PPVT), Visual motor integration tests and Miller assessment for preschoolers. Parents completed Behavior Rating Inventory of Executive Function-Preschool (BRIEF-P) and Child Behavior Checklist (CBCL). RESULTS: At preschool age, 76 (88%) children of the intervention group and 75 (83%) children of the control group participated. There were no significant differences between the intervention and the control group. However, positive interaction effects between intervention and infants with bronchopulmonary dysplasia, infants born at gestational age<28 weeks, and infants of low educated mothers were found on CBCL, CBCL and BRIEF-P, and PPVT respectively. Most interaction effects exceeded 1 standard deviation in favour of the intervention children. The 151 VLBW children performed significantly worse than the term born children on the VAT, BRIEF-P and CBCL. CONCLUSION: IBAIP effects in VLBW children did not sustain until preschool age on executive functioning, behaviour and cognition. However, the most vulnerable children had a clinical relevant profit from IBAIP. VLBW children performed worse than the term born children. This study is a follow-up at preschool age of the multi-centre RCT of IBAIP versus usual care in VLBW infants. The RCT was performed in Amsterdam, The Netherlands (IBAIP).