Applied behaviour analysis: What do teachers of students with autism spectrum disorder know

With rising numbers of school-aged children with autism educated in mainstream classrooms and applied behaviour analysis (ABA) considered the basis of best practice, teachers’ knowledge in this field has become a key concern for inclusion. Self-reported knowledge of ABA of special needs teachers (n=165) was measured and compared to their actual knowledge of ABA demonstrated in accurate responses to a multiple-choice test. Findings reported here show that teachers’ self-perceived knowledge exceeded actual knowledge and that actual knowledge of ABA was not related to training received by government agency. Implications for teacher training are discussed.

Classifying cerebral palsy: are we nearly there?

BACKGROUND: Cerebral palsy (CP) is the most common cause of physical disability in childhood in developed countries and encompasses a wide range of clinical phenotypes. Classification of CP according to movement disorder or topographical distribution is widely used. However, these classifications are not reliable nor do they accurately predict musculoskeletal pathology. More recently, the Gross Motor Function Classification System (GMFCS) has been introduced and its validity, reliability, and clinical utility have been confirmed. In 2005 it was suggested that children should be described and classified according to the GMFCS in all outcome studies involving children with CP, in the Journal of Pediatric Orthopaedics (JPO). This study aimed to describe utilization of the GMFCS in 3 journals: Journal of Bone and Joint Surgery (JBJS Am), JPO, and Developmental Medicine and Child Neurology (DMCN), over a 7-year period (2005 to 2011), and any relationship to the journal's impact factor. A secondary aim was to establish if differences in methodological quality existed between those studies utilizing GMFCS and those that did not.

METHODS: A targeted literature search of the 3 selected journals using the term "cerebral palsy" was conducted using the Medline database. Utilization of the GMFCS was assessed using report of these data in the methods or results section of the retrieved papers. The Methodological Index for Non-Randomized Studies (MINORS) was employed to evaluate the quality of papers published in JPO.

RESULTS: One hundred and fifty-four studies met the inclusion criteria and in 85 (68%) the GMFCS was used. Of these, 112 were published in JPO, of which 51 (46%) utilized the GMFCS, compared with 72% for JBJS Am, and 88% for DMCN. In the JPO, utilization of the GMFCS improved from 13% to 80%, over the 7-year study period.

CONCLUSIONS: Utilization of the GMFCS has increased rapidly over the past 7 years in the JPO but there is room for further improvement.

LEVEL OF EVIDENCE: Not applicable.

Systematic review of tools to measure outcomes for young children with autism spectrum disorder

BACKGROUND: The needs of children with autism spectrum disorder (ASD) are complex and this is reflected in the number and diversity of outcomes assessed and measurement tools used to collect evidence about children's progress. Relevant outcomes include improvement in core ASD impairments, such as communication, social awareness, sensory sensitivities and repetitiveness; skills such as social functioning and play; participation outcomes such as social inclusion; and parent and family impact.

OBJECTIVES: To examine the measurement properties of tools used to measure progress and outcomes in children with ASD up to the age of 6 years. To identify outcome areas regarded as important by people with ASD and parents.

METHODS: The MeASURe (Measurement in Autism Spectrum disorder Under Review) research collaboration included ASD experts and review methodologists. We undertook systematic review of tools used in ASD early intervention and observational studies from 1992 to 2013; systematic review, using the COSMIN checklist (Consensus-based Standards for the selection of health Measurement Instruments) of papers addressing the measurement properties of identified tools in children with ASD; and synthesis of evidence and gaps. The review design and process was informed throughout by consultation with stakeholders including parents, young people with ASD, clinicians and researchers.

RESULTS: The conceptual framework developed for the review was drawn from the International Classification of Functioning, Disability and Health, including the domains 'Impairments', 'Activity Level Indicators', 'Participation', and 'Family Measures'. In review 1, 10,154 papers were sifted - 3091 by full text - and data extracted from 184; in total, 131 tools were identified, excluding observational coding, study-specific measures and those not in English. In review 2, 2665 papers were sifted and data concerning measurement properties of 57 (43%) tools were extracted from 128 papers. Evidence for the measurement properties of the reviewed tools was combined with information about their accessibility and presentation. Twelve tools were identified as having the strongest supporting evidence, the majority measuring autism characteristics and problem behaviour. The patchy evidence and limited scope of outcomes measured mean these tools do not constitute a 'recommended battery' for use. In particular, there is little evidence that the identified tools would be good at detecting change in intervention studies. The obvious gaps in available outcome measurement include well-being and participation outcomes for children, and family quality-of-life outcomes, domains particularly valued by our informants (young people with ASD and parents).

CONCLUSIONS: This is the first systematic review of the quality and appropriateness of tools designed to monitor progress and outcomes of young children with ASD. Although it was not possible to recommend fully robust tools at this stage, the review consolidates what is known about the field and will act as a benchmark for future developments. With input from parents and other stakeholders, recommendations are made about priority targets for research.

FUTURE WORK: Priorities include development of a tool to measure child quality of life in ASD, and validation of a potential primary outcome tool for trials of early social communication intervention.

STUDY REGISTRATION: This study is registered as PROSPERO CRD42012002223.

FUNDING: The National Institute for Health Research Health Technology Assessment programme.

Systematic Review of the Measurement Properties of Tools Used to Measure Behaviour Problems in Young Children with Autism

Background
Behaviour problems are common in young children with autism spectrum disorder (ASD). There are many different tools used to measure behavior problems but little is known about their validity for the population.

Objectives
To evaluate the measurement properties of behaviour problems tools used in evaluation of intervention or observational research studies with children with ASD up to the age of six years.

Methods
Behaviour measurement tools were identified as part of a larger, two stage, systematic review. First, sixteen major electronic databases, as well as grey literature and research registers were searched, and tools used listed and categorized. Second, using methodological filters, we searched for articles examining the measurement properties of the tools in use with young children with ASD in ERIC, MEDLINE, EMBASE, CINAHL, and PsycINFO. The quality of these papers was then evaluated using the COSMIN checklist.

Results
We identified twelve tools which had been used to measure behaviour problems in young children with ASD, and fifteen studies which investigated the measurement properties of six of these tools. There was no evidence available for the remaining six tools. Two questionnaires were found to be the most robust in their measurement properties, the Child Behavior Checklist and the Home Situations Questionnaire—Pervasive Developmental Disorders version.

Conclusions
We found patchy evidence on reliability and validity, for only a few of the tools used to measure behaviour problems in young children with ASD. More systematic research is required on measurement properties of tools for use in this population, in particular to establish responsiveness to change which is essential in measurement of outcomes of intervention.

PROSPERO Registration Number

CRD42012002223

Developmental biology and cytogenetics of B. xylophilus

The pine wood nematode Bursaphelenchus xylophilus reproduces bisexually: a haploid sperm fertilizes a haploid oocyte, and the two pronuclei rearrange, move together, fuse, and begin diploid development. Early embryonic events taking place in the B. xylophilus embryo are similar to those of Caenorhabditis elegans, although the anterior-posterior axis appeares to be determined oppositely to that observed for C. elegans. Thai is, in the B. xylophilus embryo, the male pronucleus emerges at the future anterior end, whereas the female pronucleus appeares laterally. To understand the evolution of nematode developmental systems, we cloned the full length of Bx-tbb-1 (beta tubulin) from B. xylophilus cDNA and attempted to apply reverse genetics analysis to B. xylophilus. Several lengths of double stranded RNA (dsRNA) for the Bx-tbb-1 gene were synthesized by in vitro transcription, and both B. xylophilus and C. elegans were soaked in dsRNA for RNAi. Both nematodes could suck up the dsRNA, and we could detect the abnormal phenotypes caused by Bx-tbb-1 dsRNA in C. elegans, but not in B. xylophilus. We suspect that systemic RNAi might be suppressed in B. xylophilus and are attempting to establish other methods for functionally analyzing B. xylophilus genes.

Developmental Antecedents of Proactive and Reactive Rebelliousness: The Role of Parenting Style, Childhood Adversity, and Attachment

Adolescence is a time of developmental transition that for one in five young people is characterised by feelings of oppositionality, rebellion, and negativism. Despite the prevalent experience of teen turbulence and despite its significance within the phenomenological framework provided by reversal theory (RT), the childhood antecedents of rebelliousness in adolescence and adulthood have not been given extensive empirical attention within RT, although such work has been carried out using other constructs and theories. We examined recalled parenting style, childhood adversity, and attachment style in adulthood as correlates of proactive and reactive rebelliousness in a sample of 80 participants, aged 18 to 50 years. Each participant responded to a questionnaire package containing the revised Adult Attachment Scale, the Parental Bonding Instrument, the Childhood Experience of Care and Abuse Questionnaire, and the Negativism Dominance Scale. We found that paternal abusive parenting, followed by paternal parenting style, paternal neglect, and paternal antipathy were independently predictive of scores on proactive rebelliousness, the sensation-seeking form. Maternal and paternal indifferent parenting styles each were found to equivalently and independently predict scores on reactive rebelliousness, the interpersonal disaffection form. The results of this study suggest these two forms of rebelliousness may have distinctly different antecedents. A longitudinal study is needed to examine the potentially causal pathways that are suggested by the results of this cross-sectional research. We consider reversal theory explanations of these results and contrast them with complementary theoretical frameworks.

Face matching impairment in developmental prosopagnosia

Developmental prosopagnosia (DP) is commonly referred to as ‘face blindness’, a term that implies a perceptual basis to the condition. However, DP presents as a deficit in face recognition and is diagnosed using memory-based tasks. Here, we test face identification ability in six people with DP, who are severely impaired on face memory tasks, using tasks that do not rely on memory. First, we compared DP to control participants on a standardised test of unfamiliar face matching using facial images taken on the same day and under standardised studio conditions (Glasgow Face Matching Test; GFMT). DP participants did not differ from normative accuracy scores on the GFMT. Second, we tested face matching performance on a test created using images that were sourced from the Internet and so vary substantially due to changes in viewing conditions and in a person’s appearance (Local Heroes Test; LHT). DP participants show significantly poorer matching accuracy on the LHT relative to control participants, for both unfamiliar and familiar face matching. Interestingly, this deficit is specific to ‘match’ trials, suggesting that people with DP may have particular difficulty in matching images of the same person that contain natural day-to-day variations in appearance. We discuss these results in the broader context of individual differences in face matching ability.

Aims of education and well-being as absence of disorder

This thesis offers an original account of what personal well-being can be. Any account of education, it is believed, has to do with and aims at personal well-being. I approach this view on well-being not in a positive but in a negative way. I put forward some items that in certain circumstances can be taken by and called sources or forms of disorder. In the absence of such forms or sources of disorder, I assume that a certain order, prudential or moral, takes place and that constitutes the well-being of the person. The concept of ‘absence of disorder’ is introduced and argued as an educationally appropriate view of personal well-being which is the central educational aim. Therefore, ‘absence of disorder’ is positioned as the central aim of education. This concept is illuminated, for practical reasoning, by a list of seven possible forms of disorder: Comparison, Corruption, Dependency, Division, Fear, Self-disintegration and Violence. As a view of personal well-being, ‘absence of disorder’ is initially rooted in informed desire satisfaction, via the introduction of the concept of entropy. Prudentially, the agent’s informed desire is satisfied by living a life with low build up of entropy or disorder. But, in a second move such a base is also provided by the Levinasinian concept of ‘disinterest’ as a root for ‘what is to be a human’. Such ‘disinterest’ is related to the concepts of love and of ‘action for its own sake’. It is at this final approach that an attempt is made towards the approximation of the ethical and the prudential aspects of social practices. Even if only to some extent successful, the argument is directed to the following conclusion: an education aiming at ‘absence of disorder’ may promote prudential well-being and give us some confidence in simultaneously favouring moral education.

Development and implementation of a new service delivery model for children with disabilities : implications for DCD

There is a general consensus that new service delivery models are needed for children with developmental coordination disorder (DCD). Emerging principles to guide service delivery include the use of graduated levels of intensity and evidence-based services that focus on function and participation. Interdisciplinary, community-based service delivery models based on best practice principles are needed. In this case report, we propose the Apollo model as an example of an innovative service delivery model for children with DCD. We describe the context that led to the creation of a program for children with DCD, describe the service delivery model and services, and share lessons learned through implementation. The Apollo model has 5 components: first contact, service delivery coordination, community-, group- and individual-interventions. This model guided the development of a streamlined set of services offered to children with DCD, including early-intake to share educational information with families, community interventions, inter-disciplinary and occupational therapy groups and individual interventions. Following implementation of the Apollo model, waiting times decreased and numbers of children receiving services increased, without compromising service quality. Lessons learned are shared to facilitate development of other practice models to support children with DCD.

Cloning of the Bone Gla Protein gene from the teleost fish Sparus aurata (gilthead seabream). Molecular organization, developmental appearance and evolutionary implications

Tese de Doutoramento, Biologia Molecular, Faculdade de Ciências do Mar e do Ambiente, Universidade do Algarve, 2001

Inclusão de uma criança com síndrome de turner numa escola do ensino regular: um estudo de caso

Dissertação de mestrado, Educação Especial - Domínios Cognitivo e Motor, Escola Superior de Educação e Comunicação, Universidade do Algarve, 2015

Genome-wide Association Study Identifies SESTD1 as a Novel Risk Gene for Lithium Responsive Bipolar Disorder

Lithium is the mainstay prophylactic treatment for bipolar disorder (BD), but treatment response varies considerably across individuals. Patients who respond well to lithium treatment might represent a relatively homogeneous subtype of this genetically and phenotypically diverse disorder. Here, we performed genome-wide association studies (GWAS) to identify (i) specific genetic variations influencing lithium response and (ii) genetic variants associated with risk for lithium-responsive BD. Patients with BD and controls were recruited from Sweden and the United Kingdom. GWAS were performed on 2698 patients with subjectively defined (self-reported) lithium response and 1176 patients with objectively defined (clinically documented) lithium response. We next conducted GWAS comparing lithium responders with healthy controls (1639 subjective responders and 8899 controls; 323 objective responders and 6684 controls). Meta-analyses of Swedish and UK results revealed no significant associations with lithium response within the bipolar subjects. However, when comparing lithium-responsive patients with controls, two imputed markers attained genome-wide significant associations, among which one was validated in confirmatory genotyping (rs116323614, P=2.74 × 10-8). It is an intronic single-nucleotide polymorphism (SNP) on chromosome 2q31.2 in the gene SEC14 and spectrin domains 1 (SESTD1), which encodes a protein involved in regulation of phospholipids. Phospholipids have been strongly implicated as lithium treatment targets. Furthermore, we estimated the proportion of variance for lithium-responsive BD explained by common variants ('SNP heritability') as 0.25 and 0.29 using two definitions of lithium response. Our results revealed a genetic variant in SESTD1 associated with risk for lithium-responsive BD, suggesting that the understanding of BD etiology could be furthered by focusing on this subtype of BD.