Parent-report Measures Research Articles

Self-Reported Multidimensional Gender Identity in Autistic and Non-Autistic Children.

PURPOSE: The several prior studies assessing gender identity in young autistic individuals mostly included a mix of child and adolescent participants, heavily relied on parent-reported measures, and yielded mixed findings. A single parent-reported item from the Child Behavior Checklist assessing "wish to be of the opposite sex" was employed in most of these studies. Only one prior study focused specifically on children, but that study employed parent-reported measures. Using self-reported multidimensional measures, the present study assessed gender identity in autistic and non-autistic children aged 4 to 11 years (30 autistic boys, 35 non-autistic boys, 20 autistic girls, 35 non-autistic girls). Child-friendly measures were used to assess own-gender similarity, other-gender similarity, gender contentedness, and wish to be of the other gender. Vocabulary and non-verbal reasoning were also assessed. Based on descriptive statistics, compared with non-autistic boys, autistic boys showed increased gender identity variance across all four dimensions (lower own-gender similarity, higher other-gender similarity, lower gender contentedness, greater wish to be of the other gender). These group differences between autistic and non-autistic boys were medium and statistically significant for three of the four dimensions and small-to-medium and marginally significant for the remaining dimension. Autistic girls and non-autistic girls did not show consistent or significant differences in gender identity. There were no differences between the autistic and non-autistic groups in vocabulary or non-verbal reasoning in either boys or girls. Gender identity variance may emerge early in development in autistic individuals, but the trajectory may differ for boys and girls.

Parental depressiveness and children’s emotional problems

This study used panel data to examine the longitudinal effect of maternal and paternal depressiveness on children’s emotional difficulties in two-parent families in Germany. The main purpose was to examine whether the strength of this association differs depending on whether parents or children themselves assessed children’s emotional difficulties. Based on data from 1,586 children from the German Family Panel (pairfam), our findings show much stronger associations between parental depressiveness and children’s emotional difficulties when using parent-reported measures than when using child-reported measures, which indicates a negative mood bias in parent reports. The findings emphasise the importance of taking children’s self-reports into account when studying child wellbeing.

Parent-Reported Outcome Measures for Individuals with Fragile X Syndrome: Clinically Meaningful Change Thresholds.

Estimating meaningful change thresholds (MCT) on clinical outcome assessments is an important consideration when evaluating treatments. In fragile X syndrome (FXS) research, there has been no consensus on how to define MCT's on several commonly used outcome measures. The purpose of the current study was to determine clinically relevant MCT's of caregiver-rated assessments using data from a phase 3 clinical trials of arbaclofen (Berry-Kravis et al., 2017). Data were collected as a part of previous phase 3, double-blind, placebo-controlled studies of arbaclofen in individuals with FXS (Berry-Kravis et al., 2017). The two studies enrolled age groups of 5-11-years (n = 159) and 12-50-years (n = 119). The current study examines meaningful within-patient change thresholds from baseline to treatment week 8 across several measures: ABC-CFXS; PSI; Vineland-II; and a Visual Analog Scale (VAS) of Anxiety and Disruptive Behaviors. MCT's were established by using anchor-based methods, using the CGI-S and CGI-I as anchors. Examining the results of the anchor-based analyses and visual CDF plots, MCT's were observed for the pediatric study for the ABC-CFXS subscales (with a range depending on use of CGI-S or CGI-I as anchor): Irritability: 11.1-14.8 points; Hyperactivity: 6.7-8.9 points; and Socially Unresponsive/Lethargic: 6.6-8.1 points; as well both VAS subscales: Anxiety: 28.3-36.2mm; and Disruptive Behavior: 22.4-27.4mm. Such thresholds were not observed for the Vineland-II and PSI subscales. Our analysis of MCT's helps set the stage for interpreting clinical trial results in FXS. This may include use of relevant subscales of the ABC-CFXS and VAS as primary outcomes using the MCT's for response definition. This work may help define future study inclusion criteria and enable future interpretation of treatment outcome results in the field.

(In)Congruences in parent-adolescent reports of physical activity parenting practices: associations with adolescent physical activity

BackgroundInformant discrepancies in physical activity parenting practices (PAPP) are prevalent, but their effect on adolescent physical activity (PA) remains underexplored. This study aims to examine the relationship between the (in)congruence in the parent-adolescent reports of PAPP and adolescent physical activity (PA).MethodsSelf-administered questionnaires were used to collect perceptions on nine types of PAPP and adolescents’ PA levels from 373 Chinese parent-adolescent dyads. Multiple linear regression, polynomial regression, and Response Surface Analysis were employed to explore the relationship between parent-adolescent (in)congruence in PAPP reports and adolescent physical activity.ResultsOver half of the dyads exhibited incongruence in their PAPP reports, with parents generally reporting higher PAPP scores compared to adolescents. Neither parent-reported nor adolescent-reported punishment, pressuring, and restriction were significantly associated adolescent PA. In contrast, adolescent-reported disengagement, expectation, facilitation, monitoring, non-directive support, and autonomy support demonstrated stronger significant associations with their PA levels compared to parent-reported measures. Congruence in reporting expectation, facilitation, monitoring, non-directive support, and autonomy support was positively associated with adolescent PA, while incongruence in these practices showed inverse associations. In addition, adolescents’ gender-specific analyses demonstrated different informant effects on disengagement, monitoring, and non-directive support.ConclusionParent-adolescent (in)congruences on positive PAPP rather than negative PAPP showed significant relationships with adolescents’ PA levels, highlighting the importance of aligning parents’ and adolescents’ perceptions on positive PAPP to promote adolescent PA. Moreover, adolescent girls appear to be more sensitive to PAPP involving parents’ presence than boys.

Development of a novel parent-report measure of food allergy impact.

The aim of this study was to develop an outcome measure for caregivers of children with food allergy. A community-informed measure development process was utilised to generate a novel parent-reported outcome tool for caregivers of children with food allergy. We conducted focus groups with parents and caregivers of food-allergic (n = 18) children, following which a team of coders identified themes and generated an initial set of candidate items. These items were reviewed by content-area experts (n = 10) using content validity indices. We then conducted a series of cognitive interviews with parents of children with food allergy (n = 12) validating the instrument draft. We describe the results of these development steps as well as future steps to produce the final, validated form of the measure. The resultant 91-item measure is intended for parents of children ages two to 12 years. This measure fills a unique need in the field by leveraging key stakeholders' perspectives to develop and subsequently validate a novel parent-reported measure of parent and family food allergy impacts of children ages two to 12 years old.

Emotion dysregulation in autism: Severity and correlates in early childhood.

Emotion dysregulation (ED) is common and severe in older autistic youth, but is rarely the focus of early autism screening or intervention. Moreover, research characterizing ED in the preschool years (when autism is typically diagnosed) is limited. This study aimed to characterize ED in autistic children by examining (1) prevalence and severity of ED as compared to children without an autism diagnosis; and (2) correlates of ED in autistic children. A sample of 1864 parents (Mean child age = 4.21 years, SD = 1.16 years; 37% female) of 2-5 year-old children with (1) autism; (2) developmental concerns, but no autism; and (3) no developmental concerns or autism completed measures via an online questionnaire. ED was measured using the Emotion Dysregulation Inventory-Young Child, a parent report measure characterizing ED across two dimensions: Reactivity (fast, intense emotional reactions) and dysphoria (low positive affect, sadness, unease). Autistic preschoolers, compared to peers without developmental concerns, had more severe ED (+1.12 SD for reactivity; +0.60 SD for dysphoria) and were nearly four and three times more likely to have clinically significant reactivity and dysphoria, respectively. Autistic traits, sleep problems, speaking ability, and parent depression were the strongest correlates of ED in the autism sample. While more work is needed to establish the prevalence, severity, and correlates of ED in young autistic children, this study represents an important first step. Results highlight a critical need for more high-quality research in this area as well as the potential value of screening and intervention for ED in young autistic children.

Measuring Children’s Developmental Status in China Using the ECDI2030: Comparing with Direct Assessment and Teacher Report

AbstractThis study compared different approaches for monitoring progress towards Sustainable Development Goal Target 4.2, which focuses on the proportion of children who are developmentally on track. UNICEF’s Early Childhood Development Index 2030 (ECDI2030), a parent report measure, was compared with a corresponding direct assessment measure using a sample of children aged 3 and 5 (N = 309; 154 girls) in China at two time points. In the second wave, the study also investigated the correlations and agreement between the ECDI2030 and a teacher report measure for children’s development. Although Cronbach’s alpha indicated that both adult report measures had lower reliability, McDonald’s omega showed comparable reliability among the three measures when the assumption of tau-equivalence was relaxed. Moreover, both adult report measures tended to overestimate children’s developmental levels, and were less effective in capturing the development of older children compared to the direct assessment measure. The correlations between parent report and direct assessment were significant for both girls and boys, urban children, and children from higher socioeconomic quartiles in both waves. Parents’ education levels did not substantially moderate the correlations. Moreover, parent report may not predict children’s development as effectively as direct assessment. Compared to parent report, teacher report was less effective in differentiating children’s development across socioeconomic status and urbanicity. Parent and teacher judgements were more consistent on children’s early learning competencies than on children’s motor and psychosocial skills. Implications of the findings for population-based measurement of early childhood development are discussed.

Telehealth pivotal response treatment parent training: A pilot study

ABSTRACT The purpose of this pilot study was to assess the feasibility, benefits, and acceptability of a telehealth Pivotal Response Treatment (PRT) parent training program. Autistic children (N = 21; 2–6 years) with language delay and their parents participated in the 12-week program. Parent-report measures and parent–child home videos were used to assess changes in parent PRT treatment adherence and child language. Changes in child affect were used to assess intervention acceptability. At the end of the program, treatment adherence improved, and children made more intelligible utterances. Child communication skills as measured by parent-report on several standardized questionnaires also improved. This study demonstrates the benefits of telehealth parent training for improving child language and parent PRT implementation. Our findings also provide preliminary evidence of PRT acceptability. Randomized controlled trials are an important next step to evaluate the impact of telehealth parent training as a tool for overcoming barriers to accessing intervention.

Extended Child Adjustment and Parent Efficacy Scale (CAPES): inclusion of the peer relationship problems subscale and updated psychometric properties

ABSTRACT Objective The current study aimed to validate an extended version of a frequently used parent-report measure of child functioning and parent self-efficacy, the Child Adjustment and Parent Efficacy Scale (CAPES). Method A 5-item peer relationship problems subscale was developed to address the original measure’s limitation in assessing the quality of children’s peer relationships. Three additional items were added to the parent self-efficacy subscale that measures parents’ confidence in supporting children to manage peer relationship problems. Utilising data from 2,272 Australian parents of primary-school-aged children, we evaluated the psychometric properties of the extended CAPES through conducting a confirmatory factor analysis, correlational analyses of relevant measures, and calculations of internal consistency. Results A three-factor structure of the child adjustment subscale was supported, which included three subscales, namely the original emotional maladjustment subscale, the original behavioural problems subscale, and the newly developed peer relationship problems subscale. The one-factor structure of the updated parent self-efficacy subscale was also supported. All subscales displayed acceptable to excellent internal consistency, and significant correlations with relevant measures. Conclusions The extended CAPES demonstrated good validity, reliability, and readability in this initial validation study. It has great potential to be used in both research and clinical practice.

An open pilot of GROW, a responsive parenting intervention for young children with brain injuries: preliminary efficacy

Purpose Families experience stress and burden following their child’s traumatic brain injury (TBI). Parenting styles are related to parent stress and child behavior problems and executive dysfunction. Parent-child interactions are shown to be a promising intervention target. We aimed to examine the preliminary efficacy of Gaining Real life skills Over the Web (GROW) in improving responsive parenting and reducing parent stress and depression in parents of children ages 0-4 who experienced TBI. Method Participants (n = 11) were parents of children four years old or younger, at the time of enrollment, and previously hospitalized overnight for TBI. Parent-reported measures included the Center for Epidemiologic Studies Depression Scale, Parenting Stress Inventory—Short Form, and Patient-Reported Outcomes Measurement Information System. Recordings of parent-child interactions were coded using the Play and Learning Strategies coding scheme. Study hypotheses were analyzed using independent samples t-tests, chi square analyses, and linear mixed models. Results Parental warmth (t=-3.64, p = 0.001, d=.97), responsiveness (t=-2.09, p = 0.047, d = 0.70), and verbal stimulation (t=-2.30, p = 0.029, d = 0.75) increased significantly from pretreatment to posttreatment. Improvements in parental warmth were maintained 3 months posttreatment (t=-3.15, p = 0.004, d = 1.16). There were no significant changes in depression, parental stress, or quality of life. Conclusions Results provide preliminary support for the efficacy of GROW in improving responsive parenting for children following early TBI. Future directions include tailoring GROW to better fit the needs of families and testing the utility through a randomized control trial.

Validation of the Parent-report Pandemic Anxiety Scale (PAS-P) in the context of COVID-19

To be able to develop effective policy and targeted support for children and young people, it is vital to develop and validate measures that enable us to understand what aspects of pandemics are associated with anxiety and stress across a wide age range. We examined the psychometric properties of the Pandemic Anxiety Scale– Parent-report (PAS-P), which measures levels of child and adolescent pandemic-related anxieties. Factor structure, reliability, and convergent and discriminant validity of the PAS-P was assessed in a convenience sample of parents/carers (N­ = 8410) over at three time points in 2020 when COVID-19 case rates and restrictions varied. Factor structure was identified via two exploratory factor analyses (EFAs; n = 5601 and n = 1005) and then tested using confirmatory factor analysis (CFA; n = 800), measurement invariance tests, and a longitudinal CFA (n = 1651). Two factors structure for disease and consequence anxiety were observed across both EFAs and were found to have good fit in the CFAs. The PAS-P demonstrated good internal consistency and discriminant validity, as well as partial scalar invariance in latent construct measurement across child age, gender, and time. The PAS-P is a robust parent-report measure of two distinct forms of pandemic-related anxiety, suitable for reporting on children and adolescents aged 4-16 years. Although the scale has been validated in the context of the COVID-19 pandemic, it is not limited to this specific pandemic and, with minor wording modifications, may be a reliable tool in other health epidemic contexts.

Maternal Depressive Symptoms and Risk for Childhood Depression: Role of Executive Functions

Offspring of mothers with depression are at increased risk for executive function (EF) deficits and later depressive symptoms, but limited studies have examined EF as an intermediary pathway. This study examined the role of EF in mediating the association between maternal and child depressive symptoms. Data were from a longitudinal birth cohort comprising 739 participants followed from the antenatal period for 12 years. Mothers completed the Edinburgh Perinatal Depression Scale at 26 to 28 weeks' gestation and at 3 and 24 months postpartum. At ages 8.5 to 10 years, children self-reported using the Children's Depression Inventory, Second Edition. Task-based and parent-reported EF measures were collected at 4 time points between 3.5 and 8.5 years. Latent growth curve models examined antenatal depressive symptoms and their trajectory in contributing to cold (ie, cognitive) and hot (ie, affective) EFs. The extent to which EF mediated this association was then assessed. Maternal depressive symptoms did not directly predict depressive symptoms in late childhood. Antenatal depressive symptoms predicted lower cold EF (β=-.13, 95% CI [-0.25,-0.004]) and hot EF (β=-.26, 95% CI [-0.38,-0.15]). Deficits in cold EF (β=-.26, 95% CI [-0.41,-0.11]) acted as an intermediary path to depressive symptoms, whereas hot EF mediated the association between maternal and child depressive symptoms, forming an indirect path that accounted for 37.5% of the association. Deficits in hot EF may be a pathway in explaining the intergenerational transmission of depression. This finding suggests fostering EF skills as a potential strategy for at-risk children. Growing Up in Singapore Towards Healthy Outcomes (GUSTO); https://clinicaltrials.gov/study/NCT01174875; NCT01174875. We worked to ensure that the study questionnaires were prepared in an inclusive way. We worked to ensure sex and gender balance in the recruitment of human participants. We worked to ensure race, ethnic, and/or other types of diversity in the recruitment of human participants. One or more of the authors of this paper self-identifies as a member of one or more historically underrepresented racial and/or ethnic groups in science. We actively worked to promote sex and gender balance in our author group. We actively worked to promote inclusion of historically underrepresented racial and/or ethnic groups in science in our author group. While citing references scientifically relevant for this work, we also actively worked to promote sex and gender balance in our reference list. While citing references scientifically relevant for this work, we also actively worked to promote inclusion of historically underrepresented racial and/or ethnic groups in science in our reference list. The author list of this paper includes contributors from the location and/or community where the research was conducted who participated in the data collection, design, analysis, and/or interpretation of the work. One or more of the authors of this paper self-identifies as a member of one or more historically underrepresented sexual and/or gender groups in science. One or more of the authors of this paper self-identifies as living with a disability.

Executive Function and Adherence in Children and Adolescents Living with HIV: Evidence from the HIV-associated Neurocognitive Disorders in Zambia (HANDZ) Study.

Executive function (EF) may be impaired in people with human immunodeficiency virus (HIV) infection, and poor EF may affect medication adherence. However, there is little data on EF in children with HIV in sub-Saharan Africa. 208 children/adolescents with perinatally acquired HIV and 208 HIV-exposed uninfected controls were recruited in Zambia for this prospective cohort study. EF was measured using performance-based, self-report, and parental report measures. Adherence over one year of follow-up was assessed through questionnaires and viral load measurement. Children with HIV performed significantly worse on all three measures of EF. Lower parental rating of EF was associated with poorer antiretroviral therapy adherence (OR: 1.5, 95% CI = 1.02 - 2.2, p = 0.04). Children with HIV have EF impairments which may lead to consequences like poor medication adherence and treatment failure. Interventions to improve EF or compensate for impaired EF may be necessary in this population.

Fears and Worries at Nighttime in Young Children: Development and Psychometric Validation of a Parent-Report Measure (FAWN-YC).

This paper outlines the development and psychometric evaluation of the Fears and Worries at Nighttime-Young Children (FAWN-YC) scale; a parent-rated measure for children aged 3-5years. Based on previous literature, it was hypothesised that the measure would be represented by a six-factor solution, with four clusters of fear types and two behavioural manifestations of fears. Exploratory factor analysis (EFA; N = 436) and confirmatory factor analysis (CFA; N = 383), resulted in a final 17 items that loaded onto 3 factors: Nighttime Fear Focus (8 items, α = 0.92), Bedtime/Sleep Avoidance and Interference (5 items, α = 0.90), and Dark Fear (4 items, α = 0.88). Evidence of convergent validity was found through strong associations between the total score and subscales of the FAWN-YC with measures of child anxiety, fear, sleep, externalizing and conduct problems. Furthermore, there was support for divergent validity (through a very weak to no relationship with a measure of prosocial behaviours), and evidence for temporal stability was also established with 2-week test-retest reliability. Overall, the results provide strong preliminary evidence for the reliability and validity of the FAWN-YC total score and subscales. Implications for the use of the measure in research and clinical practice are discussed.

Evaluating Emotional Eating in Children from the Perspective of Parents: Psychometric Properties of the Parent Version of the Emotional Eating Scale Adapted for Children and Adolescents.

Emotional eating is associated with adverse health outcomes in children, including elevated weight status. Currently, there is not a well-validated parent-report measure of emotional eating for young children. This study assessed the reliability and validity of the 10-item parent version of the Emotional Eating Scale Adapted for Children and Adolescents (EES-C) Short-Form. The participants were 207 parents and 144 children from the southern United States. They completed the parent- and child-report EES-C Short-Form and responded to measures related to child eating behaviors, mood, and gratitude. The parent-report EES-C Short-Form demonstrated good internal consistency reliability (Cronbach's alpha = 0.94). Test-retest reliability was also supported, as evidenced by a medium correlation (ICC = 0.56, p < 0.001) between parent-rated emotional eating across two time points. Additionally, the measure demonstrated a significant correlation with a scale of emotional overeating (r = 0.25, p < 0.001)-a theoretically related construct. Supporting discriminant validity, the measure was not significantly related to a measure of parent-reported gratitude (r = 0.07, p = 0.30). A unidimensional model provided good fit for the data (CFI = 0.997, SRMR = 0.046). The results from the current study provide preliminary evidence supporting the reliability and validity of the parent version of the EES-C Short-Form. For the purpose of screening children in school or primary care settings, the EES-C Short-Form may be practical and helpful in identifying children who may be at risk of developing adverse health outcomes or more-severe eating disorder pathology.

Impact of an 8-Week, Intensive Summer Treatment Program on Improving Coercive Caregiver-Child Interactions Among Children With and Without Callous Unemotional Traits

The coercive process is the primary explanatory mechanism implicated in the maintenance of disruptive behavior disorders (DBDs), and understanding the coercive cycle may be especially important for caregivers with children with elevated callous-unemotional (CU) traits. The purpose of the current pilot study is twofold. First, we assessed whether the Summer Treatment Program (STP), an established intensive behavioral intervention for children with DBDs, reduced coercive caregiver-child interactions using a novel parent-report measure of coercive interactions. We then sought to assess whether the STP improved coercive caregiver-child interactions among children with elevated CU traits (i.e., moderated the effect). Thirty-three caregivers and their children who had been diagnosed with DBDs participated in the STP, which involved the children receiving behavior modification for 9 hours per day and caregivers attending a weekly behavioral parent training group session over the course of 8 weeks. Children (Mage = 8.51 years old, SD = 1.70, range 6–12) were primarily male (78.5%) and Hispanic/Latine (84.8%). A linear multilevel model revealed a statistically significant improvement in caregiver-child coercive interactions from pre- to posttreatment. However, child pretreatment CU traits moderated this effect such that families with children with elevated CU traits did not show improvement in caregiver-child coercive cycles following treatment. Results suggest the benefits of intensive behavioral treatment on the caregiver-child coercive cycle for many families, but the need to modify the intensity or dose of treatments for those with children with DBDs and elevated CU traits.

Attention and executive functioning in children and adolescents treated for high-risk acute lymphoblastic leukemia: A report from the Children's Oncology Group (COG).

Survivors of childhood B-acute lymphoblastic leukemia (B-ALL) are at risk for difficulties with attention and executive functioning (EF) as a late effect of treatment. The present study aimed to identify treatment and demographic factors associated with risk for difficulties with EF in youth treated for high-risk B-ALL. Children and adolescents with B-ALL treated on Children's Oncology Group (COG) protocol AALL0232 were randomized to high-dose or escalating-dose methotrexate (MTX), and either dexamethasone or prednisone during the induction phase. Neuropsychological functioning was evaluated via protocol AALL06N1, including performance-based and parent-report measures, for 177 participants (57% female, 81% white; mean age at diagnosis=8.4 years; SD=5.0) 8-24 months following treatment completion. Mean scores for all attention and EF measures were within the average range, with no significant differences as a function of MTX delivery or steroid treatment (all p >0.05). In multivariable models, participants with US public insurance exhibited significantly greater parent-reported EF difficulties than those with US private or non-US insurance (p ≤ 0.05). Additionally, participants diagnosed under 10 years of age performed significantly more poorly on measures of attention (i.e., continuous performance task, p ≤ 0.05) and EF (i.e., verbal fluency and tower planning task, p≤0.05). For survivors of pediatric B-ALL, treatment-related factors were not associated with attention or EF outcomes. In contrast, outcomes varied by demographic characteristics, including age and insurance type, an indicator of economic hardship. Future research is needed to more directly assess the contribution of socioeconomic status on cognitive outcomes in survivors.

The Early Pretending Survey (EPS): A reliable parent-report measure of pretense type development for 4- to 47-month-olds

We present a new 18-item parent-report measure of pretense development for 4- to 47-month-olds: the Early Pretending Survey (EPS). Study 1 (N = 231) demonstrated good internal reliability, and a strongly correlated 2-factor structure for 4- to 47-month-olds. Study 2 (N = 587) also showed good internal reliability, and found the two factors loaded onto a latent second-order Pretend Play factor. Study 3 (N = 84) found the EPS correlated with a researcher-led pretense experiment, however it no longer correlated when controlling for age. Additional analyses using subsamples of parents from Studies 2 and 3 showed good inter-observer reliability between parents (N = 29), and good longitudinal stability after 6 months (N = 196). Additional analyses combining Study 1–3 participants (N ≤ 902) found no item functioning differences across demographic variables, including child age, country, and socio-economic factors. Children’s EPS scores increased as they aged; girls had higher scores than boys; and children of younger parents had higher scores. Finally, we examined the age patterns of each EPS item, demonstrating when 25 %, 50 %, and 75 % of children were predicted to pass each pretense type. The EPS is useful for researchers to better understand how pretending relates to other areas of development (e.g., cognition, language). The EPS could also help parents, early years educators, and children’s media professionals decide which pretense types to use with children of different ages.

Parent-reported outcome measures evaluating communication in individuals with rare neurodevelopmental disorders: A systematic review.

Communication impairments are a leading concern for parent caregivers of individuals with rare neurodevelopmental disorders (RNDDs). Clinical trials of disease modifying therapies require valid and responsive outcome measures that are relevant to individuals with RNDDs. Identifying and evaluating current psychometric properties for communication measures is a critical step towards the selection and use of appropriate instruments. This systematic review offers (1) a description of parent-reported communication measures and (2) evidence for their psychometric properties, in RNDDs. The systematic review protocol was registered in the International Prospective Register of Systematic Reviews (PROSPERO; CRD42022334649). MEDLINE (Ovid), Embase, PsychINFO, Web of Science, CINAHL Plus, Cochrane Library, ClinicalTrials.gov, the Australian New Zealand Clinical Trials Registry were searched from inception to August 2023. Methodological assessment of quality was completed using the COnsensus-based Standards for the selection of health status Measurement INstruments (COSMIN) checklist. Parent-reported measures used in observational studies and clinical trials were identified. Data on utility, reliability and validity for RNDDs were extracted. Sixteen parent-reported communication measures were used in RNDD research, the Vineland Adaptive Behavior Scales being most commonly used. Validation data in RNDDs were identified for six of these measures. Limitations related to sample size or the scope of psychometric testing. Many communication measures have been used for RNDDs but there are few data validating their use. Valid and reliable methods of measuring communication in persons with RNDDs is a priority for future high-quality clinical trials. What is already known on the subject Communication is a critical domain for families with a child with a rare neurodevelopmental disorder (RNDD). Validated outcome measures are essential for accurate evaluation and interpretation of responses to treatments in clinical trials. What this paper adds to existing knowledge We identified 16 parent-reported communication measures that have been used with RNDDs, but only six measures had validation data for at least one RNDD. High quality evidence is accumulating, with all validation studies in this review published between 2020 to 2023. Modifications of existing measures may be required to assess communication for RNDDs. What are the clinical implications of this work? This systematic review catalogues the available psychometric data for communication measures and indicates an ongoing need for new validation studies to ensure they are fit-for-purpose for upcoming clinical trials in RNDDs. This review will inform the selection of communication measures for clinical trials and research studies.

Sensory symptoms associated with autistic traits and anxiety levels in children aged 6–11 years

BackgroundAutism spectrum conditions (ASC) and quantitative autistic traits (QATs) are associated with sensory symptoms, which may contribute to anxiety and adversely affect social and cognitive development. Although sensory symptoms can occur across all senses, the relative roles of specific sensory modalities as contributors to the autistic phenotype and to anxiety are not well understood. The objective of this study was to examine which sensory symptoms were most predictive of high anxiety.MethodsWe recruited 257 female primary caregivers of children aged 6 to 11 years (49% girls) to a questionnaire study comprising parent-report measures for classical QATs (social, communicative, and rigid), autism-related sensorimotor symptoms (visual, auditory, tactile, olfactory, gustatory, vestibular, proprioceptive, and motor), and anxiety symptoms. First, Bayesian stochastic search variable selection (SSVS) was used to identify the most probable sensorimotor predictors of specific QATs as well as diagnosed ASC. Then, the selected predictors were used in another SSVS, using anxiety symptoms as a dependent variable, to identify which of the autism-relevant sensorimotor symptoms were most robustly predictive of anxiety. Finally, the effect sizes of anxiety-related sensory symptoms were estimated with linear regressions.ResultsWe found that auditory symptoms and motor difficulties were most predictive of ASC diagnosis. Developmental motor difficulties were also strongly related to all individual QATs, whereas auditory symptoms were more selectively predictive of rigid traits. Tactile symptoms robustly predicted social interaction QATs, and proprioceptive symptoms predicted communicative QATs. Anxiety outcomes were most strongly predicted by difficulties with auditory and olfactory processing.ConclusionsThe results support the clinical importance of being alert to complaints about sounds and hearing in neurodevelopmental populations, and that auditory processing difficulties may be evaluated as an early marker of poor mental health in children with and without diagnosed autism. Olfactory processing differences appeared to be an anxiety marker less strongly associated with ASC or QATs, while motor difficulties were highly autism-relevant but not equally strongly associated with anxiety outcomes. We suggest that future studies may focus on the mechanisms and consequences of neurodevelopmental central auditory processing dysfunction and its potential relationship to anxiety disorders.