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Aim: To describe the distribution of neuroimaging patterns in a term/late preterm population‐based cohort with cerebral palsy (CP), ascertain associations between neuroimaging patterns and neonatal well‐being, estimate the proportion with antenatal or perinatal timing of neuropathology, and apply this information to the understanding of common mechanisms of brain injury and causal pathways. Method: The cohort for this observational study comprised 1348 persons born between 1999 and 2017 in Victoria, Australia. Using algorithms designed for the study, neonatal well‐being and timing of brain injury were tabulated for the whole cohort and across neuroimaging patterns and birth epochs. Results: Clinical and demographic profiles, neonatal well‐being, and timing of brain injury differed across neuroimaging patterns. An estimated 57% of the cohort had a complicated neonatal period. Timing of brain injury was antenatal in 57% and perinatal in 41%. A decrease in the relative proportions of perinatal timing of brain injury was observed over a period when the rates of CP in live births at term decreased. Interpretation: This study begins to bridge the knowledge gap about causation in CP, moving towards better description of the main mechanisms of brain injury and their contribution within CP cohorts, and facilitating the ability to monitor changes over time and the success of preventive measures. What this paper adds: In a population‐based, term/late preterm cohort with cerebral palsy, 57% had a complicated neonatal period.In the same cohort, 57% had presumed antenatal timing of brain injury.The relative proportion with perinatal injury decreased over time. What this paper adds: In a population‐based, term/late preterm cohort with cerebral palsy, 57% had a complicated neonatal period.In the same cohort, 57% had presumed antenatal timing of brain injury.The relative proportion with perinatal injury decreased over time. This original article is commented on by O'Shea on pages 829–830 of this issue. [ABSTRACT FROM AUTHOR]

Aim: To assess the risks, benefits, and resource implications of home‐blended food for children with gastrostomy tubes compared with a formula diet. Method: This prospective cohort study of children (aged 0–18 years) collected baseline data on gastrointestinal symptoms, nutritional intake, anthropometric outcomes, parent and child quality of life, and resource use. A propensity score‐weighted generalized linear mixed model was used to compare children receiving a home‐blended versus formula diet. Results: Baseline data were obtained for 180 children (2019–2021; 107 males, 73 females; mean age 9 years 7 months [SD 4 years 5 months]). Children receiving a home‐blended diet (n = 104) had similar diagnoses and age but more lived in areas of lower deprivation and parental education was higher compared to the parents of children receiving a formula diet (n = 76). Children receiving home‐blended diets had significantly better gastrointestinal scores than those receiving formula diets (B = 13.8, p < 0.001). The number of gut infections and tube blockages were similar between the two groups but with fewer stoma site infections in the group receiving home‐blended food. Children receiving a home‐blended diet had more fibre in their diet compared to children receiving a formula diet. Interpretation: Home‐blended diets should be seen as a safe option for children who are gastrostomy‐fed unless clinically contraindicated. Equality of access to home‐blended diets for children with gastrostomy should be assessed by local clinical teams. What this paper adds: Children with gastrostomy receiving a home‐blended diet had fewer gastrointestinal symptoms compared to children receiving a formula diet.Children with gastrostomy receiving a home‐blended diet had no more complications than children receiving a formula diet. What this paper adds: Children with gastrostomy receiving a home‐blended diet had fewer gastrointestinal symptoms compared to children receiving a formula diet.Children with gastrostomy receiving a home‐blended diet had no more complications than children receiving a formula diet. [ABSTRACT FROM AUTHOR]

Aim: To systematically review the effectiveness of caregiver and parent skills training programs, including caregiver‐mediated interventions, for caregivers of individuals with neurodevelopmental disorders. Method: We conducted a systematic review with a random‐effects meta‐analysis. We searched 11 electronic databases through July 2021 and used a snowball methodology to locate relevant articles of randomized controlled trials. Effect size estimates were pooled using Hedges' g from data extracted from study reports and through author requests using random‐effects meta‐analyses for three child outcome categories (child development, adaptive behavior, and problem behavior) and three caregiver outcome categories (parenting skills and knowledge, psychological well‐being, and interpersonal family relations). Results: We located 44 910 records, from which 75 randomized controlled trials involving 4746 individuals with neurodevelopmental disorders and their caregivers were included. Random‐effects meta‐analyses showed improvements in child development (g = 0.30; 99% confidence interval [CI] = 0.07–0.53) and reduction in reported problem behaviors (g = 0.41; 99% CI = 0.24–0.59), but not a statistically significant improvement in adaptive behavior (g = 0.28; 99% CI = −0.42 to 0.98). Caregivers showed improvements in parenting skills and knowledge (g = 0.72; 99% CI = 0.53–0.90), psychological well‐being (g = 0.52; 99% CI = 0.34–0.71), and interpersonal family relations (g = 0.76; 99% CI = 0.32–1.20). Interpretation: Caregiver skills training programs benefit both caregivers and children with neurodevelopmental disorders. Skills training programs improve child development and behavior, improve parenting skills, reduce caregiver mental health issues, and improve family functioning. Programs using culturally appropriate training material to improve the development, functioning, and participation of children within families and communities should be considered when caring for children with neurodevelopmental disorders. What this paper adds: Caregiver skills training programs are effective interventions for both caregivers and children.Children with neurodevelopmental disorders benefit from improvements in development and reduction of problematic behaviors.Caregivers benefit from enhanced skills and psychological well‐being.Improvements in interpersonal family relationships have also been documented. What this paper adds: Caregiver skills training programs are effective interventions for both caregivers and children.Children with neurodevelopmental disorders benefit from improvements in development and reduction of problematic behaviors.Caregivers benefit from enhanced skills and psychological well‐being.Improvements in interpersonal family relationships have also been documented. This systematic review is commented on by Lau on pages 684–685 of this issue. [ABSTRACT FROM AUTHOR]

Aim Method Results Interpretation To identify and describe assessment tools used to measure the impact of comorbidities on postoperative outcomes in children with complex chronic conditions (CCC).This was a scoping review using five electronic databases. The search was conducted in March 2022 by a medical librarian. There were no date or language restrictions. Included studies were full‐text articles published in peer‐reviewed journals that described a tool used to measure the impact of comorbidities in children with CCC to assess postoperative outcomes. A standardized data charting tool was used.A total of 2157 articles were retrieved. Five studies reporting on six comorbidity measures met inclusion criteria. All were cohort studies and were secondary analyses of data from an administrative database (n = 4) or a patient registry (n = 1). Sample sizes ranged from 645 to 25 747 participants. One paper described the assessment of reliability. Only one form of validity – predictive validity – was assessed in three papers for five measures.Findings from this scoping review revealed a paucity of comorbidity assessment tools validated for use with children with CCC; significant conceptual and measurement challenges exist in the current scientific literature. [ABSTRACT FROM AUTHOR]

Aim: To describe visual function in children with Joubert syndrome and to investigate its possible association with diagnostic and developmental aspects. Method: This retrospective cross‐sectional work included 59 patients (33 male; mean age 9 years 2 months, standard deviation 6 years 3 months, range 4 months to 23 years) diagnosed with Joubert syndrome from January 2002 to December 2020. Data about clinical (neurological, neuro‐ophthalmological, developmental/cognitive) and diagnostic (e.g. genetic testing, neuroimaging, systemic involvement) evaluations were collected in a data set during a review of medical records. Clinical and diagnostic variables were described in terms of raw counts and percentages. A χ2 test was conducted to investigate their association with neuropsychological skills. Results: Ocular motor apraxia was highly represented in our cohort (75%), with a high prevalence of refractive defects and retinal abnormalities. Developmental delay/intellectual disability was frequent (in 69.5% of the sample), associated with retinal dystrophy (p = 0.047) and reduced visual acuity both for near (p = 0.014) and for far distances (p = 0.017). Interpretation: On the basis of the relevance of oculomotor and perceptual alterations and their impact on overall and cognitive impairment, we encourage early and multidisciplinary assessment and follow‐up of visual function in children with Joubert syndrome. This would help in planning a personalized rehabilitation to sustain functional vision. Further studies will be important to explore the link between biological aspects and global functioning in children with Joubert syndrome. What this paper adds: Perceptual deficits and oculomotor impairments frequently coexist in Joubert syndrome.Retinal dysfunction may be present despite the absence of funduscopic abnormalities.Both perceptual and oculomotor impairments negatively affect cognitive development in Joubert syndrome. What this paper adds: Perceptual deficits and oculomotor impairments frequently coexist in Joubert syndrome.Retinal dysfunction may be present despite the absence of funduscopic abnormalities.Both perceptual and oculomotor impairments negatively affect cognitive development in Joubert syndrome. [ABSTRACT FROM AUTHOR]

Aim: To describe caregiver experiences, perceptions, and device preferences between a modified ride‐on car (MROC) and an Explorer Mini, including perceived changes in participation, barriers, and benefits for young children with cerebral palsy (CP) classified in Gross Motor Function Classification System (GMFCS) level V. Method: A subset of data were analyzed from a larger multisite study. Semi‐structured interviews were conducted with 10 caregivers of children with CP in GMFCS level V across a 16‐week trial with two mobility devices. Each interview was audio‐recorded, transcribed verbatim, and analysed using constant comparison methods. Results: Seven of 10 caregivers preferred the Explorer Mini over the MROC. Four themes emerged. One related to the perceived benefits and barriers of each device (ease and convenience is essential) and three related to perceived changes in participation: (1) autonomy enacted through mobility; (2) belonging and being present; and (3) participation recognized as an area of growth. Interpretation: Despite limited consideration of powered mobility for this population, caregivers of children in GMFCS level V reported similar benefits and barriers compared to children in other GMFCS levels shown in the literature. Particularly, caregivers perceived positive changes in their child's participation and recognized the ability for continued improvements in participation when using powered mobility. What this paper adds: Children with cerebral palsy classified in Gross Motor Function Classification System level V are often excluded from powered mobility use and research.Despite this, caregivers reported positive experiences for their children.Caregivers felt that participation in powered mobility led to increased autonomy and overall sense of inclusion and belonging for their children.Most caregivers preferred the Explorer Mini over the modified ride‐on car but recognized that both devices had benefits and barriers to use. What this paper adds: Children with cerebral palsy classified in Gross Motor Function Classification System level V are often excluded from powered mobility use and research.Despite this, caregivers reported positive experiences for their children.Caregivers felt that participation in powered mobility led to increased autonomy and overall sense of inclusion and belonging for their children.Most caregivers preferred the Explorer Mini over the modified ride‐on car but recognized that both devices had benefits and barriers to use. This original article is commented on by Jahan and Islam on pages 276–277 of this issue. [ABSTRACT FROM AUTHOR]

Magnetoencephalography (MEG) is a neurophysiological technique based on the detection of brain magnetic fields. Whole‐head MEG systems typically house a few hundred sensors requiring cryogenic cooling in a rigid one‐size‐fits‐all (commonly adult‐sized) helmet to keep a thermal insulation space. This leads to an increased brain‐to‐sensor distance in children, because of their smaller head circumference, and decreased signal‐to‐noise ratio. MEG allows detection and localization of interictal and ictal epileptiform discharges, and pathological high frequency oscillations, as a part of the presurgical assessment of children with refractory focal epilepsy, where electroencephalography is not contributive. MEG can also map the eloquent cortex before surgical resection. MEG also provides insights into the physiopathology of both generalized and focal epilepsy. On‐scalp recordings based on cryogenic‐free sensors have demonstrated their use in the field of childhood focal epilepsy and should become a reference technique for diagnosing epilepsy in the paediatric population. What this paper adds: Magnetoencephalography (MEG) contributes to the diagnosis and understanding of paediatric epilepsy.On‐scalp MEG recordings demonstrate some advantages over cryogenic MEG. What this paper adds: Magnetoencephalography (MEG) contributes to the diagnosis and understanding of paediatric epilepsy.On‐scalp MEG recordings demonstrate some advantages over cryogenic MEG. [ABSTRACT FROM AUTHOR]

Aim: To establish the burden of respiratory illness in cerebral palsy (CP) on the Western Australian health care system by quantifying the costs of respiratory hospitalizations in children with CP, compared with non‐respiratory hospitalizations. Method: A 2‐year (2014–2015) retrospective study using linked hospital data (excluding emergency department visits), in a population of children with CP in Western Australia aged 18 years and under (median age at hospitalization 7 years; interquartile range 5–12 years). Results: In 671 individuals (57% male) there were 726 emergency hospitalizations, and 1631 elective hospitalizations. Although there were more elective hospitalizations, emergency hospitalizations were associated with longer stays in hospital, and more days in an intensive care unit, resulting in a higher total cost of emergency hospitalizations than elective hospitalizations (total costs: emergency AU$7 748 718 vs elective AU$6 738 187). 'Respiratory' was the leading cause of emergency hospitalizations, contributing to 36% of all emergency admission costs. For a group of high‐cost inpatient users (top 5% of individuals with the highest total inpatient costs) the most common reason for hospitalization was 'respiratory'. Where non‐respiratory admissions were complicated by an additional respiratory diagnosis, length of stay was greater. Interpretation: Respiratory hospitalizations in CP are a significant driver of health care costs. In the paediatric group, they are a burden for a subgroup of children with CP. What this paper adds: Respiratory illness is the most costly area for unplanned, emergency hospitalizations for children and young people with cerebral palsy.The top 5% of individuals with the highest total inpatient costs account for a disproportionate amount of health care costs. What this paper adds: Respiratory illness is the most costly area for unplanned, emergency hospitalizations for children and young people with cerebral palsy.The top 5% of individuals with the highest total inpatient costs account for a disproportionate amount of health care costs. [ABSTRACT FROM AUTHOR]

Aim: To undertake a systematic review and meta‐analysis exploring school‐age neurodevelopmental outcomes of children after low‐grade intraventricular haemorrhage (IVH). Method: The published and grey literature was extensively searched to identify observational comparative studies exploring neurodevelopmental outcomes after IVH grades 1 and 2. Our primary outcome was neurodevelopmental impairment after 5 years of age, which included cognitive, motor, speech and language, behavioural, hearing, or visual impairments. Results: This review included 12 studies and over 2036 infants born preterm with low grade IVH. Studies used 30 different neurodevelopmental tools to determine outcomes. There was conflicting evidence of the composite risk of neurodevelopmental impairment after low‐grade IVH. There was evidence of an association between low‐grade IVH and lower IQ at school age (−4.23, 95% confidence interval [CI] –7.53, −0.92, I2 = 0%) but impact on school performance was unclear. Studies reported an increased crude risk of cerebral palsy after low‐grade IVH (odds ratio [OR] 2.92, 95% CI 1.95, 4.37, I2 = 41%). No increased risk of speech and language impairment or behavioural impairment was found. Few studies addressed hearing and visual impairment. Interpretation: This systematic review presents evidence that low‐grade IVH is associated with specific neurodevelopmental impairments at school age, lending support to the theory that low‐grade IVH is not a benign condition. What this paper adds: The functional impact of low‐grade intraventricular haemorrhage (IVH) at school age is unknown.Low‐grade IVH is associated with a lower IQ at school age.The risk of cerebral palsy is increased after low‐grade IVH.Low‐grade IVH is not associated with speech and language impairment. What this paper adds: The functional impact of low‐grade intraventricular haemorrhage (IVH) at school age is unknown.Low‐grade IVH is associated with a lower IQ at school age.The risk of cerebral palsy is increased after low‐grade IVH.Low‐grade IVH is not associated with speech and language impairment. [ABSTRACT FROM AUTHOR]

Fragile X syndrome (FXS) is a neurodevelopmental disorder caused by a full mutation (> 200 CGG repeats) in the FMR1 gene. FXS is the leading cause of inherited intellectual disabilities and the most commonly known genetic cause of autism spectrum disorder. Children with FXS experience behavioral and sleep problems, anxiety, inattention, learning difficulties, and speech and language delays. There are no approved medications for FXS; however, there are several interventions and treatments aimed at managing the symptoms and improving the quality of life of individuals with FXS. A combination of non‐pharmacological therapies and pharmacotherapy is currently the most effective treatment for FXS. Currently, several targeted treatments, such as metformin, sertraline, and cannabidiol, can be used by clinicians to treat FXS. Gene therapy is rapidly developing and holds potential as a prospective treatment option. Soon its efficacy and safety in patients with FXS will be demonstrated. What this paper adds: Targeted treatment of fragile X syndrome (FXS) is the best current therapeutic approach.Gene therapy holds potential as a prospective treatment for FXS in the future. [ABSTRACT FROM AUTHOR]

Aim: To identify, map, and describe outcome measurement domains and instruments used within a community setting to assess respiratory health in children and young people aged 1 to 18 years, diagnosed with cerebral palsy (CP). Method: A scoping review methodology informed structured searches in nine databases, grey literature, and registries, conducted in August 2021 (updated in February 2023). Articles were screened for eligibility by two independent researchers. Any outcome measurement instruments used to assess respiratory health or associated impact were extracted, categorized, and mapped to health and health‐related domains of the International Classification of Functioning, Disability, and Health. Results: Seventy‐six outcome measurement instruments were identified across 78 articles worldwide between 1970 and 2023. These were categorized into 'Body functions and structures' (n = 20), 'Activity and performance' (n = 22), and 'Participation and quality of life' (n = 19), with a further 15 mapped to 'Health care resources use'. Interpretation: No consensus of 'what' to measure and 'how' to measure respiratory health in children and young people with CP was found. Moreover, many measures were not replicable in individuals with more severe forms of CP, excluding those at increased risk of respiratory‐related morbidity and mortality. Further research is required to agree important outcome domains and associated measures in research and clinical practice. What this paper adds: A limited number and size of experimental designs were found.Seventy‐six measures were identified to assess respiratory health in cerebral palsy.No consensus was found in 'what' or 'how' to measure respiratory health.Many measures were not replicable in children and young people at risk of poorer respiratory health outcomes.Children and young people with comorbidities and learning disability were frequently excluded from studies. [ABSTRACT FROM AUTHOR]

Aim: To investigate the efficacy, safety, and impact on quality of life (QoL) of an oral formulation of 320 μg/mL glycopyrronium designed for children. Method: A double‐blind, placebo‐controlled SALIVA (Sialanar plus orAl rehabiLitation against placebo plus oral rehabilitation for chIldren and adolescents with seVere sialorrhoeA and neurodisabilities) trial was conducted. Children (3–17 years) with neurodisabilities and severe sialorrhoea (modified Teachers Drooling Scale ≥6) were randomized to 320 μg/mL glycopyrronium or placebo, in addition to non‐pharmacological standard care. Results: Of 87 participants, 44 were aged 10 years or under and 43 had cerebral palsy. The primary endpoint, change in total Drooling Impact Scale (DIS) score from baseline to day 84, was significantly greater (improved) with 320 μg/mL glycopyrronium versus placebo (median [quartile 1, quartile 3] –29.5 [−44.5, 0] vs −1 [−16, 5]; p < 0.001), an effect also observed at day 28 (median − 25 vs −2; p < 0.01). Significant reduction in bibs/clothes used per day was seen with glycopyrronium versus placebo at day 84 (median − 2 vs 0; p < 0.01). Glycopyrronium significantly improved DIS items 9 and 10 related to the extent that drooling affects the child's and family's life (p ≤ 0.03). Adverse events were reported by 77.3% and 69.8% of children with glycopyrronium and placebo respectively; the most common treatment‐related adverse event was constipation (20.5% and 16.3%). Interpretation: The formulation of 320 μg/mL glycopyrronium significantly improved drooling and reduced its impact on QoL, with good tolerability in children with neurodisabilities. What this paper adds: The formulation of 320 μg/mL glycopyrronium significantly improved Drooling Impact Scale score versus placebo at day 84.The formulation reduced the impact of drooling on the child's and family's quality of life.There were no safety or tolerability concerns with this specific formulation. [ABSTRACT FROM AUTHOR]

Hydrocephalus is rarely described in Joubert‐Boltshauser syndrome (JBTS). The aim of this study was to investigate whether this association is a chance occurrence or potentially signifies a new phenotypic subtype. The databases of Wolfson Medical Center, Sourasky Medical Center, and EB's personal collection were reviewed. Records from an additional family were obtained from RG. The patients' medical records, prenatal ultrasounds, and magnetic resonance imaging were assessed. In addition, we reviewed the medical literature for the association of ventriculomegaly/hydrocephalus (VM/HC) in JBTS. Only seven cases (from five families) were found with prenatal onset of VM/HC, diagnosed during the second trimester; three pregnancies were terminated, one was stillborn and three were born, of which one died within a week, and another died at the age of 6 years. Additional central nervous system findings included dysgenesis of the corpus callosum, delayed sulcation, polymicrogyria, and pachygyria. We found 16 publications describing 54 patients with JBTS and VM/HC: only five were diagnosed at birth and three were diagnosed prenatally. Hydrocephalus is extremely rare in JBTS. The recurrence of this association, reported in several publications in multiple family members, suggests that it might represent a new phenotypic subtype of JBTS possibly associated with specific genes or variants. Further genetic studies are needed to confirm this hypothesis. What this paper adds: The association of fetal hydrocephalus with Joubert‐Boltshauser syndrome (JBTS) is very rare but not a chance association.This association represents a new phenotypic subtype of JBTS possibly linked to specific genes or variants. [ABSTRACT FROM AUTHOR]

Aim: To examine whether antenatal diagnosis modifies relationships between neonatal brain volumes and 18‐month neurodevelopmental outcomes in children with transposition of the great arteries (TGA). Method: In a retrospective cohort of 139 children with TGA (77 antenatally diagnosed), we obtained total brain volumes (TBVs) on pre‐ (n = 102) and postoperative (n = 112) magnetic resonance imaging. Eighteen‐month neurodevelopmental outcomes were assessed using the Bayley Scales of Infant and Toddler Development, Third Edition. Generalized estimating equations with interaction terms were used to determine whether antenatal diagnosis modified associations between TBVs and neurodevelopmental outcomes accounting for postmenstrual age at scan, brain injury, and ventricular septal defect. Results: Infants with postnatal diagnosis had more preoperative hypotension (35% vs 14%, p = 0.004). The interactions between antenatal diagnosis and TBVs were significantly related to cognitive (p = 0.003) outcomes. Specifically, smaller TBVs were associated with lower cognitive scores in infants diagnosed postnatally; this association was attenuated in those diagnosed antenatally. Interpretation: Antenatal diagnosis modifies associations between neonatal brain volume and 18‐month cognitive outcome in infants with TGA. These findings suggest that antenatal diagnosis may be neuroprotective, possibly through improved preoperative clinical status. These data highlight the need to improve antenatal diagnosis rates. What this paper adds: Antenatal diagnosis of transposition of the great arteries modified relationships between neonatal brain volume and neurodevelopment.Smaller brain volumes related to poorer cognitive scores with postnatal diagnosis only.There was more preoperative hypotension in the postnatal diagnosis group. [ABSTRACT FROM AUTHOR]

Aim: To develop robust multivariable prediction models for non‐response to (1) submandibular botulinum neurotoxin A (BoNT‐A) injections and (2) concurrent submandibular and parotid (four‐gland) injections, to guide treatment decisions for drooling in children with neurodevelopmental disabilities, including cerebral palsy. Method: This was a retrospective cohort study including 262 children (155 males/107 females, median age 7 years 11 months [IQR 5 years 1 month], range 4 years 0 months – 17 years 11 months) receiving submandibular injections and 74 children (52 males/22 females, median age 7 years 7 months [IQR 4 years 3 months], range 4 years 9 months – 18 years 8 months) receiving four‐gland injections. Multivariable logistic regression analyses were used to estimate associations between candidate predictors and non‐response 8 weeks after injection. Results: Ninety‐six children (37%) were non‐responders to submandibular injections, for which developmental age was the strongest predictor (adjusted odds ratio [aOR] 2.13; 95% confidence interval [CI] 1.02–4.45 for developmental age <4 years or 4–6 years with IQ <70). Other characteristics that showed a trend towards an increased risk of non‐response were diagnosis, sex, and head position. Thirty‐four children (46%) were non‐responders to four‐gland injections, for which tongue protrusion (aOR 3.10; 95% CI 1.14–8.43) seemed most predictive, whereas multiple preceding submandibular injections (aOR 0.34; 95% CI 0.10–1.16) showed a trend towards being protective. Predictors were, however, unstable across different definitions of non‐response and both models (i.e. submandibular and four‐gland) had insufficient discriminative ability. Interpretation: Potential predictors of non‐response to BoNT‐A injections were identified. Nevertheless, the developed prediction models seemed inadequate for guidance of treatment decisions. What this paper adds: Developmental age seemed most predictive of non‐response to submandibular botulinum neurotoxin A injections.Non‐response to concurrent submandibular and parotid injections was best predicted by tongue protrusion and number of previous injections.Multivariable prediction models including these clinical characteristics were unable to discriminate well.Predictors differed when non‐response was defined using alternative outcome measures. [ABSTRACT FROM AUTHOR]

Aim: The aim of this observational study was to determine the immune status and function in young adults with cerebral palsy (CP) in comparison to typically developing individuals. Method: Blood samples from 12 individuals with CP (five males, seven females; mean age: 25 years 1 month (5 years 9 months); age range: 19–38 years) and 17 typically developing individuals (eight males, nine females; mean age: 31 years 4 months (6 years 2 months); age range: 20–40 years) were collected before, immediately after, and 1 hour after 45 minutes of frame running or running respectively. Independent t‐tests were used to compare heart rate, level of exertion, and baseline cell proportions between groups. Mixed model analysis of variance was utilized to investigate immune cell responses to exercise across groups. Results: Baseline levels of gamma delta (TCRγδ+) T‐cells were significantly higher (absolute percentage: +2.65, p = 0.028) in the individuals with CP. Several cell populations showed similar significant changes after exercise in both CP and typically developing groups. Cytotoxic (CD8+) T‐cells were only significantly elevated immediately after exercise in the typically developing participants (p < 0.01). Individuals with CP exhibited significantly lower heart rates (−11.1%, p < 0.01), despite similar ratings of perceived exertion. Interpretation: Elevated baseline TCRγδ+ T‐cells may indicate low‐grade inflammation in adults with CP. Although most of the cell populations showed typical responses to endurance exercise, the absence of response in CD8+ T‐cells in individuals with CP may indicate the need for higher intensity during exercise. What this paper adds: TCRγδ+ T‐cell baseline levels are elevated in adults with cerebral palsy (CP).The CD8+ T‐cell response to exercise was blunted in adults with CP.Exercise intensity is decisive for CD8+ T‐cell responses in individuals with CP. [ABSTRACT FROM AUTHOR]

Aim: To examine how school environment, physical functioning problems, and behavioral problems explain levels of school participation (i.e. attendance and involvement) among young people with craniofacial microsomia (CFM) and other childhood‐onset disabilities, and whether participation‐focused caregiver strategies play a role in these relationships. Method: We conducted secondary analyses of a subset of data (n = 260 families: 120 with CFM and 140 with other childhood‐onset disabilities) from the second follow‐up phase of a longitudinal cohort study. We applied structural equation modeling with data collected from the Participation and Environment Measure – Children and Youth version, the Child Behavior Checklist, and the Pediatric Quality of Life Inventory physical functioning scale. Results: Model fit was acceptable to close (comparative fit index = 0.973; root mean square error of approximation = 0.055; standardized root mean squared residual = 0.043; Tucker–Lewis index = 0.958). School environmental support had a positive effect on young people's participation attendance and involvement, and physical functioning problems had a negative effect on participation involvement. The number of disclosed caregiver strategies had a significant positive effect on the relationship between school environmental support and school participation attendance. Interpretation: Findings confirm the effect of school environmental support and physical functioning problems on school participation and highlight the role of participation‐focused caregiver strategies to intensify the positive effect of school environmental support on school participation attendance. What this paper adds: Environmental support had a positive effect on participation attendance and involvement.Physical functioning problems had a negative effect on school participation involvement.Disclosed caregiver strategies intensified the impact of environmental support on participation attendance. [ABSTRACT FROM AUTHOR]

Aim: To investigate parents' preferences and motivations for receiving and discussing prognostic genetic test results. Method: We used a cross‐sectional, interpretive description qualitative study design. We collected data through semi‐structured interviews with Australian parents, which we analysed using reflexive thematic analysis. Results: Parents (n = 32) had a child or children with a genetic neurodevelopmental condition, such as fragile X syndrome, DiGeorge (22q11.2 deletion) syndrome, or Angelman syndrome. Parents of mildly impacted or older children were tolerant to prognostic uncertainty. Parents found conversations about their child's prognosis emotional and preferred to discuss their child's potential strengths and challenges. While most were enthusiastic about prognostic tests and described many motivations for testing, the potential for prognostic information to contribute to a loss of hope and stigmatizing societal views were also discussed. Interpretation: Parents had mixed preferences and motivations for acquiring prognostic genetic information about their child, contrasting evidence in other contexts such as cancer where parents typically have minimal tolerance of uncertainty. Health professionals should consider strength‐based framing of prognostic information gained from current and emerging technologies when returning results to families. What this paper adds: Parents had varied views about receiving prognostic information on their children's neurodevelopmental condition.Some parents preferred prognostic uncertainty about their children's genetic neurodevelopmental condition. [ABSTRACT FROM AUTHOR]

Aim: To examine the effects of the Pathways and Resources for Engagement and Participation (PREP) intervention during the COVID‐19 pandemic on (1) activity performance and satisfaction, and (2) motor, cognitive, and affective body functions. Method: An interrupted time‐series design with multiple baselines across 21 young people (13 females, eight males) aged 16 to 25 years (median = 21 years 5 months) with physical disabilities was employed. The young people engaged in an 8‐week self‐chosen leisure activity (e.g. football, piano, photography) at their home or community. The Canadian Occupational Performance Measure (COPM) assessed activity performance and satisfaction weekly. Mental health problems, including affective and cognitive outcomes, were assessed weekly using the Behavior Assessment System for Children, Third Edition. Motor functions (e.g. trunk control, reaching, strength) were assessed biweekly. Linear mixed‐effects models were used. Results: The intervention had large effects on activity performance (0.78) and satisfaction (0.88) with clinically significant change in COPM scores (2.6 [95% confidence interval {CI}: 2.0–3.2] and 3.2 points [95% CI: 2.4–3.9] respectively). Young people without mental health problems at baseline benefited more from the intervention (p = 0.028). Improvements in at least one domain of body function occurred in 10 young people especially for motor outcomes. Interpretation: Results demonstrate the effectiveness of PREP during adverse times and suggest benefits going beyond participation, involving outcomes at the body‐function level. What this paper adds: Environmental‐based interventions can improve participation even during adverse times such as the COVID‐19 pandemic.Significant improvement with large effect sizes occurred in both activity performance and satisfaction.Intervention was effective for all; those without mental health problems benefited more.Improvements in body‐function outcomes were partially observed, especially in motor‐related outcomes.Body functions may improve through participation even if not targeted by the intervention. [ABSTRACT FROM AUTHOR]

Aim: To identify and analyse ways in which parents are supported to deliver rehabilitation exercises to their child after traumatic brain injury (TBI), conceptualized as strategies. Method: A systematic search was completed using seven online databases and three grey literature databases, from inception to November 2021. The included studies focused on physical rehabilitation in children after TBI with the involvement of parents as hands‐on deliverers or facilitators of rehabilitation (e.g. supervising the exercise). Intervention descriptions were reviewed to identify strategies; this was followed by fine‐grained analysis using the Behaviour Change Wheel to identify intervention components. Risk of bias was analysed using the revised Cochrane Risk‐of‐Bias Tool for Randomized Trials or the Risk Of Bias In Non‐randomized Studies ‐ of Interventions. Results: Six interventions including 211 participants and one trial protocol met the inclusion criteria. All studies included a proportion of children diagnosed with TBI and four studies included mixed samples of acquired brain injury or cerebral palsy. All interventions included elements of goal setting and instruction. Interpretation: Interventions focus heavily on the initiation of physical rehabilitation, but focus less on the longer‐term maintenance of rehabilitation delivery. Further research should integrate perspectives from parents to inform the development of new interventions. What this paper adds: Parents need support to deliver or supervise rehabilitation exercises.The interventions identified in this review supported goal setting, action planning, and learning rehabilitation exercises.Interventions focused primarily on the initiation of exercises but less on maintenance.Rehabilitation is complex and new approaches are needed to better support parents. [ABSTRACT FROM AUTHOR]

Aim: To investigate clinicoradiological features associated with epilepsy, its resolution, and drug resistance in children with cerebral palsy (CP). Method: Data were gathered from the New South Wales/Australian Capital Territory CP Register, encompassing children with CP born between 2003 and 2015 (n = 1916). Clinical features and the severity of impairments were compared among three groups: children with current epilepsy (n = 604), those with resolved epilepsy by age 5 years (n = 109), and those without epilepsy (n = 1203). Additionally, a subset of the registry cohort attending Children's Hospital Westmead (n = 256) was analysed to compare epilepsy and treatment characteristics between drug‐responsive (n = 83) and drug‐resistant groups (n = 147) using logistic regression and hierarchical cluster analysis. Results: Manual Ability Classification System levels IV and V, intellectual impairment, and vision impairment were found to be associated with epilepsy in children with CP on multivariable analysis (p < 0.01). Moderate to severe intellectual impairment and bilateral spastic CP were independent positive and negative predictors of epilepsy persistence at the age of 5 years respectively (p < 0.05). Microcephaly and multiple seizure types were predictors of drug‐resistant epilepsy (area under the receiver operating characteristic curve of 0.83; 95% confidence interval 0.77–0.9). Children with a known genetic cause (14%) and CP epilepsy surgery group (4.3%) formed specific clinical subgroups in CP epilepsy. Interpretation: Our study highlights important clinical associations of epilepsy, its resolution, and treatment response in children with CP, providing valuable knowledge to aid in counselling families and identifying distinct prognostic groups for effective medical surveillance and optimal treatment. What this paper adds: Severe motor and non‐motor impairments in cerebral palsy (CP) increase epilepsy risk.Epilepsy more likely resolves in bilateral spastic and milder CP impairments.Epilepsy in CP often manifests at an early age with multiple seizure types and high drug resistance.Children with a known genetic cause and CP epilepsy surgery group represent distinct clinical subgroups. [ABSTRACT FROM AUTHOR]

Aim: To investigate the prevalence and clinical manifestations of reading, writing, and mathematics disorders in children with cerebral palsy (CP). We explored how the clinical profile of these children differed from those with specific learning disorders (SLDs), taking into account several factors, particularly IQ scores, neuropsychological aspects, and the presence of a visual impairment. Method: A prospective cross‐sectional study was conducted in 42 children with CP (mean age 9 years 8 months; SD = 2 years 2 months) and 60 children with SLDs (mean age 10 years; SD = 1 year 7 months). Clinical characteristics, neuromotor and cognitive profiles, neuropsychological aspects (speech performance, academic skills, visual attention, phonological awareness, working memory), and signs of visual impairment (visual acuity, contrast sensitivity, visual field, oculomotor functions) were assessed. A machine learning approach consisting of a random forest algorithm, where the outcome was the diagnosis and the covariates were the clinical variables collected in the sample, was used for the analyses. Results: About 59% of the children with CP had reading, writing, or mathematics disorders. Children with CP with learning disorders had a low performance IQ, normal phonological awareness, and working memory difficulties, whereas children with SLDs had normal performance IQ, impaired phonological awareness, and mild working memory difficulties. There were no differences in verbal IQ between the two groups. Interpretation: Learning disorders are frequently associated with CP, with different clinical characteristics, compared with SLDs. Assessment of academic skills is mandatory in these children, even if the IQ is normal. At school age, specific interventions to promote academic skills in children with CP could be a major rehabilitative goal. What this paper adds: Reading, writing, and mathematics disorders in cerebral palsy have specific clinical characteristics.Their underlying mechanisms differ from those described in specific learning disorders.Working memory impairment can be considered a hallmark of learning disorders in children with cerebral palsy. [ABSTRACT FROM AUTHOR]

Aim: To examine the extent to which estimates of a latent trait or underlying construct of motor ability differ in infants born at term and preterm, based on caregiver ratings of the motor domain of PediaTrac v3.0. Method: The sample consisted of 571 caregiver–infant dyads (331 born at term, 240 born preterm), 48% female, with 51.7% of caregivers identifying as an ethnic minority. Latent trait of motor ability was estimated based on item response theory modeling. Gestational group differences (term and preterm birth) were examined at the newborn/term‐equivalent, 2‐, 4‐, 6‐, 9‐, and 12‐month time points. Results: Caregiver ratings of latent trait of motor ability were reliably modeled across the range of abilities at each time point. While the group born preterm exhibited significantly more advanced motor abilities at the term‐equivalent time point, by 6 months the group born at term was more advanced. Biological sex difference main and interaction effects were not significant. Interpretation: Caregivers provided reliable, longitudinal estimates of motor ability in infancy, reflecting important differences in the motor development of infants born at term and preterm. The findings suggest that significant motor development occurs in infants born preterm from birth to the term‐equivalent time point and provide a foundation to examine motor growth trajectories as potential predictors in the early identification of neurodevelopmental conditions and needs. What this paper adds: Longitudinal caregiver ratings of motor function in early infancy yielded reliable estimates of the latent trait of motor ability.Motor ability at the term‐equivalent time point was higher in infants born preterm than infants born at term. [ABSTRACT FROM AUTHOR]

In 2022, the International League Against Epilepsy revised their classification of epilepsy syndromes for clinicians to better understand the relationships between different epilepsy syndromes, their underlying causes, and their associated developmental and behavioral features. This review highlights portions of the current classification with an emphasis on epilepsy syndromes that readily present with developmental challenges and provides a unique framework, based on electroencephalography, to easily identify and understand these syndromes. Included in this review are a helpful categorization scheme with visual aid, descriptions of updated epilepsy syndromes, figures of relevant identifiers of syndrome and information regarding future directions toward treatment and research. Covered syndromes include developmental and epileptic encephalopathy, Dravet syndrome, Rasmussen syndrome, and infantile epileptic spasm syndrome, among others. What this paper adds: The revised epilepsy syndrome classification by the International League Against Epilepsy aims to improve the outcomes for children with epilepsy.The electroencephalography features of epilepsy syndromes are grouped based on a categorization model.This model allows clinicians to understand overlapping phenotypes and aid with both identification and diagnosis. [ABSTRACT FROM AUTHOR]

Aim: To investigate the seizure course of PCDH19 clustering epilepsy (PCDH19‐CE) in a cohort of female children in China. Method: This ambidirectional cohort study examined 113 female patients with PCDH19‐CE through multicentre collaboration. Prognostic factors for seizure freedom were evaluated by multivariate Cox regression analysis. Results: The median seizure course period from seizure onset was 6 years 6 months. Of 113 patients, 78% and 56% experienced seizure freedom for at least 1 year and at least 2 years respectively. In patients younger than 5 years (n = 30), 5 to 10 years (n = 52), and older than 10 years (n = 31), 57%, 81%, and 94% experienced at least 1 year of seizure freedom, and 32%, 52%, and 84% experienced at least 2 years of seizure freedom, respectively. However, 58% (65 out of 113) relapsed at least once after more than 1 year of seizure freedom without trigger exposure (40%) or because of common triggers, including fever (43%) and antiseizure medication (ASM) reduction (29%). There was an 84% risk of seizure relapse after ASM reduction attempts. The likelihood of seizure freedom decreased with early age at seizure onset and developmental delay. Interpretation: Patients with PCDH19‐CE exhibit increasing seizure freedom with age, but there is a risk of relapse. ASM reduction in children younger than 10 years old requires caution. Patients with early seizure onset and developmental delay have a reduced chance of seizure freedom. What this paper adds: The seizure freedom rate in PCDH19 clustering epilepsy gradually increases with age.The disease course is characterized by relapsing–remitting seizures.Antiseizure medication reduction requires caution for patients younger than 10 years of age.Patients with early seizure onset and developmental delay are less likely to achieve seizure freedom. [ABSTRACT FROM AUTHOR]

Aim: To describe the relationships between outpatient encounters, continuity of care, and unplanned hospital care in children/young people with cerebral palsy (CP). Method: In this population‐based data‐linkage cohort study we included children/young people with CP identified in the New South Wales/Australian Capital Territory CP Register (birth years 1994–2018). We measured the frequency of outpatient encounters and unplanned hospital care, defined as presentations to emergency departments and/or urgent hospital admissions (2015–2020). Continuity of outpatient care was measured using the Usual Provider of Care Index (UPCI). Results: Of 3267 children/young people with CP, most (n = 2738, 83.8%, 57.6% male) had one or more outpatient encounters (123 463 total encounters, median six outpatient encounters per year during childhood). High UPCI was more common in children/young people with mild CP (Gross Motor Function Classification System levels I–III, with no epilepsy or no intellectual disability), residing in metropolitan and areas of least socioeconomic disadvantage. Low UPCI was associated with four or more emergency department presentations (adjusted odds ratio [aOR] 2.34; 95% confidence interval [CI] 1.71–3.19) and one or more urgent hospital admissions (aOR 2.02; 95% CI 1.57–2.61). Interpretation: Children/young people with CP require frequent outpatient services. Improving continuity of care, particularly for those residing in regional/remote areas, may decrease need for unplanned hospital care. What this paper adds: Many children with cerebral palsy use multiple and frequent outpatient services.Better continuity of care is associated with living in metropolitan and less socioeconomically disadvantaged areas.Outpatient service utilization reduces at the time of transition to adult services.High outpatient utilization is associated with unplanned hospital care.Decreased continuity of care is associated with unplanned hospital care. [ABSTRACT FROM AUTHOR]

Aim: To explore the perceptions of parents of children with cerebral palsy (CP) participating in a modified sports intervention, Sports Stars Brazil. Method: Parents of children with CP (n = 15, 6–12 years old, nine males, six females), classified in Gross Motor Function Classification System levels I and II, who participated in the Sports Stars Brazil intervention, were recruited for this descriptive qualitative study. Children received the 8‐week (1 hour per week) modified sports intervention. After the intervention, parents participated in a focus group. The F‐word (fitness, family, fun, functioning, friends, and future) interpretation of the International Classification of Functioning, Disability and Health was used to guide the discussion. Participants were invited to report their experience of participating in the intervention using the F‐words for a childhood disability model. Transcripts were coded using content analysis. Results: Parents reported positive experiences of the Sports Stars Brazil intervention, including a decrease in sedentarism (fitness), improvement of motor skills (functioning), greater connection between child and family (family), better socialization (friends), increased participation in pleasurable activities (fun), and awareness of the importance of physical activity (future). All positive experiences were interconnected. Interpretation: This study presents the ability of the Sports Stars Brazil intervention to integrate all aspects of functioning and supports its role as an engaging and promising intervention option for ambulant children with CP. What this paper adds: The parents of children participating in Sports Stars Brazil perceived benefits across all F‐words.This modified sports intervention integrated all F‐words into one concept. [ABSTRACT FROM AUTHOR]

Aim: To provide a description of cerebral palsy (CP) registers globally, identify which aim to report on CP epidemiology, and report similarities and differences across topics of importance for the sustainability and collaboration between registers. Method: Representatives of all known CP registers globally (n = 57) were invited to participate. The online survey included 68 questions across aims, methodologies, output/impact, and stakeholder involvement. Responses were analysed using descriptive statistics. Results: Forty‐five registers participated, including three register networks. Twenty were newly established or under development, including 12 in low‐ and middle‐income countries (LMICs). An epidemiological aim was reported by 91% of registers. Funding is received by 85% of registers, most often from not‐for‐profit organizations. CP definitions are comparable across registers. While the minimum data set of a register network is used by most registers, only 25% of identified items are collected by all three register networks. Ninety per cent of registers measure research activities/output, and 64% measure research impact. People with lived experience are involved in 62% of registers. Interpretation: There has been a recent surge in CP registers globally, particularly in LMICs, which will improve understanding of CP epidemiology. Ongoing efforts to address identified methodological differences are essential to validate comparison of results and support register collaboration. What this paper adds: Cerebral palsy (CP) registers represent an increasing number of regions, including low‐ and middle‐income, worldwide.Most registers collect the minimum data set of a CP register network.Research activities/output and impact are measured by most registers.The majority of registers involve people with lived experience in operation or research. [ABSTRACT FROM AUTHOR]

Aim: To explore the lived experiences of individuals with fetal alcohol spectrum disorder (FASD), their caregivers, and professionals working with individuals with FASD. Method: We conducted a scoping review using qualitative methods to explore what it is like to live with FASD from the perspective of those living with FASD, their caregivers, and stakeholders experienced in working with individuals with FASD. We searched electronic databases and grey literature for research published between 2005 and 2022. Results: The 47 studies included in this scoping review show that FASD affects people's lives on a daily basis. Individuals with FASD are aware of their disability and its effects. Caregivers face daily challenges raising children with FASD and are often left unsupported by professionals. Professionals are unprepared and frustrated when supporting someone with FASD. Interpretation: FASD affects individuals, caregivers, and families on a daily basis; and they require improved support and funding. What this paper adds: Professionals are overwhelmed and frustrated because of a lack of training and organizational support with regard to fetal alcohol spectrum disorder (FASD).Extends on previous reviews with the inclusion of grey literature and professionals.FASD is a highly stigmatized disorder that affects individuals with FASD, and their caregivers and families, on a daily basis.Research is needed exploring the experiences of adults living with FASD and their siblings.There is a lack of research involving indigenous populations and their experience with FASD. [ABSTRACT FROM AUTHOR]

Aim: This paper introduces the Surveillance of Cerebral Palsy in Europe (SCPE) classification of events contributing to postneonatally acquired cerebral palsy, presents its interrater reliability, and describes the cases identified in the SCPE database. Method: The development of the classification, based on literature review and expert discussions, resulted in six main categories and 19 subcategories. The first chronological event designated as the primary event was mainly reported. Interrater reliability was assessed through online exercise providing 24 clinical vignettes representing single/complex pathways. Percent agreement and Gwet's AC1 index of reliability were estimated. Primary events were described using data of 221 children born between 2008 and 2012. Results: Thirty‐nine professionals (21 registries) participated in the reliability exercise. Substantial overall agreement was reached (0.75), with some contrast between complex (0.48, moderate agreement) and single events involved (0.89, almost perfect). The distribution of primary events showed that 32.1% were infections (category A), 23.1% head injuries (B), 15.4% related to surgery or medical interventions (C), 13.1% cerebrovascular accidents (D), 9.1% hypoxic brain damaging events of other origins (E), and 7.2% miscellaneous (F). Interpretation: This classification allows all the events involved to be recorded while consistently reporting the primary event, and may be used in different settings. What this paper adds: A standardized classification enables the description of the events contributing to postneonatal cerebral palsy (CP).The first chronological event in complex pathway leading to CP is coded.Category choice and coding of the primary event identify preventable situations.The detailed 2‐level classification is easy to use in various settings.Substantial overall interrater reliability shows that main categories can be consistently differentiated. [ABSTRACT FROM AUTHOR]

Aim: We investigated survival rates after scoliosis correction in individuals with Duchenne muscular dystrophy (DMD) and evaluated factors that can affect them. Method: This was a retrospective cohort study from 2000 to 2022 with a minimum 2‐year postoperative follow‐up. We reviewed the hospital records/spinal radiographs and analysed data with XLSTAT. Kaplan–Meier and multivariate Cox regression survival analysis was performed. Results: Forty‐three patients had a mean age at surgery of 14 years 5 months. Mean postoperative follow‐up was 10 years 10 months. There was no operative or 30‐day postoperative mortality in this group. Twenty‐four patients died because of cardiorespiratory failure. Median survivorship was 14 years 2 months, with the longest observed survival being 22 years 6 months given the limitation of the length of postoperative follow‐up. The degree of preoperative coronal imbalance and pelvic obliquity, as well as intraoperative blood loss, were factors that significantly affected survival. The impact of preoperative sagittal imbalance and extension of the fusion to the sacrum/pelvis trended towards significance. In contrast, age at surgery, preoperative/postoperative scoliosis, thoracic kyphosis, lumbar lordosis, scoliosis and pelvic obliquity flexibility or correction indices, postoperative coronal/sagittal balance, need of preoperative non‐invasive ventilation, preoperative feeding disorders, development of surgical complications, and length of hospital and intensive care unit stay were exposure variables that did not affect postsurgical survival in patients with DMD. Interpretation: Survival of up to two decades or more was possible among young patients with DMD after scoliosis correction. This was affected by factors that related to disease and deformity severity, as well as surgical morbidity. What this paper adds: There was no operative or 30‐day postoperative mortality in this group of patients with Duchenne muscular dystrophy (DMD) undergoing scoliosis correction.Survival probabilities at 5‐year, 10‐year, 15‐year, and 20‐year intervals post‐surgery were 92%, 80%, 33%, and 12% respectively.Scoliosis surgery achieved good deformity correction and a balanced spine that was maintained at follow‐up.Respiratory failure, severe pneumonia, and left ventricular failure were the leading causes of death in the study participants.Preoperative global coronal imbalance, pelvic obliquity, and intraoperative blood loss significantly predicted survival.Factors that affected survival after scoliosis surgery were associated with perioperative morbidity and disease or deformity severity in the DMD group. What this paper adds: There was no operative or 30‐day postoperative mortality in this group of patients with Duchenne muscular dystrophy (DMD) undergoing scoliosis correction.Survival probabilities at 5‐year, 10‐year, 15‐year, and 20‐year intervals post‐surgery were 92%, 80%, 33%, and 12% respectively.Scoliosis surgery achieved good deformity correction and a balanced spine that was maintained at follow‐up.Respiratory failure, severe pneumonia, and left ventricular failure were the leading causes of death in the study participants.Preoperative global coronal imbalance, pelvic obliquity, and intraoperative blood loss significantly predicted survival.Factors that affected survival after scoliosis surgery were associated with perioperative morbidity and disease or deformity severity in the DMD group. This original article is commented on by Cho on page 142 of this issue. [ABSTRACT FROM AUTHOR]

Aim: To clarify whether the Whitney Comorbidity Index (WCI) is useful in Asian adults with cerebral palsy (CP) and whether aspiration pneumonia and pressure ulcers improved the prognostic significance of the WCI. Method: This cohort study evaluated individuals aged at least 18 years with CP in Japan. We used Cox proportional hazards regression to analyse 2‐year mortality rates. The predictive performance of the Charlson Comorbidity Index, Elixhauser Comorbidity Index, and WCI were compared as comorbidity assessment criteria. Aspiration pneumonia and pressure ulcers were added to the Cox models, and their impact on hazard ratios was determined. Results: Of the 2232 adults with CP, 72 died during the 2 years. The model with a previously reported weighted WCI with aspiration pneumonia and pressure ulcers produced the best fit. Additionally, the hazard risk of 2‐year mortality for an unweighted WCI score of at least 4 was 2.56; when CP‐specific comorbidities were added, it increased to 8.94. Interpretation: This study showed that the WCI can be used in Asian adults with CP. Furthermore, assessing patient age, aspiration pneumonia, and pressure ulcers in addition to the WCI increased the predictive value for mortality. Our findings indicate that the WCI can promote valid comparisons between international populations. What this paper adds: The Whitney Comorbidity Index (WCI) is useful among adults with cerebral palsy, irrespective of ethnic differences.Assessment of aspiration pneumonia and pressure ulcers increased the WCI predictive value.The WCI helps identify adults with cerebral palsy at risk of adverse outcomes. What this paper adds: The Whitney Comorbidity Index (WCI) is useful among adults with cerebral palsy, irrespective of ethnic differences.Assessment of aspiration pneumonia and pressure ulcers increased the WCI predictive value.The WCI helps identify adults with cerebral palsy at risk of adverse outcomes. This article's abstract has been translated into Japanese. Follow the links from the abstract to view the translations. [ABSTRACT FROM AUTHOR]

Aim: To better understand parents' beliefs about causation in cerebral palsy (CP) and the emotions related to those beliefs. Method: We surveyed 226 parents of children with CP aged 1 to 18 years, recruited from the Victorian Cerebral Palsy Register, to evaluate their beliefs about the causes of CP, including genetic causes, causes specific to their own child, and their attitudes and emotions in relation to these. Results: Although 92% of participants reported that understanding the causes of their child's CP was important, uncertainty about the cause was expressed by 13%. The most frequently endorsed causal factors, in general and in their own child respectively, were intrapartum hypoxia (81%, 36%) or brain damage (69%, 22%), brain damage during pregnancy (73%, 28%), and preterm birth (66%, 28%). Genetic causes were deemed relevant by 13% of participants and hospital or professional error by 16%. Parents shared related feelings of anger (59%), sadness (80%), guilt (61%), and confusion (53%); parental anger was more likely when their child's CP was attributed to intrapartum events. Interpretation: Substantial parental interest in understanding the causes of CP, together with uncertainty about the causes, parents' causal attributions, and significant emotional sequelae, highlight a strong need for provision of information and support for families of children recently diagnosed with CP. What this paper adds: Understanding the causes of their child's cerebral palsy (CP) was important to parents.Parents most often endorsed intrapartum factors as a cause of CP.Parents reported experiencing strong emotions about the causes of their child's CP. What this paper adds: Understanding the causes of their child's cerebral palsy (CP) was important to parents.Parents most often endorsed intrapartum factors as a cause of CP.Parents reported experiencing strong emotions about the causes of their child's CP. This original article is commented on by Vasyutin on page 148 of this issue. [ABSTRACT FROM AUTHOR]

Aim: To study long‐term sequelae in children with Guillain–Barré syndrome (GBS). Method: This was a prospective observational study with children from two French tertiary centres. Data were from clinical and several standardized scales or questionnaires. Results: Fifty‐one patients were included with a median follow‐up of 6 years 4 months (range 3–20 years) after the acute phase. The sequelae rate was 67% (95% confidence interval [CI] 53–78) and did not vary with time. Most children had minor sequelae (Guillain–Barré Syndrome Disability Score [GBSDS] = 1); only one was unable to run (GBSDS = 2). The most frequent complaints were paraesthesia (43%), pain (35%), and fatigue (31%). The neurological examination was abnormal in 18% of children, autonomy was compromised in 14%, and symptoms of depression occurred in 34%. The factors associated with late‐onset sequelae were correlated with severity during the initial phase (i.e. initial GBSDS >4, odds ratio 6.6, 95% CI 1.8–33; p = 0.009). The predictive factors of more severe late‐onset conditions were initial severity (p = 0.002) and sex (female patients; p = 0.01). Interpretation: Two‐thirds of children with GBS had late‐onset sequelae following an episode, often minor, but sometimes with continuing effects on their everyday lives. Particularly affected were those who had severe GBS during the acute phase and who lost the ability to walk. What this paper adds: Two‐thirds of children with Guillain–Barré syndrome (GBS) had persistent sequelae.Sequelae were often minor, but daily repercussions of them were sometimes serious.Sequelae were significantly associated with severe GBS during the acute phase. What this paper adds: Two‐thirds of children with Guillain–Barré syndrome (GBS) had persistent sequelae.Sequelae were often minor, but daily repercussions of them were sometimes serious.Sequelae were significantly associated with severe GBS during the acute phase. [ABSTRACT FROM AUTHOR]

Despite decades of international entreaties for improvement, education about and provision of healthcare for people with disabilities remains harmfully inferior to that of the non‐disabled population. Many obstacles confound efforts to ameliorate this inequity, perhaps the most pernicious of which is negative bias on the part of providers. Narrative medicine offers a means to address healthcare attitudes towards people with disabilities, in particular negative attitudes based on 'ableism'. Through absorbing, writing, and sharing of diverse perspectives, narrative medicine kindles imagination and empathy, promoting self‐reflection. This approach enriches the students' capacity to absorb what their patients are trying to say, and to appreciate, respect, and hopefully meet the healthcare needs of people with disability. What this paper adds: Narrative medicine is a pedagogical tool to help providers listen and reflect on patients with disabilities. What this paper adds: Narrative medicine is a pedagogical tool to help providers listen and reflect on patients with disabilities. [ABSTRACT FROM AUTHOR]

The aim of this review is to revisit the meaning of common concepts and frameworks promoted to capture subjective outcomes of patients, the content of their corresponding measurements, and the preferred sources of the information of interest. This is important because conceptualizations of 'health' and the subject evaluations thereof continue to evolve. Related but distinct concepts like quality of life (QoL), health‐related QoL (HRQoL), functional status, health status, and well‐being are often used indiscriminately to assess clinical impacts of interventions and to influence decisions about patient care and policymaking. The discussion addresses and illustrates the following issues: (1) the required features of effective and valid health‐related concepts; (2) understanding underlying factors that often create confusion about QoL and HRQoL; and (3) how these concepts provide insight into, and promote, health in the context of populations with neurodisability. The hope is to illustrate how a combination of a clear research question, a hypothesis, conceptualization of the required outcomes, and operational definitions of the domains and items of interest, including item mapping, can help to achieve robust methodology and valid findings beyond the required psychometric properties. What this paper adds: The language, content, and the source of perceived health and life issues are clarified.Using the same terms for different constructs, or different terms for the same constructs, creates confusion and hinders outcome research.The challenges of using patient‐reported outcomes in neurodisability are addressed. What this paper adds: The language, content, and the source of perceived health and life issues are clarified.Using the same terms for different constructs, or different terms for the same constructs, creates confusion and hinders outcome research.The challenges of using patient‐reported outcomes in neurodisability are addressed. [ABSTRACT FROM AUTHOR]

Aim: To investigate spoken language comprehension (SLC), single‐word comprehension (SWC), functional communication development, and their determinants, in children with cerebral palsy. Method: This was a prospective cohort study in the Netherlands spanning 2 years 6 months. The main outcomes were SLC and SWC, assessed by the Computer‐Based instrument for Low motor Language Testing (C‐BiLLT) and the Peabody Picture Vocabulary Test‐III‐NL (PPVT‐III‐NL) respectively; and functional communication, measured by a subscale of the Focus on the Outcomes of Communication Under Six‐34 (FOCUS‐34). Linear mixed models were used to determine developmental trajectories, which were compared with norm and reference data. Potential determinants, for example intellectual functions, speech production, functional communication level (classified with the Communication Function Classification System, CFCS), and functional mobility, were added to assess their effects. Results: Children with cerebral palsy (n = 188; mean age 59 months, range 17–110) were monitored for 2 years 6 months. Developmental trajectories for SLC (C‐BiLLT) and SWC (PPVT‐III‐NL) were nonlinear; those for functional communication (FOCUS‐34) were linear. Compared with norm and reference groups, significantly delayed SLC, SWC, and functional communication development were found. Determinants for SLC and SWC were intellectual functions and functional communication level (CFCS); and for functional communication development (FOCUS‐34), speech production and arm–hand functioning. Interpretation: Children with cerebral palsy showed delayed SLC, SWC, and functional communication development compared with norm and reference groups. Remarkably, functional mobility was not associated with the development of SLC, SWC, or functional communication. What this paper adds: Children with cerebral palsy have delayed spoken language comprehension (SLC), single‐word comprehension (SWC), and functional communication development, compared to norm and reference data.Determinants for SLC and SWC development are intellectual functions and functional communication level.Determinants for functional communication development are speech production and arm–hand functioning.Functional mobility is not associated with SLC, SWC, or functional communication. What this paper adds: Children with cerebral palsy have delayed spoken language comprehension (SLC), single‐word comprehension (SWC), and functional communication development, compared to norm and reference data.Determinants for SLC and SWC development are intellectual functions and functional communication level.Determinants for functional communication development are speech production and arm–hand functioning.Functional mobility is not associated with SLC, SWC, or functional communication. Video Podcast: https://youtu.be/x09Tac7tsz0 [ABSTRACT FROM AUTHOR]

Aim: To explore the ingredients of early interventions provided to young children with cerebral palsy (CP) who are classified in Gross Motor Function Classification System (GMFCS) levels IV and V, and to identify the 'F‐words' addressed by the interventions. Method: Searches were completed in four electronic databases. Inclusion criteria were the original experimental studies that fitted the following PCC components: population, young children (aged 0–5 years, at least 30% of the sample) with CP and significant motor impairment (GMFCS levels IV or V, at least 30% of the sample); concept, non‐surgical and non‐pharmacological early intervention services measuring outcomes from any of the International Classification of Functioning, Disability and Health domains; and context, studies published from 2001 to 2021, from all settings and not limited to any specific geographical location. Results: Eighty‐seven papers were included for review, with qualitative (n = 3), mixed‐methods (n = 4), quantitative descriptive (n = 22), quantitative non‐randomized (n = 39), and quantitative randomized (n = 19) designs. Fitness (n = 59), family (n = 46), and functioning (n = 33) ingredients were addressed by most experimental studies, whereas studies on fun (n = 6), friends (n = 5), and future (n = 14) were scarce. Several other factors (n = 55) related to the environment, for example, service provision, professional training, therapy dose, and environmental modifications, were also relevant. Interpretation: Many studies positively supported formal parent training and use of assistive technology to promote several F‐words. A menu of intervention ingredients was provided, with suggestions for future research, to incorporate them into a real context within the family and clinical practice. What this paper adds: Family‐centred care (including coaching and caregiver‐delivered interventions) and formal parental training are effective strategies for children in GMFCS levels IV and V.Assistive technology ingredients (power, mobility, supported, sitting, stepping, and standing) may promote several 'F‐words' (functioning, fitness, family, fun, friends, and future).The lowest level of evidence was found for fun, friends, and future.Other factors (service provision, professional training, therapy dose, environmental modifications) are relevant for young children in GMFCS levels IV and V. [ABSTRACT FROM AUTHOR]

Motor features of autism have long been acknowledged by clinicians, researchers, and community stakeholders. Current DSM‐5 and ICD‐11 guidelines allow clinicians to assign a co‐occurring diagnosis of developmental [motor] coordination disorder (DCD) for autistic individuals with significant motor problems. DCD is characterized by poor motor proficiency with an onset of symptoms in early development. Studies have shown considerable overlap in the behavioral motor features observed in autism and DCD. However, others indicate that motor problems in autism and DCD may stem from different underlying sensorimotor mechanisms. Regardless of whether autism has a unique motor phenotype or an overlap with DCD, changes need to be made in the clinical pipeline to address motor problems in autism at the stages of recognition, assessment, diagnosis, and intervention. Consensus is needed to address unmet needs in research on the etiology of motor problems in autism and their overlap with DCD, to optimize clinical practice guidelines. The development of screening and assessment tools for motor problems that are valid and reliable for use with autistic individuals is essential, and an evidence‐based clinical pipeline for motor problems in autism is urgently needed. What this paper adds: Motor problems in autism are highly prevalent, yet underdiagnosed and poorly managed.An evidence‐based clinical pipeline for motor problems in autism is urgently needed. What this paper adds: Motor problems in autism are highly prevalent, yet underdiagnosed and poorly managed.An evidence‐based clinical pipeline for motor problems in autism is urgently needed. [ABSTRACT FROM AUTHOR]

Social determinants of health (SDH) influence health and social outcomes in positive and negative ways. Understanding the impact of SDH on children with cerebral palsy (CP) is essential to improve health equity, optimize health outcomes, and support children with CP and their families to thrive in society. In this narrative review, we summarize the landscape of SDH impacting children with CP and their families worldwide. In high‐income countries, children from poorer neighbourhoods are more likely to have severe comorbidities, present with spastic bilateral CP, and report lower frequency of participation in community activities. In low‐ and middle‐income countries, socioeconomic disadvantage is associated with increased risk of malnutrition, living in poorer housing conditions, not having access to proper sanitation, and living below the poverty line. Low maternal education is associated with increased likelihood of the child with CP experiencing increased severity of gross motor and bimanual functioning challenges, and poorer academic performance. Lower parental education is also associated with reduced child autonomy. On the other hand, higher parental income is a protective factor, associated with greater diversity of participation in day‐to‐day activities. A better physical environment and better social support are associated with higher participation in daily activities. Clinicians, researchers, and the community should be aware of these key challenges and opportunities. Then adopt a range of approaches that target adverse SDH/social needs and foster positive SDH in the clinical setting. What this paper adds: Understanding how social determinants of health influence health and social outcomes is a critical step towards improving health equity and outcomes.Approaches are required that target the impact of adverse SDH on access to health care for children with cerebral palsy and that work towards ensuring equity of access for all. What this paper adds: Understanding how social determinants of health influence health and social outcomes is a critical step towards improving health equity and outcomes.Approaches are required that target the impact of adverse SDH on access to health care for children with cerebral palsy and that work towards ensuring equity of access for all. [ABSTRACT FROM AUTHOR]

Aim: This study describes the process of updating the cerebral palsy (CP) common data elements (CDEs), specifically identifying tools that capture the impact of chronic pain on children's functioning. Method: Through a partnership between the American Academy for Cerebral Palsy and Developmental Medicine and the National Institute of Neurological Disorders and Stroke (NINDS), the CP CDEs were developed as data standards for clinical research in neuroscience. Chronic pain was underrepresented in the NINDS CP CDEs version 1.0. A multi‐step methodology was applied by an interdisciplinary professional team. Following an adapted CP chronic pain tools' rating system, and a review of psychometric properties, clinical utility, and compliance with inclusion/exclusion criteria, a set of recommended pain tools was posted online for external public comment in May 2022. Results: Fifteen chronic pain tools met inclusion criteria, representing constructs across all components of the International Classification of Functioning, Disability and Health. Interpretation: This paper describes the first condition‐specific pain CDEs for a pediatric population. The proposed set of chronic pain tools complement and enhance the applicability of the existing pediatric CP CDEs. The novel CP CDE pain tools harmonize the assessment of chronic pain, addressing not only intensity of chronic pain, but also the functional impact of experiencing it in everyday activities. [ABSTRACT FROM AUTHOR]

The shaken baby syndrome was originally proposed in the 1970s without any formal scientific basis. Once data generated by scientific research was available, the hypothesis became controversial. There developed essentially two sides in the debate. One side claimed that the clinical triad of subdural haemorrhage, retinal haemorrhage, and encephalopathy, or its components, is evidence that an infant has been shaken. The other side stated this is not a scientifically valid proposal and that alternative causes, such as low falls and natural diseases, should be considered. The controversy continues, but the contours have shifted. During the last 15 years, research has shown that the triad is not sufficient to infer shaking or abuse and the shaking hypothesis does not meet the standards of evidence‐based medicine. This raises the issue of whether it is fit for either clinical practice or for the courtroom; evidence presented to the courts must be unassailable. What this paper adds: There is insufficient scientific evidence to assume that an infant with the triad of subdural haemorrhage (SDH), retinal haemorrhage, and encephalopathy must have been shaken.Biomechanical and animal studies have failed to support the hypothesis that shaking can cause SDH and retinal haemorrhage.Patterns of retinal haemorrhage cannot distinguish abuse.Retinal haemorrhages are commonly associated with extracerebral fluid collections (including SDH) but not with shaking.Infants can develop SDH, retinal haemorrhage, and encephalopathy from natural diseases and falls as low as 1 foot.The shaking hypothesis and the literature on which it depends do not meet the standards of evidence‐based medicine. [ABSTRACT FROM AUTHOR]

Aim: To assess how atypical language organization after early left‐hemispheric brain lesions affects grey matter in the contralesional hemisphere. Method: This was a cross‐sectional study with between‐group comparisons of 14 patients (six female, 8–26 years) with perinatal left‐hemispheric brain lesions (two arterial ischemic strokes, 11 periventricular haemorrhagic infarctions, one without classification) and 14 typically developing age‐matched controls (TDC) with functional magnetic resonance imaging (fMRI) documented left‐hemispheric language organization (six female, 8–28 years). MRI data were analysed with SPM12, CAT12, and custom scripts. Language lateralization indices were determined by fMRI within a prefrontal mask and right‐hemispheric grey matter group differences by voxel‐based morphometry (VBM). Results: FMRI revealed left‐dominance in seven patients with typical language organization (TYP) and right‐dominance in seven patients with atypical language organization (ATYP) of 14 patients. VBM analysis of all patients versus controls showed grey matter reductions in the middle temporal gyrus of patients. A comparison between the two patient subgroups revealed an increase of grey matter in the middle frontal gyrus in the ATYP group. Voxel‐based regression analysis confirmed that grey matter increases in the middle frontal gyrus were correlated with atypical language organization. Interpretation: Compatible with a non‐specific lesion effect, we found areas of grey matter reduction in patients as compared to TDC. The grey matter increase in the middle frontal gyrus seems to reflect a specific compensatory effect in patients with atypical language organization. What this paper adds: Perinatal stroke leads to decreased grey matter in the contralesional hemisphere.Atypical language organization is associated with grey matter increases in contralesional language areas. [ABSTRACT FROM AUTHOR]

Aim: To estimate the probability of independent walking and wheeled mobility in individuals with cerebral palsy (CP) at home and in the community in relation to age and gross motor function. Method: This was a longitudinal cohort study using data reported into the combined Swedish CP follow‐up programme and national quality registry from October 2000 to October 2022. Walking, walking with aids, wheeled mobility, and assisted mobility defined independent or assisted mobility at home and in the community, based on the Functional Mobility Scale with additional data on wheelchair performance, were assessed. Results: There were 52 858 examinations reported for 6647 individuals with CP (age range 0–32 years, follow‐up period 0–22 years). Most children and adults in Gross Motor Function Classification System (GMFCS) levels I or II walked without assistive devices. The probability of dependence on others for mobility in the community was high for both children and adults in GMFCS levels III to V. Interpretation: Although independent mobility is vital for participation and social inclusion, many children and adults with CP are dependent on others for mobility. We recommend clinicians, together with families and individuals with CP, explore how to increase access to independent mobility from an early age and continuously throughout the life course. What this paper adds: •There is a high probability of independent walking in Gross Motor Function Classification System (GMFCS) levels I to II.•Mobility options vary most at home and in the community in GMFCS level III.•Being dependent on others for mobility is likely in GMFCS levels III to V. [ABSTRACT FROM AUTHOR]

Aim: This study aimed to (1) quantify attention and executive functioning in children with developmental coordination disorder (DCD), (2) assess whether some children with DCD are more likely to show attention difficulties, and (3) characterize brain correlates of motor and attention deficits. Method: Fifty‐three children (36 with DCD and 17 without) aged 8 to 10 years underwent T1‐weighted and diffusion‐weighted magnetic resonance imaging, and standardized attention and motor assessments. Parents completed questionnaires of executive functioning and symptoms of inattention and hyperactivity. We assessed regional cortical thickness and surface area, and cerebellar, callosal, and primary motor tract structure. Results: Analyses of covariance and one‐sample t‐tests identified impaired attention, non‐motor processing speed, and executive functioning in children with DCD, yet partial Spearman's rank correlation coefficients revealed these were unrelated to one another or the type or severity of the motor deficit. Robust regression analyses revealed that cortical morphology in the posterior cingulate was associated with both gross motor skills and inattentive symptoms in children with DCD, while gross motor skills were also associated with left corticospinal tract (CST) morphology. Interpretation: Children with DCD may benefit from routine attention and hyperactivity assessments. Alterations in the posterior cingulate and CST may be linked to impaired forward modelling during movements in children with DCD. Overall, alterations in these regions may explain the high rate of non‐motor impairments in children with DCD. What this paper adds: Children with developmental coordination disorder have difficulties in attention, processing speed, and executive functioning.Non‐motor impairments were not interrelated or correlated with the type or severity of motor deficit.Posterior cingulate morphology was associated with gross motor skills and inattention.Gross motor skills were also associated with left corticospinal tract morphology. [ABSTRACT FROM AUTHOR]

Aim: To characterize motor disorders in children and young people with cerebral palsy (CP). Method: This was a cross‐sectional study of 582 children and young people with CP (mean age 9 years 7 months; range 11 months–19 years 9 months; standard deviation 4 years 11 months; 340 males) attending a rehabilitation clinic at a specialized children's hospital (May 2018–March 2020). Data on motor disorders, topography, functional classifications, and non‐motor features, such as epilepsy, intellectual disability, and sensory impairments, were collected using the Australian Cerebral Palsy Register CP Description Form. Results: Fifty‐five per cent (n = 321) of children and young people with CP presented with multiple motor disorders, often affecting the same limb(s). The most common motor disorders were spasticity and dystonia (50%), spasticity only (36%), and dystonia only (6%), but 18 different combinations were identified, including choreoathetosis, ataxia, and generalized hypotonia with increased reflexes. Children with spasticity only had less severe functional deficits (p < 0.001) and lower rates of associated intellectual disability (p < 0.01) and epilepsy (p < 0.001) than those with both spasticity and dystonia. Interpretation: Multiple motor disorders in children and young people with CP are common and associated with more severe functional impairment. Accurate assessment of motor disorders is essential to guide prognosis and ensure personalized evidence‐based interventions. What this paper adds: More than half of children and young people with cerebral palsy presented with multiple motor disorders.Dystonia was identified in 60% of study participants.Dystonia was associated with more severe functional impairments and rates of non‐motor features. [ABSTRACT FROM AUTHOR]

Aim: To assess perspectives on clinical communication about sexual health in young adult males with spina bifida. Method: Semi‐structured interviews were conducted between February and May 2021 with males at least 18 years of age with spina bifida to assess their perspectives and experiences of sexual health communication with clinicians. Demographic and clinical characteristics were obtained from chart review and a survey of patients. Interviews were transcribed verbatim, and conventional content analysis was used for transcript coding. Results: Twenty individuals participated with a median age of 22 years 6 months (range 18–29 years). Sixteen had myelomeningocele. Most identified as heterosexual (n = 17) and not sexually active (n = 13). Barriers and facilitators of successful interactions were identified. Barriers for participants included general discomfort with talking about sex and variability in individual preferences for how conversations occur. Facilitators included participants' comfort with their urologist and discussing sex in relation to disability. Suggestions for improving discussions included (1) notifying individuals that discussion about sex will occur before clinic visits; (2) creating space for discussions; (3) respecting individuals' readiness to discuss; and (4) making discussions disability specific. Interpretation: Young adult males with spina bifida are interested in discussing sexual health with their clinicians. Great variability exists about conversation preferences, emphasizing the need to individualize clinical communication about sex. Current health guidelines for males may not be in line with individuals' wishes. What this paper adds: Great variability exists in individual preference around sexual health communication.Patient‐level barriers hinder successful conversations about sex.Individuals have great insight into how conversations about sex can be improved. [ABSTRACT FROM AUTHOR]

Aim: To compare the 66‐item Gross Motor Function Measure (GMFM‐66) with the reduced version of the GMFM‐66 (rGMFM‐66) with respect to the detection of clinically relevant changes in gross motor function in children with cerebral palsy (CP). Method: The study was a retrospective single centre analysis of children with CP who participated in a rehabilitation programme. Overall, 1352 pairs of GMFM‐66 and rGMFM66 measurements with a time interval of 5 to 7 months were available. To measure clinically relevant changes in gross motor function, the individual effect size (iES) was calculated. Results: The study population consisted of 1352 children (539 females), mean age 6 years 4 months (SD 2 years 4 months). The iES based on the GMFM‐66 and the rGMFM‐66 showed a significant correlation (r = 0.84, p < 0.001). The analysis of the area under the receiver operating characteristic curve showed an excellent agreement for clinically relevant gross motor improvement (Cohen's d ≥ 0.5; area under the curve = 0.90 [95% confidence interval 0.88–0.92]) or deterioration (Cohen's d ≤ −0.5; area under the curve = 0.95 [95% confidence interval 0.92–0.97]). Interpretation: Performing the rGMFM‐66 saves time compared to the full GMFM‐66. The rGMFM‐66 showed good agreement with the GMFM‐66 with respect to the detection of clinically relevant changes in gross motor function in children with CP, so its use in everyday clinical practice seems justifiable. What this paper adds: The reduced version of the 66‐item Gross Motor Function Measure (rGMFM‐66) detects clinically relevant changes in gross motor function in children with cerebral palsy.The rGMFM‐66 correlates highly with the full GMFM‐66.The rGMFM‐66 can be used in clinical practice when the time schedule is limited. [ABSTRACT FROM AUTHOR]

Aim: To compare the effect of iMOVE (Intensive Mobility training with Variability and Error) therapy with dose‐matched conventional therapy on gross motor development and secondary outcomes in young children with cerebral palsy. Method: This single‐blind, randomized controlled trial included repeated assessments of gross motor function (using the Gross Motor Function Measure) and secondary outcomes during a 12‐ to 24‐week intervention phase and at three follow‐up points after treatment. Treatment was delivered three times per week in both groups. Forty‐two children aged 12 to 36 months were stratified by age and motor function to ensure equivalence between groups at baseline. Results: Thirty‐six children completed treatment and follow‐up phases. Treatment fidelity was high and adherence was equivalent between groups (77.3% conventional therapy, 76.2% iMOVE). There were no group differences on the primary (gross motor function after 12 weeks p = 0.18; after 24 weeks p = 0.94) or any secondary (postural control p = 0.88, caregiver satisfaction p = 0.52, child engagement p = 0.98) measure after treatment or at the follow‐up points. However, one‐third of total participants exceeded predicted change after 12 weeks and 77% exceeded predicted change after 24 weeks of treatment. Interpretation: Our observations indicate a potential dose–response effect of rehabilitation therapy. We further demonstrated that individual therapeutic ingredients can be manipulated. When delivered consistently, both iMOVE and conventional therapy interventions might both be more effective than standard care. What this paper adds: Those receiving iMOVE therapy demonstrated more independent practice time, error, and child‐initiation than those receiving the dose‐matched control.iMOVE therapy was not superior to the control (conventional physical) therapy.Most participants exceeded predicted change after 24 weeks of treatment. [ABSTRACT FROM AUTHOR]