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Purpose: The aim of this study was to pilot the efficacy of rapid syllable transition (ReST) treatment when provided once per week for a 50-min treatment session for 12 weeks with five children with childhood apraxia of speech. Of central importance was the children's retention and generalization of gains from treatment as indicators of speech motor learning. Method: A multiple-baseline across-participant design was employed to investigate (a) treatment effect on the 20 treated pseudowords, (b) generalization to 40 untreated real words and 10 untreated polysyllabic word sentences, and (c) maintenance of any treatment and generalization goals to up to 4 months posttreatment. To investigate any difference between in-session performance and retention, a comparison was made between data collected during treatment and probe sessions. Results: Treatment data collected during therapy showed all children improving across their 12 treatment sessions. Three of the five children showed a treatment effect on treated pseudowords in the probe sessions, but only one child showed generalization to untreated real words, and no children showed generalization to sentences. Conclusions: ReST treatment delivered at a dose frequency of once per week was efficacious for only one of the five children. In-session treatment data were not a reliable indicator of children's learning. One session per week of ReST therapy is therefore not recommended.

Purpose: The current standard for clinical diagnosis of childhood apraxia of speech (CAS) is expert clinician judgment. The psychometric properties of this standard are not well understood; however, they are important for improving clinical diagnosis. The purpose of this study is to determine the extent to which experts agree on the clinical diagnosis of CAS using two cohorts of children with mixed speech sound disorders (SSDs). Method: Speech samples of children with SSDs were obtained from previous and ongoing research from video recordings of children aged 3-8 years (n = 36) and audio recordings of children aged 8-17 years (n = 56). A total of 23 expert, English-speaking clinicians were recruited internationally. Three of these experts rated each speech sample to provide a description of the observed features and a diagnosis. Intrarater reliability was acceptable at 85% agreement. Results: Interrater reliability on the presence or absence of CAS among experts was poor both as a categorical diagnosis (K = 0.187, 95% confidence interval [CI] [0.089, 0.286]) and on a continuous "likelihood of CAS" scale (0-100; intraclass correlation = 0.183, 95% CI [0.037, 0.347]). Reliability was similar across the video-recorded and audio-only samples. There was greater agreement on other diagnoses (such as articulation disorder) than on the diagnosis of CAS, although these too did not meet the predetermined standard. Likelihood of CAS was greater in children who presented with more American Speech-Language-Hearing Association CAS consensus features. Conclusions: Different expert raters had different thresholds for applying the diagnosis of CAS. If expert clinician judgment is to be used for diagnosis of CAS or other SSDs, further standardization and calibration is needed to increase interrater reliability. Diagnosis may require operationalized checklists or reliable measures that operate along a diagnostic continuum.

BackgroundFailure of successful transition to adult care for adolescents and young adults with chronic rheumatic diseases negatively impacts their health and wellbeing. Transition of care is a vital and complex process within pediatric rheumatology that can be difficult to execute. Use of quality improvement (QI) and clinical informatics (CI) can help implement transition programs.Local problemDespite efforts to improve transition of care within our pediatric rheumatology clinic, it has been difficult to implement and sustain good transition practices including assessment of transition readiness. Using QI methodology and CI, this study aimed to improve transition readiness assessment from 12 to 30% and sustain for one year by surveying transitioning patients yearly.MethodsA transition-focused QI team utilized methods endorsed by the Institute for Healthcare Improvement and leveraged CI to improve survey completion. Control charts of survey completion rates were tracked monthly. Descriptive statistics were used to analyze survey responses.InterventionsInterventions focused on automation of patient surveys at regularly scheduled clinic visits.Results1,265 questionnaires were administered to 1,158 distinct patients. Survey completion rose from a baseline of 12% to greater than 90% and was sustained over 18 months. Identified educational needs included health insurance, scheduling appointments, obtaining care outside of rheumatology clinic business hours, Electronic Health Record messaging, and refilling medications.ConclusionsBy leveraging CI and QI methodology, we were able to assess transition readiness in more than 90% of our patients and identify gaps in self-management. Process automation can create sustainable transition practices.

[A] dark wall of rule supports the structure of every letter, record, transcript: every proof of authority and power. I know records are compiled by winners, and scholarship is in [...]

Abstract Background Face-to-face group-based diabetes prevention programmes have been shown to be effective in many settings. Digital delivery may suit some patients, but research comparing the effectiveness of digital with face-to-face delivery is scarce. The aim was to assess if digital delivery of the English National Health Service Diabetes Prevention Programme (NHS DPP) is non-inferior to group-based face-to-face delivery in terms of weight change, and evaluate factors associated with differential change. Methods The study included those recruited to the NHS DPP in 2017–2018. Individual-level data from a face-to-face cohort was compared to two cohorts on a digital pilot who (i) were offered no choice of delivery mode, or (ii) chose digital over face-to-face. Changes in weight at 6 and 12 months were analysed using mixed effects linear regression, having matched participants from the digital pilot to similar participants from face-to-face. Results Weight change on the digital pilot was non-inferior to face-to-face at both time points: it was similar in the comparison of those with no choice (difference in weight change: -0.284 kg [95% CI: -0.712, 0.144] at 6 months) and greater in digital when participants were offered a choice (-1.165 kg [95% CI: -1.841, -0.489]). Interactions between delivery mode and sex, ethnicity, age and deprivation were observed. Conclusions Digital delivery of the NHS DPP achieved weight loss at least as good as face-to-face. Patients who were offered a choice and opted for digital experienced better weight loss, compared to patients offered face-to-face only.

She ran through paint until the sky was stained green She lived in brushes and pluses and doorways in crusts of oil and slop She drank laughter in coffee cups [...]

Summary: Adolescent idiopathic scoliosis (AIS), a sideways curvature of the spine, is sexually dimorphic, with increased incidence in females. A genome-wide association study identified a female-specific AIS susceptibility locus near the PAX1 gene. Here, we use mouse enhancer assays, three mouse enhancer knockouts, and subsequent phenotypic analyses to characterize this region. Using mouse enhancer assays, we characterize a sequence, PEC7, which overlaps the AIS-associated variant, and find it to be active in the tail tip and intervertebral disc. Removal of PEC7 or Xe1, a known sclerotome enhancer nearby, or deletion of both sequences lead to a kinky tail phenotype only in the Xe1 and combined (Xe1+PEC7) knockouts, with only the latter showing a female sex dimorphic phenotype. Extensive phenotypic characterization of these mouse lines implicates several differentially expressed genes and estrogen signaling in the sex dimorphic bias. In summary, our work functionally characterizes an AIS-associated locus and dissects the mechanism for its sexual dimorphism.

ROBERT STORR SOME YEARS BACK, a student who had attended the summer program at Skowhegan in Maine told me about the powerful impression Elizabeth Murray had made on him. One [...]

Abstract Background Childhood apraxia of speech (CAS) is a pediatric motor-based speech sound disorder that requires a specialized approach to intervention. The extant literature on the treatment of CAS commonly recommends intensive treatment using a motor-based approach, with some of the best evidence supporting the use of Dynamic Temporal and Tactile Cueing (DTTC). To date, a rigorous and systematic comparison of high and low dose frequency (i.e., frequency of therapy sessions) has not been undertaken for DTTC, resulting in a lack of evidence to guide decisions about the optimal treatment schedule for this intervention. The current study aims to fill this gap in knowledge by comparing treatment outcomes when dose frequency is varied. Methods A randomized controlled trial will be conducted to examine the efficacy of low versus high dose frequency on DTTC treatment outcomes in children with CAS. A target of 60 children, 2;6–7;11 years of age, will be recruited to participate in this study. Treatment will be provided in the community setting by speech-language pathologists who have completed specialized training administering DTTC in a research reliable manner. True randomization with concealed allocation will be used to assign children to either the low or high dose frequency group. Treatment will be administered in 1-h sessions either 4 times per week over a 6-week period (high dose) or 2 times per week over a 12-week period (low dose). To measure treatment gains, probe data will be collected before treatment, during treatment, and 1 day, 1 week, 4 weeks, and 12 weeks post-treatment. Probe data will consist of customized treated words and a standard set of untreated words to assess generalization of treatment gains. The primary outcome variable will be whole word accuracy, encompassing segmental, phonotactic, and suprasegmental accuracy. Discussion This will be the first randomized controlled trial to evaluate dose frequency for DTTC treatment in children with CAS. Trial registration ClinicalTrials.gov identifier NCT05675306, January 6, 2023.