Peabody Developmental Motor Scales Research Articles

Identifying Opportunities for Early Detection of Cerebral Palsy.

This study aimed to evaluate assessment and referral practices for the early detection and diagnosis of children at risk for or with cerebral palsy (CP) by health care and education providers in Maryland and Delaware. A secondary aim was to identify barriers for using early detection tools and identify opportunities for change to support early diagnosis and improve care. Seventy-two participants answered ≥ 50% of the survey questions. Most were occupational or physical therapists (86%) working in early intervention (61%). Eighty-eight percent indicated awareness that CP can be diagnosed by 12 months. Though 86% stated they typically suspect a diagnosis of CP between 0 and 12 months, only 19% reported that their patients received a CP diagnosis < 12 months. The Developmental Assessment of Young Children (73%) and the Peabody Developmental Motor Scales-2 (59%) were used most. Many respondents indicated never using magnetic resonance imaging (70%), the General Movements Assessment (87%), or the Hammersmith Infant Neurological Exam (69%). Participants identified clinical signs and symptoms prompting a referral for the diagnostic assessment of CP, most commonly stiffness in legs (95%), excessive head lag (93%), and persistent fisting (92%). Policy and organizational change, clinician education, and training are needed to support the implementation of CP early detection guidelines.

Characterization of Fine Motor and Visual Motor Skills in Aicardi-Goutières Syndrome.

Aicardi-Goutières syndrome is a genetic inflammatory disorder resulting in dispersed neurologic dysfunction. Despite a recognition of overall motor impairment, fine and visual motor skills are undercharacterized. We hypothesize that there is a spectrum of fine and visual motor skills in the Aicardi-Goutières syndrome population as captured by a standard outcome measure, the Peabody Developmental Motor Scales (PDMS-2), which will be proportional to overall disease severity.In a cohort of 74 subjects, the Peabody Developmental Motor Scales-2 grasping and visual-motor integration subtests were administered concurrently with the Aicardi-Goutières syndrome Severity Scale (severe [range 0-3], moderate [range 4-8], and attenuated [range 9-11]). The cohort was also compared by genotype and performance as defined by raw scores. The distribution of Peabody Developmental Motor Scales-2 scores within a genotype was assessed by interquartile ranges (IQRs).Peabody Developmental Motor Scales-2 grasping and visual-motor integration performance was the least variable in the TREX1-cohort (IQR: 10.00-12.00) versus the SAMHD1 and IFIH1 cohorts (IQR: 51.00-132.00 and 48.50-134.00, respectively). Neurologic severity highly correlated with both fine and visual motor skills (Spearman correlation: r = 0.87, 0.91, respectively). A floor effect (lowest 10% of possible scores) was observed within the severe cohort (n = 32/35), whereas a ceiling effect (top 10%) was observed in the attenuated cohort (n = 13/17).This study characterized the spectrum of fine and visual motor function in the Aicardi-Goutières syndrome population, which correlated with overall neurologic dysfunction. The Peabody Developmental Motor Scales-2 grasping and visual-motor integration showed promise as potential assessment tools in moderate and attenuated Aicardi-Goutières syndrome cohorts. A better understanding of fine and visual motor function in this population will benefit clinical care and clinical trial design.

Correlation between clinical measurement scales on gross motor function in children with spastic cerebral palsy

BackgroundChildren with cerebral palsy (CP) may have different gross motor impairments which in sequence affecting their life occupations. The purpose of the study is to investigate the correlation between Gross Motor Function Measurement-66 (GMFM-66), the test of Bruininks-Oseretsky Motor Proficiency (BOTMP), and the Peabody Developmental Motor Scale–Second Edition (PDMS-2) in young children with CP.MethodsA correlational study was applied on 50 children aged from 4 to 6 years (30 girls, 20 boys) with spastic CP.ResultsThe Pearson correlation coefficient between the GMFM scale and PDMS-2 motor quotients, and its subscales (stationary, locomotion, and object control) and also between GMFM and BOTMP gross motor quotients and its subscales (strength, agility, and body coordination) were statistically significant. Spearman’s coefficients between the grade of the Gross Motor Classification System (GMFCS) and the PDMS-2 Gross Motor Composite, BOTS-2, and its subscale results were also statistically significant.ConclusionThe three measurement scales, GMFM- 66, BOTS-2, and PDMS-2, are significantly related. Therefore, GMFCS is useful in predicting movement performance in children with CP and correlated with predictive guidance in treatment development.Trial registrationClinicalTrials.gov Identifier: NCT06124352.

Differences in Motor Development between Preterm Infants and Full-Term Preschool Children.

Although advances in obstetric and neonatal care have improved the survival of preterm infants, many studies document the increased risk of motor and sensory neurodevelopmental abnormalities that can hinder school progress. The aim of this study was to analyze the differences in gross and fine motor development in children born preterm and full term aged 3 to 6 years using the Peabody Developmental Motor Scales 2 (PDMS-II). Fifteen preterm and fifteen term children, matched for age and sex, participated in this study. They were evaluated with the PDMS-II scale. The scores obtained in the PDMS-II scale showed statistically significant differences (p < 0.05) in all subscales except for the "grasping" subscale. No dissimilarities were found between children who attended an early intervention program and those who did not participate, nor was there any correlation between week of gestation and birth weight and motor development in preschool. The results obtained show that differences are found with respect to motor development, with lower scores for those born preterm compared to children born at term. No statistically significant difference was found between preterm children who attended early intervention and those who did not. No correlation was found between motor development at preschool age and birth weight and gestational age.

Long-term outcomes of visual motor integration and motor development children with retinopathy of prematurity.

Premature infants have a risk of neurodevelopmental deficits. Little is known, however, about how retinopathy of prematurity (ROP) affects visual motor integration (VMI), which is necessary for both fine motor skills and further school abilities. Due to the systemic escape of bevacizumab in the treatment of ROP, concerns regarding the long-term neurodevelopmental effect of the drug have arisen. The aim is to evaluate VMI and motor development long-term outcomes after intravitreal bevacizumab (IVB) injection and laser treatment for ROP. Two groups of premature children were included: Bevacizumab group - 16 premature children who received IVB treatment and laser group - 23 premature children who underwent laser photocoagulation treatment in this single center cross-sectional study. At 2-6 years of age, VMI (Beery-Buktenica Developmental Test), motor development (Peabody Developmental Motor Scales-2), visual acuity, and refractive status were assessed. The incidence of abnormal visual function was significantly higher in bevacizumab group than in laser group (p = 0.022). The incidence of abnormal VMI skill was significantly higher in bevacizumab group than in laser group (p = 0.024). Incidences of abnormal gross, fine, and total motor skills were significantly higher in bevacizumab group compared to laser group (p < 0.05). Premature children who received bevacizumab for ROP demonstrated significantly lower VMI and motor development features than those with laser treatment at preschool age. Although our results suggest the relevance of bevacizumab injection in impaired VMI and motor development outcomes, general level of sickness rather than treatment might be the cause of delayed motor development.

Analysis of the acquisition of motor development and language in children with autism spectrum disorder: systematic review

The relationship between motor development and language skills has important implications for autism spectrum disorder (ASD). The aim of the present study was to analyze the acquisition of motor development and language in children with ASD. Guiding questions: Is the acquisition of language and motor development altered in children with ASD? What instruments are used to measure these aspects? Methods: A systematic review was conducted following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA). Searches were conducted in the PubMed (National Library of Medicine), Scopus and Web of Science databases using the following search terms: “Autism Spectrum Disorder” AND “Language” OR “Language Development” AND ¨Motor” OR “Motor Skills Disorders” in the period between April and June 2022. Results: A total of 1262 articles were retrieved from the electronic databases. After the discerning selection process, five articles were included in the present systematic review. Conclusion: The findings suggest that children with low motor development have a certain delay in the acquisition of language. The main instruments used for the assessment of these skills are the Mullen Scales of Early Learning, Peabody Developmental Motor Scales-2, Movement Assessment Battery for Children 2, Vineland Adaptive Behavior Scales II and British Picture Vocabulary Scale III.

Delineating clinical and developmental outcomes in STXBP1-related disorders.

STXBP1-related disorders are among the most common genetic epilepsies and neurodevelopmental disorders. However, the longitudinal epilepsy course and developmental end points, have not yet been described in detail, which is a critical prerequisite for clinical trial readiness. Here, we assessed 1281 cumulative patient-years of seizure and developmental histories in 162 individuals with STXBP1-related disorders and established a natural history framework. STXBP1-related disorders are characterized by a dynamic pattern of seizures in the first year of life and high variability in neurodevelopmental trajectories in early childhood. Epilepsy onset differed across seizure types, with 90% cumulative onset for infantile spasms by 6 months and focal-onset seizures by 27 months of life. Epilepsy histories diverged between variant subgroups in the first 2 years of life, when individuals with protein-truncating variants and deletions in STXBP1 (n = 39) were more likely to have infantile spasms between 5 and 6 months followed by seizure remission, while individuals with missense variants (n = 30) had an increased risk for focal seizures and ongoing seizures after the first year. Developmental outcomes were mapped using milestone acquisition data in addition to standardized assessments including the Gross Motor Function Measure-66 Item Set and the Grasping and Visual-Motor Integration subsets of the Peabody Developmental Motor Scales. Quantification of end points revealed high variability during the first 5 years of life, with emerging stratification between clinical subgroups. An earlier epilepsy onset was associated with lower developmental abilities, most prominently when assessing gross motor development and expressive communication. We found that individuals with neonatal seizures or early infantile seizures followed by seizure offset by 12 months of life had more predictable seizure trajectories in early to late childhood compared to individuals with more severe seizure presentations, including individuals with refractory epilepsy throughout the first year. Characterization of anti-seizure medication response revealed age-dependent response over time, with phenobarbital, levetiracetam, topiramate and adrenocorticotropic hormone effective in reducing seizures in the first year of life, while clobazam and the ketogenic diet were effective in long-term seizure management. Virtual clinical trials using seizure frequency as the primary outcome resulted in wide range of trial success probabilities across the age span, with the highest probability in early childhood between 1 year and 3.5 years. In summary, we delineated epilepsy and developmental trajectories in STXBP1-related disorders using standardized measures, providing a foundation to interpret future therapeutic strategies and inform rational trial design.

Concurrent and predictive validity of the Alberta Infant Motor Scale and the Peabody Developmental Motor Scales-2 administered to infants born preterm in Norway

BackgroundThe correlation between the Alberta Infant Motor Scale (AIMS) and the Peabody Developmental Motor Scales-2 (PDMS-2) has not previously been assessed in Norwegian infants. Our purpose was to investigate the concurrent validity of the AIMS and the PDMS-2 in a group of high-risk infants, and to investigate the predictive validity of the two tests for atypical motor function at 24 months post term age (PTA).MethodsThis is a retrospective study of the AIMS and the PDMS-2 administered to infants born preterm with gestational age ≤ 32 weeks (n = 139) who had participated in a randomized controlled trial of early parent-administered physiotherapy. The infants’ motor development had been assessed using the AIMS and the PDMS-2 at 6- and 12-months. The primary outcome was PDMS-2 at 24-months PTA. To explore the correlation between the two tests we used Spearman’s rho. Bland Altman plots were used to detect if there were systematic differences between the measurements. Receiver-operating characteristics curves were used to calculate area under the curve as an estimate of diagnostic accuracy of the AIMS and the PDMS- with respect to motor outcome at 24 months.ResultsThe correlation between the AIMS and the PDMS-2 (total motor and locomotion subscale), at 6 months, was r = 0.44 and r = 0.76, and at 12 months r = 0.56 and r = 0.80 respectively. The predictive validity for atypical motor function at 24 months, assessed using the area under the curve at 6- and at 12- months, was for the AIMS 0.87 and 0.86, respectively, and for the PDMS-2 locomotion subscale 0.82 and 0.76 respectively.ConclusionThe correlation between the AIMS and the PDMS-2 locomotion subscale, at 6- and 12- months PTA, was good to excellent in a group of infants born preterm in Norway. And the AIMS and the locomotion subscale of the PDMS-2 were equally good predictors for atypical motor outcomes at 24 months PTA. These findings indicate that the AIMS and the locomotion subscale of the PDM-2, could be used interchangeable when assessing motor development in infants at 6- or 12 months of age.Trial registrationClinicalTrials.gov NCT01089296.

Use of non-invasive transcutaneous auricular vagus nerve stimulation: neurodevelopmental and sensory follow-up.

To assess the impact of non-invasive transcutaneous auricular vagal nerve stimulation (taVNS) paired with oral feeding on long-term neurodevelopmental and sensory outcomes. We tested 21 of 35 children who as infants were gastrostomy tube (G-tube) candidates and participated in the novel, open-label trial of taVNS paired with oral feeding. To evaluate possible effects on development at 18-months after infant taVNS, we performed the Bayley-III (n = 10) and Sensory Profile (SP-2, n = 12) assessments before the COVID pandemic, and Cognitive Adaptive Test (CAT), Clinical Linguistics and Auditory Milestone (CLAMS), Ages and Stages Questionnaire (ASQ), and Peabody Developmental Motor Scales-2 gross motor tests as possible during and after the pandemic. We compared outcomes for infants who attained full oral feeds during taVNS ('responders') or received G-tubes ('non-responders'). At a mean of 19-months, taVNS 'responders' showed significantly better general sensory processing on the SP-2 than 'non-responders'. There were no differences in other test scores, which were similar to published outcomes for infants who required G-tubes. This is the first report of neurodevelopmental follow-up in infants who received taVNS-paired feeding. They had similar developmental outcomes as historical control infants failing oral feeds who received G-tubes. Our data suggests that infants who attained full oral feeds had better sensory processing.

3 Relationships between Motor Skills and Executive Functions in Preterm-Born Preschoolers

AbstractObjective:Motor skills have been linked to executive functions (EFs) in typically developing school-, and preschool-age children. Yet fine motor skills have been more consistently correlated with EFs than gross motor skills, perhaps because they are more frequently investigated. Preterm born children are vulnerable to deficits in both gross and fine motor skills, even after exclusion of neurological cases. In addition to motor skills, EFs may also be compromised in preterm born preschoolers. Because premature birth increases the odds for atypical brain development, and since adverse effects on brain functioning tend to yield increased dispersion of performance scores, we wished to determine whether fine and gross motor skills are differentially linked to performance on tasks measuring EF skills in nonhandicapped preschoolers born preterm.Participants and Methods:We studied 99 preterm (born &lt; 34 weeks) singleton preschoolers (3-4 years of age; 50 females), all graduates of the Neonatal Intensive Care Unit at William Beaumont Hospital, Royal Oak, MI. Motor skills were assessed with the Peabody Developmental Motor Scales - (Second Edition) which provide Fine and Gross Motor Quotients (FMQ, and GMQ, respectively). Three core EFs were measured: working memory, motor inhibition, and verbal fluency. Working memory skills were assessed with two Clinical Evaluation of Language Fundamentals - Preschool -Second Edition subtests: Recalling Sentences (RS) and Concepts and Following Directions (CFD). Motor inhibition and verbal fluency were assessed with the NEPSY-II Statue and Word Generation (WG) subtests, respectively. Children with a history of moderate to severe intracranial pathology or cerebral palsy were excluded.Results:We conducted linear regression analyses using scaled scores from the Statue, WG, RS, and CFD subtests as the predicted variables. Predictors of interest were the FMQ and GMQ. We adjusted for sociodemographic factors (SES and sex) and perinatal risk (gestational age, sum of antenatal complications and birth weight SD). The GMQ was significantly associated with all four EF measures (Statue, t(84) = 4.13, p &lt; .001; CFD, t(92) = 3.83, p &lt; .001; WG, t(84) = 3.38, p = .001; RS, t(90) = 3.37, p = .001). The FMQ was significantly associated with three of four EF measures (Statue, t(84) = 3.41, p = .001; CFD, t(92) = 3.97, p &lt; .001; WG, t(84) = 1.96, p = .054; RS, t(90) = 2.91, p = .005).Conclusions:Both fine and gross motor skills were associated with EF in nonhandicapped preterm-born singletons. Lower motor functioning in either motor domain was linked to reduction in performance on diverse EF measures. It should be emphasized that motor performance contributed to explaining variance in EFs even after statistical adjustment for early medical risk. In addition to the obvious conclusion that motor skills may underpin EF skills, it is likely that early risk factors not captured by the medical risk variables used in our analyses were nonetheless tapped by variability in motor performance. As preschool EFs are essential for subsequent academic performance, the significance of age-appropriate motor development in the preschool age should not be underestimated in our at-risk population.

4 Severity of Retinopathy of Prematurity and Motor Skills Development in Preschoolers

AbstractObjective:Children born preterm are at increased risk of deviation from the typical developmental trajectory. The probability of adverse developmental sequelae is increased in those with history of major perinatal complications. Retinopathy of prematurity (ROP) is a pathological disordered growth of retinal blood vessels occurring in very preterm neonates who require supplemental oxygen. ROP has been linked to intermittent hypoxemic events and blood gas derangements occurring in the Neonatal Intensive Care Unit (NICU), particularly in ventilated infants. Though presence and severity of ROP have been linked to neurodevelopmental impairment, little is known about the relationship between ROP severity and motor development in the preschool age. Because the same hypoxemic events and blood gas changes that lead to severe ROP may also lead to greater developmental deficits in motor control, we hypothesized that ROP severity will be inversely linked to the quality of motor functioning even in NICU graduates without neurological impairments.Participants and Methods:We included 95 preterm (23.6 - 33.6 weeks gestation) preschoolers (49 females, 44 members of twin pairs or triplets). The participants' age ranged from 3.3 - 4.1 years (adjusted for prematurity). ROP screening was conducted during NICU stay and rated from immature retina (0) to grade 4. Motor abilities were assessed with the Peabody Developmental Motor Scales (PDMS-2). Cases with diagnosed perinatal brain pathology (moderate to severe) or cerebral palsy were excluded from analyses.Results:We used linear mixed regression analyses with multiple gestation as a random factor. Severity of ROP was our predictor of interest, whereas socioeconomic status, sex, gestational age, and birth-weight SD served as covariates. Separate analyses were conducted using the PDMS-2 Total Motor, Fine Motor, and Gross Motor Quotients as dependent variables. ROP severity explained a unique portion of the variance in the Total Motor Quotient (F[1, 89] = 5.59, p = .02). Examination of the relationship between ROP severity and motor skill domains yielded a significant association for the Fine Motor Quotient (F[1, 89] = 6.19, p = .015) and a trend for the Gross Motor Quotient (F[1, 89] = 3.64, p = .06).Conclusions:The results of this study reveal that increase in ROP severity is linked to poorer motor skills in preterm-born preschoolers without major disabilities or perinatal diagnosis of moderate to severe brain pathology. This association was evident for both fine and gross motor skills, though only the relationship between ROP severity and the former motor index reached conventional statistical significance. Importantly, ROP severity accounted for a unique portion of the variance in motor performance, over and above the variance explained by other perinatal risk factors. This result is consistent with previous research findings indicating that ROP is linked to the occurrence of multiple, subtle hypoxemic events and 'exposure' to blood gas derangements during NICU stay in very preterm neonates who require respiratory support.

Investigating the validity of the Ages and Stages Questionnaire to detect gross motor delays in a community sample of toddlers: A cross-sectional study

ObjectiveGross motor (GM) development delays often appear in the toddler years but may not be diagnosed until school age. Early identification and intervention can improve motor outcomes. The Peabody Developmental Motor Scales - 2nd edition (PDMS-2) can assess GM skills but is resource-intensive. A parent-reported questionnaire, the Ages & Stages Questionnaire - 3rd edition (ASQ-3), may be a simple and effective way to flag potential GM delays requiring follow-up. This study aims to investigate the concurrent validity of the ASQ-3 GM subscale and the PDMS-2 GM subscale in toddlers and determine if the ASQ-3 GM subscale can accurately identify toddlers with motor delays. Study design103 healthy toddlers (12–35 months) completed the PDMS-2 GM assessment and parents completed the ASQ-3. The relationship between ASQ-3 GM and PDMS-2 GM was assessed with Kendall's Tau-b correlation, and contingency tables assessed the sensitivity, specificity, and positive and negative predicted values. ResultsThe Kendall's Tau-b correlation showed a moderate to strong correlation between the ASQ-3 GM raw scores and PDMS-2 GM quotient scores (tau-b = 0.51, p < 0.0001). The monitor zone cut-off on the ASQ-3 had a sensitivity of 67 % and specificity of 85 % for identifying children scoring >1SD on the PDMS-2. ConclusionsThe moderate-to-strong correlation between the ASQ-3-GM and PDMS-2-GM, adequate specificity and near-adequate sensitivity of the ASQ-3 combined with low resource requirements suggests that the ASQ-3 can be employed at a population level to identify toddlers with a delay and who may require referrals for further GM testing and intervention.

The relationship in early childhood body composition and physical activity levels regarding fundamental motor skill development

BackgroundFundamental motor skills (FMS) are considered essential for sport participation and might be deficit in obese children. While evidence indicates that physical activity (PA) levels impact motor skill development, the relationship between body composition, PA, and motor competence, particularly in early childhood, is not thoroughly understood. We aimed to determine if PA, fat mass (FM), and fat-free mass (FFM) are predictors of FMS.MethodsPreschoolers (n = 47) from two preschools were assessed for FMS, PA, and body composition. Peabody Developmental Motor Scales (PDMS-2) subscale scores were used to assess FMS. PA was assessed with a wrist-worn accelerometer for five days during school. FM and FFM were measured with foot-to-foot bioelectrical impedance.ResultsLinear regressions indicate significant models for stationary skills (SS) (F = 4.57, p = .004) and object manipulation skills (OMS) (F = 4.66, p = .003). FFM was the only significant predictor of SS (t = 3.98, p < .001) and OMS (t = 3.50, p = .001). FM and all intensities of PA were nonsignificant predictors in all models.ConclusionsThese results indicate that interventions that target improving or maintaining FFM may improve FMS.

Impact of home environment on motor development of infants in South India: A cross-sectional study&amp;nbsp;

Introduction: Early childhood development is a fundamentally important phase and is influenced by stimulus and nurturing. Environment influences, particularly the home environment, is considered the critical factor for optimal growth and development. Objectives: To determine the impact of home environment on motor development in South Indian infants. Method: 164 infants aged 1-11 months residing in Belagavi City, India, were recruited in a community-based cross-sectional study based on the inclusion and exclusion criteria. Structured questionnaire was used to assess the home environment followed by motor development assessment using Peabody developmental Motor Scale-2 (PDMS-2). Results: Gestational age was positively associated, while duration of breast feeding, hospitalization and weight of the infant (-1.17, p= 0.03) had negative association with the fine and total motor quotient of PDMS-2. Father’s education, occupation and income had significant positive effect on total and fine motor quotients. Outside space to play had positive association with fine motor quotient (2.28, p= 0.0154). Non availability of play material like pop up toys and child never exposed to play with floating toys, cups and socks also showed lower total quotient scores. Conclusions: Gestational age, father’s education, occupation and income had a positive effect on the motor development of South Indian infants participating in the study.

Evaluation of a computer game-assisted rehabilitation program for manual dexterity of children with cerebral palsy: Feasibility randomized control trial.

There is a need for innovation to improve the engagement and compliance of rehabilitation programs for children with upper extremity (UE) motor impairments due to cerebral palsy (CP); a computer games-based rehabilitation platform (GRP) was developed to address this need. The GRP provides engaging task-specific exercises targeting manual dexterity (object handling and manipulation). To evaluate the therapeutic value and treatment effect size of an exercise program using the GRP in children with CP. A total of 63 children with CP, aged 4 to 10 years, were recruited. The Peabody Developmental Motor Scale-2 (PDMS-2) Grasp and Visual-Motor Integration (VMI) subscores and Computer game-based Upper Extremity (CUE) assessment of manual dexterity were used to assess participants before and after a 16-week intervention program, delivered three times per week. The experimental group (XG) received a computer games-based exercise program targeting object manipulation tasks. The active control arm (CG) consisted of task-specific training similar to the tasks used in constrained induced movement therapy. There were only a few dropouts during the 16-week program, and compliance was high. Both groups showed significant improvements with medium to large effect sizes. Improvements in the PDMS-2 Grasp and VMI subscores observed in the XG were significantly greater than that in the CG. There were significant improvements (p < .01) in PDMS-2 grasp and VMI subscores for XG with moderate to large effect sizes (0.5-0.8). For CG, the Grasp and VMI subscores did improve but these changes were not statistically significant. There was a significant improvement observed in the majority of CUE object manipulation test scores for XG (p < .01) with moderate to large effect sizes (0.50-1.2) Although CG did show improvements in all CUE object manipulation test scores, the changes did not reach statistical significance (p < .01). This study demonstrates the utility of the GRP to practice a broad range of object manipulation tasks in children with CP. The present findings are positive and support further research and development. The long-term effects of the GRP program in children with CP will need to be confirmed in a future randomized controlled trial. In addition to measures of structure and function, future trials should also include outcome measures such as health-related quality of life and level of participation to validate the findings.

Eladocagene Exuparvovec Gene Therapy Improves Motor Development in Patients With Aromatic L-Amino Acid Decarboxylase Deficiency (S34.004)

<h3>Objective:</h3> Investigate the efficacy and safety of eladocagene exuparvovec in patients with aromatic l-amino acid decarboxylase deficiency (AADCd). <h3>Background:</h3> AADCd results from mutations in the dopa decarboxylase gene leading to reduced AADC enzyme activity; it is characterized by motor impairments and inability to attain developmental milestones. Eladocagene exuparvovec is a recombinant adeno-associated viral vector serotype 2 carrying the coding sequence for human AADC. <h3>Design/Methods:</h3> Eladocagene exuparvovec was infused bilaterally in the putamina of 30 children with AADC deficiency in 3 studies (AADC-CU/1601 [n=8], AADC-010 [n=10], and AADC-011 [n=12] in patients aged 18–102 months. Data were extracted on January 4, 2022. Patients receiving a total of 1.8 × 10¹¹ vg (n=21) or 2.4 × 10¹¹ vg (n=9; AADC-011) were followed for up to 120 months and assessed for motor milestone attainment using the Peabody Developmental Motor Scale, 2nd edition (PDMS-2). Specific motor skill items of the PDMS-2 were used to assess motor milestones including head control (partial or full), sitting (supported or independently), standing (with/away from support; up from cross-legged position), and walking (with/without assistance; 10 feet; taped line) Motor milestones and development were measured every 3 months for 1 year following gene therapy, then every 6–12 months for ≤120 months. <h3>Results:</h3> At baseline, no patients mastered head control or more advanced milestones. At year 1 of follow-up, patients were gaining the following skills (n): partial head control (26); full head control (15), sitting unassisted (7), supported standing (2). Progression of development was noted at years 5 and 10. By year 5, more advanced milestones were achieved (n): full head control (24), sitting unassisted (21) assisted walking (5), walking 10 feet (3), or walking up stairs (3). These abilities were maintained for as long as 10 years <h3>Conclusions:</h3> These data indicate eladocagene exuparvovec can provide a durable, positive impact on motor development in patients with AADCd. <b>Disclosure:</b> Mrs. Russell has received personal compensation for serving as an employee of PTC Therapeutics. Mrs. Russell has stock in PTC Therapeutics. Prof. ROUBERTIE has received personal compensation in the range of $500-$4,999 for serving as a Consultant for PTC. Prof. ROUBERTIE has received personal compensation in the range of $0-$499 for serving on a Scientific Advisory or Data Safety Monitoring board for PTC. Yin-Hsiu Chien has received personal compensation in the range of $500-$4,999 for serving as a Consultant for Sanofi Genzyme. Yin-Hsiu Chien has received personal compensation in the range of $500-$4,999 for serving as a Consultant for Biogen. Yin-Hsiu Chien has received personal compensation in the range of $500-$4,999 for serving as a Consultant for Novartis. Yin-Hsiu Chien has received personal compensation in the range of $500-$4,999 for serving as a Consultant for Amicus. Yin-Hsiu Chien has received personal compensation in the range of $500-$4,999 for serving on a Scientific Advisory or Data Safety Monitoring board for Sanofi. Yin-Hsiu Chien has received personal compensation in the range of $500-$4,999 for serving on a Speakers Bureau for Sanofi Genzyme. Yin-Hsiu Chien has received personal compensation in the range of $500-$4,999 for serving on a Speakers Bureau for Avexis/Norvatis. Yin-Hsiu Chien has received personal compensation in the range of $500-$4,999 for serving on a Speakers Bureau for Biogen. The institution of Yin-Hsiu Chien has received research support from Sanofi Genzyme. Ni-Chung Lee has nothing to disclose. Dr. Sierra has received personal compensation for serving as an employee of PTC Therapeutics Inc. Dr. Sierra has stock in PTC Therapeutics Inc. Dr. Turna has received personal compensation for serving as an employee of PTC Therapeutics. Dr. Turna has stock in PTC Therapeutics. Dr. Wang has received personal compensation for serving as an employee of PTC Therapeutics. Dr. Wang has received stock or an ownership interest from PTC Therapeutics, Inc.. The institution of Chun-Hwei Tai has received research support from National Taiwan University Hospital. Paul Wuh-Liang Hwu has received personal compensation in the range of $500-$4,999 for serving as a Consultant for PTC Therapeutics Inc.. Paul Wuh-Liang Hwu has received personal compensation in the range of $500-$4,999 for serving on a Scientific Advisory or Data Safety Monitoring board for PTC therapeutics. The institution of Paul Wuh-Liang Hwu has received research support from PTC therapeutics.

Influence of Modified Instructions on Locomotion Subtest Scores Using the Peabody Developmental Motor Scales, Second Edition.

The current study assessed whether changes in verbal and visual cues used to clarify requirements of the task would result in significant score differences on the Locomotion subtest of the Peabody Developmental Motor Scales, Second Edition (PDMS-2), in children aged 2.5 to 5 years. The Locomotion subtest of the PDMS-2 was administered twice, 2 to 10 days apart, to 37 children. Age-matched and gender-matched groups received instructions in both standardized and modified formats, where order depended on group assignment. Instruction type resulted in a significant change in Locomotion scores with a medium effect size and no significant interactions between instruction type and age or between instruction type and test order. Findings suggest instruction modifications using altered verbal and visual cues change PDMS-2 Locomotion subtest scores in children with typical development. These results support previous literature indicating that normative scores should not be reported if modifications were used during testing.

Hand grip strengthening exercises on fine motor skills in children with autism spectrum disorder

Background. Movement impairments in autism spectrum disorder (ASD) are manifested as both delays and deficits, with delays found in gross motor, fine motor and speech domains and deficits found in coordination, postural control and gait. Functional fine motor task is dependent on hand grip strength (HGS) which is found to be decreased. Therapeutic strategies focus primarily on sensory integration issues but HGS is not comprehensively addressed in improving fine motor skills (FMS). Objective. The aim of the study was to analyse the effect of hand grip strengthening exercises on improving FMS in children with ASD. Materials and Methods. This interventional study was carried out in 27 children with ASD who were recruited from Vidya sudha – Centre for children with special needs. After including in the study, level of FMS was assessed by Peabody Developmental Motor Scale (PDMS-2) and HGS was quantified with Martin Vigorimeter. Control group received conventional therapy and Intervention group received hand grip strengthening exercises along with routine therapy for duration of 6 weeks and HGS and level of FMS were reassessed with same outcome measures. Results. Paired t test showed statistically significant change of HGS in Intervention group and FMS of both the group with p &lt; 0.05. Unpaired t test showed statistically significant change of HGS and FMS with p &lt; 0.05 in Interventional group compared to control group. Conclusion. Hand grip strengthening exercises improves the HGS and has positive effect on FMS which can be included in the standard plan of care in rehabilitating children with ASD.

Psychometric Properties and Validation of the Italian Version of Ages & Stages Questionnaires Third Edition.

Objectives: The Ages & Stages Questionnaires Third Version (ASQ-3) identifies the risk of developmental delay in children aged 1 to 66 months. The aim of this study was to determine a reliable and valid instrument for the Italian population to enable the screening of children's development. Methods: Data from 2278 Italian children (age range: 1-66 months) were used to evaluate item discrimination power using the corrected item-total correlation. Internal consistency was analyzed by Cronbach's alpha scores and a Confirmative Factor Analysis was conducted to test the factor structure of the test. Data were also collected to examine the ASQ-3 test-retest reliability and concurrent validity, which was investigated using the Griffiths Scales of Child Development, Third Edition, the Peabody Developmental Motor Scale, Second Edition, and the Developmental Profile, Third Edition tools. In order to evaluate discriminant validity, differences between typical development children and several clinical groups have been performed. Finally, two different cut-off scores have been proposed. Results: The results showed that the questionnaires are composed of high-quality items; the original factor structure has been confirmed and strong Pearson product-moment correlation coefficients between the overall and the total for each domain (ranging from 0.73 to 0.88). The Italian version of the ASQ-3 had adequate internal consistency and a strong agreement between observations with two weeks' intervals. Moreover, the test showed a high discriminant validity due to the possibility of fully discriminating between typical development children and several clinical groups. Finally, two different cut-off scores have been identified using ROC curves in order to have a screening and a diagnostic cut-off value. Conclusion: This study evaluated the psychometric properties of the Italian adaptation of ASQ-3 questionnaires. We demonstrated the validity of the ASQ-3 and determined new cut-off scores for Italian children. Early identification and accurate assessment are important starting points to better understand and anticipate the needs of children and their link to services.

Feasibility of Family-Centered Workshops for Children Aged 18-36 Months with Language Developmental Delay

BackgroundFamily-centered workshops' effects on children with language developmental delay remain unknown. This study assessed the feasibility of workshops for children with language developmental delay. MethodsA total of 122 children aged 18-36 months with language developmental delays and their parents participated in six sessions of 2-h family-centered multidisciplinary workshops for 6 weeks. The Mandarin-Chinese Communicative Development Inventory, Peabody Developmental Motor Scale, Emotional Competency Rating Scales, Pediatric Outcomes Data Collection Instrument, Child Health Questionnaire, Pediatric Quality of Life Inventory, Caregiver Strain Index, Impact on Family Scale, PedsQL Family Impact Module, and World Health Organization Quality of Life (QOL) were administered to the children and their parents before and after the workshop. ResultsWe found improvement of emotion (P = 0.037), upper extremity and physical function (P = 0.038), and transfer and basic mobility (P = 0.019) in children and parental QOL related to children's conditions (P = 0.049), with no effect on communication ability and QOL in children and family strain and function. We also noted more significant improvement in children with pure developmental language delay than in children with nonpure developmental language delay concerning the success rates (from delayed to normal development) for expressive vocabulary (P < 0.001) and word combination (P = 0.002). Satisfaction levels toward the workshop were high. ConclusionsFamily-centered workshops improved children's emotions, functional performance, and parental QOL. Although the samples were too small to test different conditions of the developmental delay, the workshops for children with language developmental delays are acceptable and feasible.