AIM:To establish a consensus regarding the definition of trunk control, assessment tasks, and the need for technology-based evaluation of trunk control. METHOD:Twenty-six international experts completed a 3-round online Delphi study. We collected demographic information and responses concerning the definition of trunk control, assessment tasks and their organization, and technology-based assessment requirements. Agreement was rated using a 4-point Likert scale, with consensus defined as at least 75% agreement. RESULTS:Consensus was achieved for the definition (96% agreement), the categories and subcategories (77%), 37 assessment tasks, and all but one requirement for technology-based assessment. INTERPRETATION:This study established a high level of expert consensus on the definition and assessment of trunk control in children with neurological conditions, including cerebral palsy, stroke, and traumatic brain injury. Subsequent steps involve operationalizing the tasks identified and developing a technology-based assessment of trunk control.
Aim To explore lower leg muscle morphology and intramuscular fat content changes using magnetic resonance imaging (MRI) after 12 months of treatment-as-usual (TAU), after the first botulinum neurotoxin A (BoNT-A) injection in children with cerebral palsy (CP) naive to muscle tone reduction treatment.Method This prospective longitudinal study included 10 children with spastic CP (five female, median age 8 years 11 months, unilateral CP/bilateral CP 7/3, Gross Motor Function Classification System levels I/II 8/2) and 10 typically developing peers (seven female, median age 9 years 6 months). Children with CP received two MRI scans (before and 12 months after the first injection), typically developing peers received one. Muscle volume (normalized by product of weight and height), fascicle length (normalized by height), pennation angle, and intramuscular fat fraction (IFF) of medial gastrocnemius, lateral gastrocnemius, soleus, and tibialis anterior were quantified.Results After 12 months of TAU, normalized muscle volume remained unchanged while IFF increased (medial gastrocnemius: p = 0.008). Compared to typically developing peers, children with CP had smaller normalized muscle volume (tibialis anterior: p = 0.027) and similar IFF at pre-injection timepoint.Interpretation Although normalized muscle volume remained unchanged after the 12-month TAU, concurrent fat infiltration was evident in medial gastrocnemius.
AIM:To explore longitudinal associations between ophthalmological outcome and development of visual perception, fine motor precision, and visual-motor integration (VMI) in children born very preterm (gestational age <32 weeks). METHOD:This was a prospective longitudinal cohort study of 113 children (51 females, 62 males) born very preterm. Visual acuity, low-contrast visual acuity, stereopsis, and strabismus were investigated at 2 years 6 months (n = 98), 6 years 6 months (n = 84), and 12 years (n = 59). VMI was assessed at 6 years 6 months and 12 years, and visual perception and fine motor precision at 12 years, with the Beery-Buktenica Developmental Test of Visual-Motor Integration. Controls born at term were assessed at 6 years 6 months (n = 64) and 12 years (n = 46). RESULTS:For children born preterm, subnormal visual acuity at 2 years 6 months was related to both poorer VMI and fine motor precision at 12 years. Subnormal visual acuity, strabismus, and stereopsis at 6 years 6 months were also related to poorer VMI, visuo-perceptual, and fine motor measures at 12 years. These associations were not present in controls born at term. INTERPRETATION:Subnormal ophthalmological function at 2 years 6 months and 6 years 6 months in children born very preterm is related to poorer performance on VMI, visuo-perceptual, and fine motor precision tasks at 12 years, indicating that the quality of visual input interacts with developing visual-motor abilities. The results of this study provide new insights on the relationship between ophthalmological function and neurodevelopment after preterm birth.
AIM:To assess the effect of the augmented reality application called Minidocs on pain reduction during botulinum neurotoxin A (BoNT-A) injections in children with cerebral palsy (CP). METHOD:Children with CP aged 3 to 8 years undergoing BoNT-A injection were randomized to usual pain management alone (n = 41) or combined with Minidocs (n = 39). Minidocs is an augmented reality application on a digital device offering several games including active distraction, hypnotic suggestion, and counter-aggression features. The primary outcome was the child's pain during BoNT-A injection, combining patient-reported (Faces Pain Scale [FPS]) and observer-reported (Face, Legs, Activity, Cry, Consolability scale [FLACC]) outcomes. Secondary outcomes included anxiety of children (Modified Yale Preoperative Anxiety Scale) and parents (State-Trait Anxiety Inventory Form Y-1), and satisfaction with Minidocs. RESULTS:In total, 14 out of 41 (34.1%) children in the control group and 7 out of 39 (17.9%) children in the experimental group experienced pain (i.e. FPS or FLACC scores ≥4). The difference between groups was not statistically significant (odds ratio 0.36; 95% confidence interval 0.11-1.16; p = 0.087). Changes in anxiety scores from before to after injection did not differ between groups. Satisfaction with the use of Minidocs was high. INTERPRETATION:This study did not demonstrate the benefit of augmented reality on pain reduction. The counter-aggression feature is an innovation of Minidocs worth exploring. Further studies are needed to identify profiles of children who respond to non-drug therapies.
Aim To investigate the content and construct validity of the Both Hands Assessment (BoHA) for children and adolescents with bilateral cerebral palsy (CP), classified in Manual Ability Classification System (MACS) levels I to III, aged 18 months to 18 years.Method In this cross-sectional study, we included 61 adolescents with bilateral CP (37 males, 24 females, mean age 15 years 6 months, SD = 2 years 3 months) who were assessed with the BoHA to investigate content validity. Their BoHA results were combined with data from 210 children (121 males, 89 females, mean age 6 years 4 months, SD = 3 years 1 month), resulting in 271 BoHA assessments for the Rasch measurement model analyses investigating construct validity.Results After revising the bimanual item 'orients objects', the BoHA items were suitable for scoring bimanual performance in adolescents. Strong internal scale validity and reliable item and person measures were demonstrated for the 16 BoHA items, analysed separately for individuals with asymmetric (n = 94) and symmetric (n = 177) hand use. The BoHA logit measures were linked to the same reference frame, enabling comparable overall bimanual performance measures while maintaining separate item difficulty hierarchies.Interpretation The BoHA scale can measure bimanual performance in individuals with bilateral CP, classified in MACS levels I to III, aged 18 months to 18 years. This allows for monitoring development and evaluating the effectiveness of intervention from early childhood to adulthood.
The goal of this study was to assess the impact of wearing an eye patch on seizure frequency in patients with Sunflower syndrome, a reflex photosensitive epilepsy. Caregivers were instructed to record the number of handwaving episodes (HWEs) that occurred per hour under each of the following conditions: (1) no eye patch, (2) wearing an eye patch on the eye ipsilateral to the hand involved in handwaving, and (3) wearing an eye patch on the eye contralateral to the hand involved in handwaving. Patients performed each trial three times, for a total of nine 1-hour trials. Seven patients (five females, two males; age range 8 years 10 months-21 years 5 months, median age = 11 years 2 months) participated in this study. All patients experienced a decrease in HWEs while wearing an eye patch on either eye. Across all patients, the median frequency of HWEs with no eye patch (baseline) was 136 HWEs/hour. When patching the contralateral eye, the median frequency was 26 HWEs/hour. When patching the ipsilateral eye, the median frequency was 23 HWEs/hour. The findings in this study suggest that blocking sensory input to one eye may significantly reduce seizure frequency in patients with Sunflower syndrome. This may inform theories around the pathophysiology of Sunflower syndrome and provide insight into more effective treatment options.
Neonatal hypotonia presents a complex diagnostic challenge that requires timely, structured evaluation to identify underlying causes and initiate appropriate care. A narrative review was conducted based on a targeted search of PubMed and major clinical guidelines, focusing on recent advances in the diagnosis of early-onset hypotonia, spanning from clinical to new genetic tools, while also exploring different aspects of management, including new advanced therapies. A structured clinical assessment remains fundamental, especially where access to advanced investigations is limited, and can help the clinician to select the appropriate investigations to achieve a definite diagnosis. Rapid genomic technologies, including exome and genome sequencing, have significantly improved diagnostic yield. Early detection of treatable conditions can enable timely initiation of intervention. Integrating bedside assessment with genomic tools can accelerate diagnosis and improve outcomes, thus facilitating early intervention.
AIM:To investigate the relationship between quality of general movements and neurodevelopmental outcomes in 2-year-old infants born very preterm (VPT). METHOD:This was a retrospective cohort study including infants born before 32 weeks' gestation. General movements video recordings at 3 months corrected age were assessed with the Motor Optimality Score-Revised (MOS-R) and child cognitive, language, and motor development at 2 years with the Bayley Scales of Infant and Toddler Development, Third Edition. RESULTS:The study included 316 infants (52.5% male, mean [SD] gestational age 28.7 [2.3] weeks, birthweight z-score -0.14 [0.85]). The median MOS-R total score was 23. The mean cognitive composite, language, and motor scores were 102.0 (15.4), 92.1 (16.1), and 95.8 (15.3) respectively. Higher MOS-R total scores were related to better cognitive (p = 0.025) and motor development (p = 0.042). However, associations weakened when controlling for socioeconomic status, gestational age, birthweight, sex, and number of severe neonatal morbidities (i.e. severe brain lesion, necrotizing enterocolitis, sepsis, bronchopulmonary dysplasia, and retinopathy of prematurity). INTERPRETATION:While the quality of general movements at 3 months corrected age is associated with the infant's cognitive and motor development at 2 years corrected age, it might have no incremental predictive power beyond socioeconomic status and the number of severe neonatal morbidities.
Advancements in the treatment of cerebral palsy depend on animal research. Yet, most animal models have not been fully evaluated for spasticity and dystonia using clinically relevant measures of altered tone or movement patterns, which form the basis for diagnosing people with the condition. Sensory differences and pain are almost never studied in animal models. Complicating factors include the diversity of animals and injuries used to model the condition, the diversity of outcomes after acquired injury, and translating clinical measures into reliable and repeatable measurements tailored to animals, ideally using common data elements. We summarize preclinical models based on acquired injury to the nervous system in cerebral palsy research over the years and provide a comparison of developmental time courses for common laboratory animals. We encourage researchers to coalesce on consistent, reliable measurements for assessing both sensory and motor systems to ensure that animal models reflect meaningful aspects of the condition.
