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Aim: To understand the mobility experiences, supportive mobility device (SMD) use, and desired participation outcomes of individuals with cerebral palsy (CP) across the life span, and describe how perspectives of rehabilitation care and professional resources may influence mobility decision-making processes and outcomes.Method: In the second phase of an overarching study, focus groups were conducted with 164 participants (68 individuals with CP; 32 females, 36 males; mean age 17y 8mo, SD 11y 11mo, range 3-68y), 74 caregivers (50 females, 24 males), and 22 healthcare providers (14 females, eight males) across four US cities. Sessions were audio-recorded, transcribed, and analysed using constant comparison.Results: Six themes emerged. Five presented across all stakeholder groups: (1) the system is broken; (2) equipment is simultaneously liberating and restricting; (3) adaptation across the life span; (4) designed for transport, not for living; and (5) sharing our stories and sharing resources. One theme (theme 6) was specific to healthcare providers: caught in the middle.Interpretation: This qualitative study underscores the simultaneous value and frustration associated with SMDs as described by the community with CP, and recognition among all stakeholders of the need to improve connections and resource networks within the community with CP to improve SMD design and provision processes across device types and across the life span for individuals with CP.What This Paper Adds: Supportive mobility devices (SMDs) were most often equated with freedom, participation, and independence. Frustration with SMDs across the life span persisted with regard to design, function, cost, and maintenance. Stakeholders in the community with cerebral palsy are seeking greater networking and resource sharing to enhance SMD provision processes. Access to appropriate SMDs across the life span and the need for system improvement are critical. [ABSTRACT FROM AUTHOR]

Aim: To determine if pain is associated with 12-month incidence of mood affective disorders (MAD) in adults with cerebral palsy (CP).Method: Data from Optum Clinformatics® Data Mart (2013-2016) were used for this retrospective cohort study. Diagnostic codes were used to identify adults (≥18y) with CP, incident cases of MAD, and covariates (other neurodevelopmental conditions, sleep disorders, arthritis). Pain (any type, location) was identified between 1st October 2014 and 30th September 2015. The pain group was divided into new or consistent pain if they had a history of pain (i.e. consistent) in the 12 months before their first pain claim date. Crude incidence rates of MAD (expressed per 100 person-years) were estimated. Cox regression was used to estimate hazard ratio (95% confidence interval [CI]) of MAD after adjusting for covariates.Results: Adults that had new pain (n=859; incidence rate=15.5) or consistent pain (n=1303; incidence rate=17.9) had greater crude incidence rate of MAD compared to adults without pain (n=3726; incidence rate=5.9). The elevated rate of MAD remained after adjusting for covariates, for new pain (hazard ratio=2.4; 95% CI=1.9-3.0) and consistent pain (hazard ratio=2.1; 95% CI=1.7-2.7).Interpretation: Pain is associated with greater incidence of MAD in adults with CP. This association remained after accounting for potential confounding factors.What This Paper Adds: What this paper adds Pain was associated with higher 12-month incidence of mood affective disorders (MAD). The 12-month MAD incidence was similar between new and consistent pain groups. The MAD incidence remained higher adjusting for neurodevelopmental comorbidities, sleep disorders, and arthritis. [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.)

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.)

Aim: To validate the Whitney Comorbidity Index (WCI), which was recently developed to monitor disease status for adults with cerebral palsy (CP), and to identify WCI scores associated with an increased mortality risk using a representative sample of adults with CP.Method: Data from 2016 to 2018 were used from a random 20% sample from the fee-for-service Medicare database for this retrospective cohort study. The WCI was examined as unweighted (WCIunw ) and weighted (WCIw ) among adults at least 18 years old with CP. Cox regression models were developed with mortality as the outcome after adjusting for demographics. A concordance statistic (C-statistic) of at least 0.70 was considered as showing sufficient validity. The hazard ratio of mortality for each WCI score was estimated. Secondary analyses were performed for subgroups with co-occurring epilepsy and/or intellectual disabilities.Results: For the entire group (n=16 728) and subgroups, the WCI showed sufficient validity (C-statistic 0.73-0.81). For the entire group, the mortality rate was elevated for a score of 1 compared with 0 from the WCIunw (hazard ratio 3.06; 95% confidence interval [CI] 1.52-6.17) and WCIw (hazard ratio 4.08; 95% CI 1.69-9.85), and became larger with each WCI score. Results were similar for the subgroups.Interpretation: The WCI is a valid marker for health/disease status for adults with CP. Several WCI score thresholds were identified to assist in clinical decision making for preventive medicine and intervention implementation. What this paper adds The Whitney Comorbidity Index (WCI) is valid among 16 728 adults with CP. The WCI is valid for those with co-occurring epilepsy and/or intellectual disabilities. Thresholds of the WCI score were identified to assist clinical decision making. [ABSTRACT FROM AUTHOR]

Aim: To determine if walking ability and presence of intellectual disability and epilepsy change from childhood to 50 years of age in individuals with cerebral palsy (CP), and if such changes are related to age, sex, or CP subtype.Method: This was a population-based follow-up study of 142 adults born from 1959 to 1978 (82 males, 60 females; mean age 48y 4mo, range 37-58y; 44% unilateral, 35% bilateral, 17% dyskinetic, and 4% ataxic CP) listed in the CP register of western Sweden. We compared childhood data with a follow-up assessment in 2016.Results: At follow-up, walking ability had changed significantly (p<0.001). The proportion of participants walking without aids had decreased from 71% to 62%, and wheelchair ambulation increased from 18% to 25%. Walking ability was related to subtype (p=0.001), but not to age, sex, pain, fatigue, or body mass index. The proportion classified as having intellectual disability had increased from 16% to 22% (p=0.039) and the proportion with epilepsy from 9% to 18% (p=0.015). Of those with childhood epilepsy, 46% were seizure-free without medication.Interpretation: Walking ability and the presence of intellectual disability and epilepsy had changed significantly since childhood. Life-long access to specialized health care is warranted for re-evaluation of impairments, treatment, and assistance. What this paper adds Changes in impairments in individuals with cerebral palsy (CP) over time are related to CP subtype. After 50 years, walking ability in CP may have deteriorated or improved. Intellectual disability in CP may not always be detected in early childhood assessments. Epilepsy in CP may develop after childhood or may be outgrown. [ABSTRACT FROM AUTHOR]

Aim: To develop equations and software to estimate weight using segmental measures for children with cerebral palsy (CP).Method: This was a cross-sectional study. Children and adolescents with CP of both sexes from 2 to 19 years old from five cities in Argentina were included. Weight, mid-upper arm circumference (MUAC), and clinical covariables were collected. Linear regression models with weight as the dependent variable and body segment lengths as predictors were developed and compared for R2 , adjusted R2 , and the root mean square of the error.Results: In total, 381 children and adolescents (mean age 10y 5mo [SD 4y 9mo], range 2-19y; 231 males, 150 females) with a confirmed diagnosis of CP were included. Gross motor function based on the Gross Motor Function Classification System (GMFCS) was as follows: level I, 59; II, 55; III, 59; IV, 69; V, 139. The interaction between weight and other variables such as MUAC, sex, GMFCS, and age was analysed. The concordance correlation coefficient between estimated and observed weight was 0.94 (95% CI 0.93-0.95). From the results of the equations, a free software tool, named Weight Calculator CP, was developed.Interpretation: Weight in children with CP can be predicted using MUAC, GMFCS, and age. Weight Calculator CP can be used in clinical practice when direct weight cannot be obtained. What this paper adds Equations can be used to estimate weight in children with cerebral palsy via body segments. Weight can be estimated according to age and gross motor function. The average difference between estimated and observed weights was 119g. [ABSTRACT FROM AUTHOR]

Aim: To explore the feasibility and possible effects of low-frequency repetitive transcranial magnetic stimulation (rTMS) delivered to the supplementary motor area (SMA) on tic severity and motor system neurophysiology in children with Tourette syndrome.Method: Ten children with Tourette syndrome (eight males, two females; 9-15y) participated in this open-label, phase 1 clinical trial. Treatment consisted of 1800 low-frequency (1Hz) neuronavigated robotic rTMS (100% resting motor threshold) to the SMA, bilaterally for 15 sessions. The primary outcome was a change in Yale Global Tic Severity Scale (YGTSS) total score from baseline to posttreatment. Secondary outcome measures included changes in magnetic resonance spectroscopy metabolite concentrations, TMS neurophysiology measures, TMS motor maps, and clinical assessments (anxiety, depression) from baseline to the end of treatment.Results: The YGTSS score decreased from baseline after treatment (p<0.001; Cohen's d=2.9). All procedures were well-tolerated.Interpretation: Robot-driven, neuronavigated bilateral rTMS of the SMA is feasible in children with Tourette syndrome and appears to reduce tic severity. What this paper adds Repetitive transcranial magnetic stimulation (rTMS) is feasible to use in children with Tourette syndrome. rTMS is tolerated by children with Tourette syndrome. Precise targeting of the supplementary motor area using functional magnetic resonance imaging is also feasible in these children. [ABSTRACT FROM AUTHOR]

Aim: To identify and map studies that have assessed the effect of interventions on lower-limb macroscopic muscle-tendon morphology in children with spastic cerebral palsy (CP).Method: We conducted a literature search of studies that included pre- and post-treatment measurements of lower-limb macroscopic muscle-tendon morphology in children with spastic CP. Study quality was evaluated and significant intervention effects and effect sizes were extracted.Results: Twenty-eight articles were identified. They covered seven different interventions including stretching, botulinum neurotoxin A (BoNT-A), strengthening, electrical stimulation, whole-body vibration, balance training, and orthopaedic surgery. Study quality ranged from poor (14 out of 28 studies) to good (2 out of 28). Study samples were small (n=4-32) and studies were variable regarding which muscles and macroscopic morphological parameters were assessed. Inconsistent effects after intervention (thickness and cross-sectional area for strengthening, volume for BoNT-A), no effect (belly length for stretching), and small effect sizes were reported.Interpretation: Intervention studies reporting macroscopic muscle-tendon remodelling after interventions are limited and heterogeneous, making it difficult to generalize results. Studies that include control groups and standardized assessment protocols are needed to improve study quality and data synthesis. Lack or inconclusive effects at the macroscopic level could indicate that the effects of interventions should also be evaluated at the microscopic level.What This Paper Adds: Muscle-targeted interventions to remodel muscle morphology are not well understood. Studies reporting macroscopic muscle remodelling following interventions are limited and heterogeneous. Passive stretching may preserve but does not increase muscle length. The effects of isolated botulinum neurotoxin A injections on muscle volume are inconsistent. Isolated strengthening shows no consistent increase in muscle volume or thickness. [ABSTRACT FROM AUTHOR]

Aim: To investigate the utility of the Insight Inventory (a structured clinical inventory completed by caregivers) for assessment of children with cerebral visual impairment; and to investigate effectiveness of tailored habilitational strategies derived from the responses to the Insight Inventory.Method: Fifty-one eligible children (26 males, 25 females; mean age 9y 5mo, SD 3y, range 5-16y) were recruited from Great Ormond Street Hospital, London. They underwent baseline assessment including neuro-ophthalmological and neuropsychological evaluations, and parent- and child-reported ratings on a questionnaire-based measure of quality of life. Parents also completed the Insight Inventory. On the basis of responses to the Inventory, families received individualized habilitational strategies. Follow-up assessments 6 months later included repeating the Insight Inventory and quality of life questionnaires.Results: Correlations were found between the Insight Inventory and the Wechsler Intelligence Scale for Children, Fourth Edition, the Beery-Buktenica Test of Visual-Motor Integration, and the Benton Facial Recognition Test, suggesting that the Insight Inventory is an effective tool to estimate visual-perceptual difficulties. At 6 months follow-up, caregiver reports indicated significant improvements in the quality of life of children below the age of 12 years.Interpretation: The Insight Inventory is a simple questionnaire which covers practical aspects of cognitive visual function in everyday life. It provides in-depth information about the aspects that children struggle with. It can also guide programmes of individualized habilitation strategies, which may enhance the quality of life of younger children.What This Paper Adds: Questionnaire scores demonstrate biologically plausible correlations with formal neuropsychological tests of visual function. After administration of matched practical habilitational strategies, younger children showed improvement in quality of life and functional vision scores. [ABSTRACT FROM AUTHOR]

Aim: To evaluate the properties of the Infant Motor Activity Log (IMAL), a caregiver-report for frequency and quality of use of more affected upper extremity in infants with neurological and functional impairments.Method: This was a prospective cohort study of 66 children (34 females, 32 males) aged 6 to 24 months (mean age [SD] 13.7mo [5.3]) with neurological and functional impairments and a confirmed cerebral palsy diagnoses after 2 years, and 51 age-matched typically developing children. The IMAL was administered at baseline and 4 weeks later. Typically developing infants were tested with randomly assigned 'more affected' upper extremity. Psychometric properties were evaluated using Spearman's correlation coefficient, Cronbach's alpha, and Jonckheere-Terpstra tests.Results: In the children with impairments, the IMAL showed internal consistency (alpha≥0.88) for the How Well Scale (HWS) and How Often Scale (HOS). Test-retest reliability was 0.64 (HOS) and 0.70 (HWS), demonstrating stability over time. Correlation with Bayley Scales of Infant and Toddler Development, Third Edition more affected arm raw scores were 0.70 (HOS) and 0.72 (HWS) (p<0.001) demonstrating construct validity. Both scale scores decreased with increasing Gross Motor Function Classification System and Mini-Manual Ability Classification System (p<0.001) levels, supporting discriminative validity. Discrimination between typically developing infants and infants with impairments was high (HWS: area under the receiver operating characteristic curve [AUC] 0.96, 95% confidence interval [CI] 0.94-0.99 and HOS AUC=0.95, CI 0.92-0.99).Interpretation: The IMAL is a valid and reliable discriminative caregiver measure of upper limb performance and may complement measures of capacity in infants with neurological and functional impairments.What This Paper Adds: The Infant Motor Activity Log (IMAL) is a valid and reliable measure of caregiver perception of upper limb function. The IMAL fills a measurement gap for infant motor performance in children with impairments. The IMAL discriminates among motor function levels. [ABSTRACT FROM AUTHOR]

Aim: To analyse the development of motor skill and executive function in school-aged children with and without developmental coordination disorder (DCD).Method: Using a longitudinal design, 186 children (86 males, 100 females) aged 6 to 11 years at Time 1 were tested over a 2-year period, 52 of whom were diagnosed with DCD at Time 1 (27 males, 25 females; mean age 8y 5mo, SD 1y 6mo) using DSM-5 criteria. The McCarron Assessment of Neuromuscular Development assessed motor status at Time 1 and at 2-year follow-up (Time 2). Executive function was assessed using a well-validated measure, the Groton Maze Learning Test.Results: The DCD cohort at Time 1 had moderate incidence of executive function deficit (41%). Most importantly, at a group level, children with persisting DCD (across Times 1 and 2) also showed significantly lower levels of executive function than children with typical motor development at both time points. At an individual level, around 26% of children in this group had persisting executive function deficits relative to normal ranges of performance.Interpretation: Children with persisting DCD are at significant risk of executive function issues. The combination of motor and cognitive issues as a potential risk factor in the longer-term development of children is discussed.What This Paper Adds: Around half of children initially diagnosed with developmental coordination disorder (DCD) had the same diagnosis at 2-year follow-up. 41% of children with DCD have impaired executive function. Children with persisting DCD show poorer executive function than those with typical motor development or remitting DCD. [ABSTRACT FROM AUTHOR]

Aim: To describe the trajectory of clinical signs in children who developed human immunodeficiency virus encephalopathy (HIVE) after starting early antiretroviral therapy (ART).Method: This was a retrospective case-cohort description of HIVE among Cape Town participants from the Children with HIV Early AntiRetroviral treatment (CHER) trial. Criteria for HIVE diagnosis were at least two of: (1) acquired central motor deficit, (2) impaired brain growth, and (3) failure to attain or loss of developmental milestones in the absence of an alternative aetiology.Results: Of 133 surviving participants who initiated ART at a median age of 9 weeks and who were followed until a median age of 6 years, 20 (12%) developed HIVE at a median age 31 months (interquartile range 19-37). In these, the first neurological deterioration was noticed at a median age of 19 months, when 16 were on ART and nine had undetectable HIV viral load for a median of 12 months. Signs of upper motor neurons were present in 18, of whom 12 resolved and four had persistent spastic diplegia; 19 had motor delay, of whom 14 resolved; 12 had language delay, of whom 11 resolved; and 16 had impaired brain growth, of whom only five recovered. For the 16 participants already on ART at HIVE diagnosis, regimens were not altered in response to diagnosis.Interpretation: HIVE may occur despite early ART initiation and virological suppression and then resolve on unchanged ART, most likely as intrathecal inflammation subsides.What This Paper Adds: Despite suppressive antiretroviral therapy, children can develop human immunodeficiency virus encephalopathy, The most common manifestations are motor deficits and impaired brain growth. Most experience improvement, with many resolving without additional intervention. [ABSTRACT FROM AUTHOR]

Aim: This study compared the cost-effectiveness of botulinum neurotoxin A (BoNT-A) injections with two-duct ligation of the submandibular glands as treatment for severe drooling after one treatment cycle.Method: The study was part of a larger, partly single-blinded, randomized clinical trial (trialregister.nl identifier NTR3537). Data were collected between 2012 and 2017. Evaluation was at 32 weeks after one treatment cycle. Fifty-seven patients with cerebral palsy or other neurological, non-progressive disorders and severe drooling classified as having a drooling frequency ≥3 or a drooling severity ≥2, in whom conservative treatment was deemed ineffective, were randomized to treatment by BoNT-A or two-duct ligation. An incremental cost-effectiveness ratio (ICER) was calculated using the success rates as the measure of benefit. Treatment success was defined as a decrease ≥50% from baseline to 32 weeks in the subjective visual analogue scale for the severity of drooling or the objective drooling quotient.Results: Fifty-three patients were analysed (22 females, 31 males; mean age 11y, range 8-22y). Average costs for one treatment cycle, which included one BoNT-A injection, were €1929 (standard error 62) for BoNT-A and €3155 (standard error 99) for two-duct ligation. Treatment success was in favour of two-duct ligation (63% vs 27%; number needed to treat 3). The ICER was €34 per 1% gain in treatment success in favour of two-duct ligation versus BoNT-A.Interpretation: The additional cost of two-duct ligation is to some extent offset by a larger treatment success rate compared with BoNT-A.What This Paper Adds: Botulinum neurotoxin A (BoNT-A) is less expensive per percentage of success than two-duct ligation. The additional cost of two-duct ligation over BoNT-A is offset by greater treatment success. [ABSTRACT FROM AUTHOR]

Aim: To examine the efficacy of Hand-Arm Bimanual Intensive Therapy (HABIT) on daily functioning, unimanual dexterity, and bimanual performance of children with bilateral cerebral palsy (CP) compared with customary care.Method: Forty-one children with bilateral CP, aged 4 to 16 years, classified in levels I to III of the Manual Ability Classification System, were randomly assigned to HABIT (90h) (n=21) or to customary care (4.5h) (n=20). Participants' daily functioning (Pediatric Evaluation of Disability Inventory [PEDI], Canadian Occupational Performance Measure [COPM]), unimanual dexterity (Jebsen-Taylor Test of Hand Function, Box and Blocks Test [BBT]), and bimanual performance (Both Hands Assessment) were assessed pre-, post-, and 6 months after the intervention. Linear mixed-effects models were used for inferential analysis.Results: Children participating in HABIT showed greater improvements in daily functioning (COPMperformance : χ12 =9.50, p<0.01; COPMsatisfaction : χ12 =5.07, p<0.05; PEDIfunctional skills : χ12 =6.81, p<0.01; PEDIcaregiver assistance : χ12 =6.23, p<0.05) and in the dexterity of the dominant hand (BBT: χ12 =3.99, p<0.05) compared with children maintaining customary care. Group or time effects did not explain any variance in bimanual performance or in the dexterity of the non-dominant hand.Interpretation: HABIT may be beneficial for children with bilateral CP, with benefits evidenced for daily functioning outcomes.What This Paper Adds: Hand-Arm Bimanual Intensive Therapy (HABIT) improved daily functioning of children with bilateral cerebral palsy (CP). Bimanual performance, measured by the Both Hands Assessment, did not change after HABIT in children with bilateral CP. Children with asymmetric and symmetric hand use exhibited similar improvements after HABIT. [ABSTRACT FROM AUTHOR]

Aim: To evaluate pain prevalence and characteristics in children and adolescents with predominant dyskinetic and mixed (dyskinetic/spastic) cerebral palsy (CP) motor types.Method: Seventy-five participants with a diagnosis of CP and confirmed dyskinetic or mixed (dyskinetic/spastic) motor type took part in a multisite cross-sectional study. The primary outcome was carer-reported pain prevalence (preceding 2wks) measured using the Health Utilities Index-3. Secondary outcomes were chronicity, intensity, body locations, quality of life, and activity impact.Results: Mean participant age was 10 years 11 months (SD 4y 2mo, range 5-18y). There were 44 males and 31 females and 37 (49%) had predominant dyskinetic CP. Pain was prevalent in 85% and it was chronic in 77% of participants. Fifty-two per cent experienced moderate-to-high carer-reported pain intensity, which was significantly associated with predominant dyskinetic motor types (p=0.008). Pain occurred at multiple body locations (5 out of 21), with significantly increased numbers of locations at higher Gross Motor Function Classification System levels (p=0.02). Face, jaw, and temple pain was significantly associated with predominant dyskinetic motor types (p=0.005). Poorer carer proxy-reported quality of life was detected in those with chronic pain compared to those without (p=0.03); however, chronic pain did not affect quality of life for self-reporting participants.Interpretation: Pain was highly prevalent in children and adolescents with predominant dyskinetic and mixed (dyskinetic/spastic) motor types, highlighting a population in need of lifespan pain management.What This Paper Adds: Chronic pain prevalence in children and adolescents with predominant dyskinetic and mixed (dyskinetic/spastic) motor types is high. Pain occurs across multiple body locations in predominant dyskinetic and mixed (dyskinetic/spastic) motor types. Less recognized locations of pain include the face, jaw, and temple for predominant dyskinetic motor types. [ABSTRACT FROM AUTHOR]

Aim: The purpose of this scoping review was to describe and map the literature available on the participation of young people (0-21y) with disabilities and/or chronic conditions living in low- and middle-income countries (LMICs).Method: A systematic search and selection process identified 78 publications. Descriptive data were extracted using a data-charting form, and studies were mapped using the family of participation-related constructs framework.Results: The findings demonstrated that, although the published evidence is steadily increasing, the participation research on this vulnerable population is still either absent or very scarce in the majority of LMICs, and very little is known about the participation of children with chronic health conditions. Most studies included in this review focused on attendance or 'being there'.Interpretation: Although attendance is an important aspect, more needs to be done to understand children's experiences or involvement while attending, thus capturing both dimensions of participation.What This Paper Adds: There is an increasing trend in research on participation patterns of children with disabilities in low- and middle-income countries. Most research focuses on children's attendance, or 'being there'. We know very little about children's involvement, or experience, while attending daily activities. [ABSTRACT FROM AUTHOR]

Aim: To evaluate the effect of progressive resistance training of the ankle plantarflexors on gait efficiency, activity, and participation in adolescents with cerebral palsy (CP).Method: Sixty-four adolescents (10-19y; 27 females, 37 males; Gross Motor Function Classification System [GMFCS] levels I-III) were randomized to 30 sessions of resistance training (10 supervised and 20 unsupervised home sessions) over 10 weeks or usual care. The primary outcome was gait efficiency indicated by net nondimensional oxygen cost (NNcost). Secondary outcomes included physical activity, gross motor function, participation, muscle strength, muscle and tendon size, and muscle and tendon stiffness. Analysis was intention-to-treat.Results: Median attendance at the 10 supervised sessions was 80% (range 40-100%). There was no between-group difference in NNcost at 10 (mean difference: 0.02, 95% confidence interval [CI] -0.07 to 0.11, p=0.696) or 22 weeks (mean difference: -0.08, 95% CI -0.18 to 0.03, p=0.158). There was also no evidence of between-group differences in secondary outcomes at 10 or 22 weeks. There were 123 adverse events reported by 27 participants in the resistance training group.Interpretation: We found that 10 supervised sessions and 20 home sessions of progressive resistance training of the ankle plantarflexors did not improve gait efficiency, muscle strength, activity, participation, or any biomechanical outcome among adolescents with CP.What This Paper Adds: Thirty sessions of progressive resistance training of the ankle plantarflexors over 10 weeks did not improve gait efficiency among ambulatory adolescents with cerebral palsy. Resistance training did not improve muscle strength, activity, or participation. Ninety percent of participants experienced an adverse event. Most adverse events were expected and no serious adverse events were reported. [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.)

Aim: To assess effects of growth on lower limb maximal isometric muscle strength (MIMS) development in children with developmental coordination disorder (DCD).Method: This observational study used hand-held dynamometry to evaluate MIMS (hip abductors, flexors, extensors; knee flexors and extensors; ankle dorsiflexors) in children with DCD (n=33, 12 females, 21 males, 6-12y, mean [SD] age 9y [2y]). Regression analysis compared changes in MIMS for similar changes in growth (height or body mass) for children with DCD and typically developing children (pre-existing database, n=183), controlling for age and sex.Results: For the same height gain, the gain in muscle strength was 37.3% to 69.2% less in children with DCD compared with typically developing children, with significantly lower slopes (p-value between <0.001-0.042) in all muscle groups tested except knee extensors and ankle dorsiflexors in females. Strength gains related to body mass gains were not different for children with DCD compared to typically developing children.Interpretation: Even when growing at a similar rate, children with DCD do not develop muscle strength gains at the same rate as their typically developing peers.What This Paper Adds: Strength gains with growth (height) may be reduced in children with developmental coordination disorder. [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.)

Aim: To evaluate the feasibility, acceptability, and preliminary effects of Participation and Environment Measure-Plus (PEM+) 2.0, an optimized version of a web-based, participation-focused, care-planning tool.Method: Twenty-two caregivers of children aged 0 to 5 years receiving rehabilitation services, who reported dissatisfaction with their child's participation, had internet access, and could read and write English, were recruited for this 2-week, single-arm pilot trial. Feasibility was assessed through retention rates, completion time, percentage of care plans developed relative to caregiver reported need, and independent completion of PEM+ 2.0. Acceptability was assessed by the Usefulness, Satisfaction, and Ease of Use Questionnaire. Preliminary effects were assessed by two items on caregiver reported impact of PEM+ 2.0 on confidence for addressing their child's participation.Results: Eighteen caregivers completed at least one iteration of PEM+ 2.0; of those, 17 were female and 15 were 30 to 39 years old. The median completion time was 12.99 minutes (quartile 1, 6.30; quartile 3, 17.33), mean care plan creation relative to need was 50% (standard deviation [SD] 31), and 17 completed PEM+ 2.0 independently. Mean acceptability scores were 3.80 to 4.97 (SD 1.25-1.97) and mean preliminary effect scores were 4.61 to 4.72 (SD 1.85-2.24), out of 7.0. There were strong and significant positive associations between two of the three estimates of PEM+ 2.0 acceptability and caregiver confidence (r=0.577-0.793, p<0.01).Interpretation: Electronic health tools have the potential for facilitating family-centered care in pediatric rehabilitation. PEM+ 2.0 is a feasible tool within pediatric rehabilitation and has potential to be an acceptable tool for improving caregiver confidence for promoting their child's participation in valued activities.What This Paper Adds: Participation and Environment Measure-Plus (PEM+) 2.0 is feasible for collaboratively engaging caregivers in the care-planning process. Caregivers perceived PEM+ 2.0 as helpful for thinking about their child's participation and what can be done to improve it. Feasibility and acceptability results will inform PEM+ 2.0 optimizations. [ABSTRACT FROM AUTHOR]

The aim of this case series was to assess and characterize cognitive abilities, autistic traits, and adaptive behaviour in Tatton-Brown-Rahman syndrome (TBRS). The sample included 18 individuals with a clinical and genetic diagnosis of TBRS (11 males, seven females; mean age 17y 7mo, SD 9y 5mo, range 7y 2mo-33y 10mo). The British Ability Scales, Third Edition and the Autism Diagnostic Observation Schedule, Second Edition (ADOS-2) were administered to all participants. The Social Responsiveness Scale, Second Edition and the Vineland Adaptive Behaviour Scales, Third Edition were completed by a parent/caregiver. The majority of participants (n=15) had intellectual disability and General Conceptual Ability scores ranged from 39 to 76 (mean 53.17, SD 12.13). Participants displayed a profile of better verbal ability compared with non-verbal reasoning ability and spatial ability. Autistic traits were prevalent and eight participants scored above the cut-off on the ADOS-2, although symptoms were less pronounced in older individuals. Adaptive functioning was impaired but commensurate with intellectual ability. Overall, TBRS is associated with an uneven cognitive profile and a high prevalence of autistic traits. This has implications for identifying appropriate services and support that may be beneficial for individuals with TBRS. WHAT THIS PAPER ADDS: Tatton-Brown-Rahman syndrome is associated with intellectual disability and impaired adaptive functioning. Autistic traits were prevalent within the sample. Lower intellectual ability and adaptive behaviour were associated with greater severity of autistic traits. [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.)

Aim: To determine neurocognitive performance and behavioural problems in children with Panayiotopoulos syndrome.Method: All 18 children (10 females, 8 males; mean age 4y 7mo; SD 1y 10mo) diagnosed with Panayiotopoulos syndrome at the Kempenhaeghe Epilepsy Center in the Netherlands between 2010 and 2017 were analysed retrospectively. All underwent a neuropsychological/behavioural assessment, an academic assessment, and a 24-hour electroencephalogram.Results: Mean full-scale IQ (93.5; range 76-123; p=0.04) and performance IQ (93.2; range 76-126; p=0.04) were within the normal range, although significantly lower compared to the normative mean. Verbal IQ (96.3; range 76-118) and processing speed (96.1; range 74-114) were not significantly lower. Simple auditory/visual reaction times, visual attention, visual-motor integration, and verbal memory were significantly lower compared to normative values. On average, patients with Panayiotopoulos syndrome were 8 months behind in arithmetic speed and 11 months behind in reading speed for the number of months in school. Behavioural questionnaires revealed significantly higher scores on reported internalizing behavioural problems.Interpretation: Children with Panayiotopoulos syndrome demonstrated diffuse cognitive dysfunction in full-scale IQ, performance IQ, visual attention, visual-motor integration, and verbal memory. A high incidence of internalizing behavioural problems was reported. This strongly suggests neuropsychological and behavioural comorbidity in children with Panayiotopoulos syndrome.What This Paper Adds: Children with Panayiotopoulos syndrome are at risk for cognitive deficits in various cognitive domains. Children with Panayiotopoulos syndrome are also prone to internalizing behavioural problems. Mild-to-severe academic underachievement was present in more than half of the children with Panayiotopoulos syndrome. [ABSTRACT FROM AUTHOR]

Aim: To explore how patients with cerebral palsy (CP) perceive their gait and evaluate associations between subjective gait perception and: objective gait parameters, endurance, pain, and fatigue.Method: Sixty-two patients (21 females and 41 males; mean [SD] age 20y [5y 1mo], range 15-29y) performed a clinical gait analysis. Self-selected walking speed, Gait Profile Score, and Gait Variable Score were calculated. Subjective gait perception was assessed with a visual analogue scale using the question: 'On a scale from 0 (worst) to 10 (optimal), how would you describe your walking today?'. A 6-minute walk test (6MWT) measured endurance; the 36-Item Short Form Health Survey (SF-36) evaluated quality of life. T-tests, Pearson correlations, and univariate and multiple linear regression models were used to compare and find associations between the data.Results: Overall mean (SD) subjective gait perception was 7.5 (1.8) and was significantly higher for patients in Gross Motor Function Classification System (GMFCS) level I (7.9 [1.5]) than for patients in GMFCS levels II and III (5.9 [2.0]). Positive correlations were found between subjective gait perception and gait scores, walking speed, 6MWT distance, and SF-36 score. Only walking speed was a significant predictor of subjective gait perception.Interpretation: Subjective gait perception was influenced by GMFCS level and linked partially with the walking speed. The gait quality did not explain subjective gait perception. It is important to combine subjective and objective gait scores to develop personalized therapeutic goals.What This Paper Adds: Subjective gait perception is influenced by the physical impairment levels of patients with cerebral palsy. Subjective gait perception and objective gait scores are associated. Walking speed is the only predictor of gait perception. [ABSTRACT FROM AUTHOR]

Aim: To evaluate cortical excitability with transcranial magnetic stimulation (TMS) in children with new-onset epilepsy before and after antiepileptic drugs (AEDs).Method: Fifty-five drug-naïve patients (29 females, 26 males; 3-18y), with new-onset epilepsy were recruited from 1st May 2014 to 31st October 2017 at the Child Neurology Department, Queen Silvia's Children's Hospital, Gothenburg, Sweden. We performed TMS in 48 children (23 females, 25 males; mean [SD] age 10y [3y], range 4-15y) with epilepsy (27 generalized and 21 focal) before and after the introduction of AEDs. We used single- and paired-pulse TMS. We used single-pulse TMS to record resting motor thresholds (RMTs), stimulus-response curves, and cortical silent periods (CSPs). We used paired-pulse TMS to record intracortical inhibition and facilitation at short, long, and intermediate intervals.Results: There were no differences in cortical excitability between children with generalized and focal epilepsy at baseline. After AED treatment, RMTs increased (p=0.001), especially in children receiving sodium valproate (p=0.005). CSPs decreased after sodium valproate was administered (p=0.050). As in previous studies, we noted a negative correlation between RMT and age in our study cohort. Paired-pulse TMS could not be performed in most children because high RMTs made suprathreshold stimulation impossible.Interpretation: Cortical excitability as measured with RMT decreased after the introduction of AEDs. This was seen in children with both generalized and focal epilepsy who were treated with sodium valproate, although it was most prominent in children with generalized epilepsy. We suggest that TMS might be used as a prognostic tool to predict AED efficacy.What This Paper Adds: Resting motor threshold (RMT) correlated negatively with age in children with epilepsy. No differences in cortical excitability were noted between patients with generalized and focal epilepsy. Treatment with antiepileptic drugs decreased cortical excitability as measured with transcranial magnetic stimulation (TMS). Decreased cortical excitability with increased RMT was recorded, especially after sodium valproate treatment. Paired-pulse TMS was difficult to perform because of high RMTs in children. [ABSTRACT FROM AUTHOR]

Aim: To: (1) investigate the relationship between upper-limb impairment and health-related quality of life (HRQoL) for children with cerebral palsy and (2) develop a mapping algorithm from the Cerebral Palsy Quality of Life Questionnaire for Children (CPQoL-Child) onto the Child Health Utility 9D (CHU9D) measure.Method: The associations between physical and upper-limb classifications and HRQoL of 76 children (40 females, 36 males) aged 6 to 15 years (mean age 9 years 7 months [SD 3y]) were assessed. Five statistical techniques were developed and tested, which predicted the CHU9D scores from the CPQoL-Child total/domain scores, age, and sex.Results: Most participants had mild impairments. The Manual Ability Classification System (MACS) level was significantly negatively correlated with CHU9D and CPQoL-Child (r=-0.388 and r=-0.464 respectively). There was a negative correlation between the Neurological Hand Deformity Classification (NHDC) and CPQoL-Child (r=-0.476, p<0.05). The generalized linear model with participation, pain domain, and age had the highest predictive accuracy.Interpretation: The weak negative correlations between classification levels and HRQoL measures may be explained by the restricted range of impairment levels of the participants. The MACS and NHDC explained the impact of upper-limb impairment on HRQoL better than the other classifications. The generalized linear model with participation, pain, and age is the suggested mapping algorithm. The suggested mapping algorithm will facilitate the use of CPQoL-Child for economic evaluation and can be used to conduct cost-utility analyses.What This Paper Adds: The Manual Ability Classification System and Neurological Hand Deformity Classification were the best predictors of health-related quality of life measures. Age and Cerebral Palsy Quality of Life Questionnaire for Children participation and pain domain scores can predict Child Health Utility 9D scores. [ABSTRACT FROM AUTHOR]

Aim: To assess the reliability and predictive validity of the developmental and socio-emotional scales of the Standardized Infant NeuroDevelopmental Assessment (SINDA).Method: To assess reliability, two sets of three assessors forming eight assessor-pairs independently rated the developmental and socio-emotional scales of 60 infants. To evaluate predictive validity, 223 infants (gestational age 30wks [range 23-41wks]; 117 males, 106 females) attending a non-academic outpatient clinic were assessed by different assessors with SINDA's neurological, developmental, and socio-emotional scales. Atypical neurodevelopmental outcome at a corrected age of 24 months or older implied a Bayley Mental or Psychomotor Developmental Index score of less than 70 or neurological disorder (including cerebral palsy). Behavioural and emotional disorders were classified according to the International Classification of Diseases, 10th Revision. Predictive values were calculated from SINDA (2-12mo corrected age, median 7mo) and typical versus atypical outcome, and for intellectual disability only (Mental Developmental Index <70).Results: Assessors highly agreed on the developmental and socio-emotional assessments (developmental scores: Spearman's rank correlation coefficient ρ=0.972; single socio-emotional behaviour items: Cohen's κ=0.783-0.896). At 24 months or older, 65 children had atypical outcome. Atypical neurological scores predicted atypical outcome (sensitivity 83%, specificity 96%); atypical developmental scores predicted intellectual disability (sensitivity 77%, specificity 92%). Atypical emotionality and atypical self-regulation were associated with behavioural and emotional disorders.Interpretation: SINDA's three scales are reliable, and have a satisfactory predictive validity for atypical developmental outcome at 24 months or older in a non-academic outpatient setting. SINDA's developmental scale has promising predictive validity for intellectual disability. SINDA's socio-emotional scale is a tool for caregiver counselling.What This Paper Adds: Standardized Infant NeuroDevelopmental Assessment (SINDA)'s developmental and socio-emotional scales have excellent interrater reliability. Replication of the satisfactory validity of SINDA's neurological scale for atypical outcome. [ABSTRACT FROM AUTHOR]

Aim: To determine if genetic variation associated with decreased dopamine neurotransmission predicts a decrease in motor development in a convenience cohort study of infants born extremely-low-birthweight (ELBW).Method: Four hundred and ninety-eight infants born ELBW had genome-wide genotyping and a neurodevelopmental evaluation at 18 to 22 months of age, corrected for preterm birth. A polygenic risk score (PRS) was created to combine into one predictor variable the hypothesized influences on motor development of alleles at seven independent single nucleotide polymorphisms previously associated with relative decreases in both dopamine neurotransmission and motor learning, by summing the number of alleles present in each infant (range=0-14). The motor development outcome was the Psychomotor Development Index (PDI) of the Bayley Scales of Infant Development, Second Edition. The linear regression models were adjusted for seven clinical and four genetic ancestry covariates. The mean PRS of infants with cerebral palsy (CP) was compared to those without CP.Results: PRS was inversely related to PDI (p=0.011). Each 1-point increase in PRS resulted in an average decrease in PDI of 1.37 points. Patients with CP did not have a greater mean PRS than those without (p=0.67), both with and without adjustment for covariates.Interpretation: Genetic variation that favors a decrease in dopamine neurotransmission predisposes to a decrease in motor development in infants born ELBW, but not to the diagnosis of CP.What This Paper Adds: Genetic variation in dopamine neurotransmission was associated with a decrease in motor development in infants born at an extremely-low-birthweight. It does not predispose to the diagnosis of cerebral palsy. [ABSTRACT FROM AUTHOR]

Aim: To compare long-term psychosocial and functional outcomes of young adults with uncomplicated childhood-onset epilepsy (COE) to population norm controls utilizing a controlled prospective cohort study.Method: Psychosocial and functional outcomes were assessed at 10-year follow-up. Fifty-three young adults (27 males, 26 females) with COE (n=21 remission; 18y 1mo-30y 9mo; mean age 23y 4mo [SD 3y 4mo]; mean age of epilepsy onset 12y [SD 3y 2mo]) were compared to 55 (23 males, 32 females) first-degree cousin controls (18y 5mo-29y 8mo; mean age 23y 6mo [SD 3y]). Seizure remission status and baseline comorbidities (attention-deficit/hyperactivity disorder [ADHD], depressive disorders, anxiety disorders, and academic problems) were examined as possible risk factors for significant differences in functional outcomes.Results: Poorer functional outcomes, indicated by patient rated cognition and overall disability, were evident among young adults with epilepsy compared to controls (all p<0.05). These difficulties were due to baseline comorbid ADHD and academic problems. Remission status was not related to measured cognition and overall disability.Interpretation: Psychosocial outcomes of young adults with COE were similar to controls. In contrast, functional outcomes were worse in epilepsy across cognition and overall disability. Baseline comorbid ADHD and academic problems were identified as risk factors at 10-year follow-up suggesting that these early recognized comorbidities at or near diagnosis have long-term impacts.What This Paper Adds: Young adults with childhood-onset epilepsy (COE) and controls have similar psychosocial outcomes 10 years after diagnosis. Young adults with COE report greater limitations in cognition and overall disability than controls. Baseline presence of attention-deficit/hyperactivity disorder and academic problems significantly affect cognitive and overall disability scores. [ABSTRACT FROM AUTHOR]

Aim: To examine self- and proxy-reported symptoms of depression in children with epilepsy.Method: This was a prospective longitudinal cohort study of children with epilepsy. Participants were treated at six Canadian tertiary-care centers and followed over 28 months with repeated assessments of child self-reported symptoms of depression using the Children's Depression Inventory Short-Form (CDI-S). Trajectories of symptoms of depression were estimated using linear mixed effects (LME) modeling.Results: At baseline, 477 children had complete data (mean age [SD] 11y 5mo [2y 1mo], range 7y 7mo-15y 1mo; 234 females, 243 males). Mean CDI-S T score at baseline was 45.7 (SD=7.5) and at 28 months was 44.9 (SD=8.2), both were within the 'average' range. Results from LME modeling revealed mean raw CDI-S score of 1.897, corrected for age 10 years (corresponding to T scores slightly below the normed mean of 50), with no significant change over three measurements (slope=-0.113, p=0.135), indicating that CDI-S scores were stable over 28 months. Children with high initial CDI-S scores had lower subsequent scores, as demonstrated by the correlation of -0.827 between intercept and slope (p<0.001). Parents reported comparable findings.Interpretation: Self- and proxy-reported symptoms of depression were generally low and stable over an extended follow-up period. Normalization of scores was seen upon repeated assessment, even in children with higher scores of symptoms of depression at one point. These findings speak to the value and importance of repeated assessment over time.What This Paper Adds: In children with epilepsy, self- and proxy-reported symptoms of depression were generally low and stable over 28 months. The trajectory of symptoms of depression was not associated with seizure severity, whether considering the frequency or type of seizures. Parents' reports of symptoms of depression were comparable to the children's self-evaluations. [ABSTRACT FROM AUTHOR]

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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.)

Aim: To determine whether, and how, neonatal intensive care unit (NICU) parents want to receive early neurodevelopmental screening information about their child's future risk of cerebral palsy and other disabilities.Method: This was a qualitative interview study. Parents of hospitalized infants born preterm completed semi-structured interviews. Data were analysed using a directed content analysis approach.Results: Thematic saturation was achieved after 19 interviews. Four themes characterized parent perceptions of early neurodevelopmental screening: (1) acceptability: most parents were in favour of neurodevelopmental screening if parents could refuse; (2) disclosure of results: parents want emotional preparation for results, especially false positives; (3) emotional burden of uncertainty: parents of children in the NICU balance taking their infant's illness 'day by day' and preparing for an uncertain future. Parents expressed distress with screening that increased uncertainty about the future; and (4) disability: prior experience with disability informs parent concerns.Interpretation: Parents interpret the risks and benefits of NICU developmental screening through the lens of prior experiences with disability. Most expressed interest in screening and emphasized a desire for autonomy, pretest counselling, and emotional preparation.What This Paper Adds: Most parents with infants in the neonatal intensive care unit expressed interest in early screening for developmental disability. Prior experience with disability informed concerns about specific deficits. Parents emphasized a desire for autonomy, pretest counselling, and emotional preparation. [ABSTRACT FROM AUTHOR]

Aim: To develop a detailed priority list of family-centred rehabilitation goals on the activity level within the International Classification of Functioning, Disability and Health (ICF) chapters d4 'Mobility' and d5 'Self-care' in a paediatric population with a broad range of health conditions.Method: Twenty-two months after implementing a systematic, family-centred, goal-setting process, the rehabilitation goals of 212 inpatients were retrospectively allocated to the most detailed level of ICF categories by two independent researchers. The overall frequencies of these goals were calculated and stratified by health condition, functional independence, and age.Results: Ninety-three females and 119 males were included in the study (mean age 10y 9mo, SD 4y 5mo, range 2y 1mo-21y 5mo). The five most frequent rehabilitation goals were ICF codes d4500 'Walking short distances' (11%), d4200 'Transferring oneself while sitting' (9%), d5400 'Putting on clothes' (7%), d451 'Going up and down stairs' (6%), and d4153 'Maintaining a sitting position' (5%). These top goals varied in the subgroups with regard to the underlying health condition, functional independence, and age.Interpretation: The findings of this study are not generalizable due to the large heterogeneity in priorities. However, they can be used to incorporate families' needs into future research designs and the development of new technologies.What This Paper Adds: Walking short distances is the most frequent mobility/self-care goal of paediatric rehabilitation. The top goals depend on health condition, functional independence, and age. Priorities vary considerably between children undergoing rehabilitation. Rehabilitation goals need to be assessed individually for each child. [ABSTRACT FROM AUTHOR]

Aim: To compare the rate of falls between adults with and without cerebral palsy (CP).Method: We used primary care data on 1705 adults with CP and 5115 adults without CP matched for age, sex, and general practice attended. We compared odds of experiencing a fall between adults with and without CP using conditional logistic regression. We compared the rate of falls using a negative binomial model.Results: Participants were 3628 males (53%) and 3192 females (47%) (median age 29y, interquartile range 20-42y) at the start of follow-up. Follow-up was 14 617 person-years for adults with CP and 56 816 person-years for adults without CP. Of adults with CP, 15.3% experienced at least one fall compared to 5.7% of adults without CP. Adults with CP had 3.64 times (95% confidence interval [CI] 2.98-4.45) the odds of experiencing a fall compared to adults without CP. The rate of falls was 30.5 per 1000 person-years and 6.7 per 1000 person-years for adults with and without CP respectively (rate ratio 5.83, 95% CI 4.84-7.02) INTERPRETATION: Adults with CP are more likely to fall, and fall more often, than adults without CP. The causes and consequences of falls in adults with CP need examination.What This Paper Adds: Twenty adults with CP and 5.3 adults without CP experienced at least one fall per 1000 person-years. Adults with CP experienced 30.5 falls per 1000 person-years compared to 6.7 falls per 1000 person-years among adults without CP. Adults with CP had 3.64 times the odds of experiencing a fall compared to adults without CP. Adults with CP experienced 5.83 times more falls than adults without CP. [ABSTRACT FROM AUTHOR]

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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.)

Aim: To identify maternal, obstetric, and neonatal risk factors related to perinatal arterial ischaemic stroke (PAIS) diagnosed within 28 days after birth and to understand the underlying pathophysiology.Method: For case and control ascertainment, we used active surveillance in 345 paediatric hospitals and a population-based perinatal database for quality assurance of hospital care. We analysed complete cases of PAIS using logistic regression. Multivariate analysis was guided by a directed acyclic graph.Results: After exclusion of records with missing data, we analysed 134 individuals with PAIS and 576 comparison individuals. In univariate analysis, male sex, preterm birth (<37wks gestational age), small for gestational age (SGA), low umbilical artery pH (<7.1), low 5-minute-Apgar score (<7), multiple pregnancies, hypoxia, intubation/mask ventilation, nulliparity, Caesarean section, vaginal-operative delivery, chorioamnionitis, and oligohydramnios were associated with an increased risk. Mutual adjustment yielded male sex (odds ratio [OR] 1.81; 95% confidence interval [CI] 1.20-2.73), multiple birth (OR 3.22; 95% CI 1.21-8.58), chorioamnionitis (OR 9.89; 95% CI 2.88-33.94), preterm birth (OR 1.86; 95% CI 1.01-3.43), and SGA (OR 3.05; 95% CI 1.76-5.28) as independent risk factors.Interpretation: We confirmed the increased risk in males and the role of chorioamnionitis and SGA for PAIS, pointing to the importance of inflammatory processes and fetal-placental insufficiency. Multiple birth and preterm birth were additional risk factors.What This Paper Adds: Chorioamnionitis and small for gestational age (SGA) precede perinatal arterial ischaemic stroke (PAIS). Chorioamnionitis and SGA are independent risk factors for PAIS. Inflammatory processes and fetal-placental insufficiency are the likely underlying mechanisms. Multiple birth and preterm birth are additional risk factors. [ABSTRACT FROM AUTHOR]

Aim: To compare language, speech, and voice of children born preterm and at term, and determine relevant predictors of outcome.Method: Three hundred infants (150 males, 150 females; 149 born at <30wks' gestation, 151 term-born) were prospectively recruited at birth from the Royal Women's Hospital. We administered the Preschool Language Scales, Fifth Edition, Diagnostic Evaluation of Articulation and Phonology, Grade Roughness Breathiness Asthenia Strain Scale, and Pediatric Voice Handicap Index at 3 years, and compared groups. We examined hypothesized predictors in children born preterm: gestational age at birth, birthweight, sex, chronic lung disease, high social risk, multilingualism, neurodevelopmental diagnosis, and oromotor feeding.Results: Children born preterm had poorer language than children born at term (coefficient -5.43). Speech and voice were similar between groups (coefficients -0.70 to 1.63). Chronic lung disease predicted voice (coefficient 6.05); male sex (coefficients 4.54-6.18), high social risk (coefficient -6.02 to -9.30), and neurodevelopmental diagnosis (coefficients -16.42 to -20.61) predicted language.Interpretation: Children born before 30 weeks' gestation had poorer language than children born at term. Children born preterm with neurodevelopmental disabilities or high social risk experience poorer language outcomes, and would benefit from enrichment of their language environment.What This Paper Adds: Speech and voice outcomes were similar between children born preterm and at term. Male sex, high social risk, and neurodevelopmental diagnosis predicted language outcomes. [ABSTRACT FROM AUTHOR]

Aim: To compare hospital admission patterns after the first year of life in Australian children with developmental disabilities and children with no known disability, according to maternal country of birth and Indigenous status.Method: This was a retrospective cohort study using linked data across health, disability, and hospital admission databases. The study investigated 656 174 children born in Western Australia between 1983 and 2008 with a total of 1 091 834 records of hospital admissions.Results: Children with no known disability born to Indigenous mothers had the highest rate of hospital admissions compared to children of non-Indigenous mothers. Children of foreign-born mothers from low-income countries had the highest rate of hospital admissions if disability was present. Children with cerebral palsy (CP) with or without associated intellectual disability had the highest rate of hospital admissions among children with developmental disability, especially if mothers were foreign-born.Interpretation: Children with CP and intellectual disability, particularly from minority backgrounds (Indigenous Australian and foreign-born mothers), were at higher risk of being admitted to hospital after the first year of life.What This Paper Adds: Hospital admissions in Australian children with and without disabilities differ according to maternal country of birth. Hospital admission rates in children without a developmental disability were greatest for Australian-born Indigenous children. Disabled Australian-born children of foreign-born mothers from low-income countries had the highest hospital admission rates. Hospital admission risk was greatest for Australian-born children with cerebral palsy, especially if mothers were foreign-born. [ABSTRACT FROM AUTHOR]

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Aim: To determine factors associated with acquisition of a sitting position in patients with spinal muscular atrophy type 1 (SMA1) treated with nusinersen.Method: Using data from the registry of patients with SMA1 treated with nusinersen, we compared the subgroups of sitters and non-sitters after 14 months of therapy as a function of baseline level, SMN2 copy number, age at treatment initiation, and improvement at 2 and 6 months post-treatment initiation. We used Hammersmith Infant Neurological Examination, Section 2 (HINE-2) and Children's Hospital of Philadelphia Infant Test of Neuromuscular Disorders for motor evaluation.Results: Fifty children (22 females, 28 males), mean age 22 months (SD 20.7; range 2.5-102.8mo) were treated. Data on sitting position acquisition were collected for 47 patients at month 14. Fifteen patients were able to sit unassisted; 11 of 15 had a baseline HINE-2 score of at least 2 points and 11 of 14 had an improvement over baseline of at least 2 points at month 6. Patients who improved by 2 or more points at month 6 were three times more likely to be sitters at month 14 than those who did not.Interpretation: High baseline motor function and improvement in HINE-2 score after 6 months of treatment are associated with the probability of acquiring a sitting position in patients with SMA1 treated with nusinersen.What This Paper Adds: Fifteen of 47 patients with spinal muscular atrophy could sit unaided 14 months after treatment with nusinersen. The number of SMN2 copies were not predictive of acquisition of a sitting position. Baseline condition and clinical response after 6 months of treatment were most predictive of sitting position acquisition. [ABSTRACT FROM AUTHOR]

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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.)

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.)

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.)

Aim: To investigate the interdependence between risk factors associated with long-term intellectual development in individuals with tuberous sclerosis complex (TSC).Method: The Tuberous Sclerosis 2000 Study is a prospective longitudinal study of individuals with TSC. In phase 1 of the study, baseline measures of intellectual ability, epilepsy, cortical tuber load, and mutation were obtained for 125 children (63 females, 62 males; median age=39mo). In phase 2, at an average of 8 years later, intellectual abilities were estimated for 88 participants with TSC and 35 unaffected siblings. Structural equation modelling was used to determine the risk pathways from genetic mutation through to IQ at phase 2.Results: Intellectual disability was present in 57% of individuals with TSC. Individuals without intellectual disability had significantly lower mean IQ compared to unaffected siblings, supporting specific genetic factors associated with intellectual impairment. Individuals with TSC who had a slower gain in IQ from infancy to middle childhood were younger at seizure onset and had increased infant seizure severity. Structural equation modelling indicated indirect pathways from genetic mutation, to tuber count, to seizure severity in infancy, through to IQ in middle childhood and adolescence.Interpretation: Early-onset and severe epilepsy in the first 2 years of life are associated with increased risk of long-term intellectual disability in individuals with TSC, emphasizing the importance of early and effective treatment or prevention of epilepsy.What This Paper Adds: Intellectual disability was present in 57% of individuals with tuberous sclerosis complex (TSC). Those with TSC without intellectual disability had significantly lower mean IQ compared to unaffected siblings. Earlier onset and greater severity of seizures in the first 2 years were observed in individuals with a slower gain in intellectual ability. Risk pathways through seizures in the first 2 years predict long-term cognitive outcomes in individuals with TSC. [ABSTRACT FROM AUTHOR]