Search

The CP phenotype for each cohort was scored on the following criteria as either high confidence (fulfilled >=5 criteria), moderate (3-4), or low confidence (<=2): (i) CP defined with well acknowledged definition; (ii) clinical diagnosis of CP confirmed at time of recruitment; (iii) inclusion/exclusion criteria of the study met the clinical description of CP; (iv) CP clinically confirmed at >= age four; (v) motor disorders of CP well described; (vi) individual phenotyping data available. B Results b : Among the sample of children, 30 did not have CP and 45 had spastic CP (GMFCS levels I-V; 29 bilateral CP; 16 unilateral CP: 8 right, 8 left). B Background and Objective(s) b : The compounding effects of adverse health/function factors as individuals with cerebral palsy (CP) age into and through adulthood can create multiple pathways to complex medical profiles, but the patterns of comorbidities among adults with CP remains understudied. B Background and Objective(s) b : Cerebral palsy (CP) results in impairment of muscle function including muscles of the face, mouth, and throat, which may cause oropharyngeal dysphagia (OPD) and affects 85% of children with CP. B Background and Objective(s) b : The QEDIN-CP network provides training and support for early screening to fast-track infants to early intervention according to the cerebral palsy (CP) early detection guideline. [Extracted from the article]

The article presents abstracts on medical topics which includes researches related to torsional deformities in ambulatory children with cerebral palsy, avascular necrosis following reconstructive hip surgery in children and foot posture in children with cerebral palsy.

Aim: (1) To systematically review the literature on developmental outcomes from infancy to adolescence of children with complex congenital heart disease (CHD) who underwent early surgery; (2) to run a meta-regression analysis on the Bayley Scales of Infant Development, Second Edition Mental Developmental Index and Psychomotor Developmental Index (PDI) of infants up to 24 months and IQs of preschool-aged children to adolescents; (3) to assess associations between perioperative risk factors and outcomes.Method: We searched pertinent literature (January 1990 to January 2019) in PubMed, Embase, CINAHL, and PsycINFO. Selection criteria included infants with complex CHD who had primary surgery within the first 9 weeks of life. Methodological quality, including risk of bias and internal validity, were assessed.Results: In total, 185 papers met the inclusion criteria; the 100 with high to moderate methodological quality were analysed in detail. Substantial heterogeneity in the group with CHD and in methodology existed. The outcome of infants with single-ventricle CHD was inferior to those with two-ventricle CHD (respectively: average scores for PDI 77 and 88; intelligence scores 92 and 98). Perioperative risk factors were inconsistently associated with developmental outcomes.Interpretation: The literature on children undergoing surgery in early infancy suggests that infants with a single ventricle are at highest risk of adverse developmental outcomes. [ABSTRACT FROM AUTHOR]

An abstract of the article "Baseline findings from a pilot study to evaluate the use of postural support, constructed using Appropriate paper-based technology (APT), to be used with children with Cerebral Palsy in Kenya" by Rachel Lindoewood and others is presented.

Aim: To identify the research on childhood disability service adaptations and their impact on children and young people with long‐term disability during the COVID‐19 pandemic. Method: A mapping review was undertaken. We searched the World Health Organization Global COVID‐19 database using the search terms 'children', 'chronic/disabling conditions', and 'services/therapies'. Eligible papers reported service changes for children (0–19 years) with long‐term disability in any geographical or clinical setting between 1st January 2020 and 26th January 2022. Papers were charted across the effective practice and organization of care taxonomy of health system interventions and were narratively synthesized; an interactive map was produced. Results: Reduction of face‐to‐face care and usual provision had a huge impact on children and families. Adoption of telehealth provided continuity for the care and management of some conditions. There was limited evidence of changes to mental health services, transitions of care, social care, or child‐reported satisfaction or acceptability of service changes. Interpretation: The long‐term impacts of service change during the pandemic need full evaluation. However, widespread disruption seems to have had a profound impact on child and carer health and well‐being. Service recovery needs to be specific to the individual needs of children with a disability and their families. This should be done through coproduction to ensure that service changes meet needs and are accessible and equitable. [ABSTRACT FROM AUTHOR]

Aim: Taping is popular in a variety of paediatric clinical settings. The purpose of this study was to investigate the effectiveness of taping on the rehabilitation of children with cerebral palsy (CP).Method: We used MEDLINE, Embase, PubMed, CINAHL, and the Cochrane Central Register of Controlled Trials as the electronic databases for the review. We reviewed all relevant studies published up to May 2015. We also analysed pertinent secondary references. We used Sackett's Levels of Evidence and the guidelines for critical review of McMaster University to criticize the reviewed articles.Results: Nine papers met the inclusion criteria. Five of these were randomized controlled trials, three were case series, and one was a single case study. Four papers were high quality according to the methodological critical forms of this review, and two of these found that taping was effective in increasing activity in children with CP.Interpretation: Although benefits of taping were found in the population, and functional gains according to the International Classification of Functioning, Disability and Health were obtained, the evidence was not conclusive. Randomized controlled trials with larger sample sizes and with more specific taping procedures are required to strengthen the evidence for the effectiveness of taping on children with CP. [ABSTRACT FROM AUTHOR]

The article presents abstracts of articles on topics including upper limb orthoses for children with cerebral palsy, motor development in infants with asymmetric brain injury, and risk of cerebral palsy in infants.

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 synthesize the evidence about the main intervention characteristics of cognitive behavioral therapies (CBTs) for individuals with cerebral palsy and identify barriers and facilitators to their success, focusing on aspects of feasibility and markers of success. Method: A scoping review methodology informed a literature search for papers published between 1991 and 2021. Articles were screened, reviewed, and categorized using the DistillerSR systematic review software, and critically appraised for quantitative and/or qualitative criteria. Results: Out of 1265 publications identified, 14 met the inclusion criteria. Elements associated with the specific study participant characteristics (46% female; aged 6–65 years), type of CBT techniques used (third‐wave [n = 6], cognitive [n = 3], cognitive and behavioral [n = 2], biofeedback training [n = 2]), and features of the study context and methodological quality (two randomized clinical trials and small sample sizes [n ≤ 12]), were identified. Most studies had psychological targets of intervention (n = 10) and secondary physiological (n = 3) or social (n = 2) objectives. Feasibility indicators were described in nearly one‐third of the papers. Interpretation: This study highlights the high flexibility within CBT interventions, enabling their adaptation for individuals with cerebral palsy. However, relatively little, and only low‐certainty evidence was identified. More high‐quality research in terms of specific CBT techniques, optimal treatment doses, and detailed population characteristics are needed. [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]

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: To compare the efficacy of 0, 5, and 20 sessions of transcranial direct current stimulation (tDCS) for reducing symptoms of autism spectrum disorder (ASD). Method: Thirty‐six male children with ASD (mean age 2 years 3 months, SD 4 months, age range 1 years 6 months–2 years 11 months) were balanced and stratified by age, sex, and baseline severity of ASD, to: (1) a control group that received 20 sessions of sham tDCS; (2) a 5‐session tDCS group (5‐tDCS) that received 5 sessions of active tDCS followed by 15 sessions of sham tDCS; and (3) a 20‐session tDCS group (20‐tDCS) that received 20 sessions of active tDCS. All groups participated in the special school activity of Khon Kaen Special Education Center, Thailand. The primary outcome was autism severity as measured by the Childhood Autism Severity Scale. Results: The 5‐tDCS and 20‐tDCS groups evidenced greater reductions in autism severity than the control group at days 5 and 14, and months 6 and 12. There were no significant differences in the outcome between the 5‐ and 20‐tDCS groups at any time point. Within‐group analysis showed clinically meaningful improvements starting at month 6 for the participants in the control group, and clinically meaningful improvements starting on day 5 in both active tDCS groups, all of which were maintained to month 12. Interpretation: The 5‐ and 20‐session tDCS seems to reduce autism severity faster than sham tDCS. These effects maintained at least for 1 year. What this paper adds: Twenty sessions of transcranial direct current stimulation (tDCS) were not superior to five sessions.Sham tDCS with a special school activity can reduce autism severity starting at 6 months after treatment.The benefits observed for 5 and 20 sessions of tDCS last for at least 12 months. What this paper adds: Twenty sessions of transcranial direct current stimulation (tDCS) were not superior to five sessions.Sham tDCS with a special school activity can reduce autism severity starting at 6 months after treatment.The benefits observed for 5 and 20 sessions of tDCS last for at least 12 months. This original article is commented on by Wilson on pages 730–731 of this issue. [ABSTRACT FROM AUTHOR]

Aim: To estimate the global prevalence of intellectual developmental disorder (IDD) and the IDD prevalence–genotype association in Becker muscular dystrophy (BMD) or Duchenne muscular dystrophy (DMD) according to the affected isoforms of the DMD gene: Dp427, Dp140, Dp71. Method: Systematic searches in MEDLINE, Scopus, Web of Science, and the Cochrane Library were conducted from inception of each database to March 2022. Observational studies that determined the prevalence of IDD in the population with BMD or DMD were included. Meta‐analyses of IDD prevalence and prevalence ratios of the IDD–genotype association were conducted. Results: Forty‐nine studies were included. The prevalence of IDD in BMD was 8.0% (95% confidence interval 5.0–11.0), and in DMD it was 22.0% (18.0–27.0). Meta‐analyses of IDD–genotype association showed a deleterious association between IDD and the number of isoforms affected in DMD, with a prevalence ratio = 0.43 (0.28–0.64) and 0.17 (0.09–0.34) for Dp140+/Dp71+ versus Dp140−/Dp71+ and Dp140+/Dp71+ versus Dp140−/Dp71− comparisons respectively. However, in BMD, there was no association for Dp140+/Dp71+ versus Dp140−/Dp71+. Interpretation: There is a high prevalence of IDD in BMD and DMD. Moreover, the number of isoforms affected is strongly and negatively associated with the prevalence of IDD in DMD. What this paper adds: The global prevalence of intellectual developmental disorder (IDD) was 8% in Becker muscular dystrophy and 22% in Duchenne muscular dystrophy (DMD).The global prevalence of IDD in DMD was 12%, 29%, and 84% in participants with Dp427−/Dp140+/Dp71+, Dp427−/Dp140−/Dp71+, and Dp427−/Dp140−/Dp71− genotypes respectively.In DMD, 12% and 22% of participants had abnormal performance IQ and verbal IQ values respectively. What this paper adds: The global prevalence of intellectual developmental disorder (IDD) was 8% in Becker muscular dystrophy and 22% in Duchenne muscular dystrophy (DMD).The global prevalence of IDD in DMD was 12%, 29%, and 84% in participants with Dp427−/Dp140+/Dp71+, Dp427−/Dp140−/Dp71+, and Dp427−/Dp140−/Dp71− genotypes respectively.In DMD, 12% and 22% of participants had abnormal performance IQ and verbal IQ values respectively. [ABSTRACT FROM AUTHOR]

Aim: To test the efficacy of an integrated microfinance/livelihood and community‐based rehabilitation (IMCBR) programme in improving health‐related quality of life (HRQoL) and motor function of children with cerebral palsy (CP) and gain in social capital to their ultra‐poor families in rural Bangladesh. Method: This was an open‐label cluster randomized control trial. Children with CP aged 5 years or under were randomly allocated to three arms; Arm A: IMCBR; Arm B: community‐based rehabilitation (CBR); and Arm C: care‐as‐usual. The CBR was modified with phone follow‐up followed by home‐based CBR at 2.5 months post‐enrolment because of the COVID‐19 pandemic. Intention‐to‐treat analysis was performed. Results: Twenty‐four clusters constituting 251 children–primary caregivers' dyads were assigned to three arms (Arm A = 80; Arm B = 82; Arm C = 89). Between baseline and endline, the percentage mean change in the physical functioning domain of HRQoL was highest in Arm A (30.0%) with a significant mean difference between Arm A and Arm B (p = 0.015). Improvement in the mean social capital score was significantly higher in Arm A compared to Arm C (p < 0.001). Interpretation: The findings suggest that IMCBR could improve the HRQoL of children with CP and the social capital of their ultra‐poor families. Long‐term follow‐up of the trial participants and future exploration of such interventions are essential. The integrated livelihood and CBR programme holds potential to improve health and well‐being of children with CP and their ultra‐poor families. What this paper adds: Half of the families who received livelihoods were impacted by a cold‐wave, suggesting the need for a more disaster‐resilient livelihood asset.The integration of livelihood with community‐based rehabilitation programme helps to improve health‐related quality of life of children with cerebral palsy and the social capital of their ultra‐poor families. What this paper adds: Half of the families who received livelihoods were impacted by a cold‐wave, suggesting the need for a more disaster‐resilient livelihood asset.The integration of livelihood with community‐based rehabilitation programme helps to improve health‐related quality of life of children with cerebral palsy and the social capital of their ultra‐poor families. This original article is commented on by Fianto on pages 727–728 of this issue. [ABSTRACT FROM AUTHOR]

Aim: To summarize the literature on the prevalence of pediatric hearing loss in low‐ and middle‐income countries (LMICs). Method: A systematic review initially identified 2833 studies, of which 122 met the criteria for inclusion. Eighty‐six of those studies included diagnoses and were included in a meta‐analysis. Results: The meta‐analysis indicated a 1% (95% confidence interval = 0.8–2.0) prevalence of childhood hearing loss across LMICs. There was significant heterogeneity between studies and evidence of publication bias. The prevalence of mild and moderate cases of hearing loss was higher than more severe cases and there were fewer cases of mixed hearing loss compared to conductive or sensorineural hearing loss. No differences were identified between the prevalence of unilateral versus bilateral hearing loss or hearing loss according to sex. The quality of the studies, age of participants, and location of data collection may have influenced the results. High variability in the reporting of etiology made the causes of hearing loss unclear. Interpretation: The literature indicates that 1% of children in LMICs have hearing losses. However, most studies missed children with acquired hearing loss, which may lead to under‐reporting of global prevalence. This systematic review is an initial step toward developing and implementing population‐appropriate treatment and prevention programs for childhood hearing loss in LMICs. What this paper adds: The prevalence of childhood hearing loss in low‐ and middle‐income countries is 1%.Reporting of hearing loss etiology was highly variable. [ABSTRACT FROM AUTHOR]

Aim: To describe the development of social function in children with cerebral palsy (CP) classified in all levels of the Communication Function Classification System (CFCS). Method: This prospective, longitudinal population‐based cohort study recruited children with CP born in Queensland, Australia. Social functioning was measured using the Pediatric Evaluation of Disability Inventory (PEDI) social function domain at 2 years, 2 years 6 months, 3 years, 4 years, and 5 years, and the PEDI Computer Adaptive Test (PEDI‐CAT) social/cognitive domain at 8 to 12 years. Results: Seventy‐four children provided 356 observations. PEDI‐CAT social/cognitive scaled scores at 8 to 12 years were (mean [SD] n) CFCS level I, 68.6 (2.7) 45; CFCS level II, 64.0 (3.4) 10; CFCS level III, 63.5 (3.7) 4; CFCS level IV, 56.8 (5.0) 9; CFCS level V, 47.2 (5.8) 6. Scores within expected range for age (not less than 2 SD below mean) at 8 to 12 years were achieved by 35 (78%) children in CFCS level I and four (14%) in CFCS levels II to V. Forty‐nine per cent of children scored at least two standard deviations below the population mean on a proxy measure of fluid intelligence. Intellectual impairment was associated with lower PEDI‐CAT social/cognitive scaled scores in univariable analysis (β = −8.3, 95% confidence interval − 10.91 to −5.63; p < 0.001) but had a smaller effect when modelled together with CFCS. Interpretation: Social function attained by 8 to 12 years of age was strongly related to level of communication function (CFCS). The small number of children classified in CFCS levels II to V necessitates caution when viewing these individual CFCS level trajectories. What this paper adds: There is a strong relation between social functioning and Communication Function Classification System (CFCS) levels.At 8 to 12 years, 35 out of 45 children in CFCS level I met social functioning age expectations.Twenty‐five out of 29 in CFCS levels II to V had social functioning below that expected for age.CFCS and age were more strongly associated with development of social functioning than Gross Motor Function Classification System or Manual Ability Classification System and age. What this paper adds: There is a strong relation between social functioning and Communication Function Classification System (CFCS) levels.At 8 to 12 years, 35 out of 45 children in CFCS level I met social functioning age expectations.Twenty‐five out of 29 in CFCS levels II to V had social functioning below that expected for age.CFCS and age were more strongly associated with development of social functioning than Gross Motor Function Classification System or Manual Ability Classification System and age. This original article is commented on by Eslinger on pages 596–597 of this issue. [ABSTRACT FROM AUTHOR]

Aim: To study long‐term clinical and cognitive outcomes of patients with anti‐N‐methyl‐d‐aspartate receptor encephalitis (NMDAR‐E), an acute autoimmune neurological disease with severe acute presentations. Method: In this French multicentre retrospective observational cohort study, patients no older than 18 years with a follow‐up of at least 2 years were included. Data from clinical and cognitive assessments were collected. Results: Eighty‐one patients were included (57 females, 24 males; median age 10 years 7 months [range 1–18 years], median follow‐up 40 months [range 25–53 months]). At last follow‐up, 35 patients (45%) had cognitive impairment, 48 (70%) had academic difficulties, and 65 (92%) needed rehabilitation. Seventy‐one patients (88%) had a modified Rankin Scale score of no more than 2. A higher number of symptoms at diagnosis was associated with cognitive impairment (p = 0.01), while an abnormal electroencephalogram at diagnosis increased the risk of academic difficulties (p = 0.03). Interpretation: Although most children with NMDAR‐E seemed to recover from motor disabilities, more than 45% had cognitive and academic difficulties. The initial severity of symptoms seems to have an impact on cognition and academic performances. What this paper adds: Forty‐five per cent of patients had cognitive impairment at ≥2 years diagnosis of anti‐N‐methyl‐d‐aspartate receptor encephalitis (NMDAR‐E).Seventy per cent of patients had academic difficulties at ≥2 years diagnosis of NMDAR‐E.Ninety‐two per cent of patients needed rehabilitative care at ≥2 years diagnosis of NMDAR‐E.A high number of symptoms at diagnosis were associated with cognitive impairment. What this paper adds: Forty‐five per cent of patients had cognitive impairment at ≥2 years diagnosis of anti‐N‐methyl‐d‐aspartate receptor encephalitis (NMDAR‐E).Seventy per cent of patients had academic difficulties at ≥2 years diagnosis of NMDAR‐E.Ninety‐two per cent of patients needed rehabilitative care at ≥2 years diagnosis of NMDAR‐E.A high number of symptoms at diagnosis were associated with cognitive impairment. [ABSTRACT FROM AUTHOR]

In the last decade, there has been a dramatic increase in the number of families resorting to internet‐based public appeals to fund access to novel, highly expensive, or experimental therapies for rare disorders. Medical crowdfunding may provide a means to fund treatments or interventions, but it raises individual and societal ethical questions. In this review, we consider the ethical challenges crowdfunding poses in paediatric neurology, drawing on the example of gene therapy for spinal muscular atrophy. We discuss physician responsibilities, and how neurologists should respond to crowdfunding that they encounter in clinical practice. We also briefly consider actions that can be taken by clinicians, charities, and crowdfunding websites to reduce harms. The best way to mitigate these harms may be to target the high costs and restrictive criteria that limit access to many novel treatments, and to optimize treatment utility, for instance by newborn screening. What this paper adds: Crowdfunding is a social phenomenon arising from families' inability to access desired treatment.Treatments sought by crowdfunding range from those that are clearly beneficial (but unaffordable) to those that would be ineffective and potentially harmful.Crowdfunding carries a range of harms and risks to families and children and has wider social impact. What this paper adds: Crowdfunding is a social phenomenon arising from families' inability to access desired treatment.Treatments sought by crowdfunding range from those that are clearly beneficial (but unaffordable) to those that would be ineffective and potentially harmful.Crowdfunding carries a range of harms and risks to families and children and has wider social impact. [ABSTRACT FROM AUTHOR]

Aim: To validate a novel equation to estimate weight from mid‐upper arm circumference (MUAC) among children with cerebral palsy (CP) in rural Bangladesh. Method: Children with CP aged 2 to 18 years registered in the Bangladesh CP Register were randomly selected. Data on sociodemographics, Gross Motor Function Classification System level, and anthropometric measurements were extracted. Bland–Altman plots with a 95% agreement limit and Lin's concordance correlation coefficient with 95% confidence intervals (CI) were reported to measure agreement between observed and estimated weight. Percentage error was used to determinate the method's accuracy. Results: There were 497 participants with a mean age at assessment of 9 years (SD 4 years 11 months) (47.7% female). Lin's concordance correlation coefficient between the observed and estimated weights was 0.90 (95% CI 0.89–0.92). Bland–Altman plots showed a reasonable accuracy of the equation in the study cohort. The mean percentage error of the equation was 5.04%. The average difference between observed and estimated weights was −1.02 kg (SD 5.1). The differences between observed and estimated weights were significantly greater among children with weight‐for‐age, height‐for‐age, or BMI‐for‐age z‐scores less than or equal to −4. Interpretation: It is possible to predict the weight of children with CP from MUAC with sufficient accuracy. The equation can be used for populations in low‐resources and low‐ and middle‐income countries. What this paper adds: The equations predict the weight of children with cerebral palsy from their mid‐upper arm circumference reasonably accurately.The difference between observed and estimated weights ranged between 0 kg and ± 5 kg in 81.5% of children.Sex and Gross Motor Function Classification System level did not affect the accuracy of the equations.The equations were less accurate for estimating the weight of severely undernourished children. What this paper adds: The equations predict the weight of children with cerebral palsy from their mid‐upper arm circumference reasonably accurately.The difference between observed and estimated weights ranged between 0 kg and ± 5 kg in 81.5% of children.Sex and Gross Motor Function Classification System level did not affect the accuracy of the equations.The equations were less accurate for estimating the weight of severely undernourished children. [ABSTRACT FROM AUTHOR]

Aim: To explore the phenotypic spectrum and refine the genotype–phenotype correlation of DYNC1H1‐related epilepsy. Method: The clinical data of 15 patients with epilepsy in our cohort and 50 patients with epilepsy from 24 published studies with the DYNC1H1 variants were evaluated. Results: In our cohort, 13 variants were identified from 15 patients (seven males, eight females). Twelve variants were de novo and seven were new. Age at seizure onset ranged from 3 months to 4 years 5 months (median age 1 year). Common seizure types were epileptic spasms, focal seizures, tonic seizures, and myoclonic seizures. Mild‐to‐severe developmental delay was present in all patients. Six patients were diagnosed with West syndrome and one was diagnosed with epileptic encephalopathy with continuous spikes and waves during slow sleep (CSWS). Collectively, in our cohort and published studies, 17% had ophthalmic diseases, 31% of variants were located in the stalk domain, and 92% patients with epilepsy had a malformation of cortical development (MCD). Interpretation: The phenotypes of DYNC1H1‐related epilepsy included multiple seizure types; the most common epileptic syndrome was West syndrome. CSWS is a new phenotype of DYNC1H1‐related epilepsy. One‐third of the variants in patients with epilepsy were located in the stalk domain. Most patients had a MCD and developmental delay. What this paper adds: Nearly 40% of patients with DYNC1H1 variants had epilepsy.Ninety‐two percent of patients with DYNC1H1‐related epilepsy had malformation of cortical development.More than 10% of patients with DYNC1H1‐related epilepsy were diagnosed with West syndrome.Continuous spikes and waves during slow sleep could be a new phenotype of DYNC1H1 variants.One‐third of the variants in patients with epilepsy were located in the stalk domain. What this paper adds: Nearly 40% of patients with DYNC1H1 variants had epilepsy.Ninety‐two percent of patients with DYNC1H1‐related epilepsy had malformation of cortical development.More than 10% of patients with DYNC1H1‐related epilepsy were diagnosed with West syndrome.Continuous spikes and waves during slow sleep could be a new phenotype of DYNC1H1 variants.One‐third of the variants in patients with epilepsy were located in the stalk domain. [ABSTRACT FROM AUTHOR]

Aim: Neurodevelopment is a key outcome for many childhood trials and observational studies. Clinically important decisions may rest on finding relatively small differences in neurodevelopment between groups receiving complex and costly interventions. Our purpose was to determine whether studies which measure neurodevelopment report the numbers, training, and auditing of assessors and, for multiple assessor studies, whether the results were adjusted and if so by which method?Method: Electronic searches were conducted using Medline, Embase, Cinahl, PsycINFO, and the Cochrane Library. A study was eligible if it reported neurodevelopmental outcome in children resident in the UK, less than or equal to 18 years and was published between 2000 and 2015. Trials and observational studies were included.Results: Three hundred and seven full papers were reviewed: 52% of papers did not report the number of assessors used; 21% used a single assessor; and 27% used multiple assessors. Thirty-five per cent mentioned that assessors were trained in the use of the neurodevelopmental tool; 13% of assessors were audited; and only 1% of studies adjusted statistically for the number of assessors.Interpretation: At the very least, the quality of reporting the use of assessors in these research publications is poor, while at worst, the variability of assessors may mask the true relationship between an intervention/observation and neurodevelopmental outcome. [ABSTRACT FROM AUTHOR]

Sunflower syndrome is a rare photosensitive epilepsy which has received little attention in recent medical literature. The historical cases documenting the epilepsy's stereotyped handwaving motion in front of light characterized the behavior as self‐inducing seizures via mimic of stroboscopic effect. However, the relationship between handwaving episodes and attendant generalized electroencephalogram abnormalities, and an appreciation of the compulsive attraction the sun and other light sources hold for these patients, suggest the handwaving motion may be a part of the seizure rather than a mechanism of self‐induction. The lack of awareness of Sunflower syndrome often leads to misdiagnosis. The seizures are often refractory to traditional anticonvulsant medication, and patients resort to behavioral intervention, such as hats and sunglasses, to reduce handwaving episodes. Further study is required to determine the syndrome's natural history and to identify more effective treatment options. What this paper adds: Sunflower syndrome is a rare condition that is often misdiagnosed.Awareness of the clinical and electroencephalogram characteristics of Sunflower syndromemay reduce the prevalence of misdiagnosis. What this paper adds: Sunflower syndrome is a rare condition that is often misdiagnosed.Awareness of the clinical and electroencephalogram characteristics of Sunflower syndromemay reduce the prevalence of misdiagnosis. [ABSTRACT FROM AUTHOR]

Aim: To describe the standardized neurodevelopmental outcomes after the first year of life in children with congenital Zika syndrome (CZS) and those exposed to Zika virus (ZIKV) during fetal life, but without microcephaly at birth. Method: This scoping review included observational studies about the standardized neurodevelopmental outcome in children with CZS or exposed to ZIKV, but without microcephaly, assessed after 12 months of age. The databases searched were MEDLINE/Pubmed, LILACS, Scielo, Scopus, PsycINFO, CINAHL, and Embase. Risk of bias was assessed with the Joanna Briggs Institute Critical Appraisal Checklists. Results: Seventeen papers were included: 12 focused on children with CZS, four on children born without microcephaly, and one described both. Only one of the studies about CZS reported a child with microcephaly and typical development; the remainder described a severe pattern of global developmental delay and cerebral palsy. The prevalence of epilepsy was 74.6%. In the reports about children born without microcephaly, 6.9% to 8.7% had some domain with a score below −2 SD, and three children developed autism spectrum disorder. Interpretation: CZS is associated with severe global developmental delay and cerebral palsy after 1 year of age. In children born without microcephaly, although most have typical development, some may be at risk for impairments. [ABSTRACT FROM AUTHOR]

Aim: To synthesize existing evidence on the effectiveness of speech‐language teleinterventions delivered via videoconferencing to users of augmentative and alternative communication (AAC) devices. Method: A systematic literature search was conducted in 10 electronic databases, from inception until August 2021. Included were speech‐language teleinterventions delivered by researchers and/or clinicians via videoconferencing to users of AAC devices, without restrictions on chronological age and clinical diagnosis. The quality of the studies included in the review was appraised using the Downs and Black checklist and the Single‐Case Experimental Design Scale; risk of bias was assessed using the Risk Of Bias In Non‐Randomized Studies ‐ of Interventions and the single‐case design risk of bias tools. Results: Six teleinterventions including 25 participants with a variety of conditions, such as Down syndrome, autism, Rett syndrome, and amyotrophic lateral sclerosis met the inclusion criteria. Five studies used a single‐case experimental design and one was a cohort study. Teleinterventions included active consultation (n = 2), functional communication training (n = 2), brain–computer interface (n = 1), and both teleintervention and in‐person intervention (n = 1). All teleinterventions reported an increase in participants' independent use of AAC devices during the training sessions compared to baseline, as well as an overall high satisfaction and treatment acceptability. Interpretation: Speech‐language teleinterventions for users of AAC devices show great potential for a successful method of service delivery. Future telehealth studies with larger sample sizes and more robust methodology are strongly encouraged to allow the generalization of results across different populations. What this paper adds: Individuals can learn to use augmentative and alternative communication (AAC) devices independently during tele‐AAC interventions.Service providers and recipients reported an overall high satisfaction and acceptability for AAC services delivered via teleinterventions.Speech‐language teleinterventions may be an effective method of providing AAC intervention services. What this paper adds: Individuals can learn to use augmentative and alternative communication (AAC) devices independently during tele‐AAC interventions.Service providers and recipients reported an overall high satisfaction and acceptability for AAC services delivered via teleinterventions.Speech‐language teleinterventions may be an effective method of providing AAC intervention services. [ABSTRACT FROM AUTHOR]

Aim: To develop a core set of outcome domains to be measured in clinical studies on lower limb orthopaedic surgery for ambulant children with cerebral palsy (CP) that represents the priorities of an international multi‐stakeholder group (children, parent/carers, and health professionals). Method: Potential outcome domains were identified through literature review and qualitative interviews with key stakeholders. These were scored in an international two‐round Delphi survey, using a 9‐point Likert scale. A final consensus meeting with key stakeholders agreed on the most important outcome domains and refined the core outcome set (COS). Results: One hundred and sixty‐one health professionals and 36 individuals with CP and their parents/carers rated 21 of 41 outcomes as important in the Delphi survey. The final consensus group agreed 19 outcomes within eight domains to be included in the final COS: pain and fatigue, lower limb structure, motor function, mobility (daily life activities), gait‐related outcomes, physical activity, independence, and quality of life. Interpretation: A COS for lower limb orthopaedic surgery for children with CP was developed. Incorporating this in the design of future clinical studies will provide a more holistic assessment of the impact of treatment while allowing meaningful comparisons and future synthesis of results from primary studies. What this paper adds: Eight core outcome domains were identified as important to measure in future clinical research.Key stakeholders perceived pain, balance and fall, and independence as very important outcomes.Six contextual factors were identified as essential in surgical decision‐making. What this paper adds: Eight core outcome domains were identified as important to measure in future clinical research.Key stakeholders perceived pain, balance and fall, and independence as very important outcomes.Six contextual factors were identified as essential in surgical decision‐making. This original article is commented on by Shrader on pages 153–154 of this issue. [ABSTRACT FROM AUTHOR]

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Aim: To describe the clinical course of pain intensity in individuals with cerebral palsy (CP) resulting from usual care or specific interventions. Method: We conducted an exploratory prognostic systematic review searching electronic databases from inception to 31st December 2021. Evidence from low and moderate risk‐of‐bias studies was synthesized. Results: We retrieved 2275 citations; 18 studies met the inclusion criteria and 10 were synthesized. The course of pain intensity in children with CP receiving usual care was stable over 15 weeks (χ2[2] = 1.8, p = 0.5). Children who received continuous intrathecal baclofen (CITB) reported significant pain intensity reduction (visual analogue scale [VAS] = −4.2 out of 10, 95% confidence interval [CI] = −6.3 to −2.1]) 6 months postinsertion but similar children receiving usual care had no significant change over 6 months (VAS = 1.3 out of 10, 95% CI = −1.3 to 3.6). Children receiving botulinum neurotoxin A (BoNT‐A) injections had significant decreases in pain after 1 month (numeric rating scale = −6.5, 95% CI = −8.0 to −5.0). Adults with chronic pain receiving usual care reported stable pain intensity over time; pain intensity improved in ambulatory adults exercising and those treated surgically for cervical myelopathy. Interpretation: The course of pain intensity in individuals with CP is unclear. Evidence suggests that children and adults receiving usual care had stable pain intensity over the short or long term. Interventions (CITB and BoNT‐A in children and exercise and surgical treatment for cervical myelopathy in adults) had pain intensity reduction. Larger study samples are needed to confirm these results. What this paper adds: Pain intensity was stable in children with cerebral palsy (CP) receiving usual care.Adults with CP and chronic pain receiving usual care had stable, persistent pain intensity.Children receiving continuous intrathecal baclofen via pump and botulinum neurotoxin A reported significantly lower pain intensities.Adults with chronic pain and dyskinetic CP and cervical myelopathy reported significantly lower pain intensity with exercise or cervical decompression.Limited high‐quality evidence exists describing non‐procedural pain changes in individuals with CP. What this paper adds: Pain intensity was stable in children with cerebral palsy (CP) receiving usual care.Adults with CP and chronic pain receiving usual care had stable, persistent pain intensity.Children receiving continuous intrathecal baclofen via pump and botulinum neurotoxin A reported significantly lower pain intensities.Adults with chronic pain and dyskinetic CP and cervical myelopathy reported significantly lower pain intensity with exercise or cervical decompression.Limited high‐quality evidence exists describing non‐procedural pain changes in individuals with CP. [ABSTRACT FROM AUTHOR]

Assisted reproductive technology (ART) includes fertility treatment in which either eggs or embryos are handled outside a female's body to promote successful pregnancies and healthy offspring. Current ART procedures encompass in vitro fertilization with or without intracytoplasmic sperm injection. The most common complication of ART is related to the consequences of multiple pregnancy, which can be prevented or minimized by reducing the number of embryos transferred to the uterus, commonly single embryo transfer. ART has been shown to be variably associated with adverse short‐ and long‐term perinatal outcomes, including cerebral palsy, autism, neurodevelopmental imprinting disorders, and cancer. However, there is uncertainty as to whether reported problems are related to the ART procedure itself, to factors related to infertility, to other medical and environmental factors, or a combination thereof. From a pathophysiological perspective, whether ART alters epigenetic mechanisms of gene expression, leading to later developmental, medical, and behavioral disorders, is an area of active investigation. With the meticulously conducted short‐ and long‐term outcome studies completed so far, overall, and after controlling for multiple gestations and preterm delivery, the results suggest that ART is a safe procedure, offering hope to many parent(s) wishing for a healthy child. This paper highlights ART methods and the risk factors and confounders in the interpretation of short‐ and long‐term outcome data, providing the reader with a means to evaluate findings and conclusions of outcome studies. What this paper adds: Assisted reproductive technology (ART) is a relatively safe procedure.Single embryo implantation optimizes outcome.Informed consent, including the risks and benefits of ART, should be required.Ongoing longitudinal studies are necessary to fully understand ART outcomes. [ABSTRACT FROM AUTHOR]

Aim: To assess the clinical utility of exome sequencing for patients with developmental and epileptic encephalopathies (DEEs). Method: Over 2 years, patients with DEEs were recruited for singleton exome sequencing. Parental segregation was performed where indicated. Results: Of the 103 patients recruited (54 males, 49 females; aged 2 weeks–17 years), the genetic aetiology was identified in 36 out of 103 (35%) with management implications in 13 out of 36. Exome sequencing revealed pathogenic or likely pathogenic variants in 30 out of 103 (29%) patients, variants of unknown significance in 39 out of 103 (38%), and 34 out of 103 (33%) were negative on exome analysis. After the description of new genetic diseases, a molecular diagnosis was subsequently made for six patients or through newly available high‐density chromosomal microarray testing. Interpretation: We demonstrate the utility of exome sequencing in routine clinical care of children with DEEs. We highlight that molecular diagnosis often leads to changes in management and informs accurate prognostic and reproductive counselling. Our findings reinforce the need for ongoing analysis of genomic data to identify the aetiology in patients in whom the cause is unknown. The implementation of genomic testing in the care of children with DEEs should become routine in clinical practice. What this paper adds: The cause was identified in 35% of patients with developmental and epileptic encephalopathies.KCNQ2, CDKL5, SCN1A, and STXBP1 were the most frequently identified genes.Reanalysis of genomic data found the cause in an additional six patients.Genetic aetiology was identified in 41% of children with seizure onset under 2 years, compared to 18% with older onset.Finding the molecular cause led to management changes in 36% of patients with DEEs. What this paper adds: The cause was identified in 35% of patients with developmental and epileptic encephalopathies.KCNQ2, CDKL5, SCN1A, and STXBP1 were the most frequently identified genes.Reanalysis of genomic data found the cause in an additional six patients.Genetic aetiology was identified in 41% of children with seizure onset under 2 years, compared to 18% with older onset.Finding the molecular cause led to management changes in 36% of patients with DEEs. [ABSTRACT FROM AUTHOR]

Aim: To compare the risks of adverse events 3 months after Onabotulinumtoxin‐A and Lanbotulinumtoxin‐A injections in children with cerebral palsy (CP) and to identify risk factors and associations. Method: A total of 1037 children (682 males, 355 females; mean age 5 years 2 months [SD 3 years]; age range 2 years–17 years 10 months) with CP underwent 1013 Onabotulinumtoxin‐A injections and 418 Lanbotulinumtoxin‐A injections from 2012 to 2021. Information was recorded in a purpose‐built database. Results: The adverse event rates of Onabotulinumtoxin‐A and Lanbotulinumtoxin‐A were reported as 13.92% and 11.96% respectively. Most adverse events were mild and self‐limiting. Children in Gross Motor Function Classification System (GMFCS) levels IV to V had a higher risk of adverse events than those in GMFCS levels I to III (odds ratio [OR] [95% confidence interval {CI}] = 3.65 [1.56, 5.40], p < 0.01). The history of recent illness and higher dose increased the likelihood of adverse events (OR [95% CI] = 2.00 [1.55, 3.00] and 2.20 [1.53, 3.07] respectively, p < 0.01). Sex, age, and the number of injections had no significant effect on adverse event rates (p > 0.05). The incidence of upper respiratory tract infection and lower respiratory tract infection after injections was weakly correlated with the incidence before injections (r = 0.36 and r = 0.27 respectively, p < 0.01). Interpretation: Occurrence of adverse events was similar between Onabotulinumtoxin‐A and Lanbotulinumtoxin‐A in children with CP. Dose, GMFCS level, and health background were risk factors. What this paper adds: The prevalence of adverse events was similar between Onabotulinumtoxin‐A and Lanbotulinumtoxin‐A in children with cerebral palsy (CP).The prevalence of adverse events increased with the severity of CP and the injected dose.Sex, age, and number of injections had no significant effect on the prevalence of adverse events. What this paper adds: The prevalence of adverse events was similar between Onabotulinumtoxin‐A and Lanbotulinumtoxin‐A in children with cerebral palsy (CP).The prevalence of adverse events increased with the severity of CP and the injected dose.Sex, age, and number of injections had no significant effect on the prevalence of adverse events. This original article is commented on by Hastings‐Ison and Graham on pages 10–11 of this issue. [ABSTRACT FROM AUTHOR]

Copyright of Developmental Medicine & Child Neurology is the property of Wiley-Blackwell and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use. This abstract may be abridged. No warranty is given about the accuracy of the copy. Users should refer to the original published version of the material for the full abstract. (Copyright applies to all Abstracts.)