Reducing disparities in behavioral health treatment in pediatric primary care: a randomized controlled trial comparing Partnering to Achieve School Success (PASS) to usual ADHD care for children ages 5 to 11 - study protocol.

BACKGROUND: Integrating behavioral health services into pediatric primary care can improve access to care, especially for children marginalized by poverty and racial/ethnic minority status. In primary care, a common presenting concern is attention-deficit/hyperactivity disorder (ADHD). Services in primary care for marginalized children with ADHD typically include medication alone; therapy to improve skills and build relationships is less available. This study evaluates the effectiveness of a behavioral intervention offered through primary care for marginalized families coping with ADHD (Partnering to Achieve School Success, PASS) compared to treatment as usual (TAU). METHOD: Three hundred participants will be randomly assigned to PASS or TAU. Participants include children ages 5 to 11 who have ADHD and are from economically marginalized families. PASS is a personalized, enhanced behavioral intervention that includes evidence-based behavior therapy strategies and enhancements to promote family engagement, increase caregiver distress tolerance, and provide team-based care to improve academic and behavioral functioning. TAU includes services offered by primary care providers and referral for integrated behavioral health or community mental health services. Outcomes will be assessed at mid-treatment (8 weeks after baseline), post-treatment (16 weeks), and follow-up (32 weeks) using parent- and teacher-report measures of service use, child academic, behavioral, and social functioning, parenting practices, family empowerment, and team-based care. Mixed effects models will examine between-group differences at post-treatment and follow-up. Analyses will examine the mediating role of parenting practices, family empowerment, and team-based care. Subgroup analyses will examine differential effects of intervention by child clinical characteristics and socioeconomic factors. DISCUSSION: This study is unique in targeting a population of children with ADHD marginalized by low socioeconomic resources and examining an intervention designed to address the challenges of families coping with chronic stress related to poverty. TRIAL REGISTRATION: This study was registered on clinicaltrials.gov (NCT04082234) on September 5, 2019, prior to enrollment of the first participant. The current version of the protocol and IRB approval date is October 4, 2023. Results will be submitted to ClinicalTrials.gov no later than 30 days prior to the due date for the submission of the draft of the final research report to the Patient-Centered Outcomes Research Institute.

Nonverbal Cognitive Skills in Children With Aicardi Goutières Syndrome.

BACKGROUND AND OBJECTIVES: Aicardi Goutières syndrome (AGS) is type I interferonopathy characterized by severe neurologic impairment. Although many children with AGS demonstrate motor and expressive language deficits, the magnitude of receptive language impairment is uncharacterized. We sought to characterize cognitive function in AGS-affected children using assessment tools with reduced dependence on motor abilities and compare cognitive testing outcomes with overall severity and parental assessment of adaptive behavior. METHODS: We performed a cross-sectional study. Children were recruited as part of the Myelin Disorders Biorepository Project at the Children's Hospital of Philadelphia. We included individuals with a confirmed diagnosis of AGS. We administered the Leiter International Performance Scale, third edition (Leiter-3), and the Vineland Adaptive Behavior Scale, third edition (VABS-3), in the context of research encounters. Motor skills were categorized by AGS Severity Scale mobility levels. Descriptive statistics and Spearman's rank correlation were used to compare assessments. Mann-Whitney and Kruskal-Wallis tests with correction with Dunn's multiple comparison test were used to compare test performance between mobility groups. RESULTS: Cognitive and adaptive behavior performance was captured in 57 children. The mean age at encounters was 8.51 (SD 5.15) years. The median (IQR) Leiter-3 score was 51 (interquartile range [IQR] 60), with administration failure in 20 of 57 (35%) individuals. On the VABS-3, the Motor Domain (median 29, IQR 36.25) was more impacted than the Communication (median 50, IQR 52), Daily Living Skills (median 52, IQR 31), and Socialization (median 54, IQR 40) Domains (p < 0.0001). The AGS Scale correlated with VABS-3 (r = 0.86, p < 0.0001) and Leiter-3 (r = 0.87, p < 0.0001). There was correlation between VABS-3 Domains and Leiter-3 (r-range 0.83-0.97). Gross motor and fine motor categories, respectively, correlated with VABS-3 (H = 39.37, p < 0.0001; U = 63, p < 0.0001) and Leiter-3 (H = 40.43, p < 0.0001; U = 66, p < 0.0001). Within each gross motor and fine motor category of the AGS Scale, a subset of children scored within normal IQ range. DISCUSSION: Parental assessment of function by the VABS-3 correlated with directly assessed performance measures. Our data underscore the potential value of VABS-3 and Leiter-3 as tools to assess psychometric function in AGS. With a deeper understanding of our patients' abilities, we can better guide clinicians and families to provide appropriate support and personalized interventions to empower children with leukodystrophies to maximize their communication and educational potential.

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.

Implementation fidelity, student outcomes, and cost-effectiveness of train-the-trainer strategies for Masters-level therapists in urban schools: results from a cluster randomized trial.

BACKGROUND: Little is known about the effectiveness and cost-effectiveness of train-the-trainer implementation strategies in supporting mental health evidence-based practices in schools, and about the optimal level of support needed for TT strategies. METHODS: The current study is part of a larger type 2 hybrid cluster randomized controlled trial. It compares two train-the-trainer strategies, Train-the-Trainer (TT) and Train-the-Trainer plus ongoing consultation for trainers (TT +) on the delivery of a group cognitive behavioral treatment protocol for anxiety disorders. Participants were 33 therapists, 29 supervisors, and 125 students who were at risk for anxiety disorders from 22 urban schools. Implementation outcomes were implementation fidelity and treatment dosage. Student outcomes were child- and parent-reported symptoms of anxiety, child-reported symptoms of depression, and teacher-reported academic engagement. We estimated the cost of implementing the intervention in each condition and examined the probability that a support strategy for supervisors (TT vs TT +) is a good value for varying values of willingness to pay. RESULTS: Therapists in the TT and TT + conditions obtained similarly high implementation fidelity and students in the conditions received similar treatment dosages. A mixed effects modeling approach for student outcomes revealed time effects for symptoms of anxiety and depression reported by students, and emotional disaffection reported by teachers. There were no condition or condition × times effects. For both conditions, the time effects indicated an improvement from pre-treatment to post-treatment in symptoms of anxiety and depression and academic emotional engagement. The average cost of therapist, supervisor, and consultant time required to implement the intervention in each condition was $1002 for TT and $1431 for TT + (p = 0.01). There was a greater than 80% chance that TT was a good value compared to TT + for all values of willingness to pay per one-point improvement in anxiety scores. CONCLUSIONS: A TT implementation approach consisting of a thorough initial training workshop for therapists and supervisors as well as ongoing supervision for therapists resulted in adequate levels of fidelity and student outcomes but at a lower cost, compared to the TT + condition that also included ongoing external expert consultation for supervisors. TRIAL REGISTRATION: ClinicalTrials.gov identifier: NCT02651402.

Motor training for young children with cerebral palsy: A single-blind randomized controlled trial.

AIM: To compare the effect of iMOVE (Intensive Mobility training with Variability and Error) therapy with dose-matched conventional therapy on gross motor development and secondary outcomes in young children with cerebral palsy. METHOD: This single-blind, randomized controlled trial included repeated assessments of gross motor function (using the Gross Motor Function Measure) and secondary outcomes during a 12- to 24-week intervention phase and at three follow-up points after treatment. Treatment was delivered three times per week in both groups. Forty-two children aged 12 to 36 months were stratified by age and motor function to ensure equivalence between groups at baseline. RESULTS: Thirty-six children completed treatment and follow-up phases. Treatment fidelity was high and adherence was equivalent between groups (77.3% conventional therapy, 76.2% iMOVE). There were no group differences on the primary (gross motor function after 12 weeks p = 0.18; after 24 weeks p = 0.94) or any secondary (postural control p = 0.88, caregiver satisfaction p = 0.52, child engagement p = 0.98) measure after treatment or at the follow-up points. However, one-third of total participants exceeded predicted change after 12 weeks and 77% exceeded predicted change after 24 weeks of treatment. INTERPRETATION: Our observations indicate a potential dose-response effect of rehabilitation therapy. We further demonstrated that individual therapeutic ingredients can be manipulated. When delivered consistently, both iMOVE and conventional therapy interventions might both be more effective than standard care. WHAT THIS PAPER ADDS: Those receiving iMOVE therapy demonstrated more independent practice time, error, and child-initiation than those receiving the dose-matched control. iMOVE therapy was not superior to the control (conventional physical) therapy. Most participants exceeded predicted change after 24 weeks of treatment.

The effects of Fe, Mg, and Pt-doping on the improvement of Ni stabilized on Al2O3-CeO3 catalysts for methane dry reforming.

Herein, the promotional effects of Mg, Fe, and Pt on Ni-based catalysts supported on Al2O3-CeO2 (Ni/Al2O3-CeO2) were investigated in the dry reforming of methane (DRM) reaction. The interaction of a suitable amount of MgO and FeO with Ce2O3 stabilized in the catalysts was demonstrated by the temperature-programmed desorption of CO2 (CO2-TPD). Ce2O3 has a high basicity for adsorbing CO2, generating a monoclinic Ce2O2CO3 species in the DRM reaction. Surface oxygen ions were also produced by adding MgO and FeO, as demonstrated by the temperature-programmed reduction of H2 (H2-TPR). Monoclinic Ce2O2CO3 and surface oxygen may both be used to oxidize and remove the carbon that was deposited, maintaining the high activity and stability of the metal Ni and Pt catalysts. The high dispersion and synergistic interactions between the platinum and oxide phases, which are associated with the decrease in reduction temperature and the rise in the number of basic sites, are responsible for the increased activity of Pt with M-Ni/Al2O3-CeO2 catalysts. The co-doped Ni/Al2O3-CeO2 catalysts with Mg and Fe significantly enhanced the activity (more than 80% methane and 84% CO2 conversion), the selectivity toward syngas (∼90%), and maintained the H2/CO ratio at about 0.97 at 700 °C.

A comparison of two group cognitive behavioral therapy protocols for anxiety in urban schools: appropriateness, child outcomes, and cost-effectiveness.

Background: Cognitive behavioral therapy (CBT) for pediatric anxiety is efficacious for reducing anxiety symptoms and improving functioning, but many children are unable to access CBT for anxiety in community settings. Schools are an important setting in which children access mental health care, including therapy for anxiety. In this setting, therapy is usually delivered by Masters-level therapists. Objectives: Friends for Life (FRIENDS), a 12-session, manualized, group CBT program for anxiety has demonstrated effectiveness when implemented in schools. However, prior research has also found challenges regarding feasibility and cultural fit when delivering FRIENDS in the urban school context. To address these challenges, we adapted FRIENDS for implementation in the school setting so that it might be more feasible and culturally appropriate for low-income, urban schools in the United States, while maintaining the core components of treatment. The current study uses a mixed-method approach to compare the effectiveness, cost-effectiveness, and perceived appropriateness of FRIENDS and CATS when delivered by Masters-level therapists with train-the-trainer support. Materials and methods: First, we compared change scores for student outcomes (i.e., child-report MASC-2 total score, parent-report MASC-2 total score, teacher-report Engagement and Disaffection subscale scores) from pre- to post- treatment between students receiving FRIENDS and students receiving CATS to assess whether the two conditions resulted in equivalent outcomes. Second, we compared the cost and cost-effectiveness between the groups. Finally, we used an applied thematic analysis to compare appropriateness of the interventions as perceived by therapists and supervisors. Results: The mean change score for the child-reported MASC-2 was 1.9 (SE = 1.72) points in the FRIENDS condition and 2.9 (SE = 1.73) points in the CATS condition; results indicated that the conditions were similar in their treatment effects, and symptom reductions were small in both groups. The modified protocol, CATS, was shown to cost significantly less to implement compared to FRIENDS and showed greater cost-effectiveness. Finally, compared to therapists and supervisors in the CATS condition, therapists and supervisors in the FRIENDS condition more strongly described aspects of the intervention that were not appropriate for their context and in need of more extensive adaptations. Conclusion: Relatively brief, group CBT for anxiety, with adaptations to improve cultural fit, is a promising approach to treat youth anxiety symptom when delivered by school-based therapists with train-the-trainer implementation support.

Exploration of Gross Motor Function in Aicardi-Goutières Syndrome.

Background: Aicardi-Goutières syndrome (AGS) is a rare genetic disorder characterized by a spectrum of motor abilities. While the Aicardi-Goutières syndrome severity score favors severely impacted individuals, there is an unmet need to define tools measuring function across the Aicardi-Goutières syndrome spectrum as potential outcome assessments for future clinical trials. Methods: Gross Motor Function Measure-88 (GMFM-88) and AGS Severity Scale were administered in individuals affected by Aicardi-Goutières syndrome (n = 71). We characterized the performance variability by genotype. Derived versions of the GMFM-88, including the GMFM-66, GMFM-66 item set (GMFM-66IS), and GMFM-66 Basal&Ceiling (GMFM-66BC) were calculated. The Aicardi-Goutières syndrome cohort was divided into severe (AGS Severity Scale score <4) or attenuated (≥4). Performance on the AGS Severity Scale highly correlated with total GMFM-88 scores (Spearman Correlation: R = 0.91). To assess variability of the GMFM-88 within genotypic subcohorts, interquartile ranges (IQRs) were compared. Results: GMFM-88 performance in the TREX1 cohort had least variability while the SAMHD1 cohort had the largest IQR (4.23 vs 81.8). Floor effect was prominent, with most evaluations scoring below 20% (n = 46, 64.79%), particularly in TREX1- and RNASEH2-cohorts. Performance by the GMFM-66, GMFM-66IS, and GMFM-66BC highly correlated with the full GMFM-88. The Aicardi-Goutières syndrome population represents a broad range of gross motor skills. Conclusions: This work identified the GMFM-88 as a potential clinical outcome assessment in subsets of the Aicardi-Goutières syndrome population but underscores the need for additional validation of outcome measures reflective of the diverse gross motor function observed in this population, including low motor function. When time is limited by resources or patient endurance, shorter versions of the GMFM-88 may be a reasonable alternative.

Development of an Online Training Platform and Implementation Strategy for School-Based Mental Health Professionals in Rural Elementary Schools: A Mixed-Methods Study.

Children in rural settings are less likely to receive mental health services than their urban and suburban counterparts and even less likely to receive evidence-based care. Rural schools could address the need for mental health interventions by using evidence-based practices within a tiered system of supports such as positive behavioral interventions and supports. However, very few school professionals, with or without mental health training, have received training on evidence-based practices. Rural schools need implementation strategies focused on training to prepare school personnel for the implementation of interventions with fidelity. Little is known about training strategies that are feasible and appropriate for the rural school context. User-centered design is an appropriate framework for the development of training strategies for professionals in rural schools because of its participatory approach and the development of products that fit the context where they are going to be used. The purpose of the study was to develop and assess components of an online training platform and implementation strategy based on the user-centered design. Quantitative and qualitative data from 25 participants from an equal number of schools in rural areas of Pennsylvania were used in the study. A mixed-methods design utilizing complementary descriptive statistics and theme analyses indicated that the training platform and implementation strategy were perceived as highly acceptable, appropriate, feasible and usable by school professionals. The resulting training platform and implementation strategy will fill a void in the training literature in rural schools.

Analysis and process evaluation of metal dopant (Zr, Cr)-promoted Ga-modified ZSM-5 for the oxidative dehydrogenation of propane in the presence and absence of CO2.

A systematic study of the comparative performances of 4% Ga-, 4% Zr-, and 1% Cr-impregnated H-ZSM-5 catalysts for oxidative dehydrogenation of propane in the presence and absence of CO2 is presented. It was found that methane, ethene, propene, butene, pentene, and BTX are the major products from all of these catalysts at various reaction temperatures (400-550 °C), WHSV, 4 kgcat s molC3H8 -1 and feed gas, C3H8/N2 = 2.5/97.5 and C3H8/CO2/N2 = 2.5/5/92.5; flow rate, 75 mL min-1 under atmospheric pressure for 10 h. The combination of material characterization and catalytic testing revealed that Ga-, Zr-, and Cr-doped H-ZSM-5 are excellent catalysts for this process, helping to achieve around 61% CO2 conversion. The co-doped Ga/H-ZSM-5 catalysts significantly enhanced the activity (65% propane and 61% CO2 conversion at 550 °C) among all the tested catalysts, with approximately 100% total selectivity 62% towards BTX and 26% towards propene, but with lower selectivity for methane, ethene, and pentene (i.e., light hydrocarbons). TPR profiles indicated that the redox cycle between Cr(iii)O6 and Cr(vi)O4 played an important role in the dehydrogenation of C3H6 over Cr/Zr-Ga/H-ZSM-5. CO2 could oxidize a part of the Cr(iii) species to Cr(vi) species under the reaction conditions used.

Locomotor learning in infants at high risk for cerebral palsy: A study protocol.

Background: Physical disability in individuals with cerebral palsy (CP) creates lifelong mobility challenges and healthcare costs. Despite this, very little is known about how infants at high risk for CP learn to move and acquire early locomotor skills, which set the foundation for lifelong mobility. The objective of this project is to characterize the evolution of locomotor learning over the first 18 months of life in infants at high risk for CP. To characterize how locomotor skill is learned, we will use robotic and sensor technology to provide intervention and longitudinally study infant movement across three stages of the development of human motor control: early spontaneous movement, prone locomotion (crawling), and upright locomotion (walking). Study design: This longitudinal observational/intervention cohort study (ClinicalTrials.gov Identifier: NCT04561232) will enroll sixty participants who are at risk for CP due to a brain injury by one month post-term age. Study participation will be completed by 18 months of age. Early spontaneous leg movements will be measured monthly from 1 to 4 months of age using inertial sensors worn on the ankles for two full days each month. Infants who remain at high risk for CP at 4 months of age, as determined from clinical assessments of motor function and movement quality, will continue through two locomotor training phases. Prone locomotor training will be delivered from 5 to 9 months of age using a robotic crawl training device that responds to infant behavior in real-time. Upright locomotor training will be delivered from 9 to 18 months of age using a dynamic weight support system to allow participants to practice skills beyond their current level of function. Repeated assessments of locomotor skill, training characteristics (such as movement error, variability, movement time and postural control), and variables that may mediate locomotor learning will be collected every two months during prone training and every three months during upright training. Discussion: This study will develop predictive models of locomotor skill acquisition over time. We hypothesize that experiencing and correcting movement errors is critical to skill acquisition in infants at risk for CP and that locomotor learning is mediated by neurobehavioral factors outside of training.Project Number 1R01HD098364-01A1.ClinicalTrials.gov Identifier: NCT04561232.

Natural history and neurodevelopmental outcomes in perinatal stress induced hyperinsulinism.

Objective: To describe perinatal stress induced hyperinsulinism (PSIHI), determine the prevalence of neurodevelopmental differences, and identify risk factors for poor developmental prognosis. Methods: Subjects with a history of hyperinsulinism (HI) and perinatal stress and in whom resolution of the HI was demonstrated were included. Medical record review, caregiver interview, and three validated developmental assessments were completed. Results: Of the 107 subjects (75% male), 36% were born between 32 and 37 weeks. Median age of hypoglycemia presentation was 0 days. Median age at HI diagnosis was 12 days (IQR 13.5). Median length of time for initiation of definitive treatment was 14 days (IQR 14).Caregiver interviews were completed for 53 of 79 eligible subjects. Developmental concerns were reported by 51%. Neurodevelopmental assessments were completed by caregivers of 37 of the 53 enrolled subjects. The proportion of subjects scoring >1 SD and >2 SD away from the mean in the direction of concern on the major composite scores was significantly greater than in the general population (40.5% vs. 15.8%, P ≤ 0.0001 and 18.9% vs. 2.2%, P ≤ 0.0001, respectively).Male sex, small for gestational age status (SGA), and treatment with continuous feeds were associated with assessment scores >1 SD from the mean (P < 0.05). SGA and preeclampsia were associated with assessment scores >2 SD from the mean (P < 0.05). Conclusion: While the majority of infants presented with hypoglycemia in the first day of life, diagnosis and treatment occurred 12-14 days later. Children with PSIHI are at high risk of neurodevelopmental deficits and are more likely to perform below average on developmental assessment.

Visceral adiposity is related to insulin sensitivity and inflammation in adolescents with obesity and mild sleep disordered breathing.

OBJECTIVES: To evaluate the relationships between adipose tissue distribution, insulin secretion and sensitivity, sleep-disordered breathing, and inflammation in obese adolescents. METHODS: Cross-sectional study of 56 obese adolescents who underwent anthropometric measures, dual-energy X-ray absorptiometry, overnight polysomnography, oral glucose tolerance test (OGTT) and frequently sampled intravenous glucose tolerance test. Correlation and regression analyses were used to assess relationships between adiposity, insulin secretion and sensitivity, measures of sleep-disordered breathing (oxyhemoglobin nadir, SpO2; apnea hypopnea index, AHI; arousal index, AI; maximum end-tidal CO2; non-REM sleep duration), and inflammation (high-sensitivity C-reactive protein, hsCRP). RESULTS: Subjects (55% female) were mean (SD) 14.4 (2.1) years, with BMI Z-score of 2.3 (0.4). AHI was >5 in 10 (18%) subjects and 1< AHI ≤5 in 22 (39%). Visceral adipose tissue area (VAT) was positively correlated with OGTT 1 and 2 h insulin and 1 h glucose, and hsCRP (r=0.3-0.5, p≤0.007 for each). VAT was negatively correlated with sensitivity to insulin (r=-0.4, p=0.005) and SpO2 nadir (r=-0.3, p=0.04) but not with other sleep measures. After adjustment for BMI-Z, sex, population ancestry, age, and sleep measures, VAT remained independently associated with insulin measures and 1 h glucose, but no other measures of glycemia. SAT was not associated with measures of glycemia or insulin resistance. CONCLUSIONS: Among adolescents with obesity, visceral adiposity was associated with insulin resistance, SpO2 nadir, and inflammation. The independent association of visceral adiposity with insulin resistance highlights the potential role of VAT in obesity-related chronic disease.

Development and evaluation of a remote training strategy for the implementation of mental health evidence-based practices in rural schools: pilot study protocol.

BACKGROUND: An increasing number of schools in rural settings are implementing multi-tier positive behavioral interventions and supports (PBIS) to address school-climate problems. PBIS can be used to provide the framework for the implementation of evidence-based practices (EBPs) to address children's mental health concerns. Given the large service disparities for children in rural areas, offering EBPs through PBIS can improve access and lead to better long-term outcomes. A key challenge is that school personnel need technical assistance in order to implement EBPs with fidelity and clinical effectiveness. Providing ongoing on-site support is not feasible or sustainable in the majority of rural schools, due to their remote physical location. For this reason, remote training technology has been recommended for providing technical assistance to behavioral health staff (BHS) in under-served rural communities. OBJECTIVES: The purpose of this study is to use the user-centered design, guided by an iterative process (rapid prototyping), to develop and evaluate the appropriateness, feasibility, acceptability, usability, and preliminary student outcomes of two online training strategies for the implementation of EBPs at PBIS Tier 2. METHODS: The study will employ a pragmatic design comprised of a mixed-methods approach for the development of the training platform, and a hybrid type 2, pilot randomized controlled trial to examine the implementation and student outcomes of two training strategies: Remote Video vs. Remote Video plus Coaching. DISCUSSION: There is a clear need for well-designed remote training studies focused on training in non-traditional settings. Given the lack of well-trained mental health professionals in rural settings and the stark disparities in access to services, the development and pilot-testing of a remote training strategy for BHS in under-served rural schools could have a significant public health impact. ETHICS AND DISSEMINATION: The project was reviewed and approved by the institutional review board. Results will be submitted to ClinicalTrials.gov and disseminated to community partners and participants, peer-reviewed journals, and academic conferences. TRIAL REGISTRATION: ClinicialTrials.gov, NCT05034198 and NCT05039164.

Study protocol: cluster randomized trial of consultation strategies for the sustainment of mental health interventions in under-resourced urban schools: rationale, design, and methods.

BACKGROUND: The school is a key setting for the provision of mental health services to children, particularly those underserved through traditional service delivery systems. School-wide Positive Behavioral Interventions and Supports (PBIS) is a tiered approach to service delivery based on the public health model that schools use to implement universal (Tier 1) supports to improve school climate and safety. As our prior research has demonstrated, PBIS is a useful vehicle for implementing mental and behavioral health evidence-based practices (EBPs) at Tier 2 for children with, or at risk for, mental health disorders. Very little research has been conducted regarding the use of mental health EBPs at Tier 2 or how to sustain implementation in schools. METHODS/DESIGN: The main aim of the study is to compare fidelity, penetration, cost-effectiveness, and student outcomes of Tier 2 mental health interventions across 2 sustainment approaches for school implementers in 12 K-8 schools. The study uses a 2-arm, cluster randomized controlled trial design. The two arms are: (a) Preparing for Sustainment (PS)-a consultation strategy implemented by school district coaches who receive support from external consultants, and (b) Sustainment as Usual (SAU)-a consultation strategy implemented by school district coaches alone. Participants will be 60 implementers and 360 students at risk for externalizing and anxiety disorders. The interventions implemented by school personnel are: Coping Power Program (CPP) for externalizing disorders, CBT for Anxiety Treatment in Schools (CATS) for anxiety disorders, and Check-in/Check-out (CICO) for externalizing and internalizing disorders. The Interactive Systems Framework (ISF) for Dissemination and Implementation guides the training and support procedures for implementers. DISCUSSION: We expect that this study will result in a feasible, effective, and cost-effective strategy for sustaining mental health EBPs that is embedded within a multi-tiered system of support. Results from this study conducted in a large urban school district would likely generalize to other large, urban districts and have an impact on population-level child mental health. Trial registration ClinicalTrials.gov identifier number NCT04869657. Registered May 3, 2021.

Development of a neurologic severity scale for Aicardi Goutières Syndrome.

BACKGROUND AND PURPOSE: Aicardi Goutières Syndrome (AGS) is a severe, autoinflammatory leukodystrophy characterized by global neurologic dysfunction. Our goal was to create an easy-to-apply scale relevant to the unique developmental challenges associated with AGS. METHODS: All individuals were recruited through our natural history study. Individuals were classified by AGS severity as mild, moderate, or severe, and clinical encounters were assigned a composite score for neurologic function calculated from the sum of three functional classification scales. Through expert consensus, we identified 11 key items to reflect the severity of AGS across gross motor, fine motor, and cognitive skills to create the AGS Scale. There was strong interrater reliability. The AGS scale was applied across available medical records to evaluate neurologic function over time. The AGS scale was compared to performance on a standard measure of gross motor function (Gross Motor Function Measure-88, GMFM-88) and a putative diagnostic biomarker of disease, the interferon signaling gene expression score (ISG). RESULTS: The AGS scale score correlated with severity classifications and the composite neurologic function scores. When retrospectively applied across our natural history study, the majority of individuals demonstrated an initial decline in function followed by stable scores. Within the first 6 months of disease, the AGS score was the most dynamic. The AGS scale correlated with performance by the GMFM-88, but did not correlate with ISG levels. CONCLUSIONS: This study demonstrates the utility of the AGS scale as a multimodal tool for the assessment of neurologic function in AGS. The AGS scale correlates with clinical severity and with a more labor-intensive tool, GMFM-88. This study underscores the limitations of the ISG score as a marker of disease severity. With the AGS scale, we found that AGS neurologic severity is the most dynamic early in disease. This novel AGS scale is a promising tool to longitudinally follow neurologic function in this unique population.

Therapy and Psychotropic Medication Use in Young Children With Autism Spectrum Disorder.

BACKGROUND AND OBJECTIVES: Guidelines suggest young children with autism spectrum disorder (ASD) receive intensive nonpharmacologic interventions. Additionally, associated symptoms may be treated with psychotropic medications. Actual intervention use by young children has not been well characterized. Our aim in this study was to describe interventions received by young children (3-6 years old) with ASD. The association with sociodemographic factors was also explored. METHODS: Data were analyzed from the Autism Speaks Autism Treatment Network (AS-ATN), a research registry of children with ASD from 17 sites in the United States and Canada. AS-ATN participants receive a diagnostic evaluation and treatment recommendations. Parents report intervention use at follow-up visits. At follow-up, 805 participants had data available about therapies received, and 613 had data available about medications received. RESULTS: The median total hours per week of therapy was 5.5 hours (interquartile range 2.0-15.0), and only 33.4% of participants were reported to be getting behaviorally based therapies. A univariate analysis and a multiple regression model predicting total therapy time showed that a diagnosis of ASD before enrollment in the AS-ATN was a significant predictor. Additionally, 16.3% of participants were on ≥1 psychotropic medication. A univariate analysis and a multiple logistic model predicting psychotropic medication use showed site region as a significant predictor. CONCLUSIONS: Relatively few young children with ASD are receiving behavioral therapies or total therapy hours at the recommended intensity. There is regional variability in psychotropic medication use. Further research is needed to improve access to evidence-based treatments for young children with ASD.

Orofacial Manifestations of Stickler Syndrome: An Analysis of Speech Outcome and Facial Growth After Cleft Palate Repair.

PURPOSE: The purpose of this study was to characterize airway problems, speech outcomes, and facial growth in patients with Stickler syndrome undergoing cleft palate repair. METHODS: A retrospective, longitudinal study was performed at the Children's Hospital of Philadelphia on 25 patients with Stickler syndrome and 53 nonsyndromic patients with clefts of the secondary palate repaired between 1977 and 2000. Airway problems were characterized by the incidence of Pierre Robin Sequence (PRS) and the necessity for surgical airway management. Speech was analyzed using the Pittsburgh weighted values for speech symptoms associated with velopharyngeal incompetence (VPI). Longitudinal anthropometric measurements represented up to 12 years of longitudinal cephalofacial growth. RESULTS: Seventy-two percent of patients with Stickler syndrome were diagnosed with PRS, 55.6% of whom required surgical airway management. Conversely, 20.8% of nonsyndromic patients were diagnosed with PRS (P < 0.0001), 18% of whom required surgical intervention (P < 0.05). Speech outcomes were poorer in patients with Stickler syndrome with 40% demonstrating borderline VPI and 13.3% demonstrating VPI, compared with 21.8% and 9.1%, respectively, in the nonsyndromic group. Both groups exhibited significantly shallower upper and mid facial depths and wider upper facial breadths when compared with normal standards of facial growth. Although there was a tendency toward decreased facial depths in patients with Stickler syndrome relative to nonsyndromic patients, the differences were nonsignificant. CONCLUSIONS: Patients with Stickler syndrome show significant potential for early airway compromise and a poorer prognosis for speech outcome after cleft palate repair. Their cephalofacial growth does not differ significantly from that of nonsyndromic cleft palate patients.

Implementation of targeted mental health interventions in urban schools: Preliminary findings for impact of training strategy on program fidelity.

School-based mental health programs are increasingly recognized as methods by which to improve children's access to evidence-based practices (EBPs), particularly in urban under resourced communities. School-wide positive behavior interventions and supports (PBIS) is one approach to integrating mental health services into school-based programming; however, school providers require training and support to implement programs as intended. We have conducted a randomized controlled trial to compare two models for training school-based personnel to deliver group EBPs to children at high risk of developing internalizing or externalizing problems. School personnel (N = 24) from 6 schools in a large urban school district were trained with either a basic training and consultation strategy, or an enhanced training and consultation strategy. Preliminary findings show that the enhanced strategy resulted in 9% higher content fidelity than the basic strategy. School personnel who were switched to the basic strategy had slightly lower content fidelity for the last two years of the trial and school personnel who continued to receive basic consultation during the step-down phase saw their fidelity decline. The two conditions did not differ with regard to process fidelity.