Changes in the early communicative behaviors of young children with significant cognitive and motor developmental delays in a two-year span.

INTRODUCTION: This study examines longitudinal changes in communicative behavior of young children with significant cognitive and motor developmental delays (SDD) and determines their individual communicative trajectories. A second focus of this study is the relation of changes in communicative behavior with motor skills. METHODS: Data consists of codes resulting from a self-developed coding scheme used on observations of 23 children in three different settings and responses on a questionnaire. First, group trends were determined to find out whether communication-related variables tend to significantly change over the course of two years. Furthermore, these findings were contrasted with the individual trajectories of the children. Next, the association of initial communicative skills and (the acquisition of) specific motor skills with the change in their communicative functioning was studied. Wilcoxon Signed Ranks and correlational analyses were used to answer the research questions. RESULTS: Out of sixteen different variables related to communicative behavior, ten changed significantly over the course of two years. Children with more focus on prompt on the first datapoint showed a significantly larger increase of signs of functionality. Still, all children showed highly individual trajectories. Children with better motor skills on the first datapoint showed a significantly larger increase in communication rate. CONCLUSIONS: Results show that if a myriad of detailed variables are taken into account children with significant cognitive and motor developmental disabilities do change regarding their communicative functioning, but that they tend to all show unique developmental trajectories. Children with stronger skills in some aspects of communication and motor functioning, can be considered advantaged regarding their communicative development.

The effect of manual movements on stuttering in individuals with down syndrome.

PURPOSE: Stuttering may disrupt the speech of individuals with Down syndrome (DS), but standard stuttering therapies may be less adapted to these clients' needs. This study examined if their strength in gesture use can lead to the development of a new stuttering therapy. METHOD: Eighteen individuals with DS who stutter participated in an experimental task. During this task, they produced sentences in three different conditions: once without the ability to use gestures, once while moving the mouth of a hand puppet synchronous with their speech, and once while making beat gestures along their speech. Stuttering frequency was measured and compared between conditions while controlling for the effect of articulation rate. RESULTS: The experimental hand puppet and beat condition did not affect the stuttering frequency, but the covariate articulation rate did. An exploratory posthoc analysis showed that the articulation rate decreased during the experimental hand puppet and beat condition. Manual movements in the present task might only induce fluency through articulation rate reduction. However, analyses at individual level show significant interindividual variability. CONCLUSION: Individual analyses show that effect on stuttering frequency cannot be attributed entirely to articulation rate reduction and that beat gestures might still play a role. However, at this point, there is not enough direct evidence to implement beat gestures in current stuttering therapy.

Piloting attachment psychoeducation provided to parents of children with severe disabilities: Testing the feasibility of a digital micro-intervention.

BackgroundA digital micro-intervention offering attachment psychoeducational videos was explored regarding its feasibility in parents of children with severe disabilities. Method: A mixed-methods study (including daily diaries and one-time questionnaires) with 16 parents (75.0% female) of children with severe disabilities (up to 10 years of age) was done during a three-week intervention. Results: Parents were positive about the video series and almost no drop-out occurred. The videos stimulated their learning and thinking and offered parents some personal meaning. Preliminary efficacy tests showed no major changes in parents' parenting self-efficacy (PSE) or perceptions of statements on parent-child attachment. Conclusions: The study showed promising results regarding the micro-interventions' acceptability and implementation. The limited efficacy testing did not show major changes in parents' PSE. Further research is needed to investigate the differential relevance of the micro-intervention, based on parents' needs, as well as its optimal embeddedness in a broader intervention trajectory.

Best practice recommendations on the application of seclusion and restraint in mental health services: An evidence, human rights and consensus-based approach.

WHAT IS KNOWN ON THE SUBJECT?: Seclusion and restraint still regularly occur within inpatient mental health services. The Council of Europe requires the development of a policy on for instance age limits, techniques and time limits. However, they only define the outer limits of such a policy by indicating when rights are violated. Within these limits, many choices remain open. Staff and service managers lack clarity on safe and humane procedures. Research literature provides limited and contradictory insights on these matters. WHAT THIS PAPER ADDS TO EXISTING KNOWLEDGE?: The study resulted in 77 best practice recommendations on the practical application of restraint and seclusion as last resort intervention in inpatient youth and adult mental health services, including forensic facilities. To our knowledge, this is the first study in which the development of recommendations on this topic is not only based on scientific evidence, but also on an analysis of European human rights standards and consensus within and between expert-professionals and experts-by-experience. This approach allowed to develop for the first time recommendations on time limits, asking for second opinion, and registration of seclusion and restraint. WHAT ARE THE IMPLICATIONS FOR PRACTICE?: The 77 recommendations encourage staff to focus on teamwork, safety measures, humane treatment, age and time limits, asking for second opinion, observation, evaluation and registration when applying seclusion and restraint as last resort intervention. The implementation of the best practice recommendations is feasible provided that they are combined with a broad preventive approach and with collaboration between service managers, staff (educators) and experts-by-experience. Under these conditions, the recommendations will improve safety and humane treatment, and reduce harm to both service users and staff. ABSTRACT: INTRODUCTION: Seclusion and restraint still regularly occur within inpatient mental health services. Professionals lack clarity on safe and humane procedures. Nevertheless, a detailed policy on for instance age limits, techniques and time limits is required. AIM: We developed recommendations on the humane and safe application of seclusion, physical intervention and mechanical restraint in inpatient youth and adult mental health services, including forensic facilities. METHOD: After developing a questionnaire based on a rapid scientific literature review and an analysis of human rights sources stemming from the Council of Europe, 60 expert-professionals and 18 experts-by-experience were consulted in Flanders (Belgium) through a Delphi-study. RESULTS: After two rounds, all but one statement reached the consensus-level of 65% in both panels. The study resulted in 77 recommendations on teamwork, communication, materials and techniques, maximum duration, observation, evaluation, registration, second opinion and age limits. DISCUSSION: Combining an evidence, human rights and consensus-based approach allowed for the first time to develop recommendations on time limits, asking for second opinion and registration. IMPLICATIONS FOR PRACTICE: When combined with a preventive approach and collaboration between service managers, staff (educators) and experts-by-experience, the recommendations will improve safety and humane treatment, and reduce harm to service users and staff.

Describing the communicative profiles of young children with a significant cognitive and motor developmental delay.

The communicative behavior of young children with significant cognitive and motor developmental delays is generally considered to be limited, idiosyncratic and non-intentional. At present, changes between and within children over time regarding their communicative behavior are hard to detect. This article describes an exploratory observational study that draws on data from the first data point of 38 children who are participating in a longitudinal project on the developmental trajectories of children with significant cognitive and motor developmental delays. The aims of this study were to (a) describe the participants' communicative behavior in detail with communication-related variables that reflect differences across individuals, (b) create summarizing variables and (c) explore whether subgroups of children can be detected. A self-developed coding scheme and descriptive statistics combined with correlational analyses were used, followed by a principal component analysis and visual inspection of the outcome of this analysis. The within-group differences related to communicative behavior was characterized using 16 variables. Based on these variables, three overarching components were formulated: communication proficiency, Expressions of Discomfort and Rejection and Differentiation According to Focus. All participating children were found to be unique in terms of their component scores and the relationship among their component scores.

Gesture-speech integration is related to vocabulary skills in children with developmental language disorder, Williams syndrome and typical development.

BACKGROUND: Individuals with developmental language disorder or Williams syndrome are reported to use more gestures than individuals with typical development. However, these two groups differ considerably in visuospatial and language skills, two skills that are hypothesized to shape gesture rate. AIMS: We first examined whether children with both developmental language disorder and children Williams syndrome do indeed use more gestures. Our second aim was to disentangle the role of vocabulary and visuospatial skills in the use of supplementary gestures (i.e., containing unique information). To account for participant heterogeneity, analyses included both group comparisons and vocabulary and visuospatial skills at an individual level. As a third aim, the role of visuospatial skills was further examined in relation to gestures containing spatial content. METHODS & PROCEDURES: In a cross-sectional group design, three participant groups watched and then retold a cartoon: children with typical development (n = 25), children with developmental language disorder (n = 25) and children/young people with Williams syndrome (n = 14). Their narrations were transcribed and hand gestures were coded based on gesture-speech integration (redundant, adds information to particular lexical items, gives information that is entirely absent from speech) and spatial content. Participants' expressive vocabulary and visuospatial skills were measured. OUTCOMES & RESULTS: Between-group comparisons showed that individuals with developmental language disorder or Williams syndrome did indeed use more gestures. Poisson loglinear modelling demonstrated that a relative higher use of supplementary gestures was determined by lower expressive vocabulary skills. Neither the group distinction nor visuospatial skills shaped the supplementary gesture rate nor spatial gesture rate. CONCLUSIONS & IMPLICATIONS: Regardless of neurodevelopmental condition or typical development, a higher use of supplementary gestures was influenced by expressive vocabulary skills. Children with lower vocabulary skills spontaneously capitalized on the multimodality of communication to express constituents that were not present in their verbal speech. This finding is a promising starting point for future gesture intervention studies examining whether implicit modelling of gesture use can encourage gestures even more in these children and if this allows them to achieve higher linguistic complexity. On a methodological note, the observed intra-group skill variability demonstrates that group comparisons need to be complemented with correlational measures accounting for skills at an individual level. WHAT THIS PAPER ADDS: What is already known on the subject Children with developmental language disorder and children with Williams syndrome are more inclined to use gestures than typically developing children. Research conducted in adults with typical development points towards the role of lexical and visuospatial skills in gesture use, but it is unclear how these skills shape gesture use in children with atypical development. What this paper adds to existing knowledge This study compares the rate of gestures that convey meaning that is not expressed in speech between the three aforementioned populations. Novel is the inclusion of the group distinction, individual lexical skills, and visuospatial skills in one encompassing statistical model. What are the potential or actual clinical implications of this work? The inclination to use gestures that replace speech is related to lexical skills. Visuospatial skills do not seem to play a role and should not be considered as a factor when thinking about gesture intervention. Understanding how gestures relate to specific skills is a first step to understanding how gesture interventions can bolster language production.

Changes in the social-emotional functioning of young children with a significant cognitive and motor developmental delay across a two-year period.

Children with a significant cognitive and motor developmental delay (SDD) are vulnerable for the development of (future) behavioral and mental health problems. To support children within this target group, knowledge on their social-emotional development is necessary. Therefore, in this paper, an explorative assessment of the changes in the social-emotional functioning of children with SDD over a two-year period was done. Yearly semi-structured interviews with one or more primary caregiver(s) of 25 children were conducted and analyzed on 13 domains of social-emotional functioning, according to an adapted version of the Scale for Emotional Development - Revised (SED-R). This study showed a lot of variation in individual and group patterns of change across the two-year period. Charting the social-emotional development in this target group is challenging and requires further in-depth analysis of individual trajectories as well as more fine-grained and long-term data collection.

The social-emotional functioning of young children with a significant cognitive and motor developmental delay.

Children with a significant cognitive and motor developmental delay (SDD) are vulnerable for the development of (future) behavioral and mental health problems. This paper aims to assess the social-emotional functioning of these children, both globally and in various domains. Semi-structured interviews with one or more primary caregiver(s) of 45 children were conducted and analyzed on 13 domains of social-emotional functioning, according to the Scale for Emotional Development - Revised (SED-R). The SED-R scoring system was slightly adapted in the current study in order to elucidate more subtle differences between children. A general delayed social-emotional development was found, with children functioning within different phases across domains and certain domains generally showing higher or lower scores. To capture the emotional needs of children with SDD, a more disaggregated scoring system seems valuable so that both the global level and the level per domain can be taken in consideration in designing interventions.

The relation between gestures and stuttering in individuals with Down syndrome.

BACKGROUND: Evidence shows that neurotypical individuals who stutter use fewer gestures than those who do not stutter. Presently, no research exists about the interaction of stuttering and gestures in individuals with Down syndrome. METHOD: Twenty-nine individuals with Down syndrome (7-19 years) of whom 16 stuttered and 13 spoke fluently and 20 neurotypical children (3-10 years) of whom 8 stuttered and 12 spoke fluently participated in this study. In spontaneous speech transcriptions, stuttering events and gestures were coded. RESULTS: Comparisons of gesture frequency during stuttered and fluent speech inside the Down syndrome and neurotypical group show that the Down syndrome group uses significantly more gestures during stuttered than during fluent speech while no significant difference is seen in the neurotypical group. CONCLUSIONS: There is some preliminary evidence that individuals with Down syndrome try to compensate for their stuttering events, however, analyses on word level are necessary to confirm a successful compensation.

'We can't do it alone': Perceived social support in parents of children with a significant cognitive and motor developmental delay.

BACKGROUND: It is generally acknowledged that parent social support is an important target for intervention. To explore the specific needs of parents of young children with a significant cognitive and motor developmental delay (SDD), we aim to chart the sources and perceived sufficiency of the social support they receive. METHOD: Within the context of a broader project, 42 parents of a young child with SDD filled out a questionnaire on contextual factors, including information on social support. RESULTS: The majority of parents reported to receive (more than) sufficient support. Insufficiency is primarily reported with regard to practical support. Apart from the partner, grandparents of the child were the most common source of support. CONCLUSIONS: This study confirms the importance of professional support and work-life balance within this specific target group. Specific attention for practical support needs seems warranted considering the high basic care needs of these children.

Resilience and quality of life in young adults with a 22q11.2 deletion syndrome: a patient's perspective.

In the transition period between adolescence and young adulthood, individuals with 22q11.2DS are at an increased risk of developing severe psychiatric disorders. Various studies have focused on detecting risk factors, but until now protective factors are still understudied in 22q11.2DS. The current case-control study focuses on the role of resilience and quality of life (QoL) in young adults with 22q11.2DS and behavioural problems, in comparison with persons with an intellectual disability (ID) without a known genetic disorder. Self-report (and caregiver report) standardized questionnaires were used. Predictive general linear models were constructed to compare the resilience and quality of life across both groups (22q11.2DS vs ID-group) and to analyse the association between personal characteristics in both groups. Young adults with a 22q11.2DS show less resilience compared with both the general population norms and young adults with ID. Only a subscale of resilience (Acceptance of self and life) contributes to the reported level of QoL. Reported health problems are not related to resilience, but have an important effect on QoL. Our results suggest different factors are underlying resilience and the relation with QoL in 22q11.2DS and ID in general. These factors deserve more research and are important to take into account in clinical practice.

Lessons learned: A critical reflection on child- and contextual variables related to the development of children with a significant cognitive and motor developmental delay.

BACKGROUND: The scientific study of young children with a significant cognitive and motor developmental delay is challenging due to the children's complex disabilities and high demands on family life and professional support. We aim to critically reflect on the measurement and analysis of child- and contextual variables within an ongoing research project on these children's developmental trajectories. METHOD: The OJKO-project tracked the development of a sample of children (n = 45) between the age of 6 months and 4 years with a significant cognitive and motor developmental delay, in Belgium and the Netherlands. RESULTS: The complexity of the children's disabilities and daily life context, and subsequent challenges in measurement and analysis of variables, were confirmed and reflected upon. CONCLUSIONS: Due to the uniqueness and complexity of this target group, research should be characterized by creativity, perseverance and substantial modesty in the immediate generalization of results.

Influence of gestures on the intelligibility and comprehensibility of utterances with stuttering events in individuals with Down syndrome.

INTRODUCTION: Individuals with Down syndrome (DS) often stutter, which can affect their speech intelligibility. Previous research has shown that manual signs can enhance speech intelligibility and comprehensibility. It remains unclear to what extent spontaneous hand gestures, such as iconic and beat gestures, may enhance intelligibility and/or comprehensibility during utterances with stuttering events. METHODS: Eleven individuals with DS who stutter provided video-recorded speech samples. From these samples, 60 utterances containing a stuttering event were selected. In half of them, the stuttering events were accompanied by gestures; in the other half, the stuttering events were not accompanied by gestures. The samples were shown to 250 assessors who were blind to the study's goals. Each sample was shown in three visibility conditions: 1) video-with-audio, 2) video-with-audio but with the speaker's mouth covered and 3) audio-only. The assessors rated speech intelligibility on a 7-point Likert scale and transcribed the speech sample. The effect of gesture production, gesture type and the visibility conditions on comprehensibility and intelligibility was examined with a hierarchical multiple linear regression. RESULTS: When a speaker had used a gesture during a stuttering event, the Likert scale score increased with 0.47 and the accuracy of transcription with 9.07%. There was no difference in effect between the different gesture types. Despite the effect from gesture use, there was no effect of the visibility conditions on the Likert scale or transcription score. CONCLUSIONS: Gestures positively affect intelligibility and comprehensibility of utterances with stuttering events in individuals with DS by altering the speech production. The possibility of beat gestures as a therapy method should be examined, with caution for the development of maladaptive behaviours.

In search of a novel way to analyze early communicative behavior.

The aim of this study was to develop a coding scheme that enables researchers and practitioners to conduct a detailed analysis of the communicative behavior of young children with significant cognitive and motor developmental delays. Currently, there is a paucity of methods to do conduct such an analysis. For the study, video observations of three different scenarios from 38 children with significant cognitive and motor developmental delays aged between 12 and 54 months, were used. Findings from the video observations served as the primary means for development of the coding scheme, which comprises three main categories - context, partner behavior, and individual behavior - and several subcategories. The coding scheme was used to document the early expressive communicative behavior of persons with significant cognitive and motor developmental delays in a detailed manner. This fine-grained information is necessary to differentiate children based on their communicative abilities, to monitor their communicative development longitudinally, and to inform person-centered communicative interventions.

Using an ecological approach to grasp the complexity of social inclusion around a person with a disability.

BACKGROUND: Mapping social inclusion often focuses merely on a specific aspect of interpersonal relationships or community participation and is often executed from one perspective (either the person with a disability or a proxy). OBJECTIVE: This paper explores whether the ecological model of Simplican, Leader, Kosciulek and Leahy (2015) can serve as a basis to develop an instrument that maps a variety of influencing enabling and disabling variables on both interpersonal relations and community participation, including the input of persons with a disability, network members and professionals. METHODS: Directed content analysis was used to apply the ecological model to 14 cases of persons with a disability in independent supported living. RESULTS: The ecological model proved to be a useful framework to map enabling and disabling factors of social inclusion in individual cases. Additions were made to the model, including adding a new level of 'dynamics' to grasp the dynamic reality of social inclusion. Different informants gave partly overlapping information, but the integration of the three perspectives was an added value for understanding social inclusion in all its complexities. CONCLUSIONS: This paper serves as a first step in developing an in-depth instrument to map social inclusion.

An experiment on measuring awareness of stuttering in individuals with Down syndrome.

BACKGROUND: Awareness of stuttering is likely to depend upon the development of the metalinguistic skill to discriminate between fluent speech and stuttering and the ability to identify one's own speech as fluent or stuttered. Presently, little is known about these abilities in individuals with Down syndrome (DS). PURPOSE: This study investigates whether individuals with DS and typically developing (TD) children who stutter and who do not stutter differ in their ability to discriminate between fluent speech and stuttering. The second purpose of this study is to discover if this ability is correlated with their self-identification ability. METHOD: An experiment to investigate awareness with tasks for discrimination of stuttering and self-identification was developed. It was administered to 28 individuals (7-19 years) with DS, 17 of them stutter and 11 do not, and 20 TD children (3-10 years), 8 of them stutter and 12 do not. Skills to discriminate stuttering were compared between these groups and correlated with self-identification within these groups. The influence of stuttering severity and developmental/chronological age on their ability to discriminate was also investigated. RESULTS: The ability to discriminate does not differ significantly between the DS and TD group, but is highly influenced by developmental age. This ability correlates with self-identification but only for the TD individuals who speak fluently. CONCLUSION: The ability to discriminate matures around the age of 7 and conscious awareness may rely on this ability. Differences between the present findings and earlier studies suggest that differentiation in levels and types of awareness is warranted.

Looking back, looking forward: Methodological challenges and future directions in research on persons with profound intellectual and multiple disabilities.

BACKGROUND: Within the context of the Special Interest Research Group (SIRG) on Persons with Profound Intellectual and Multiple Disabilities (PIMD), researchers often discuss the methodological problems and challenges they are confronted with. The aim of the current article was to give an overview of these challenges. METHODS: The challenges are centred on six topics. These reflect the main components of a study's design: (a) participant demarcation, (b) participant recruitment, (c) data collection and instruments, (d) data analysis, (e) ethics/including the "voice" of persons with PIMD and (f) theoretical models. RESULTS: Next, to describing the specific challenges, possible solutions and pathways to address them are discussed. These are illustrated by recent studies by the authors and other researchers in the field. CONCLUSIONS: The current contribution wants to stimulate further discussion and exchange of ideas, and the development of creative research techniques.

Assistive technology for persons with profound intellectual disability: a european survey on attitudes and beliefs.

BACKGROUND: Persons with profound intellectual disability (PID) are mostly not able to use assistive technology (AT) independently. Caregivers play an important mediating role in implementing AT in the daily life of persons with PID. Both first-order barriers, extrinsic to caregivers, and second-order barriers, intrinsic to caregivers, influence the attitudes and behaviors of caregivers with regard to AT-use. It could be asked if increased knowledge on and experience with AT may impact the effect of first- and second-order barriers. This study investigated how knowledge and experience influence the professional caregivers' beliefs about which factors may impact the AT use in persons with PID and their intentions to use AT for persons with PID. METHODS: A questionnaire on the experienced limitations and successes in using AT was developed. The questionnaire was send to professionals working with or responsible for persons with PID in various countries in Europe. In total the answers of 195 respondents were included in this study. RESULTS AND CONCLUSIONS: This study's results demonstrate that AT is used for various reasons in persons with PID, mostly to support communication and interaction or for fun or relaxation. Based on the answers of the respondents can be concluded that both experience and knowledge of caregivers seem to influence first- and second-order barriers. Besides, a possibility to overcome the second-order barriers is to provide professionals with possibilities to increase their knowledge and experience.IMPLICATIONS FOR REHABILITATIONAT for persons with PID is mostly used for communication and interaction or for fun and relaxation.Professional caregivers belief that AT-use may positively influence various aspects in the life of persons with PID, especially communication and interaction, active engagement and participation in activities, and self-esteem of the person.Caregivers need to have sufficient experience in order to rate the barriers of AT-use as less limited in the group of persons with PID.In order to overcome the barriers experienced in implementing AT in persons with PID, knowledge of caregivers is essential.

Family-centered practices in home-based support for families with children with an intellectual disability: Judgments of parents and professionals.

BACKGROUND: The realization of the family-centered approach (FCA) in home-based support (HBS) for families with children with an intellectual disability (ID) in Flanders was investigated, and parents' and family workers' perspectives were compared. The relation between parents' educational level, the family worker's education, and his/her experience in HBS; and parents' and family workers' judgments on the realization of the FCA was considered. METHOD: Parents (N = 58 families) and family workers (N = 46) completed the helpgiving practices scale and the enabling practices scale. RESULTS: The FCA was largely present, parents rated its realization higher than family workers. Considering family workers' answers, parents' educational level appeared an important factor for parental autonomy. CONCLUSIONS: The study confirms recent research on the realization of the FCA. Including different perspectives, a nuanced view on the realization of the FCA was obtained. Further research on the concrete meaning, interpretation, and elaboration of the FCA is needed.

An investigation into the relationship between Quality of pantomime gestures and visuospatial skills.

While children with developmental language disorder or Williams syndrome appear to use hand gestures to compensate for specific cognitive and communicative difficulties, they have different cognitive strength-weakness profiles. Their semantic and visuospatial skills potentially affect gesture quality such as iconicity. The present study focuses on untangling the unique contribution of these skills in the quality of gestures. An explicit gesture elicitation task was presented to 25 participants with developmental language disorder between 7 and 10 years of age, 25 age-matched peers with typical development, and 14 participants with Williams Syndrome (8-23 years). They gestured pictures of objects without using speech (pantomime). The iconicity, semantic richness, and representation technique of the pantomimes were coded. Participants' semantic association and visuospatial skills were formally assessed. Iconicity was slightly lower in individuals with Williams syndrome, which seems related to their visuospatial deficit. While semantic saliency was similar across participant groups, small differences in representation technique were found. Partial correlations showed that visuospatial skills and semantic skills were instrumental in producing clear pantomimes. These findings indicate that clinicians aiming to enhance individuals' natural iconic gestures should consider achieved iconicity, particularly in individuals with low visuospatial skills.