The impact of managing a child’s chronic fatigue syndrome/ myalgic encephalopathy (CFS/ME) on family relationships

Although there is a large body of research literature concerning the impact of childhood chronic illness on family relationships, very little of this has specifically focussed on the impact of childhood Chronic Fatigue Syndrome/Myalgic Encephalopathy (CFS/ME). The current study therefore aims to investigate the impact of having a child with CFS/ME on family relationships from the perspective of parents. This study takes a qualitative approach. Semi-structured interviews were used to explore 18 parents’ experiences of having a child with CFS/ME and the impact this has had on family relationships. Interview data were transcribed and analysed using thematic analysis. The analysis led to the identification of 5 main themes: “Long and Difficult Journey”, “Uncertainty”, “Isolation and Restriction”, “Focus on the unwell person at the expense of family life” and “Parental roles”. They felt that life had become a constant struggle and often felt exhausted. They felt uncertainty, isolation and restriction. These parents talked about feeling like family life had become focussed on their unwell child, such that some expressed a need to escape from the situation. They also described how, as a consequence of managing their child’s CFS/ME, family members could often feel marginalised and the illness could be seen as a destructive force. However, many parents also described working as a team and feeling that family relationships had benefitted in some way. The results of this study are in line with findings of other studies into other chronic illnesses. In addition, this is the first study to focus exclusively on the impact of CFS/ME on family relationships from the perspective of the parents, and is unique in that there is a dual focus on both mothers and fathers. Clinicians should be aware that parents of children with CFS/ME may need additional support, such as information as to what to expect at various stages of the illness and where to access the relevant support

“It's not one size fits all”; the use of videoconferencing for delivering therapy in a Specialist Paediatric Chronic Fatigue Service

Background: There are few specialist paediatric Chronic Fatigue Syndrome (CFS/ME) services in the UK. Therefore, the distance some families have to travel to reach these services can be a barrier to accessing evidence-based treatment. Videoconferencing technology such as Skype provides a means of delivering sessions remotely. This study aimed to explore the views of children and young people, their parents, and healthcare professionals of treatment delivered by videoconferencing in a specialist paediatric CFS/ME team. Method: To explore the experiences of the participants, a qualitative design was selected. Twelve young people (age 9–18), and 6 parents were interviewed about their experience of treatment sessions delivered via videoconferencing within a specialist CFS/ME service. A focus group explored the views of healthcare professionals (N = 9) from the service. Thematic analysis was used. Results: Three themes were identified from the data: “Challenges and concerns”, “Benefits” and “Treatment provision”. Challenges and concerns that participants identified were; difficulties experienced with technology; a sense of a part of communication being lost with virtual connections; privacy issues with communicating online and feeling anxious on a screen. Participants felt that benefits of videoconferencing were; improving access to the chronic fatigue service; convenience and flexibility of treatment provision; a sense of being more open online and being in the comfort of their own home. In terms of treatment provision participants talked about videoconferencing as a part of a hierarchy of communication; the function of videoconferencing within the context of the chronic fatigue service; additional preparation needed to utilise videoconferencing and an assumption that videoconferencing is “part of young people's lives”. Conclusions: Although the experience of sessions provided by videoconferencing was different to sessions attended in person, participants tended to be positive about videoconferencing as an alternative means of accessing treatment, despite some barriers. Videoconferencing could be an additional option within an individualised care plan, but should not be an alternative to face to face support. Keywords: Chronic fatigue syndrome, Myalgic encephalomyelitis, Skype, Video-conferencing, Adolescent

What stops children with a chronic illness accessing health care: A mixed methods study in children with Chronic Fatigue Syndrome/Myalgic Encephalomyelitis (CFS/ME)

Background: Paediatric Chronic Fatigue Syndrome/Myalgic Encephalomyelitis (CFS/ME) is relatively common and disabling with a mean time out of school of more than one academic year. NICE guidelines recommend referral to specialist services immediately if severely affected, within 3 months if moderately affected and within 6 months if mildly affected. However, the median time-to-assessment by a specialist service in the UK is 18 months. This study used a mixed-methods approach to examine factors associated with time taken to access specialist services. Methods. Time-to-assessment was analysed as a continuous "survival-time" variable in Cox regression models using data from self-completed assessment forms for children attending a regional specialist CFS/ME service between January 2006 and December 2009. Semi-structured interviews about barriers experienced in accessing healthcare for their child were conducted with nine parents of children aged < 17 years (8 individual and one parent couple). Interviews were digitally recorded and analysed using "thematic analysis". Results: 405 children were assessed between 2006 and 2009 and information on school attendance was available on 388. Only 1/125 with severe CFS/ME and 49/263 (19%) with mild to moderate CFS/ME were seen within NICE recommended timeframe. Increased fatigue was associated with shorter time to assessment (HR = 1.15; 95% CI 1.03, 1.29 per unit increase in Chalder fatigue score; P = 0.01). Time-to-assessment was not associated with disability, mood, age or gender. Parents described difficulties accessing specialist services because of their own as well as their GP's and Paediatrician's lack of knowledge. They experienced negative attitudes and beliefs towards the child's condition when they consulted GPs, Paediatricians and Child Psychiatrists. Parents struggled to communicate an invisible illness that their child and not themselves were experiencing. Conclusions: GPs, Child Psychiatrists and Paediatricians need more knowledge about CFS/ME and the appropriate referral pathways to ensure timeliness in referral to specialist services. © 2011 Webb et al; licensee BioMed Central Ltd

Developmental Programming Mediated by Complementary Roles of Imprinted Grb10 in Mother and Pup

Developmental programming links growth in early life with health status in adulthood. Although environmental factors such as maternal diet can influence the growth and adult health status of offspring, the genetic influences on this process are poorly understood. Using the mouse as a model, we identify the imprinted gene Grb10 as a mediator of nutrient supply and demand in the postnatal period. The combined actions of Grb10 expressed in the mother, controlling supply, and Grb10 expressed in the offspring, controlling demand, jointly regulate offspring growth. Furthermore, Grb10 determines the proportions of lean and fat tissue during development, thereby influencing energy homeostasis in the adult. Most strikingly, we show that the development of normal lean/fat proportions depends on the combined effects of Grb10 expressed in the mother, which has the greater effect on offspring adiposity, and Grb10 expressed in the offspring, which influences lean mass. These distinct functions of Grb10 in mother and pup act complementarily, which is consistent with a coadaptation model of imprinting evolution, a model predicted but for which there is limited experimental evidence. In addition, our findings identify Grb10 as a key genetic component of developmental programming, and highlight the need for a better understanding of mother-offspring interactions at the genetic level in predicting adult disease risk

Antagonistic roles in fetal development and adult physiology for the oppositely imprinted Grb10 and Dlk1 genes

BACKGROUND: Despite being a fundamental biological problem the control of body size and proportions during development remains poorly understood, although it is accepted that the insulin-like growth factor (IGF) pathway has a central role in growth regulation, probably in all animals. The involvement of imprinted genes has also attracted much attention, not least because two of the earliest discovered were shown to be oppositely imprinted and antagonistic in their regulation of growth. The Igf2 gene encodes a paternally expressed ligand that promotes growth, while maternally expressed Igf2r encodes a cell surface receptor that restricts growth by sequestering Igf2 and targeting it for lysosomal degradation. There are now over 150 imprinted genes known in mammals, but no other clear examples of antagonistic gene pairs have been identified. The delta-like 1 gene (Dlk1) encodes a putative ligand that promotes fetal growth and in adults restricts adipose deposition. Conversely, Grb10 encodes an intracellular signalling adaptor protein that, when expressed from the maternal allele, acts to restrict fetal growth and is permissive for adipose deposition in adulthood. RESULTS: Here, using knockout mice, we present genetic and physiological evidence that these two factors exert their opposite effects on growth and physiology through a common signalling pathway. The major effects are on body size (particularly growth during early life), lean:adipose proportions, glucose regulated metabolism and lipid storage in the liver. A biochemical pathway linking the two cell signalling factors remains to be defined. CONCLUSIONS: We propose that Dlk1 and Grb10 define a mammalian growth axis that is separate from the IGF pathway, yet also features an antagonistic imprinted gene pair. ELECTRONIC SUPPLEMENTARY MATERIAL: The online version of this article (doi:10.1186/s12915-014-0099-8) contains supplementary material, which is available to authorized users

Dose reduction of biologic therapy in inflammatory arthritis: A qualitative study of patients' perceptions and needs

© 2018 John Wiley & Sons, Ltd. Objective: Successful biologic disease-modifying anti-rheumatic drug (bDMARD) dose reduction appears increasingly possible from clinical trials. The present study aimed to understand the patient perspective of bDMARD dose reduction. Methods: Patients with rheumatoid arthritis, ankylosing spondylitis or psoriatic arthritis who were self-administering subcutaneous bDMARDs therapy at two National Health Service trusts participated in semi-structured interviews. To capture multiple experiences, patients were purposefully sampled for a range of age, gender, disease duration, reducing/not reducing bDMARDs and either within 3–12months of bDMARD initiation or ≥12months and in remission/low disease activity. Inductive thematic analysis was utilized. Results: Fifteen patients were interviewed (six on dose reduction). Five overarching themes were identified. When thinking about dose reduction, patients reflected on their difficult life before bDMARDs (“Where I was then”) compared with their transformative effects (“Where I am now”). All raised concerns that a dose reduction would take them back to where they used to be (“Fears for the future”) and most believed it to be a cost-cutting exercise. Most had “Hopes for the future”, that a reduction would lower their risk of side effects, and release funds for other patients. They wanted a clear rationale for reduction, collaborative decision making, and control over flexible dosing (“Information needs”). Conclusion: Patients were fearful of reducing the dose of their bDMARDs, having previously experienced uncontrollable symptoms. However, most were willing to try, provided that there was a clear rationale and that it was in their best interests, with opportunities for collaboration and dose control. These patient perspectives will inform the provision of patient information to guide clinical discussions

Correction: What stops children with a chronic illness accessing health care: a mixed methods study in children with Chronic Fatigue Syndrome/Myalgic Encephalomyelitis (CFS/ME)

Correction to: BMC Health Services Research (2011) 11:308. https://doi.org/10.1186/1472-6963-11-308.Following a report of a publications review jointly commissioned by the Health Research Authority and the University of Bristol, the authors would like to correct the following element of the ethics statement provided in the original article [1]:Ethical permission for the qualitative part of the study was granted by the North Somerset and South Bristol Research Ethics Committee (REC Reference number 09/H0106/81). The study was also approved by the Research and Development department of the RNHRD. The North Somerset & South Bristol Research Ethics Committee decided that the collection and analysis of these data for service evaluation did not require ethical review by an NHS research ethics committee or approval from the NHS R&D Office (REC reference number 07/Q2006/48).The original article [1] has been corrected.ReferencesWebb et al. What stops children with a chronic illness accessing health care: a mixed methods study in children with Chronic Fatigue Syndrome/Myalgic Encephalomyelitis (CFS/ME). BMC Health Serv Res. 2011;11:308. https://doi.org/10.1186/1472-6963-11-308