Expanding the phenotype of cerebellar-facial-dental syndrome: Two siblings with a novel variant in BRF1.

Cerebellofaciodental syndrome (MIM #616202) is an autosomal recessive condition characterized by intellectual disability, microcephaly, cerebellar hypoplasia, dysmorphic features, and short stature. To date, eight patients carrying biallelic BRF1 variants have been reported. Here, we describe two siblings with congenital microcephaly and corpus callosum hypoplasia, pre and postnatal growth retardation, congenital heart defect and severe global developmental delay. We also detected additional findings not previously reported in this syndrome, including bilateral sensorineural hearing impairment and inner ear malformation. Whole exome sequencing identified a novel homozygous missense variant (c.654G>C, p.[Trp218Cys]) in BRF1, predicted to affect the protein structure. Expression assessment showed extremely low BRF1 protein expression caused by the identified variant, supporting its causal involvement. The description of new patients with cerebellofaciodental syndrome is essential to better delineate the phenotypic and genotypic spectrum of the disease.

Effects of a scalable home-visiting intervention on child development in slums of urban India: evidence from a randomised controlled trial.

BACKGROUND: An estimated 63.4 million Indian children under 5 years are at risk of poor development. Home visits that use a structured curriculum to help caregivers enhance the quality of the home stimulation environment improve developmental outcomes. However, achieving effectiveness in poor urban contexts through scalable models remains challenging. METHODS: Using a cluster randomised controlled trial, we evaluated a psychosocial stimulation intervention, comprising weekly home visits for 18 months, in urban slums of Cuttack, Odisha, India. The intervention is complementary to existing early childhood services in India and was run and managed through a local branch of a national NGO. The study ran from August 2013 to July 2015. We enrolled 421 children aged 10-20 months from 54 slums. Slums were randomised to intervention or control. Primary outcomes were children's cognitive, receptive language, expressive language and fine motor development assessed using the Bayley-III. Prespecified intent-to-treat analysis investigated impacts and heterogeneity by gender. TRIAL REGISTRATIONS: ISRCTN89476603, AEARCTR-0000169. RESULTS: Endline data for 378 (89.8%) children were analysed. Attrition was balanced between groups. We found improvements of 0.349 of a standard deviation (SD; p = .005, stepdown p = .017) to cognition while impacts on receptive language, expressive language and fine motor development were, respectively, 0.224 SD (p = .099, stepdown p = .184), 0.192 SD (p = .085, stepdown p = .184) and 0.111 (p = .385, stepdown p = .385). A child development factor improved by 0.301 SD (p = .032). Benefits were larger for boys. The quality of the home stimulation environment also improved. CONCLUSIONS: This study shows that a potentially scalable home-visiting intervention is effective in poor urban areas.

Evolution of the wealth gap in child development and mediating pathways: Evidence from a longitudinal study in Bogota, Colombia.

Large gaps in cognition and language on the Bayley-III between the top and bottom household wealth quartiles in 1,330 children aged 6-42 months in a representative sample of low- and middle-income families in Bogota were previously shown. Maternal education and the home environment mediated these wealth effects, whereas height-for-age mediated a small amount of the language deficit only. At ages 6-8 years, we relocated 72% of the children and assessed their IQ on the WISC-V, school achievement, and behavior to investigate the evolution of the wealth gaps and potential mediators. The wealth gap in IQ at 6-8 years was significantly larger than that in a factor combining Bayley-III language and cognition at 6-42 months; whereas the gap in achievement was larger but not significantly. Moreover, in cross-sectional analysis, the IQ gap increased from 6 to 8 years reaching over 1 SD. In contrast, the gap in behavior was not significant in either childhood stage. Parental education and early home environment remained major mediators of the wealth gap in IQ and achievement at 6-8 years; later home environment and attending private education also had an effect; and early height-for-age was no longer significant. The home environment partly mediated the effect of parental education on wealth. All mediators combined explained most of the variance in the wealth gap; the remaining gaps being not significant. Results highlight the importance of the early home environment and suggest that interventions focusing on that should have long-term benefits. Also, continued intervention through to 8 years may be desirable. A video abstract of this article can be viewed at https://youtu.be/_U53iXNww3I.

[Context and results of early childhood development in 12 to 59 months old children living in Mexico]. / Contexto y resultados del desarrollo infantil temprano en niños y niñas de 12 a 59 meses en México.

OBJECTIVE: To evaluate early childhood development (ECD) and its determinants in 12 to 59 months old children residents of communities <100 000 inhabitants. MATERIALS AND METHODS: The Encuesta Nacional de Salud y Nutrición of communities <100 000 inhabitants (Ensanut 100k) evaluated language level, access to ECD care services and standardized indicators of the eight quality of the development environment. We report indicator prevalence and standardized language scores according to variables of interest. RESULTS: 20.7% of children attended eight wellchild care visits within the first year of life, 13.0% received an ECD assessment, 75.0% receive support for learning, 23.4% have books and 57.7% experiment violent discipline. Improved language levels are associate with socioeconomic capacities, maternal education, preschool attendance, support for learning and household books. Children exposed to more protective factors present a language level 1.5 standard deviations higher than their peers exposed to more risk factors. CONCLUSIONS: There is a need to increase the coverage of ECD care services and to improve early development opportunities within households.

OBJETIVO: Evaluar el desarrollo infantil temprano (DIT) y sus determinantes en niños/as de 12 a 59 meses residentes en localidades de menos de 100 000 habitantes. MATERIAL Y MÉTODOS: . La Encuesta Nacional de Salud y Nutrición en localidades con menos de 100 000 habitantes (Ensanut 100k) evaluó el nivel de lenguaje, acceso a servicios de atención al DIT e indicadores de calidad del contexto de desarrollo. Se estiman prevalencias de indicadores y puntajes estandarizados de lenguaje según variables de interés. RESULTADOS: 20.7% de los niños/as asistió a ocho consultas del niño sano en su primer año, 13.0% recibió evaluación de DIT, 75.0% recibe apoyo al aprendizaje, 23.4% cuenta con libros y 57.7% sufre disciplina violenta. Mejores niveles de lenguaje se asocian con las capacidades económicas, escolaridad materna, asistencia a preescolar, apoyo al aprendizaje y acceso a libros. Los niños/as expuestos a más factores protectores presentan nivel de lenguaje 1.5 DE mayor que en niños/as con más factores de riesgo. CONCLUSIONES: Se requiere aumentar la cobertura de atención al DIT y mejorar las oportunidades de desarrollo en hogares.

Observing home-visiting quality at scale with the home visit rating scales and a supervisor checklist in Peru.

As home-visiting programs expand, there is a need to develop cost-effective tools to monitor their quality at scale. We compare the Home Visit Rating Scales (HOVRS), an instrument to measure home-visit process quality widely used in the United States, to a checklist designed for the Peruvian national home-visiting program, Cuna Mas. Both instruments were administered to a sample of 554 home visits and an equal number of mother-child dyads by their 176 home visitors. While the HOVRS was scored on video recordings of the visits by trained coders, the checklist was scored live by Cuna Mas supervisors. We assessed the validity of both measures in their first application in rural Peru, compared their performance, and selected a subset of items in the checklist to propose a simplified, shorter, and more cost-efficient instrument. To this end, we reorganized checklist items into constructs that best mapped those covered by the HOVRS and selected the highest performing according to predefined criteria. We found that both the HOVRS and the checklist had high reliability and acceptable levels of validity. We argue that the simplified checklist could prove useful for quality monitoring of service delivery of at-scale home-visiting programs and as a tool to support in-service training.

A medida que los programas de visita a casa se expanden, hay una necesidad de desarrollar herramientas de costo eficaz para vigilar la calidad de los mismos a una escala. Comparamos las Escalas de Evaluación de Visitas a Casa (HOVRS), un instrumento para medir la calidad de los procesos de visita a casa ampliamente usado en los Estados Unidos, con una lista de control diseñada para el programa nacional de visitas a casa de Perú, Cuna Mas. Los 176 visitadores a casa le administraron ambos documentos a un grupo muestra de 554 visitas a casa y a un número equivalente de díadas mamá-niño. Los codificadores entrenados evaluaron HOVRS por medio de grabaciones de video de las visitas, mientras que la lista de control fue evaluada en persona por los supervisores de Cuna Mas. Nosotros evaluamos la validez de ambas medidas en su primera puesta en práctica en el Perú rural, comparamos su rendimiento y seleccionamos un subgrupo de categorías en la lista de control para proponer un instrumento simplificado, más corto y de costo más eficaz. Con este fin, reorganizamos las categorías de la lista de control en segmentos que mejor cubren aquellas que están presentes en HOVRS y seleccionamos las que presentan mayor rendimiento de acuerdo con criterios predefinidos. Nos dimos cuenta de que tanto HOVRS como la lista de control presentaban una alta confiabilidad y niveles aceptables de validez. Sostenemos que la lista de control simplificada pudiera demostrar utilidad en cuanto a la calidad de la supervisión del ofrecimiento del servicio de programas de visitas a casa a una escala y como una herramienta para apoyar la capacitación en el empleo.

Du fait que les programmes de visite à domicile se développement, on voit un besoin de développer également des outils rentables afin de contrôler leur qualité à grande échelle. Nous comparons les Echelles d'Evaluation de la Visite à Domicile (en anglais Home Visit Rating Scales, soit HOVRS), un instrument destiné à mesurer la qualité du processus de visite à domicile largement utilisé aux Etats-Unis, à une checklist conçue pour le programme national péruvien de visite à domicile, Cuna Mas. Les deux instruments ont été utilisés avec un échantillon de 554 visites à domiciles et une nombre égal de dyades mère-enfant par leurs 176 visiteurs à domicile. Alors que la HOVRS était évaluée au moyen d'enregistrements vidéo des visites par des évaluateurs entraînés, la checklist a été évaluée en temps réel par les superviseurs de Cuna Mas. Nous avons évalué la validité des deux mesures dans leur première application dans le Pérou rural, comparé leur performance, et sélectionné un sous-groupe d'éléments de la checklist afin de proposer un instrument simplifié, plus court et plus rentable. Pour ce faire nous avons réorganisé les éléments de la checklist en structures cartographiant le mieux ceux couverts par les HOVRS et sélectionné les plus performants selon des critères prédéfinis. Nous avons trouvé qu'à la fois les HOVRS et la checklist faisaient preuve d'une fiabilité élevée et de niveaux de validité acceptables. Nous concluons que la checklist simplifiée pourrait s'avérer utile pour le contrôle de la qualité des prestations de service de programmes de visites à domicile à grande échelle et en tant qu'outil utilisé pour soutenir la formation continue.

Impacts 2 years after a scalable early childhood development intervention to increase psychosocial stimulation in the home: A follow-up of a cluster randomised controlled trial in Colombia.

BACKGROUND: Poor early childhood development (ECD) in low- and middle-income countries is a major concern. There are calls to universalise access to ECD interventions through integrating them into existing government services but little evidence on the medium- or long-term effects of such scalable models. We previously showed that a psychosocial stimulation (PS) intervention integrated into a cash transfer programme improved Colombian children's cognition, receptive language, and home stimulation. In this follow-up study, we assessed the medium-term impacts of the intervention, 2 years after it ended, on children's cognition, language, school readiness, executive function, and behaviour. METHODS AND FINDINGS: Study participants were 1,419 children aged 12-24 months at baseline from beneficiary households of the cash transfer programme, living in 96 Colombian towns. The original cluster randomised controlled trial (2009-2011) randomly allocated the towns to control (N = 24, n = 349), PS (N = 24, n = 357), multiple micronutrient (MN) supplementation (N = 24, n = 354), and combined PS and MN (N = 24, n = 359). Interventions lasted 18 months. In this study (26 September 2013 to 11 January 2014), we assessed impacts on cognition, language, school readiness, executive function, and behaviour 2 years after intervention, at ages 4.5-5.5 years. Testers, but not participants, were blinded to treatment allocation. Analysis was on an intent-to-treat basis. We reassessed 88.5% of the children in the original study (n = 1,256). Factor analysis of test scores yielded 2 factors: cognitive (cognition, language, school readiness, executive function) and behavioural. We found no effect of the interventions after 2 years on the cognitive factor (PS: -0.031 SD, 95% CI -0.229-0.167; MN: -0.042 SD, 95% CI -0.249-0.164; PS and MN: -0.111 SD, 95% CI -0.311-0.089), the behavioural factor (PS: 0.013 SD, 95% CI -0.172-0.198; MN: 0.071 SD, 95% CI -0.115-0.258; PS and MN: 0.062 SD, 95% CI -0.115-0.239), or home stimulation. Study limitations include that behavioural development was measured through maternal report and that very small effects may have been missed, despite the large sample size. CONCLUSIONS: We found no evidence that a scalable PS intervention benefited children's development 2 years after it ended. It is possible that the initial effects on child development were too small to be sustained or that the lack of continued impact on home stimulation contributed to fade out. Both are likely related to compromises in implementation when going to scale and suggest one should not extrapolate from medium-term effects of small efficacy trials to scalable interventions. Understanding the salient differences between small efficacy trials and scaled-up versions will be key to making ECD interventions effective tools for policymakers. TRIAL REGISTRATION: ISRCTN18991160.

Admixture in Latin America: geographic structure, phenotypic diversity and self-perception of ancestry based on 7,342 individuals.

The current genetic makeup of Latin America has been shaped by a history of extensive admixture between Africans, Europeans and Native Americans, a process taking place within the context of extensive geographic and social stratification. We estimated individual ancestry proportions in a sample of 7,342 subjects ascertained in five countries (Brazil, Chile, Colombia, México and Perú). These individuals were also characterized for a range of physical appearance traits and for self-perception of ancestry. The geographic distribution of admixture proportions in this sample reveals extensive population structure, illustrating the continuing impact of demographic history on the genetic diversity of Latin America. Significant ancestry effects were detected for most phenotypes studied. However, ancestry generally explains only a modest proportion of total phenotypic variation. Genetically estimated and self-perceived ancestry correlate significantly, but certain physical attributes have a strong impact on self-perception and bias self-perception of ancestry relative to genetically estimated ancestry.

Characterisation and expression of myogenesis regulatory factors during in vitro myoblast development and in vivo fasting in the gilthead sea bream (Sparus aurata).

The aim of this study was to characterise a primary cell culture isolated from fast skeletal muscle of the gilthead sea bream. Gene expression profiles during culture maturation were compared with those obtained from a fasting-refeeding model which is widely used to modulate myogenesis in vivo. Myogenesis is controlled by numerous extracellular signals together with intracellular transcriptional factors whose coordinated expression is critical for the appropriate development of muscle fibres. Full-length cDNAs for the transcription factors Myf5, Mrf4, Pax7 and Sox8 were cloned and sequenced for gilthead sea bream. Pax7, sox8, myod2 and myf5 levels were up-regulated during the proliferating phase of the myogenic cultures coincident with the highest expression of proliferating cell nuclear antigen (PCNA). In contrast, myogenin and mrf4 transcript abundance was highest during the differentiation phase of the culture when myotubes were present, and was correlated with increased myosin heavy chain (mhc) and desmin expression. In vivo, 30days of fasting resulted in muscle fibre atrophy, a reduction in myod2, myf5 and igf1 expression, lower number of Myod-positive cells, and decreased PCNA protein expression, whereas myogenin expression was not significantly affected. Myostatin1 (mstn1) and pax7 expression were up-regulated in fasted relative to well-fed individuals, consistent with a role for Pax7 in the reduction of myogenic cell activity with fasting. The primary cell cultures and fasting-feeding experiments described provide a foundation for the future investigations on the regulation of muscle growth in gilthead sea bream.

Identifying victimization clusters across people with intellectual disabilities: A latent class analysis.

BACKGROUND: Research has shown high rates of victimization among people with intellectual disabilities (ID), but victimization clusters have been barely explored. OBJECTIVE: We address the gap by examining how reported victimization experiences are grouped into different classes and identifying differences in the characteristics of the individuals in each class. METHODS: We conducted a cross-sectional self-report study with a sample of adults with an ID diagnosis (n = 260). We gathered data about the participants' victimization experiences and socio-demographics, and then subjected the data to latent class analysis (LCA). RESULTS: Three different classes were detected: High victimization (n = 27, 10.4 %); medium victimization, low sexual (n = 97, 37.3 %); and low victimization (n = 136, 52.3 %). The results highlight the experiences of sexual and physical victimization among the high-victimization class, in which women are overrepresented, and physical victimization among the medium-victimization class. The study also found that experiences of assault and bias attacks occur to a varying extent across all three classes. The LCA and poly-victimization methods showed substantial agreement but also differences when identifying the most victimized participants. In addition, we detected significant differences between classes in gender, type of school attended, place of residence, legal incapacity, type of support needed, secondary disability and poly-victimization status. CONCLUSION: We identified distinct underlying ingroup patterns of victimization and sociodemographic inter-class differences that contribute to a better understanding of victimization within the population in question. The results have prevention and intervention implications for caregivers and providers of services for people with ID.

Better at home or in residential care? Victimization of people with intellectual disabilities at the hands of caregivers.

BACKGROUND: People with intellectual disabilities often need assistance of some kind in their everyday life. Support needs can increase the risk of their victimization at the hands of professional and family caregivers. This paper explores the differences in caregiver victimization between participants living in residential care settings and those who are not. METHOD: A sample of 260 adults (59.2 % men) with an intellectual disability diagnosis were assessed using an adaptation of the Juvenile Victimization Questionnaire comparing prevalence, sum and variety scores. RESULTS: More than half of the sample (59.2 %) experienced some form of caregiver victimization throughout their lifetime, with physical abuse, verbal abuse, and neglect being the most frequently reported forms. Participants in residential care settings experienced significantly more caregiver victimization incidents and a broader range of victimization forms than their counterparts outside residential care. Significant differences were found based on the individuals' place of residence and gender. Details are provided on the last victimization incident, the perpetrator, the psychological and physical consequences of the victimization, and the reporting rates. CONCLUSIONS: This study outlines high rates of lifetime caregiver victimization, with those who live in residential care settings at particular risk. Further research is needed to gain a deeper understanding of the nuances of caregiver victimization and to prevent abuse in caregiving contexts.

Insulin-like growth factors effects on the expression of myogenic regulatory factors in gilthead sea bream muscle cells.

Gilthead sea bream (Sparus aurata) is a widely cultured fish; however, muscle development regulation is poorly known. Myogenesis can be activated by the myogenic regulatory factors (MRFs: MyoD, Myf5, myogenin and MRF4) and by endocrine signals from the growth hormone (GH)/insulin-like growth factors (IGFs) axis. We cultured gilthead sea bream myocytes to better understand the role of IGFs in muscle growth and differentiation through the regulation of MRFs expression. First, we studied the expression pattern during culture development of IGFs and IGF-I splice variants. The expression of igf-II was highest at the beginning of the culture and decreased when the cells started to differentiate, similarly to that observed for total igf-I. Igf-Ib showed a paralleled expression pattern as that of total igf-I, whereas igf-Ic was more stable during culture progression. Next, we analyzed the expression of IGFs and MRFs after incubation of cells at day 4 with GH, IGF-I, IGF-II and combinations of them at 3, 6 and 18 h. IGF-II increased myod2 and myf5 expression, genes involved in early muscle cell proliferation. Moreover, IGF-I caused an increase on mrf4 and myogenin expression, both involved in the later stages of development corresponding to differentiation. Regarding the regulation of IGFs expression, igf-I was stimulated by GH and IGF-II alone and combined, whereas igf-II expression was increased in response to IGF-I, suggesting a nice model of crossed regulation. Overall, the present model could be very useful to understand the different regulatory roles of these endocrine and transcription factors on fish myogenesis.

Experiences of Bias Victimization Among People With Intellectual Disabilities.

Research has drawn attention to the stigma and high rates of victimization among people with intellectual disabilities (ID) and an overlap between bias and non-bias victimization. However, studies of bias events or hate crime involving persons with ID are scarce. Using a self-report measure, we analyze lifetime bias victimization in a sample of 260 adults diagnosed with ID (age M = 41.7, SD = 12.0; 59.2% men), of whom 92 experienced bias victimization (age M = 41.2, SD = 11.9; 54.3% men), and compare the number of different types of victimization and the poly-victimization status between bias and non-bias victims. We also examine the following features: the victim, offender(s), victim-offender relationship, and location. Results show that bias victims experience a higher number of different types of victimization than non-bias victims (M = 7.74 and 4.96, respectively; p <.001, rrb=.37, ξ=.42) and are four times more likely to be poly-victims than non-bias victims (odds ratio [OR] = 4.37; 95% CI, p <.001). Most of the victims experienced a number of bias victimization episodes (89.1%; n = 82), and more than a quarter were injured (27.2%, n = 25) as a result of the victimization. All the bias victimizations by strangers were carried out in public places, as were most of the bias victimizations by acquaintances. Schoolmates and work colleagues perpetrated attacks at school and in the workplace, respectively. More than half of the victims, 63% (n = 58), spoke of the experience with someone, but only one reported it to the authorities. The paper provides a valuable descriptive and bivariate analysis of bias victimization of people with ID. The findings will help to understand bias violence against this population, highlighting the need for targeted and effective interventions.

A Remote Parenting Program and Parent and Staff Perspectives: A Randomized Trial.

OBJECTIVES: To assess impact and implementation of remote delivery of a parenting program following suspension of in-person visits during the coronavirus disease 2019 pandemic. METHODS: Impact of remote delivery of the Reach Up parenting program on parenting practices was evaluated by randomized trial in Jamaica. Mothers with children aged 5 to 24 months who met 1 of 7 at-risk criteria were enrolled at health centers. Participants were randomly assigned to intervention or control using random number tables generated by a statistician. Intervention comprised a manual for parents with illustrated play activities, phone calls, and short message service messages. The control group received usual care. Parent practices were measured using an adapted Family Care Indicators telephone-administered questionnaire by interviewers unaware of group assignment. Qualitative interviews were conducted with staff and parents in Jamaica and Brazil and staff in Ecuador to identify facilitators and barriers to remote delivery of Reach Up. RESULTS: Two hundred forty-seven participants were assessed at endline (control n = 130; intervention n = 117). Intervention increased parent activities that support child development, effect size 0.34 SD (95% confidence interval 0.03-0.53), and use of praise, odds 2 times higher with intervention. There were no benefits to interactive language or play materials. Qualitative results showed parents appreciated program continuation and felt motivated to help their child, and methods were acceptable to staff. Barriers included poor mobile phone access, difficulty contacting parents, and feedback limitations without in-person contact. CONCLUSIONS: Remote delivery methods have potential to contribute to scaling of parenting programs.

The Reach Up Parenting Program, Child Development, and Maternal Depression: A Meta-analysis.

BACKGROUND AND OBJECTIVES: Evidence is needed on effective approaches to build parents' ability to promote child development feasible in low- and middle-income countries. Our objective was to synthesize impact of the Reach Up early childhood parenting program in several low- and middle-income countries and examine moderation by family and implementation characteristics. METHODS: Systematic search using PubMed and Academic Search Elite/EBSCO Host. Randomized controlled trials of the Reach Up program from 1985 to February 2022 were selected. Data were extracted by 2 independent researchers. Primary outcomes were child cognitive, language, and motor development. Secondary outcomes were home stimulation and maternal depressive symptoms. We synthesized pooled effect sizes using random effect inverse-variance weighting and effect modification by testing pooled subgroup effect estimates using the χ2 test for heterogeneity. RESULTS: Average effect size across 18 studies ranged from 0.49 (95% confidence interval [CI] 0.32 to 0.66) for cognition, 0.38 (CI 0.24 to 0.51) for language, 0.27 (CI 0.13 to 0.40) for motor development, 0.37 (CI 0.21 to 0.54) for home stimulation, and -0.09 (CI -0.19 to 0.01) for maternal depressive symptoms. Impacts were larger in studies targeted to undernourished children, with mean enrollment older than age 12 months and intervention duration 6 to 12 months. Quality of evidence assessed with the Cochrane Assessment of Risk of Bias and GRADE system was moderate. Instruments used to assess child development varied. In moderator analyses, some subgroups included few studies. CONCLUSIONS: Reach Up benefits child development and home stimulation and is adaptable across cultures and delivery methods. Child and implementation characteristics modified the effects, with implications for scaling.

The creation of the Global Scales for Early Development (GSED) for children aged 0-3 years: combining subject matter expert judgements with big data.

INTRODUCTION: With the ratification of the Sustainable Development Goals, there is an increased emphasis on early childhood development (ECD) and well-being. The WHO led Global Scales for Early Development (GSED) project aims to provide population and programmatic level measures of ECD for 0-3 years that are valid, reliable and have psychometrically stable performance across geographical, cultural and language contexts. This paper reports on the creation of two measures: (1) the GSED Short Form (GSED-SF)-a caregiver reported measure for population-evaluation-self-administered with no training required and (2) the GSED Long Form (GSED-LF)-a directly administered/observed measure for programmatic evaluation-administered by a trained professional. METHODS: We selected 807 psychometrically best-performing items using a Rasch measurement model from an ECD measurement databank which comprised 66 075 children assessed on 2211 items from 18 ECD measures in 32 countries. From 766 of these items, in-depth subject matter expert judgements were gathered to inform final item selection. Specifically collected were data on (1) conceptual matches between pairs of items originating from different measures, (2) developmental domain(s) measured by each item and (3) perceptions of feasibility of administration of each item in diverse contexts. Prototypes were finalised through a combination of psychometric performance evaluation and expert consensus to optimally identify items. RESULTS: We created the GSED-SF (139 items) and GSED-LF (157 items) for tablet-based and paper-based assessments, with an optimal set of items that fit the Rasch model, met subject matter expert criteria, avoided conceptual overlap, covered multiple domains of child development and were feasible to implement across diverse settings. CONCLUSIONS: State-of-the-art quantitative and qualitative procedures were used to select of theoretically relevant and globally feasible items representing child development for children aged 0-3 years. GSED-SF and GSED-LF will be piloted and validated in children across diverse cultural, demographic, social and language contexts for global use.

Protocol for validation of the Global Scales for Early Development (GSED) for children under 3 years of age in seven countries.

INTRODUCTION: Children's early development is affected by caregiving experiences, with lifelong health and well-being implications. Governments and civil societies need population-based measures to monitor children's early development and ensure that children receive the care needed to thrive. To this end, the WHO developed the Global Scales for Early Development (GSED) to measure children's early development up to 3 years of age. The GSED includes three measures for population and programmatic level measurement: (1) short form (SF) (caregiver report), (2) long form (LF) (direct administration) and (3) psychosocial form (PF) (caregiver report). The primary aim of this protocol is to validate the GSED SF and LF. Secondary aims are to create preliminary reference scores for the GSED SF and LF, validate an adaptive testing algorithm and assess the feasibility and preliminary validity of the GSED PF. METHODS AND ANALYSIS: We will conduct the validation in seven countries (Bangladesh, Brazil, Côte d'Ivoire, Pakistan, The Netherlands, People's Republic of China, United Republic of Tanzania), varying in geography, language, culture and income through a 1-year prospective design, combining cross-sectional and longitudinal methods with 1248 children per site, stratified by age and sex. The GSED generates an innovative common metric (Developmental Score: D-score) using the Rasch model and a Development for Age Z-score (DAZ). We will evaluate six psychometric properties of the GSED SF and LF: concurrent validity, predictive validity at 6 months, convergent and discriminant validity, and test-retest and inter-rater reliability. We will evaluate measurement invariance by comparing differential item functioning and differential test functioning across sites. ETHICS AND DISSEMINATION: This study has received ethical approval from the WHO (protocol GSED validation 004583 20.04.2020) and approval in each site. Study results will be disseminated through webinars and publications from WHO, international organisations, academic journals and conference proceedings. REGISTRATION DETAILS: Open Science Framework https://osf.io/ on 19 November 2021 (DOI 10.17605/OSF.IO/KX5T7; identifier: osf-registrations-kx5t7-v1).

Characteristics and Prevalence of Lifetime Sexual Victimization Among a Sample of Men and Women with Intellectual Disabilities.

This study examines the prevalence and characteristics of sexual victimization experiences suffered by people with intellectual disabilities (ID). The sample consisted of 260 adults with an ID diagnosis (154 men and 106 women), ranging in age from 20 to 71 years (M = 41.69, SD = 12.05). The results showed that 35% of the sample had been sexually victimized at some point in their life. Being a woman, being declared legally incapable, and having comorbid mental health diagnoses were the most relevant characteristics of sexual victims with ID. Fondling was the most reported victimization, and rape showed the greatest gender differences, with a higher risk for women with ID of being raped compared to their male counterparts (odds ratio = 4.28, p < .05). The offender was generally a known male adult, and the percentage of incidents reported to the authorities was very low (7.4%). The psychological consequences of abuse were general distress, anxiety, and depressive symptomatology. Intervention and prevention programs targeting this population, as well as the training of professionals and caregivers, are essential to deal with sexual victimization and to protect and ensure the quality of life of people with ID.

Lifetime Victimization and Poly-Victimization in a Sample of Adults With Intellectual Disabilities.

People with intellectual disabilities appear to be more vulnerable to victimization. However, few studies have assessed victimization in these groups and those that do exist are highly heterogeneous and tend to focus only on specific forms of violence. This study attempts to shed light on the phenomenon of victimization among adults with intellectual disabilities by assessing victimization and poly-victimization throughout their life course. The sample consisted of 260 adults (154 men and 106 women) with an intellectual disability diagnosis, recruited from the Catalan Federation for People with Intellectual Disabilities (DINCAT) in Spain. They ranged in age from 20 to 71 years (M = 41.69, SD = 12.05). Victimization experiences were assessed by means of an adaptation of the retrospective version of the Juvenile Victimization Questionnaire (JVQ). The results showed that 96.9% of the participants had suffered some kind of victimization throughout their lives. With respect to the types of victimization, the most frequent were common victimization (87.7%), witnessing and indirect victimization (67.3%), victimization by caregivers (59.2%), sexual victimization (35%), and electronic victimization (23.5%). Women and early adults tended to experience higher rates of victimization. The poly-victimized group experienced 13 or more incidents of victimization throughout their lives. This study highlights the elevated rates of lifetime victimization among people with intellectual disabilities. It adds to previous evidence that special protection programs are required to address this issue and emphasizes the need for prevention and intervention measures in this particularly vulnerable group.

Early Stimulation and Nutrition: The Impacts of a Scalable Intervention.

Early childhood development is becoming the focus of policy worldwide. However, the evidence on the effectiveness of scalable models is scant, particularly when it comes to infants in developing countries. In this paper, we describe and evaluate with a cluster-Randomized Controlled Trial an intervention designed to improve the quality of child stimulation within the context of an existing parenting program in Colombia, known as FAMI. The intervention improved children's development by 0.16 of a standard deviation (SD) and children's nutritional status, as reflected in a reduction of 5.8 percentage points of children whose height-for-age is below -1 SD.

Assessing the performance of the Caregiver Reported Early Development Instruments (CREDI) in rural India.

Although many education and health programs aim to improve early childhood development, it is challenging to assess developmental levels of infants and small children through large household surveys. The Caregiver Reported Early Development Instruments (CREDI) has been proposed as an adaptable, practical, and low-cost instrument for measuring the developmental status of children under 3 years of age at scale, as it is relatively short and collected by caregiver report. This study employed the CREDI to measure the development of a sample of 994 children ages 22-35 months in rural India and compared the results to those obtained using the Bayley Scales of Infant and Toddler Development (Bayley-III), a reliable and widely used instrument, albeit one not always suited to large-scale data collection efforts given its length, cost, and complexity of administration. The CREDI validation exercise showed that caregivers can provide assessments in keeping with the more interactive (hence more time-consuming and training-intensive) Bayley-III instrument. Noteworthy, there was no indication that concordance of the instruments differed by education of the caregiver. This is important as it points to alternate feasible tools to measure child development outcomes through large-scale surveys.