Relationship between early infant motor repertoire and neurodevelopment on the hammersmith infant neurological examination in a developmentally vulnerable First Nations cohort.

AIM: To implement a culturally-adapted screening program aimed to determine the ability of infant motor repertoire to predict early neurodevelopment on the Hammersmith Infant Neurological Examination (HINE) and improve Australian First Nations families' engagement with neonatal screening. METHODS: A prospective cohort of 156 infants (55 % male, mean (standard deviation [SD]) gestational age 33.8 (4.6) weeks) with early life risk factors for adverse neurodevelopmental outcomes (ad-NDO) participated in a culturally-adapted screening program. Infant motor repertoire was assessed using Motor Optimality Score-revised (MOS-R), captured over two videos, 11-13+6 weeks (V1; <14 weeks) and 14-18 weeks (V2; ≥14 weeks) corrected age (CA). At 4-9 months CA neurodevelopment was assessed on the HINE and classified according to age-specific cut-off and optimality scores as; developmentally 'on track' or high chance of either adverse neurodevelopmental outcome (ad-NDO) or cerebral palsy (CP). RESULTS: Families were highly engaged, 139/148 (94 %) eligible infants completing MOS-R, 136/150 (91 %), HINE and 123 (83 %) both. Lower MOS-R at V2 was associated with reduced HINE scores (ß = 1.73, 95 % confidence interval [CI] = 1.03-2.42) and high chance of CP (OR = 2.63, 95%CI = 1.21-5.69) or ad-NDO (OR = 1.38, 95%CI = 1.10-1.74). The MOS-R sub-category 'observed movement patterns' best predicted HINE, infants who score '4' had mean HINE 19.4 points higher than score '1' (95%CI = 12.0-26.9). Receiver-operator curve analyses determined a MOS-R cut-off of <23 was best for identifying mild to severely reduced HINE scores, with diagnostic accuracy 0.69 (sensitivity 0.86, 95%CI 0.76-0.94 and specificity 0.40, 95 % CI 0.25-0.57). A trajectory of improvement on MOS-R (≥2 point increase in MOS-R from 1st to 2nd video) significantly increased odds of scoring optimally on HINE (OR = 5.91, 95%CI 1.16-29.89) and may be a key biomarker of 'on track' development. INTERPRETATION: Implementation of a culturally-adapted program using evidence-based assessments demonstrates high retention. Infant motor repertoire is associated with HINE scores and the early neurodevelopmental status of developmentally vulnerable First Nations infants.

Efficacy of Early Intervention for Infants With Cerebral Palsy in an LMIC: An RCT.

OBJECTIVE: To test efficacy of a parent-delivered multidomain early intervention (Learning through Everyday Activities with Parents [LEAP-CP]) for infants with cerebral palsy (CP) compared with equal-dose of health advice (HA), on (1) infant development; and (2) caregiver mental health. It was hypothesized that infants receiving LEAP-CP would have better motor function, and caregivers better mental health. METHODS: This was a multisite single-blind randomized control trial of infants aged 12 to 40 weeks corrected age (CA) at risk for CP (General Movements or Hammersmith Infant Neurologic Examination). Both LEAP-CP and HA groups received 15 fortnightly home-visits by a peer trainer. LEAP-CP is a multidomain active goal-directed intervention. HA is based on Key Family Practices, World Health Organization. Primary outcomes: (1) infants at 18 months CA: Pediatric Evaluation of Disability Inventory-Computer Adaptive Test (PEDI-CAT mobility); and (2) caregiver: Depression Anxiety and Stress Scale. RESULTS: Of eligible infants, 153 of 165 (92.7%) were recruited (86 males, mean age 7.1±2.7 months CA, Gross Motor Function Classification System at 18 m CA: I = 12, II = 25, III = 9, IV = 18, V = 32). Final data were available for 118 (77.1%). Primary (PEDI-CAT mobility mean difference = 0.8 (95% CI -1.9 to 3.6) P = .54) and secondary outcomes were similar between-groups. Modified-Intention-To-Treat analysis on n = 96 infants with confirmed CP showed Gross Motor Function Classification System I and IIs allocated to LEAP-CP had significantly better scores on PEDI-CAT mobility domain (mean difference 4.0 (95% CI = 1.4 to 6.5), P = .003) compared with HA. CONCLUSIONS: Although there was no overall effect of LEAP-CP compared with dose-matched HA, LEAP-CP lead to superior improvements in motor skills in ambulant children with CP, consistent with what is known about targeted goal-directed training.

The Pediatric Subjective Global Nutrition Assessment Classifies More Children With Cerebral Palsy as Malnourished Compared With Anthropometry.

BACKGROUND: Nutrition assessment is multidimensional; however, much of the literature examining the nutritional status of children with cerebral palsy (CP) focuses on a single dimension. OBJECTIVE: The aim of the study was to evaluate nutritional status in children and adolescents with CP by comparing results from the Pediatric Subjective Global Nutrition Assessment (SGNA) with results from traditional anthropometric measures. DESIGN: This study was a cross-sectional observational study. PARTICIPANTS/SETTING: This study was conducted in a tertiary hospital outpatient setting in Brisbane, Australia, from February 2017 to March 2018. A total of 89 children (63 boys) with CP aged between 2 and 18 years of age were included. All Gross Motor Function Classification System levels were observed. The majority of children were in Gross Motor Function Classification System I and II (57, 64%) compared with Gross Motor Function Classification System III to V (32, 36%). Children with feeding tubes and those acutely unwell or hospitalized were excluded. MAIN OUTCOME MEASURES: Children were classified as well nourished, moderately malnourished, or severely malnourished by dietitians using the SGNA. Weight, height, body mass index (BMI), triceps skinfold thickness, subscapular skinfold thickness, and mid upper arm circumference were measured and converted to z scores to account for age and sex differences. Moderate malnutrition was defined by z scores -2.00 to -2.99 and severe malnutrition as ≤-3.00 z scores. STATISTICAL ANALYSIS PERFORMED: Multinomial logistic analyses were used to compare results from the SGNA and each single measurement. Continuous outcomes were transformed into z scores. Agreement was assessed with 2 categories: not malnourished and malnourished. Comparison statistics included percent agreement, sensitivity, and specificity. RESULTS: More children were classified as moderately or severely malnourished by SGNA than any of the anthropometric z score cutoffs. The majority of children were well nourished (n = 63) with 20 (22%) moderately malnourished and 6 (7%) severely malnourished by SGNA. The SGNA classified 11 children as malnourished that were not classified as malnourished by BMI. Children with moderate or severe malnutrition by SGNA had lower weight (P < .001, P < .001), BMI (P < .001, P < .001), mid upper arm circumference (P < .001, P < .001), triceps skinfold thickness (P = .01, P = .007), and subscapular skinfold thickness (P = .005, P = .02) z scores than well-nourished children. CONCLUSION: The SGNA identified more potentially malnourished children including children classified as well nourished by the single measurements such as BMI, height, and weight. The SGNA provided a clinically useful multidimensional approach to nutrition assessment for children with CP.

Development and validation of a screening tool for feeding/swallowing difficulties and undernutrition in children with cerebral palsy.

AIM: To develop and validate a screening tool for feeding/swallowing difficulties and/or undernutrition in children with cerebral palsy (CP). METHOD: This cross-sectional, observational study included 89 children with CP (63 males, 26 females; median age 6y 0mo; interquartile range 4y 0mo-8y 11mo), across all Gross Motor Function Classification System levels. Children with feeding tubes were excluded. Children were classified as well-nourished or moderately to severely undernourished, using the paediatric Subjective Global Nutrition Assessment. Eating and drinking abilities were classified using the Eating and Drinking Ability Classification System (EDACS) from mealtime observation and videofluoroscopic swallow studies when indicated. Parents/caregivers answered 33 screening questions regarding their child's feeding/swallowing abilities and nutritional status. The diagnostic ability of each question for identifying children with feeding/swallowing difficulties and undernutrition was calculated and the combination of questions with the highest sensitivity and specificity identified. RESULTS: Feeding difficulties impacted on swallow safety in 26 children (29%) and 26 children (29%) were moderately or severely undernourished. The 4-item final tool had high sensitivity and specificity for identifying children with feeding/swallowing difficulties (81% and 79% respectively) and undernutrition (72% and 75% respectively). The tool successfully identified 100 per cent of children with severe undernutrition and 100 per cent of those classified as EDACS level IV or V. INTERPRETATION: Screening for feeding/swallowing difficulties and undernutrition will enable early identification, assessment, and management for those children in need. WHAT THIS PAPER ADDS: A screening tool with high sensitivities and specificities for identifying children with feeding/swallowing difficulties and undernutrition. The tool identified 100 per cent of children with severe undernutrition. The tool identified 100 per cent of children in Eating and Drinking Ability Classification System levels IV or V.

Community-based parent-delivered early detection and intervention programme for infants at high risk of cerebral palsy in a low-resource country (Learning through Everyday Activities with Parents (LEAP-CP): protocol for a randomised controlled trial.

INTRODUCTION: Cerebral palsy (CP) is the most common childhood physical disability, with 80% estimated to be in low-middle-income countries. This study aims to (1) determine the accuracy of General Movements (GMs)/Hammersmith Infant Neurological Examination (HINE) for detecting CP at 18 months corrected age (CA); (2) determine the effectiveness of a community-based parent-delivered early intervention for infants at high risk of CP in West Bengal, India (Learning through Everyday Activities with Parents for infants with CP; LEAP-CP). METHODS: This study comprises two substudies: (1) a study of the predictive validity of the GMs and HINE for detecting CP; (2) randomised, double-blinded controlled trial of a novel intervention delivered through peer trainers (Community Disability Workers, CDW) compared with health advice (15 fortnightly visits). 142 infants at high risk of CP ('absent fidgety' GMs; 'high risk score' on HINE) aged 12-40 weeks CA will be recruited to the intervention substudy, with infants randomised based on a computer-generated sequence. Researchers will be masked to group allocation, and caregivers and CDWs naïve to intervention status. Visits will include therapeutic modules (goal-directed active motor/cognitive strategies and LEAP-CP games) and parent education. Health advice is based on the Integrated Management of Childhood Illness, WHO. Infants will be evaluated at baseline, post intervention and 18 months CA. The primary hypothesis is that infants receiving LEAP-CP will have greater scaled scores on the Pediatric Evaluation of Disability Inventory-Computer Adaptive Test (mobility domain) at 18 months compared with health advice. Secondary outcomes include infant functional motor, cognitive, visual and communication development; infant growth; maternal mental health. ETHICS AND DISSEMINATION: This study is approved through appropriate Australian and Indian ethics committees (see in text) with families providing written informed consent. Findings from this trial will be disseminated through peer-reviewed journal publications and conference presentations. TRIAL REGISTRATION NUMBER: 12616000653460p; Pre-results.

Oropharyngeal Dysphagia and Cerebral Palsy.

OBJECTIVES: To determine the progression of oropharyngeal dysphagia (OPD) in preschool-aged children with cerebral palsy (CP) according to gross motor function. It was hypothesized that fewer children would have OPD at 60 months compared with 18 to 24 months (predominately Gross Motor Function Classification System [GMFCS] I-II). METHODS: Longitudinal population-based cohort of 179 children (confirmed CP diagnosis, born in Queensland in 2006-2009, aged 18-60 months at study entry [mean = 34.1 months ± 11.9; 111 boys; GMFCS I = 46.6%, II = 12.9%, III = 15.7%, IV = 10.1%, and V = 14.6%]). Children had a maximum of 3 assessments (median = 3, total n = 423 assessments). OPD was classified by using the Dysphagia Disorders Survey part 2 and rated from video by a certified pediatric speech pathologist. GMFCS was used to classify children's gross motor function. RESULTS: OPD prevalence reduced from 79.7% at 18 to 24 months to 43.5% at 60 months. There were decreasing odds of OPD with increasing age (odds ratio [OR] = 0.92 [95% confidence interval (CI) 0.90 to 0.95]; P < .001) and increasing odds with poorer gross motor function (OR = 6.2 [95% CI 3.6 to 10.6]; P < .001). This reduction was significant for children with ambulatory CP (GMFCS I-II, OR = 0.93 [95% CI 0.90 to 0.96]; P < .001) but not significant for children from GMFCS III to V (OR [III] = 1.0 [95% CI 0.9 to 1.1]; P = .897; OR [IV-V] = 1.0 [95% CI 1.0 to 1.1]; P = .366). CONCLUSIONS: Half of the OPD present in children with CP between 18 and 24 months resolved by 60 months, with improvement most common in GMFCS I to II. To more accurately detect and target intervention at children with persisting OPD at 60 months, we suggest using a more conservative cut point of 6 out of 22 on the Dysphagia Disorders Survey for assessments between 18 and 48 months.

Parent-reported indicators for detecting feeding and swallowing difficulties and undernutrition in preschool-aged children with cerebral palsy.

AIM: To determine the most accurate parent-reported indicators for detecting (1) feeding/swallowing difficulties and (2) undernutrition in preschool-aged children with cerebral palsy (CP). METHOD: This was a longitudinal, population-based study, involving 179 children with CP, aged 18 to 60 months (mean 34.1mo [SD 11.9] at entry, 111 males, 68 females [Gross Motor Function Classification System level I, 84; II, 23; III, 28; IV, 18; V, 26], 423 data points). Feeding/swallowing difficulties were determined by the Dysphagia Disorders Survey and 16 signs suggestive of pharyngeal phase impairment. Undernutrition was indicated by height-weight and skinfold composite z-scores less than -2. Primary parent-reported indicators included mealtime duration, mealtime stress, concern about growth, and respiratory problems. Other indicators were derived from a parent feeding questionnaire, including 'significant difficulty eating and drinking'. Data were analysed using multilevel mixed-effects regression and diagnostic statistics. RESULTS: Primary parent-reported indicators associated with feeding/swallowing were 'moderate-severe parent stress' (odds ratio [OR]=3.2 [95% confidence interval {CI} 1.3-7.8]; p<0.01), 'moderate-severe concern regarding growth' (OR=4.5 [95% CI 1.7-11.9]; p<0.01), and 'any respiratory condition' (OR=1.8 [95% CI 1.4-5.8]; p<0.01). The indicator associated with undernutrition was 'moderate-severe concern regarding growth' (height-weight OR=13.5 [95% CI 3.0-61.3]; p<0.01; skinfold OR=19.1 [95% CI 3.7-98.9]; p<0.01). 'Significant difficulty eating and drinking' was most sensitive/specific for feeding outcome (sensitivity=58.6%, specificity=100.0%), and 'parent concern regarding growth' for undernutrition (sensitivity=77.8%, specificity=77.0%). INTERPRETATION: Parent-reported indicators are feasible for detecting feeding and swallowing difficulties and undernutrition in children with CP, but need formal validation. WHAT THIS PAPER ADDS: Parent-reported indicators can detect feeding/swallowing difficulties and undernutrition in children with cerebral palsy. Most accurate screening questions were 0-10 scales for 'difficulty eating' and 'difficulty drinking'. Supplementation of these scales with additional indicators would improve detection.

The Eating and Drinking Ability Classification System in a population-based sample of preschool children with cerebral palsy.

AIM: To determine (1) the reproducibility of the Eating and Drinking Ability Classification System (EDACS); (2) EDACS classification distribution in a population-based cohort with cerebral palsy (CP); and (3) the relationships between the EDACS and clinical mealtime assessment, other classifications, and health outcomes. METHOD: This was a cross-sectional population-based cohort study of 170 children with CP at 3 years to 5 years (mean 57.6mo, standard deviation [SD] 8.3mo; 105 males, n=65 females). Functional abilities were representative of a population sample (Gross Motor Function Classification System level I=74, II=34, III=21, IV=18, V=23). The EDACS was the primary classification of mealtime function. The Dysphagia Disorders Survey was the clinical mealtime assessment. Gross motor function was classified using the Gross Motor Function Classification System. RESULTS: EDACS classification had 88.3% intrarater agreement (κ=0.84, intraclass correlation coefficient=0.95; p<0.001) and 51.7% interrater agreement (κ=0.36, intraclass correlation coefficient=0.79; p<0.001). In total, 56.5% of children were classified as EDACS level I. There was a strong stepwise relationship between the Dysphagia Disorders Survey and EDACS (r=0.96, p<0.001). Parental stress (odds ratio=1.3, p=0.05) and feeding tubes (odds ratio=6.4, p<0.001) were significantly related to more limited function on the EDACS. INTERPRETATION: The EDACS presents a viable adjunct to clinical assessment of feeding skills in children with CP for use in surveillance trials and clinical practice. A rating addendum would be a useful contribution to the tool to enhance reproducibility.

Body composition, diet, and physical activity: a longitudinal cohort study in preschoolers with cerebral palsy.

BACKGROUND: Altered body composition in children with cerebral palsy (CP) could be due to differences in energy intake, habitual physical activity (HPA), and sedentary time. OBJECTIVE: We investigated the longitudinal relation between the weight-for-age z score (WZ), fat-free mass (FFM), percentage of body fat (%BF), and modifiable lifestyle factors for all Gross Motor Function Classification System (GMFCS) levels (I-V). DESIGN: The study was a longitudinal population-based cohort study of children with CP who were aged 18-60 mo (364 assessments in 161 children; boys: 61%; mean ± SD recruitment age: 2.8 ± 0.9 y; GMFCS: I, 48%; II, 11%; III, 15%; IV, 11%; and V, 15%). A deuterium dilution technique or bioelectrical impedance analysis was used to estimate FFM, and the %BF was calculated. Energy intake, HPA, and sedentary time were measured with the use of a 3-d weighed food diary and accelerometer wear. Data were analyzed with the use of a mixed-model analysis. RESULTS: Children in GMFCS group I did not differ from age- and sex-specific reference children with typical development for weight. Children in GMFCS group IV were lighter-for-age, and children in GMFCS group V had a lower FFM-for-height than those in GMFCS group I. Children in GMFCS groups II-V had a higher %BF than that of children in GMFCS group I, with the exception of orally fed children in GMFCS group V. The mean %BF of children with CP classified them as overfat or obese. There was a positive association between energy intake and FFM and also between HPA level and FFM for children in GMFCS group I. CONCLUSIONS: Altered body composition was evident in preschool-age children with CP across functional capacities. Gross motor function, feeding method, energy intake, and HPA level in GMFCS I individuals are the strongest predictors of body composition in children with CP between the ages of 18 and 60 mo.

Oropharyngeal dysphagia in children with cerebral palsy: comparisons between a high- and low-resource country.

PURPOSE: There is paucity of research investigating oropharyngeal dysphagia (OPD) in young children with cerebral palsy (CP), and most studies explore OPD in high-resource countries. This study aimed at determining the proportion and severity of OPD in preschool children with CP in Bangladesh, compared to Australia. METHOD: Cross-sectional, comparison of two cohorts. Two hundred and eleven children with CP aged 18-36 months, 81 in Bangladesh (mean = 27.6 months, 61.7% males), and 130 in Australia (mean = 27.4 months, 62.3% males). The Dysphagia Disorders Survey (DDS) - Part 2 was the primary OPD outcome for proportion and severity of OPD. Gross motor skills were classified using the Gross Motor Function Classification System (GMFCS), motor type/distribution. RESULTS: (i) Bangladesh sample: proportion OPD = 68.1%; severity = 10.4 SD = 7.9. Australia sample: proportion OPD = 55.7%; severity = 7.0 SD = 7.5. (ii) There were no differences in the proportion or severity of OPD between samples when stratified for GMFCS (OR = 2.4, p = 0.051 and ß = 1.2, p = 0.08, respectively). CONCLUSIONS: Despite overall differences in patterns of OPD between Bangladesh and Australia, proportion and severity of OPD (when adjusted for the functional gross motor severity of the samples) were equivalent. This provides support for the robust association between functional motor severity and OPD proportion/severity in children with CP, regardless of the resource context. Implications for Rehabilitation The proportion and severity of OPD according to gross motor function level were equivalent between high- and low-resource countries (LCs). Literature from high-resource countries may be usefully interpreted by rehabilitation professionals for low-resource contexts using the GMFCS as a framework. The GMFCS is a useful classification in LCs to improve earlier detection of children at risk of OPD and streamline management pathways for optimal nutritional outcomes. Rehabilitation professionals working in LCs are likely to have a caseload weighted towards GMFCS III-V, with less compensatory OPD management options available (such as non-oral nutrition through tubes).

Longitudinal Growth, Diet, and Physical Activity in Young Children With Cerebral Palsy.

OBJECTIVES: To describe the longitudinal relationship between height-for-age z score (HZ), growth velocity z score, energy intake, habitual physical activity (HPA), and sedentary time across Gross Motor Function Classification System (GMFCS) levels I to V in preschoolers with cerebral palsy (CP). METHODS: Children with CP (n = 175 [109 (62.2%) boys]; mean recruitment age 2 years, 10 months [SD 11 months]; GMFCS I = 83 [47.2%], II = 21 [11.9%], III = 28 [15.9%], IV = 19 [10.8%], V = 25 [14.2%]) were assessed 440 times between the age of 18 months and 5 years. Height/length ratio was measured or estimated via knee height. Population-based standards were used to calculate HZ and growth velocity z-score by age and sex categories. Feeding method (oral or tube) and gestational age at birth (GA) were collected from parents. Three-day ActiGraph and food diary data were used to measure HPA/sedentary time ratio and energy intake, respectively. Oropharyngeal dysphagia was rated with the Dysphagia Disorder Survey (part 2, Pediatric). Analysis was undertaken with mixed-effects regression models. RESULTS: For GMFCS level I, height and growth velocity did not differ from population-level growth standards. Children in levels II to V were significantly shorter, and those in levels III to V grew significantly more slowly than those in level I. There was a significant positive association between HZ and GA at all GMFCS levels. Energy intake, HPA, sedentary time, Dysphagia Disorder Survey score, and feeding method were not significantly associated with either height or growth velocity once GMFCS level was accounted for. CONCLUSIONS: Functional status and GA should be considered when assessing the growth of a child with CP. Research into interventions aimed at increasing active movement in GMFCS levels III to V and their efficacy in improving growth and health outcomes is warranted.

Longitudinal Study of Oropharyngeal Dysphagia in Preschool Children With Cerebral Palsy.

OBJECTIVES: To determine changes in prevalence and severity of oropharyngeal dysphagia (OPD) in children with cerebral palsy (CP) and the relationship to health outcomes. DESIGN: Longitudinal cohort study. SETTING: Community and tertiary institutions. PARTICIPANTS: Children (N=53, 33 boys) with a confirmed diagnosis of CP assessed first at 18 to 24 months (Assessment 1: mean age ± SD, 22.9±2.9 mo corrected age; Gross Motor Function Classification System [GMFCS]: I, n=22; II, n=7; III, n=11; IV, n=5; V, n=8) and at 36 months (Assessment 2). INTERVENTIONS: Not applicable. MAIN OUTCOME MEASURES: OPD was classified using the Dysphagia Disorders Survey (DDS) and signs suggestive of pharyngeal dysphagia. Nutritional status was measured using Z scores for weight, height, and body mass index (BMI). Gross motor skills were classified on GMFCS and motor type/distribution. RESULTS: Prevalence of OPD decreased from 62% to 59% between the ages of 18 to 24 months and 36 months. Thirty percent of children had an improvement in severity of OPD (greater than smallest detectable change), and 4% had worse OPD. Gross motor function was strongly associated with OPD at both assessments, on the DDS (Assessment 1: odds ratio [OR]=20.3, P=.011; Assessment 2: OR=28.9, P=.002), pharyngeal signs (Assessment 1: OR=10.6, P=.007; Assessment 2: OR=15.8, P=.003), and OPD severity (Assessment 1: ß=6.1, P<.001; Assessment 2: ß=5.5, P<.001). OPD at 18 to 24 months was related to health outcomes at 36 months: low Z scores for weight (adjusted ß=1.2, P=.03) and BMI (adjusted ß=1.1, P=.048), and increased parent stress (adjusted OR=1.1, P=.049). CONCLUSIONS: Classification and severity of OPD remained relatively stable between 18 to 24 months and 36 months. Gross motor function was the best predictor of OPD. These findings contribute to developing more effective screening processes that consider critical developmental transitions that are anticipated to present challenges for children from each of the GMFCS levels.

Food and fluid texture consumption in a population-based cohort of preschool children with cerebral palsy: relationship to dietary intake.

AIM: To determine the texture constitution of children's diets and its relationship to oropharyngeal dysphagia (OPD), dietary intake, and gross motor function in young children with cerebral palsy (CP). METHOD: A cross-sectional, population-based cohort study comprising 99 young children with CP (65 males, 35 females) aged 18 to 36 months (mean age 27mo; Gross Motor Function Classification System [GMFCS] level I, n=45; II, n=13; III, n=14; IV, n=10; V, n=17). CP subtypes were classified as spastic unilateral (n=35), spastic bilateral (n=49), dyskinetic (n=5), and other (n=10), in accordance with the criteria of the Surveillance of Cerebral Palsy in Europe. Habitual dietary intake of food textures, energy, and water were determined from parent-completed 3-day weighed food records. Parent-reported feeding ability of food textures was reported on the Pediatric Evaluation of Disability Inventory and a feeding questionnaire. OPD was classified based on clinical feeding assessment using the Dysphagia Disorders Survey (rated by a certified assessor, KAB) and a subjective Swallowing Safety Recommendation (classified by a paediatric speech pathologist, KAB). RESULTS: Food/fluid textures were modified for 39% of children. Children with poorer gross motor function tended to receive a greater proportion of energy from fluids (GMFCS levels IV-V: ß=0.9, p=0.002) in their diets and fewer chewable foods (level III: ß=-0.7, p=0.03; levels IV-V: ß=-1.8, p<0.001) compared to level I to II participants. Fluids represented a texture for which children frequently had OPD and the texture most frequently identified as unsafe (or recommended for instrumental assessment). INTERPRETATION: These findings indicate that swallowing safety, feeding efficiency, and energy/water intake should be considered when providing feeding recommendations for children with CP.

Clinical signs suggestive of pharyngeal dysphagia in preschool children with cerebral palsy.

This study aimed to determine the discriminative validity, reproducibility, and prevalence of clinical signs suggestive of pharyngeal dysphagia according to gross motor function in children with cerebral palsy (CP). It was a cross-sectional population-based study of 130 children diagnosed with CP at 18-36 months (mean=27.4, 81 males) and 40 children with typical development (TD, mean=26.2, 18 males). Sixteen signs suggestive of pharyngeal phase impairment were directly observed in a videoed mealtime by a speech pathologist, and reported by parents on a questionnaire. Gross motor function was classified using the Gross Motor Function Classification System. The study found that 67.7% of children had clinical signs, and this increased with poorer gross motor function (OR=1.7, p<0.01). Parents reported clinical signs in 46.2% of children, with 60% agreement with direct clinical mealtime assessment (kappa=0.2, p<0.01). The most common signs on direct assessment were coughing (44.7%), multiple swallows (25.2%), gurgly voice (20.3%), wet breathing (18.7%) and gagging (11.4%). 37.5% of children with TD had clinical signs, mostly observed on fluids. Dysphagia cut-points were modified to exclude a single cough on fluids, with a modified prevalence estimate proposed as 50.8%. Clinical signs suggestive of pharyngeal dysphagia are common in children with CP, even those with ambulatory CP. Parent-report on 16 specific signs remains a feasible screening method. While coughing was consistently identified by clinicians, it may not reflect children's regular performance, and was not sufficiently discriminative in children aged 18-36 months.

Validity and reproducibility of measures of oropharyngeal dysphagia in preschool children with cerebral palsy.

AIM: The aim of the study was to determine the best measure to discriminate between those with oropharyngeal dysphagia (OPD) and those without OPD, among young children with cerebral palsy (CP). METHOD: We carried out a cross-sectional population-based study involving 130 children with CP aged between 18 months and 36 months (mean 27.4mo; 81 males, 49 females) classified according to the Gross Motor Function Classification Scale (GMFCS) as level I (n=57), II (n=15), III (n=23), IV (n=12), or V (n=23). Forty children with CP (mean 28.5mo; 21 males,19 females, eight for each GMFCS level) were included in the reproducibility sub-study, and 40 children with typical development (mean 26.2mo; 18 males, 22 females) were included in the validity sub-study. OPD was assessed using the Dysphagia Disorders Survey (DDS), Pre-Speech Assessment Scale (PSAS), and Schedule for Oral Motor Assessment (SOMA). We analysed reproducibility using inter- and intrarater agreement (percentage) and reliability (kappa values and intraclass correlation coefficients). Construct validity was assessed as concordance between measures (SOMA, DDS, and PSAS). In the absence of a criterion standard measure for OPD, prevalence was estimated using latent class variable analysis. Data from the children with typical development were used to propose modified OPD cut-points for discriminative validity. RESULTS: All measures had strong agreement (>85%) for inter- and intrarater reliability. The SOMA had the best specificity (100.0%), but lacked sensitivity (53.0%), whereas the DDS and PSAS had high sensitivity (each 100.0%) but lacked specificity (47.1% and 70.6% respectively). OPD prevalence when calculated using the web-based estimation was 65.4%, which was similar to the estimate from the modified cut-points. INTERPRETATION: Using the sample of children with typical development and modified cut-points, OPD prevalence was lower than estimates with standard scoring. We propose using these modified cut-points when administering the DDS, PSAS or SOMA in young children with CP.

Motor severity in children with cerebral palsy studied in a high-resource and low-resource country.

OBJECTIVES: To compare the patterns of motor type and gross motor functional severity in preschool-aged children with cerebral palsy (CP) in Bangladesh and Australia. METHODS: We used comparison of 2 prospective studies. A total of 300 children with CP were aged 18 to 36 months, 219 Australian children (mean age, 26.6 months; 141 males) recruited through tertiary and community services, and 81 clinic-attendees born in Bangladesh (mean age, 27.5 months; 50 males). All children had diagnosis confirmed by an Australian physician, and birth and developmental history collected on the Physician Checklist. All children were classified by the same raters between countries using the Gross Motor Function Classification System (GMFCS), and motor type and distribution. RESULTS: There were more children from GMFCS I-II in the Australian sample (GMFCS I, P < .01; III, P < .01; V, P = .03). The patterns of motor type also differed significantly with more spasticity and less dyskinetic types in the Australian sample (spasticity, P < .01; dystonia, P < .01; athetosis, P < .01). Birth risk factors were more common in the Bangladesh sample, with risk factors of low Apgar scores (Australia, P < .01), lethargy/seizures (Australia, P = .01), and term birth (Bangladesh, P = .03) associated with poorer gross motor function. Cognitive impairments were significantly more common in the Bangladesh children (P < .01), and visual impairments more common in Australia (P < .01). CONCLUSIONS: Patterns of functional severity, motor type, comorbidities, etiology, and environmental risk factors differed markedly between settings. Our results contribute to understanding the patterns of CP in low-resource settings, and may assist in optimizing service delivery and prioritizing appropriate early interventions for children with CP in these settings.

Oropharyngeal dysphagia in preschool children with cerebral palsy: oral phase impairments.

PURPOSE: This study aimed to document the prevalence and patterns of oral phase oropharyngeal dysphagia (OPD) in preschool children with cerebral palsy (CP), and its association with mealtime duration, frequency and efficiency. METHODS: Cross-sectional population-based cohort study of 130 children diagnosed with CP at 18-36 months ca (mean = 27.4 months, 81 males) and 40 children with typical development (mean = 26.2, 18 males). Functional abilities of children with CP were representative of a population sample (GMFCS I = 57, II = 15, III = 23, IV = 12, V = 23). Oral phase impairment was rated from video using the Dyspahgia Disorders Survey, Schedule for Oral Motor Impairment, and Pre-Speech Assessment Scale. Parent-report was collected on a feeding questionnaire. Mealtime frequency, duration and efficiency were calculated from a three day weighed food record completed by parents. Gross motor function was classified using the Gross Motor Function Classification System (GMFCS). RESULTS: Overall, 93.8% of children had directly assessed oral phase impairments during eating or drinking, or in controlling saliva (78.5% with modified cut-points). Directly assessed oral phase impairments were associated with declining gross motor function, with children from GMFCS I having a 2-fold increased likelihood of oral phase impairment compared to the children with TD (OR = 2.0, p = 0.18), and all children from GMFCS II-V having oral phase impairments. Difficulty biting (70%), cleaning behaviours (70%) and chewing (65%) were the most common impairments on solids, and difficulty sipping from a cup (60%) for fluids. OPD severity and GMFCS were not related to mealtime frequency, duration or efficiency, although children on partial tube feeds had significantly reduced mealtime efficiency. CONCLUSIONS: Oral phase impairments were common in preschool children with CP, with severity increasing stepwise with declining gross motor function. The prevalence and severity of oral phase impairments were significantly greater for most tasks when compared to children with typical development, even for those with mild CP. Children who were partially tube fed had significantly lower feeding efficiency, so this could be a useful early indicator of children needing supplementation to their nutrition (through increasing energy density of foods/fluids, or tube feeds).

Oropharyngeal dysphagia and gross motor skills in children with cerebral palsy.

OBJECTIVES: To determine the prevalence of oropharyngeal dysphagia (OPD) and its subtypes (oral phase, pharyngeal phase, saliva control), and their relationship to gross motor functional skills in preschool children with cerebral palsy (CP). It was hypothesized that OPD would be present across all gross motor severity levels, and children with more severe gross motor function would have increased prevalence and severity of OPD. METHODS: Children with a confirmed diagnosis of CP, 18 to 36 months corrected age, born in Queensland between 2006 and 2009, participated. Children with neurodegenerative conditions were excluded. This was a cross-sectional population-based study. Children were assessed by using 2 direct OPD measures (Schedule for Oral Motor Assessment; Dysphagia Disorders Survey), and observations of signs suggestive of pharyngeal phase impairment and impaired saliva control. Gross motor skills were described by using the Gross Motor Function Measure, Gross Motor Function Classification System (GMFCS), Manual Ability Classification System, and motor type/ distribution. RESULTS: OPD was prevalent in 85% of children with CP, and there was a stepwise relationship between OPD and GMFCS level. There was a significant increase in odds of having OPD, or a subtype, for children who were nonambulant (GMFCS V) compared with those who were ambulant (GMFCS I) (odds ratio = 17.9, P = .036). CONCLUSIONS: OPD was present across all levels of gross motor severity using direct assessments. This highlights the need for proactive screening of all young children with CP, even those with mild impairments, to improve growth and nutritional outcomes and respiratory health.

Longitudinal cohort protocol study of oropharyngeal dysphagia: relationships to gross motor attainment, growth and nutritional status in preschool children with cerebral palsy.

INTRODUCTION: The prevalence of oropharyngeal dysphagia (OPD) in children with cerebral palsy (CP) is estimated to be between 19% and 99%. OPD can impact on children's growth, nutrition and overall health. Despite the growing recognition of the extent and significance of health issues relating to OPD in children with CP, lack of knowledge of its profile in this subpopulation remains. This study aims to investigate the relationship between OPD, attainment of gross motor skills, growth and nutritional status in young children with CP at and between two crucial age points, 18-24 and 36 months, corrected age. METHODS AND ANALYSIS: This prospective longitudinal population-based study aims to recruit a total of 200 children with CP born in Queensland, Australia between 1 September 2006 and 31 December 2009 (60 per birth-year). Outcomes include clinically assessed OPD (Schedule for Oral Motor Assessment, Dysphagia Disorders Survey, Pre-Speech Assessment Scale, signs suggestive of pharyngeal phase impairment, Thomas-Stonell and Greenberg Saliva Severity Scale), parent-reported OPD on a feeding questionnaire, gross motor skills (Gross Motor Function Measure, Gross Motor Function Classification System and motor type), growth and nutritional status (linear growth and body composition) and dietary intake (3 day food record). The strength of relationship between outcome and exposure variables will be analysed using regression modelling with ORs and relative risk ratios. ETHICS AND DISSEMINATION: This protocol describes a study that provides the first large population-based study of OPD in a representative sample of preschool children with CP, using direct clinical assessment. Ethics has been obtained through the University of Queensland Medical Research Ethics Committee, the Children's Health Services District Ethics Committee, and at other regional and organisational ethics committees. Results are planned to be disseminated in six papers submitted to peer reviewed journals, and presentations at relevant international conferences.

Clinimetrics of measures of oropharyngeal dysphagia for preschool children with cerebral palsy and neurodevelopmental disabilities: a systematic review.

AIM: The aim of this study was to determine the psychometric properties and clinical utility of objective measures of oropharyngeal dysphagia (OPD) in children with cerebral palsy or neurodevelopmental disabilities aged 12 months to 5 years. METHOD: Five electronic databases were searched to identify measures of OPD. The Consensus-based Standards for the Selection of Measurement Instruments (COSMIN) Checklist was used to assess psychometric properties and a Modified CanChild Outcome Rating Form was used for clinical utility. RESULTS: Nine measures of OPD from 27 papers were assessed: the Brief Assessment of Motor Function - Oral Motor Deglutition Scale; the Behavioral Assessment Scale of Oral Functions in Feeding; the Dysphagia Disorders Survey; the Feeding Behaviour Scale; the Functional Feeding Assessment, modified; the Gisel Video Assessment; the Oral Motor Assessment Scale; the Pre-Speech Assessment Scale; and the Schedule for Oral Motor Assessment. INTERPRETATION: The Schedule for Oral Motor Assessment and the Functional Feeding Assessment, modified, proved to be the strongest measures based on published psychometric properties of validity and reliability. The Schedule for Oral Motor Assessment and the Dysphagia Disorders Survey were found to have the strongest clinical utility. Further studies to test the psychometric properties of existing measures, in particular predictive validity, responsiveness, and test-retest reliability, would be beneficial for selecting an appropriate measure for both clinical and research contexts.