A qualitative study exploring the perceptions of health among pre-teen girls from disadvantaged communities in Dublin.

There are disparities in health outcomes between youth from higher and lower socioeconomic backgrounds, and girls are especially vulnerable to changes in health-related behaviours as they develop. Therefore, this study explored how girls from disadvantaged communities in Dublin, Ireland, make sense of 'being healthy.' A phenomenological qualitative design was implemented. Three focus groups were conducted (n = 22, 10-12 years) and data were analysed using thematic analysis. Food and physical appearance featured prominently within the girls' definitions of health. Girls and their families from low-SES backgrounds may experience more difficulties with time scarcity as well as environmental barriers to a healthy lifestyle.

The burden and impact of recurrent abdominal pain - exploring the understanding and perception of children and their parents.

Background: Recurrent abdominal pain (RAP) is a common complaint for children and can result in a significantly lower quality of life due to the extent it can interfere with normal life. RAP can also significantly impact the quality of life of parents. This study sought to qualitatively explore parents' and children's understanding and perceptions of the burden and impact of RAP. Methods: Semi-structured interviews were conducted with a sample of parent/child dyads or families (N = 5) engaging with a psychology service. Findings: The findings of the inductive thematic analysis revealed four emergent themes common to both parents and children: (1) Perception, understanding and identification of RAP, (2) Contributing factors, (3) Coping mechanisms/pain management strategies, and (4) Impact and burden of RAP. Conclusions: These findings have important clinical implications regarding the identification and management of RAP and may also contribute to improving communication between clinicians, parents and children by providing insight from multiple perspectives into how RAP is experienced.

Living through liminality? Situating the transitional experience of parents of children with mucopolysaccharidoses.

BACKGROUND: Mucopolysaccharidoses (MPS) are rare inherited metabolic disorders that come under category three of life-limiting conditions. Children born with this condition show no symptoms at birth, but its effects show as a progressive disease in subsequent years. The severity of the condition varies according to the specific type, ranging from very mild symptoms to, in most cases, complex healthcare needs, including mental and physical disabilities. AIMS: This study aimed to elucidate the meanings of the transition experience of being a parent of a child with MPS. Van Gennep's three-stage rite of passage theory and Turner's theory of liminality were utilised to understand these families' transition experiences are learning to live with their child's ongoing progressive illness trajectory. METHODS: A qualitative design utilising hermeneutic phenomenology was used. Longitudinal qualitative in-depth interviews were carried out with eight parents at a three-time point over 17-months period. RESULTS: This study provided an interpretation of the lived experience of parents of children and young adults with MPS. As such, it embraces a liminal experience of living with a rare life-limiting illness and the unique passage to becoming a parent of a child with MPS. Parents reported their experience of transition from being the parent of a normal healthy child to be the parent of a child with MPS. They described their transition experience as multi-faceted and complex, neither linear nor time-bound, but rather cyclical. CONCLUSION: The rites of passage conceptual framework helped to identify specific and significant unmet supportive and social care needs of these families and their children. This information will enhance the development of a substantial support system to meet the family's emotional, psychological and social needs during the illness transitions from diagnosis and throughout their illness journey.

IMPACT study: measuring the impact of caregiving on families and healthcare professionals of children and adults living with mucopolysaccharidoses in Ireland.

Introduction: Disease trajectories are often uncertain among individuals living with mucopolysaccharidoses (MPS) due to the progressive nature of the illness and the goal of care. This study investigated the impact on caregivers and care providers of children and adults living with MPS. Methods: The study used a cross-sectional design and a convenience sampling strategy which involved two sequential study components. The stage 1 quantitative component included three validated scales: the abbreviated World Health Organization Quality of Life (WHOQOL-BREF), the Paediatric Inventory for Parents (PIP) and the 14-item Resilience Scale (RS-14). The stage 2 qualitative component consisted of two focus groups with healthcare professionals (HCPs) (n = 9) working with children and adults living with MPS across three clinical sites in Ireland. Data were collected between November 2017 and July 2019. Results: A total of 31 parents identified as caregivers participated in this study. The mean quality of life (QoL) score was 93.81, indicating a significantly high QoL. The PIP frequency total mean was 102.74 and difficulty mean 104.94. The mean score for the RS-14 was notably high, 81.42 out of a maximum of 98. The majority of the results showed high levels of concern for the future, with just under 50% finding themselves very often feeling scared that their child's condition will deteriorate or that their child will die and finding these thoughts very difficult. The healthcare professionals' (HCPs) perceptions were focused on the complexity of MPS, coping strategies, managing expectations and support services. Conclusion: The overall findings of the study reinforced the need for sustained and enhanced psychological support to ensure both families of children and adults living with MPS and the HCPs are supported in the continued delivery of quality patient care and outcomes. Subjective and objective measures from family caregivers and HCPs yield results that can decrease stress and improve psychological support. Plain language summary: Impact of caregiving on families and healthcare professionals of children and adults living with mucopolysaccharidoses in Ireland Mucopolysaccharidoses (MPS) is a group of one of the many rare inherited metabolic disorders that come under category three of life-limiting conditions. Children born with this genetic condition show no change at birth, but effects start to show in subsequent years as it is a progressive disease. The severity of the condition varies according to the specific type, ranging from very mild symptoms to, in most cases, multisystemic, restricted growth or mental and physical disabilities. Recent developments in treatments for some forms of MPS have dramatically changed the quality of life (QoL) for patients. Other forms of treatment are currently under investigation and development. This study aimed to provide a detailed and reliable evidence base on the impact of caregiving for patients living with MPS on family caregivers and healthcare providers. Paper questionnaires were completed by the family caregivers of children and adults living with MPS. These three questionnaires focused on measuring QoL, parental stress and anxiety, and resilience among these families. Two focus group interviews were carried out with healthcare professionals working with children and adults living with MPS across three clinical sites in Ireland.A total of 31 parents completed the questionnaires in this study, indicating a significantly high QoL and notably high resilience. The majority of the parental stress results were related to concern for the future, with just half of the families finding themselves very often scared that their child's condition will deteriorate or that their child will die and find these thoughts very difficult. The healthcare providers also spoke about the complexity of MPS, coping strategies, managing expectations and support services for the families of children and adults living with MPS. This study provided evidence for clinicians and policymakers to improve the availability of appropriate healthcare provisions for people living with MPS and their families.

Rare Disease Research Partnership (RAinDRoP): a collaborative approach to identify research priorities for rare diseases in Ireland.

Background: The Rare Disease Research Partnership (RAinDRoP) was established in 2018 to bring together a wide variety of diverse voices in the rare disease community in Ireland and form a research partnership. This approach enabled clinicians, patients, carers and researchers to work together to identify top research priorities for rare diseases, which focused on a life-course perspective rather than a disease-specific need.                                                                                                           Methods: A participatory multiple phase approach was used to identify research priorities for rare diseases. The research process involved three main phases: Phase I, Public Consultation Survey on Research in Rare Diseases in Ireland (PCSRRDI); Phase II, Research Prioritisation Workshop (RPW); Phase III, Follow-up Public Consultation and Prioritisation Survey (FWPCPS). Results: In total, 240 individuals completed the phase I PCSRRDI, which comprised of a cross-section of health care professionals, researchers and people living with rare diseases. One thousand and fifteen statements were collected, reflecting issues and shared challenges in rare diseases. A shortlisting step by step was used to identify any statements that had received a total score of above 50% into 10-12 researchable questions or statements per the theme for the phase II workshop. Phase II was focused on three main themes: (1) Route to Diagnosis, (2) Living with Rare Disease, (3) Integrated and Palliative Care. In total, 62 individuals attended the overall workshop; 42 participated in the prioritisation sessions. A cross-section of health care professionals, researchers and people living with rare diseases were engaged at each workshop. Seventy-five individuals completed the final phase III public ranking by priority responses, and they ranked the top 15 research priorities defined by the multi-stakeholders at the phase II consensus meeting. Conclusions: This study identified priorities for rare diseases research aimed at improving the health and wellbeing of people living with rare diseases.

A network analysis of the causal attributions for obesity in children and adolescents and their parents.

Illness attributions inform how people understand illness and relate to psychological outcomes. Parental attributions may impact children's adjustment to illness. This study investigated child, adolescent and parental causal attributions in paediatric obesity and illustrates the relationships between these attributions using network analysis. A cross-sectional design using the diagram network analytic method. Thirty children and 25 parents generated individual causal attribution maps. Network theory was used to analyse causal effects and results were visualised using network models. The results indicated large individual variation in the networks. Activity and eating-related variables featured strongly in child and parent networks. Children viewed their activity while parents regarded their child's eating behaviours as the main drivers of obesity. The characterisation of some children along an internalising dimension was supported in both networks. Habitual cluster behaviours and difficulties in regulation were identified in the child network. Parents ascribed significance to the future impact of obesity on body image and mood. Challenges in parent management were also indicated. Obesity is a heterogeneous condition, requiring a tailored treatment approach. Therapeutic directions were identified in the areas of activity, food intake, sleep hygiene and parent management. Attributional processes represent a potential mechanism to tailor obesity treatment. Further research is needed to establish the relationship between attributions, treatment engagement and outcome.

European adolescent substance use: the roles of family structure, function and gender.

OBJECTIVES: The aim of this study was, first, to explore family structure and measures of family functioning in relation to adolescent substance use and secondly, to establish if these relationships differed according to gender or according to the city of origin of the sample. DESIGN, SETTING, PARTICIPANTS: The study surveyed pupils aged 14-15 years in representative samples drawn from five European cities: Newcastle upon Tyne, Dublin, Rome, Bremen and Groningen. Data were obtained on 3984 participants in relation to their substance use, living with both biological parents, confiding in parents and grandparents. and supervision, as well as other variables representing delinquency, social class and drug availability. RESULTS: Living with both parents was associated with reduced levels of drug use in four cities but not in Dublin, due perhaps to the high availability from peers in that city. It was not associated with reduced levels of regular drinking. The effect of confiding in mother was evident in all cities and in relation to substance use in general. However, when a delinquency variable was added to the logistic regressions, its significance in relation to polydrug use disappeared. Supervision was somewhat more important in relation to male than female drug use. CONCLUSION: Living with both parents is a less robust barrier to substance use than qualitative aspects of family life, particularly attachment to mothers. The latter is a robust inhibitor of substance use irrespective of regional differences in drug availability, weakening only in the face of more generally problematic behaviour. Perhaps because of their greater tendency to risk-taking or rule breaking, supervision appears more important for male than female drug use. These findings underscore the role of families, but especially that of mothers, in regulating the substance-related behaviour of young people.