Content validation of observer-reported sickle cell pain diaries (SCPD-CS and SCPD-CN): results from interviews with caregivers

Background Patients with sickle cell disease (SCD) experience daily pain and acute episodes known as sickle cell pain crises (SCPCs). The Sickle Cell Pain Diary-Caregiver Report (SCPD-C) is an observer-reported diary for use by caregivers of children ages < 12 years with SCD. This study reports on the content validity of the SCPD-C. Methods The SCPD-C was developed based on a literature review, measurement expert input, and a patient advisory board including clinicians. Three rounds of interviews (including both concept elicitation and cognitive debriefing methodologies) were conducted with caregivers of children with SCD aged < 12 to evaluate the content validity of the SCPD-C. Results Across three rounds of interviews, caregivers confirmed concepts in the SCPD-C and described observed impacts that were important and were added. Overall, caregivers evaluated the SCPD-C as easy to understand, with some minor adaptations for clarity. Additionally, the diary was split into two versions based on the child’s age and school enrollment status (SCPD-CS for school-aged and SCPD-CN for non-school age children). Conclusions Caregivers provided valuable input that led to important additions and changes to the measures. The SCPD-CS and SCPD-CN are appropriate and fit-for-purpose observer-reported outcome measures of SCPC-related pain frequency and severity, and impacts on health-related quality of life.

Quality of Life in Patients With Velopharyngeal Insufficiency in West China

Objective This study aimed to investigate the quality of life (QOL) of patients with cleft lip and palate and velopharyngeal insufficiency (VPI) in relation to sex, age, age at initial cleft lip surgery, and age at initial cleft palate surgery. Design This is a cross-sectional study. Setting The study was conducted in a tertiary medical center. Participants The participants were caregivers of 72 patients with cleft lip and palate and VPI aged 4 to 20 years. Main Outcome Measure(s) Participants completed the Chinese version of the caregiver report of the VPI Effects on Life Outcomes (VELO) questionnaire. The Mann–Whitney U test was used to evaluate the patients’ sex, age, age at initial cleft lip repair, and age at initial cleft palate repair in relation to VELO total score and domains. Spearman correlation analysis was completed including all study variables. Associations between the study variables and the VELO total score were tested using a generalized linear mixed model. Results In the univariate analysis, patients’ age and age at initial cleft palate surgery influenced the QOL of patients with VPI. There were no differences in the VELO total score or domains based on sex or age at first cleft lip surgery. In the generalized linear mixed model, patients older than 8 years had higher VELO total scores. Conclusions By caregiver report, the QOL of patients under age 8 years with VPI was lower than older patients. In addition, the caregiver impact domain was higher for parents of children who had their initial cleft palate surgery at age 2 years or younger.

Predictors of Satisfaction with Autism Treatment Services During COVID-19

The COVID-19 pandemic has created unprecedented challenges and disruptions for autistic individuals receiving specialized treatment services. This caregiver-report survey study (n = 339) explored predictors of satisfaction with autism services during COVID-19 to improve perceived support for these families. Specifically, we investigated whether service delivery medium (telehealth vs. in person), child’s emotional functioning, and caregiver stress would predict satisfaction with the most highly utilized services. Satisfaction ratings for ABA/behavioral, speech/language, and occupational therapy were lower when delivered via telehealth as compared to in person. Caregivers who reported higher emotional dysregulation in their children were less satisfied with behavioral therapy services. These results provide a critical caregiver-informed perspective on factors influencing satisfaction with specialized autism services during COVID-19.

Content validation of observer-reported sickle cell pain diaries (SCPD-CS and SCPD-CN): results from interviews with caregivers

Background Patients with sickle cell disease (SCD) experience daily pain and acute episodes known as sickle cell pain crises (SCPCs). The Sickle Cell Pain Diary - Caregiver Report (SCPD-C) is an observer-reported diary for use by caregivers of children ages < 12 years with SCD. This study reports on the content validity of the SCPD-C. Methods The SCPD-C was developed based on a literature review, measurement expert input, and a patient advisory board including clinicians. Three rounds of interviews (including both concept elicitation and cognitive debriefing methodologies) were conducted with caregivers of children with SCD aged < 12 to evaluate the content validity of the SCPD-C. Results Across three rounds of interviews, caregivers confirmed concepts in the SCPD-C and described observed impacts that were important and were added. Overall, caregivers evaluated the SCPD-C as easy to understand, with some minor adaptations for clarity. Additionally, the diary was split into two versions based on the child’s age and school enrollment status (SCPD-CS for school-aged and SCPD-CN for non-school age children). Conclusions Caregivers provided valuable input that led to important additions and changes to the measures. The SCPD-CS and SCPD-CN are appropriate and fit-for-purpose observer-reported outcome measures of SCPC-related pain frequency and severity, and the impacts on health-related quality of life.

Caregiver Stress and the Association Between Clinically Observed and Caregiver-Reported Sensory Responsiveness

Date Presented 04/13/21 Sensory processing challenges are measured by caregiver report and clinician observation and affect many people with autism spectrum disorder and their caregivers. It is unclear how caregiver stress influences the correlation between scores on caregiver report and observational measures. The present analysis explores correlations between two formats of tools to assess sensory processing and indicates that caregiver stress moderates the association between sensory-seeking scores on the measures. Primary Author and Speaker: Emily Campi Additional Authors and Speakers: Abdullah S. Alsaeed, Kecia Doyle, Shanda N. Geil, and Kristen A. Pickett Contributing Authors: Noah T. Herzog

Patterns of Cognition, Communication, and Adaptive Behavior in Children With Developmental Disabilities

Young children with developmental disabilities (DD) exhibit a range of strengths and weaknesses in cognitive, language, and adaptive skills. Identifying individual patterns of abilities across these domains is important for informing interventions. This study examines how 129 toddlers with significant developmental delays and less than 10 spoken words perform across different developmental domains and assessment methods (i.e., caregiver report and clinician-administered tests). Children exhibited statistically and clinically meaningful strengths and weaknesses across developmental domains, which may have important implications for differential interventions. Caregiver-reported and clinician-rated measures of cognition, language and adaptive functioning were highly related. However, the relation between caregiver report and clinician ratings was weaker for a subgroup of children with relatively more limited expressive language compared to other children in the sample.

The Toddler Autism Symptom Inventory: Use in diagnostic evaluations of toddlers

Although symptoms of autism are present early in life and early diagnosis can lead to better outcomes, there is a dearth of validated caregiver-report interviews designed for children under the age of 3 years. We developed the Toddler Autism Symptom Inventory, a semi-structured interview designed to assess the presence and absence of skills and symptoms in children aged 12–36 months. Reliability and validity of items and a cutoff score for likelihood of autism spectrum disorder were established. Specificity and sensitivity of this cutoff were confirmed with a cross-validation sample. The Toddler Autism Symptom Inventory effectively identified most children with autism without excessive false positives. The Toddler Autism Symptom Inventory is a developmentally appropriate caregiver interview for use in diagnostic evaluations of children under age 3 years that offers clearly operationalized diagnostic criteria and a cutoff for autism likelihood for very young children. Lay abstract Determining whether a young child has an autism spectrum disorder requires direct observation of the child and caregiver report of the child’s everyday behaviors. There are few interviews for parents that are specifically designed for children under 3 years of age. The Toddler Autism Symptom Inventory is a new interview that asks caregivers of children age 12–36 months about symptoms of possible autism spectrum disorder. The Toddler Autism Symptom Inventory uses a cutoff score to indicate likelihood for autism spectrum disorder; this cutoff score appears to accurately identify most children who are diagnosed with autism spectrum disorder without identifying too many who do not have autism spectrum disorder. The Toddler Autism Symptom Inventory interview can help clinicians to determine whether a young child shows symptoms suggestive of an autism spectrum disorder.

Feasibility pilot of an adapted parenting program embedded within the Thai public health system

Background This feasibility pilot of the Parenting for Lifelong Health for Young Children program in Thailand aimed to: 1) explore the feasibility of study evaluation approaches; 2) assess the feasibility of delivering an adapted program; 3) report indicative effects on child maltreatment and related outcomes; and 4) examine intervention content associated with key mechanisms of change perceived by caregivers and facilitators. Method Sixty primary caregivers of children aged 2–9 years were recruited for an 8-week parenting program embedded within the local health system. Mixed-methods approaches included quantitative caregiver-report and observational data from standardized instruments, and qualitative data from individual and group interviews with caregivers and program facilitators. Analyses involved Wilcoxon signed-rank tests, paired t-tests, Friedman’s ANOVA, and thematic analysis. Results Participants reported that most (65%) were grandparents or great-grandparents. Study retention and response rates were high, and enrolled caregivers attended an average of 93% of sessions. Primary outcomes showed caregiver-reported pre-post reductions in overall child maltreatment (d = − 0.58, p < 0.001), as well as reductions in physical (d = − 0.58, p < 0.001) and emotional abuse (d = − 0.40, p < 0.001). Combined caregiver report and observational assessments using the HOME Inventory showed reductions in abusive and harsh parenting (d = − 0.52, p < 0.001). Secondary outcomes demonstrated decreases in child neglect; dysfunctional parenting; poor child monitoring and supervision; parental sense of inefficacy; child behavior problems; daily report on child problem behavior; parent overall depression, anxiety, and stress; and attitudes supporting physical punishment and harsh discipline. There were increases in overall positive parenting, daily positive parenting behavior, as well as HOME Inventory assessments on parent-child relationships. Thematic analyses from interviews and focus group data identified six key program themes associated with strengthened parent-child relationships, reduced child behavior problems, improved attitudes and strategies toward discipline, and improved management of parental stress. Conclusions This study represents one of few evaluations to test the feasibility of an evidence-based parenting program embedded within routine public health service delivery in a low- or middle-income country. Findings show preliminary effectiveness in reducing child maltreatment, improvements on 22 of 24 secondary outcomes, and perceived mechanisms of change that support quantitative findings. Prospects are promising for program scalability, pending randomized controlled trial results. Trial registration 11/01/2019, ClinicalTrials.gov, ID# NCT03539341.