Background and aims: Information on the neurodevelopmental outcomes of children born very preterm is required for multiple purposes. Reliable and up-to-date data sources are lacking. The overall aim of this thesis is to evaluate the validity and usability of the neurodevelopmental outcome data of very preterm children available from current data sources. The specific objectives were: 1) to examine the validity of outcome data recorded during routine follow-up assessment 2) to explore early childhood social-communication difficulties exhibited by very preterm children 3) to assess the stability over time of neurodevelopmental diagnoses made in early childhood. Methods: Three studies were conducted to meet the objectives. For studies 1 and 2, I recruited children born at < 30 weeks’ gestation at 2 years corrected age (age corrected for prematurity) from 13 participating study sites. In study 1, I compared the agreement between the neurodevelopmental outcomes of 190 children recorded at their routine NHS assessments and data obtained by a research assessment using the Bayley Scales of Infant Development, 3rd edition. In study 2, the social-communication skills of 141 children were determined using the parent-completed Quantitative Checklist of Autism in Toddlers (Q-CHAT) questionnaire and compared to published results from the general population. In study 3, I conducted a systematic review and using meta-analytic methods, I calculated the pooled sensitivity and specificity of early developmental assessment in identifying school-age cognitive deficit from 24 studies. Conclusions: 1) Compared with research assessment, routine NHS follow-up assessment had a low sensitivity but high specificity for identifying children with neurodevelopmental impairment. 2) Very preterm children display greater early childhood social-communication difficulties and autistic behaviour than the general population as measured by their parents on the Q-CHAT. 3) Early neurodevelopmental assessment has high specificity but low sensitivity for identifying later school-age cognitive deficits.

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The purpose of this study was to improve the care of children with VSD in rural Thailand. Based on the literature review, these children experienced long waiting times or surgery and had difficulty in gaining or maintaining optimal weight prior to surgery. An ethnographic approach was used to explore and understand the aregivers’ problems in their cultural context; especially the experiences of how the amilies managed the care of their child, in order to overcome the issues of gaining or maintaining optimal weight prior to surgery. Ten family case studies resulted from semi-structured interviews, field notes, observations, and photographs from the ten main caregivers of children with VSD. Ten health professionals who dealt with these children were interviewed to ascertain any differences in perspective between the professionals and the caregivers with respect to nutritional management. Data were collected between January and May 2012 in a egional hospital and at the homes of the participating caregivers. A thematic approach was used to analyse the data. The triangulated findings generated themes indicating hat both health professionals and caregivers had insufficient knowledge in taking care of children with VSD in rural Thailand, who were waiting for surgery. The key issues elated to the caregivers’ insufficient knowledge were the limited knowledge of health professionals, inadequate resources at the clinic and the clinical environment did not provide useful information. Furthermore, some of the knowledge the caregivers used was inappropriate for supporting weight gain in their child. Drawing on the literature and findings, the content for nutritional preoperative information package for the aregivers to help enable their child to gain or maintain optimal weight prior to surgery has been designed. The findings suggested that, particularly in the rural areas, any written materials to support the caregivers should be designed for those who have a eading age of at least primary school level.

This thesis is a piece of Creative Analytic Practice which uses auto/ethnographic storytelling to dis/orientate autism, childhood, and dis/ability. Dis/orientation is a theoretical tool I developed through, and with, the thesis borrowing from Critical Disability Studies’ dis/humanism and Ahmed’s orientation in queer phenomenology. Dis/humanism both desires the human and rejects it at the same time; the vital and giddy experience of dis/orientation is both to be drawn towards a comfortable orientation and at once to reject its seductive temptation as a final resting place or single terrain when encountering lives lived as ‘autistic’ and ‘child’. All orientations to autism, childhood and dis/ability are the effects of repeated and often concealed actions over time which I argue are temporary and changeable, given time and dis/orientating labour. Herein lies the hopeful call of dis/orientation throughout this thesis. Ethnographic fieldwork took place with children and families at home, school, nursery, public spaces and a muddy allotment over an 18 month period. Stories of the fieldwork are told as both method of inquiry and analytical tool. Three analysis chapters respond to my research questions exploring the ‘talking’, ‘being’ and ‘becoming’ of autism, childhood, and dis/ability. I argue that through talk, autism becomes rhizomatic in families' lives and is shaped by, and shaping of, collective identity work, emotional labour, and the neuro-governance of everyday life in neoliberal, commodified times. I argue that some orientations make some beings and becomings of childhood's labelled autistic more im/possible than others. Most significantly, I claim that a turn to embodiment outside of Cartesianism is valuable to theory, policy, practice and research in recognising the potential ‘becomings’ of disabled children’s childhoods in which they exceed subjectivation as ‘autistic’ or ‘child’ with and through their bodies in everyday life.

Pregnancy induces numerous changes in the physiology of the mother as she adapts to the metabolically demanding fetus developing within her womb. It is during this period of intense physiological stress that she begins to develop the maternal behaviours that will ultimately support the nurturing of the offspring after parturition and protect them from harm. In human pregnancies, women are particularly susceptible to mood disorders which may reflect an enhanced vulnerability induced by pregnancy. The placenta is the key organ of pregnancy driving the physiological and potentially the behavioural changes required for a successful pregnancy. Placental dysfunction may contribute to maternal mood disorders by mis-programming maternal behaviour, a relationship that can be explored in a tractable animal model. Imprinted genes are expressed in the placenta and have been implicated in the regulation of key endocrine lineage of the mouse placenta. Phlda2 is a maternally expressed imprinted gene that regulates one such lineage, the Spongiotrophoblast (SpT), which is a source of placental lactogens and a number of other hormones. Placental lactogens belong to the same hormone family as prolactin and some members of this family mediate their action via the Prolactin Receptor (Prlr). Both prolactin and the Prlr have been directly shown to be required for the establishment of maternal care behaviour in rodents. This led to the hypothesis that Phlda2 might modulate maternal care in rodents by regulating the size of the placental endocrine compartment. To test this hypothesis, we used three cohorts of Wildtype (WT) dams carrying fetuses that possessed either a 50% reduction in the SpT, resulting from a double dose of Phlda2 (Mouseline: Phlda2 +/+ BACx1, TG; 2X) or a 200% increase in the SpT, resulting from a null dose of Phlda2 (Mouseline: Phlda2 -/+), KO; 0X) or 100% of the WT size of the SpT, resulting from a single dose of Phlda2 (Mouseline: Phlda2 +/+, WT; 1X) generated using Recipient Embryo Transfer (RET). Maternal behaviour was studied postpartum and a biomolecular characterisation was performed during pregnancy using microarray and Quantitative Polymerase Chain Reaction (qPCR) to look at changes in maternal gene expression and a histological approach used to examine maternal neurogenesis in the Sub-ventricular Zone (SVZ) of the lateral ventricles and Sub-Granular Zone (SGZ) of the hippocampus. Additionally, a study was made of male offspring carrying the Phlda2 transgene (TG), which were previously shown to be low birth weight, their non-transgenic litter mates (NON-TG) and a fully WT cohort of mice to ask whether placental endocrine dysfunction in utero programmed altered behaviour later in life. The results of this first study showed distinct changes in pup retrieval, nest building and the dam’s grooming and licking behaviour with dams exposed to the smaller endocrine compartment spending less time with their pups. Specific maternal brain regions showed altered transcriptional profiles at Embryonic Day (E) 16.5 of pregnancy and there was a significant reduction in neurogenesis. While there were no differences in anxiety or locomotor activity levels in the offspring cohorts, there were significant changes in hedonic response in both the TG and NON-TG offspring. Together, these data provide the first evidence that imprinted genes can influence both maternal care behaviour and offspring behavioural outcomes via the placental endocrine compartment. This work has wider implications since human studies have shown that elevated placental PHLDA2 is a common features of human growth restricted pregnancies. There is a a co-occurrence of low birth weight and maternal mood disorders with mothers experiencing prenatal depression having a three-fold increased risk of a Low Birth Weight (LBW) baby. Aberrant imprinting in the placenta could account for this co-occurrence.

Background and aim: Present at birth, a cleft lip and/or palate (CL±P) - affecting 1 in 700 births - requires a number of interventions (e.g. surgical, speech and language therapy) from birth until late adolescence. Increased psychosocial problems are reported in the literature for children and young people (CYP) with additional condition(s) to a CL±P (Feragen & Stock, 2014). However, the presence of a learning disability (LD) in addition to a CL±P remains qualitatively unexplored. How CYP with LDs and CL±P experience their different appearance, treatment, and their friendships, is unknown, making it difficult to ascertain their need for further support/interventions. This study therefore explored their experiences. Method: The triangulation of data sources resulted in 39 semi-structured interviews with 15 CYP with clefts (aged 10-16) with and without LDs, 15 parent interviews and nine interviews with Healthcare Professionals. Thematic Analysis was used to determine patterns across the data. Results: Nine superordinate themes were identified, and overlapped between participant groups; self-image, struggles, resilience, double vulnerability (of having LDs and a cleft), tensions, good practice, and promotion of rights. Struggles were particularly apparent for CYP with LDs/learning needs, and their parents. Resilience was most strongly asserted by CYP without LDs and their parents. However, key mediating factors affected experiences of appearance, treatment and friendships, namely how familial needs were met in hospital and school. Results led to an interpretative framework, and conceptual flow-diagram of the findings. Conclusion: Individualised care and support in hospitals and schools is pivotal to ensure inclusion, and improve the experiences of CYP with LDs and CL±P.

The number of children with life-limiting and life-threatening conditions in the UK is rising. In response to the reality that many children are living longer and complexities of care are increasing, providers of palliative care services are tasked with developing flexible approaches to delivery of care. Children’s hospices are key players in this. However, evidence suggests that only a small percentage of parents of children with life-limiting and life-threatening conditions choose to access hospice services. This study explored parents’ perspectives of existing hospice and palliative care services in one region of England. Barriers and facilitators to accessing services, the types and characteristics of hospice services parents wanted and how best these services could be delivered to meet the needs of the population of children with life-limiting and life-threatening conditions were considered. A two phase qualitative study underpinned by a constructivist grounded theory methodology was employed for the study. In Phase 1 focus groups were used to collect data from twenty four parents of children accessing services at the hospice. In Phase 2 in-depth semi-structured interviews were conducted with seven parents of children who did not use services at the hospice and with a further seven parents who had either previous experience of the hospice or were using a hospice outside of the region. Data derived from the interviews were analysed using a grounded theory method. Three main themes were generated: Coming ‘Home’; This is Living Now; and Moving Forward. A theory of place bonding was developed which reflects how affective bonds develop over time between parents of children with life-limiting conditions and the hospice. The five dimensions of place bonding: place familiarity, place identity, place belongingness and place association led to a deeper sense of place rootedness and established bonds with the hospice. Finding a place where they belonged and where they felt at ‘home’ made the decision to accept help in caring for their child with a life-limiting condition more manageable. The thesis concludes by acknowledging the importance of place bonding and its potential to influence practice for children’s hospices and also in providing a platform for service development for other respite situations for children and young people with life-limiting conditions.

This thesis explores the experience of Cystic Fibrosis (CF) in young people and healthcare professionals across three papers. Firstly, a systematic literature review explores the experiences of peer support for young people with CF, including peer support with CF peers, non-CF peers, impact of segregation, diagnosis disclosure, need for and purpose of peer support and parental and peer relationships. The need for social support in the lives of young people with CF was highlighted. The findings emphasised the need to continue addressing social needs of youth with CF, particularly finding ways to reduce the risk of cross-infection if youth choose to pursue friendships with CF peers. Limitations to the evidence base prevented reciprocal associations, bi-directional relationships and changes over time to be examined. Future research needs to consider the paucity of literature on peer support for young people with CF and potential avenues for future research are discussed. The second paper presents findings from an empirical study, qualitatively exploring the lived experiences of healthcare professionals (HCPs) working with children and adolescents with CF and the transition to adult services. This study was undertaken according to the principles of interpretative phenomenological analysis (IPA), with semi-structured interviews conducted with seven participants. Three superordinate themes emerged from the data, all dynamically intertwined and represent the interplay between both professional and (inter)-personal dynamics within CF care. Implications for clinical practice and future research are discussed. The third paper discusses implications for theory and clinical practice emerging from the first two papers. Discussion emphasises how this study examined an under-represented area and expands the opportunity to bring HCPs’ experiences and social needs of young people with CF to the foreground. This paper concludes with personal reflections on the research process and outcomes.

This thesis focused on the high levels of social vulnerability experienced by individuals with Williams Syndrome (WS). The investigation began with parent interviews about social approach behaviour, with parents emphasising the lack of awareness of social boundaries that many individuals with WS display. The qualitative analysis also highlighted the within-syndrome variability in the parental accounts, prompting discussion on the heterogeneity of the WS social profile. Based on the atypical social approach behaviour described by parents, the subsequent studies addressed issues of personal space and interpersonal distance. Using a parent report questionnaire, it was found that children with WS were more likely to violate the personal space of others. This was followed up with a stop-distance paradigm which showed that children with WS failed to regulate their distance based on familiarity, and stood the same distance from a stranger as they did their parent, which was not the case for typically developing individuals. Given these findings, the research progressed to explore the issue of trust in WS. It was found that children with WS displayed higher levels of trust behaviour, compared to their mental age matched typically developing peers and struggled to decipher trustworthiness from faces. Taken together, these findings seem to suggest that children with WS could be experiencing high levels of social vulnerability on a daily basis. It is widely accepted that this social vulnerability continues into adulthood, with increased levels of both independence and isolation posing a new set of challenges. The subsequent chapter probed the level of insight that adults with WS had about their own vulnerability. Using the Social Vulnerability Questionnaire, it was found that adults with WS consistently reported lower levels of vulnerability, compared to parent reports. This emphasised the need for multi-informant methods, and called for interventions which target self-awareness in order to increase intervention efficacy. The final chapters looked at how this social vulnerability manifests in the online environment. It was found that adults with WS frequently use the Internet and the majority visit social networking sites every day or almost every day, with little parental supervision or oversight. These individuals were more likely to agree to engage in socially risky behaviours (e.g. meeting an “online friend” in person) compared to risky behaviours that were not social in nature when online (e.g. giving out passwords). A case study interview with an adult with WS and their parent highlighted that this individual held a broad definition of what a friend was and found they used the Internet as a tool to expand their social network, which was of great concern to their parent. The findings included in this thesis provide in-depth information relating to social vulnerability in WS and offer the first insights into online social behaviour and online vulnerability in adults with WS. The theoretical and real-world implications of these findings are emphasised throughout and a number of suggestions are made to help the research progress towards intervention development.

Problems in family interaction have long been identified as risk factors for child anxiety disorders. Communication and confiding, more specifically, appear relevant to both anxiety and somatisation. There is a substantial literature linking parent-child communication to healthy development, for example regarding language and regulation of emotion. Conversely, there is evidence, chiefly from work among adolescents and adults, that secrecy or inhibited disclosure is associated with adverse physical and psychological outcomes. It appears plausible that, among preadolescents, too, expression of feelings may alleviate anxiety, while inhibited communication may exacerbate stress and somatic symptoms. As few studies have explored these relationships in community samples, the present study set out to do so, under the aegis of a larger piece of research among 7-11 year olds. The two-stage study involved administration of questionnaires to 2566 children and 1368 parents, followed by interviews with a subsample of primary caregivers and children in 145 families. Based on a review of the literature, it was hypothesised that levels of child communication would be associated with anxiety and somatic symptoms, after taking into account other relevant child and family factors, but that the strength of these relationships would vary by gender, and the aspect of communication in question. A new scale, suitable for 7-11 year olds, was developed in order to measure different aspects of communication. As anticipated, aspects of communication were associated with anxiety and somatic symptoms, and contributed to explaining variance in somatic symptoms for both sexes, even after controlling for anxiety and other relevant factors, including parental control and child depressive symptoms. In line with hypotheses, inhibition was more strongly associated with symptoms than was open communication, as was perceived caregiver responsiveness. Girls reported higher levels of communication than boys, but also slightly higher levels of inhibition, which was a stronger predictor of anxiety for girls than boys. These results are discussed in the context of previous studies, and in terms of their implications for practice. Limitations of the study are discussed and suggestions made for future research.