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Journal of Intellectual Disability Research 881
doi: 10.1111/jir.12155
volume 58 part 10 pp 881–882 october 2014
Editorial
Developmental trajectories in behavioural phenotypes
As the conceptualisation of behavioural phenotypes has evolved a number of themes have emerged which, regardless of the syndrome or the behavioural, cognitive or emotional difference of interest, will shape future research by virtue of their central importance. This year’s Society for the Study of Behavioural Phenotypes meeting takes place in New York with a focus on the theme of developmental trajectories of behavioural phenotypes across genetic syndromes. This theme cuts across clinical and research agendas and has proved to be influential in determining the scale and design of future research projects that might inform best practice. The papers selected for this special edition and the SSBP abstracts reflect the importance of this research for clinical service delivery. Prognosis is at the heart of parental concerns once a diagnosis is received and the study by de Groot-van der Mooren et al. (2014) of the neonatal diagnosis of Down syndrome alludes to the importance of the impact of information giving at an early stage and the way in which this can be actively managed. Evers et al. (2014) provide further detail of the mental health issues that are more common in 22q.11.2 deletion syndrome and thus enrich the prediction for future need and support. Similarly, Antshel et al. (2014) analyse longitudinal data on aspects of academic achievement in the same syndrome. It is these descriptive and longitudinal studies that, in combination, will enable researchers and clinicians to develop and implement syndrome
sensitive interventions in the future in response to the known risk marker of genetic disorder and provide more detailed prognosis at an early stage. The form of available interventions will need to reflect syndrome characteristics as well as draw on mainstream assessments and interventions. An example of a syndrome sensitive intervention that is applicable in adulthood in Williams syndrome, but more typically emphasised in childhood, is described by Fisher et al. (2014). In this study the authors seek to address the possible negative consequences of excessive friendliness in this syndrome by adapting and implementing a stranger awareness training programme. Such an intervention may prevent exploitation and is particularly important for people with Williams syndrome. A second example of the integration of mainstream and syndrome sensitive interventions is described in the abstract of the Tom Oppé lecture that Stewart Einfeld has been invited to deliver at this year’s SSBP meeting (Oral Abstracts 2014). This overview of an early intervention strategy, involving Triple P parent training modified according to the behavioural phenotypes associated with specific genetic disorders, is an example of the way ahead. Such a strategy requires clinical experience and expertise in the delivery of this type of intervention alongside in depth knowledge of behavioural phenotypes. If successful, it is not inconceivable that early intervention of this kind can favourably influence the later outcome for many people who have genetic
© 2014 MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd
Journal of Intellectual Disability Research
volume 58 part 10 october 2014
882 Editorial
syndromes. That is one goal shared by those attending this year’s SSBP meeting and encapsulated in the combination of clinical and research expertise that will no doubt be clearly evident in the Tom Oppé lecture. Chris Oliver Professor of Neurodevelopmental Disorders School of Psychology University of Birmingham Birmingham UK
References Antshel K., Hier B., Fremont W., Faraone S. V. & Kates W. (2014) Predicting reading comprehension academic achievement in late adolescents with velo-cardio-facial
(22q11.2 deletion) syndrome (VCFS): a longitudinal study. Journal of Intellectual Disability Research 58, 926– 39. doi: 10.1111/jir.12134 Evers L. J. M., van Amelsvoort T. A. M. J., Candel M. J. J. M., Boer H., Engelen J. J. M. & Curfs L. M. G. (2014) Psychopathology in adults with 22q11 deletion syndrome and moderate and severe intellectual disability. Journal of Intellectual Disability Research 58, 915–25. doi: 10.1111/jir.12117 Fisher M. H. (2014) Evaluation of a stranger safety training programme for adults with Williams syndrome. Journal of Intellectual Disability Research 58, 903–14. doi: 10.1111/jir.12108 de Groot-van der Mooren M. D., Gemke R. J. B. J., Cornel M. C. & Weijerman M. E. (2014) Neonatal diagnosis of Down syndrome in the Netherlands: suspicion and communication with parents. Journal of Intellectual Disability Research 58, 953–61. doi: 10.1111/ jir.12125 (2014), Oral Abstracts. Journal of Intellectual Disability Research 58, 883–89.
© 2014 MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd
Journal of Intellectual Disability Research 881
doi: 10.1111/jir.12155
volume 58 part 10 pp 881–882 october 2014
Editorial
Developmental trajectories in behavioural phenotypes
As the conceptualisation of behavioural phenotypes has evolved a number of themes have emerged which, regardless of the syndrome or the behavioural, cognitive or emotional difference of interest, will shape future research by virtue of their central importance. This year’s Society for the Study of Behavioural Phenotypes meeting takes place in New York with a focus on the theme of developmental trajectories of behavioural phenotypes across genetic syndromes. This theme cuts across clinical and research agendas and has proved to be influential in determining the scale and design of future research projects that might inform best practice. The papers selected for this special edition and the SSBP abstracts reflect the importance of this research for clinical service delivery. Prognosis is at the heart of parental concerns once a diagnosis is received and the study by de Groot-van der Mooren et al. (2014) of the neonatal diagnosis of Down syndrome alludes to the importance of the impact of information giving at an early stage and the way in which this can be actively managed. Evers et al. (2014) provide further detail of the mental health issues that are more common in 22q.11.2 deletion syndrome and thus enrich the prediction for future need and support. Similarly, Antshel et al. (2014) analyse longitudinal data on aspects of academic achievement in the same syndrome. It is these descriptive and longitudinal studies that, in combination, will enable researchers and clinicians to develop and implement syndrome
sensitive interventions in the future in response to the known risk marker of genetic disorder and provide more detailed prognosis at an early stage. The form of available interventions will need to reflect syndrome characteristics as well as draw on mainstream assessments and interventions. An example of a syndrome sensitive intervention that is applicable in adulthood in Williams syndrome, but more typically emphasised in childhood, is described by Fisher et al. (2014). In this study the authors seek to address the possible negative consequences of excessive friendliness in this syndrome by adapting and implementing a stranger awareness training programme. Such an intervention may prevent exploitation and is particularly important for people with Williams syndrome. A second example of the integration of mainstream and syndrome sensitive interventions is described in the abstract of the Tom Oppé lecture that Stewart Einfeld has been invited to deliver at this year’s SSBP meeting (Oral Abstracts 2014). This overview of an early intervention strategy, involving Triple P parent training modified according to the behavioural phenotypes associated with specific genetic disorders, is an example of the way ahead. Such a strategy requires clinical experience and expertise in the delivery of this type of intervention alongside in depth knowledge of behavioural phenotypes. If successful, it is not inconceivable that early intervention of this kind can favourably influence the later outcome for many people who have genetic
© 2014 MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd
Journal of Intellectual Disability Research
volume 58 part 10 october 2014
882 Editorial
syndromes. That is one goal shared by those attending this year’s SSBP meeting and encapsulated in the combination of clinical and research expertise that will no doubt be clearly evident in the Tom Oppé lecture. Chris Oliver Professor of Neurodevelopmental Disorders School of Psychology University of Birmingham Birmingham UK
References Antshel K., Hier B., Fremont W., Faraone S. V. & Kates W. (2014) Predicting reading comprehension academic achievement in late adolescents with velo-cardio-facial
(22q11.2 deletion) syndrome (VCFS): a longitudinal study. Journal of Intellectual Disability Research 58, 926– 39. doi: 10.1111/jir.12134 Evers L. J. M., van Amelsvoort T. A. M. J., Candel M. J. J. M., Boer H., Engelen J. J. M. & Curfs L. M. G. (2014) Psychopathology in adults with 22q11 deletion syndrome and moderate and severe intellectual disability. Journal of Intellectual Disability Research 58, 915–25. doi: 10.1111/jir.12117 Fisher M. H. (2014) Evaluation of a stranger safety training programme for adults with Williams syndrome. Journal of Intellectual Disability Research 58, 903–14. doi: 10.1111/jir.12108 de Groot-van der Mooren M. D., Gemke R. J. B. J., Cornel M. C. & Weijerman M. E. (2014) Neonatal diagnosis of Down syndrome in the Netherlands: suspicion and communication with parents. Journal of Intellectual Disability Research 58, 953–61. doi: 10.1111/ jir.12125 (2014), Oral Abstracts. Journal of Intellectual Disability Research 58, 883–89.
© 2014 MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd
