Increased levels of anti-BSA antibodies in children with Down syndrome.

Introduction: Autoimmune diabetes occurs more often in the first 2 years of life in children with Down syndrome (DS) compared with the general population. We previously observed increased frequencies of islet autoantibodies, including insulin autoantibodies (IAA), in children with DS. Assays for IAA using 125I-labelled insulin require competition to overcome cross reactivity with antibodies to the cow's milk protein, bovine serum albumin (BSA). 125I-IAA assay results suggested that levels of antibodies to BSA may also be increased in children with DS. The aim of this study therefore was to determine whether the levels of anti-BSA antibodies differed in children with DS compared with controls. Methods: Samples were available from two populations with DS: one from the UK, (UK DS cohort n=106, 58 male, median age 12.5 years) and one from Estonia (Estonian DS cohort: n=121, 65 male, median age 9.75 years). A UK control population was provided by sex and age-matched healthy siblings of probands participating in the Bart's Oxford (BOX) family study of type 1 diabetes. A competitive-displacement radiobinding assay (RBA) and a Dissociation Enhanced Lanthanide Fluoroimmunoassay (DELFIA) were developed to measure and confirm anti-BSA antibody levels. HLA class II genotype was analysed by PCR using sequence specific primers (PCR-SSP). Results: Overall, levels of anti-BSA antibodies were increased in those with DS compared with controls (p<0.0001) but this was not HLA associated. Conclusion: Increased levels of anti-BSA antibodies may reflect a defect in immune maturation or increased gut permeability in children with DS, increasing their risk of developing autoimmunity.

The importance of school in the management of Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS): issues identified by adolescents and their families.

Paediatric Myalgic Encephalomyelitis (ME)/Chronic Fatigue Syndrome (CFS) is a disabling condition. Schools play a key role in adolescents' experiences with managing ME/CFS. However, little is known about the experiences of adolescents with ME/CFS (and their families) in schools. This paper is an incidental qualitative study, which combines data from two independent ME/CFS studies: study 1 researched ethnic minority adolescents with ME/CFS; study 2 explored Acceptance and Commitment Therapy for adolescents with ME/CFS who had not recovered after one year. Participants included: adolescents with ME/CFS; their families; and medical professionals (ME/CFS specialists and non-specialists). Adolescents, their families, and ME/CFS medical professionals were recruited from a UK specialist paediatric ME/CFS service. Non-ME/CFS medical professionals were recruited from the same region. Semi-structured qualitative interviews and focus groups were undertaken. Participants' views on schools from each study were combined and thematic analysis was used to identify themes. Fifteen adolescents with ME/CFS (11-17 years old), sixteen family members, and ten medical professionals (GPs, school nurses and ME/CFS specialists) were interviewed. Four key themes were found: (1) adolescents identified school was important for aiding ME/CFS recovery, especially educationally and socially; (2) families described varying levels of support from schools and local authorities with help managing ME/CFS - some described significant practical and emotional difficulties to accessing education, whereas others recounted examples of positive supportive strategies, particularly when teachers had previous experience or knowledge of ME/CFS; (3) parents thought three-way communication between schools, healthcare and families could improve support; (4) participants felt schools were an appropriate place for knowledge building and raising awareness of ME/CFS amongst teachers and pupils, to aid improved supportive measures. In conclusion, this paper provides rich data that highlights the importance of education and the realistic fears and hurdles for adolescents with ME/CFS remaining engaged in education and the impact on their future. Some families described positive strategies in school, which were viewed as helpful to manage ME/CFS in the classroom. These strategies could be implemented alongside knowledge building initiatives and improved communication between healthcare and education. There is a need to further investigate useful strategies and determine how teachers can be best supported in implementing them.

Qualitative study of the acceptability and feasibility of acceptance and commitment therapy for adolescents with chronic fatigue syndrome.

Background: Paediatric chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME) is disabling and relatively common. Although evidenced-based treatments are available, at least 15% of children remain symptomatic after one year of treatment. Acceptance and commitment therapy (ACT) is an alternative therapy option; however, little is known about whether it is an acceptable treatment approach. Our aim was to find out if adolescents who remain symptomatic with CFS/ME after 12 months of treatment would find ACT acceptable, to inform a randomised controlled trial (RCT) of ACT. Methods: We recruited adolescents (diagnosed with CFS/ME; not recovered after one year of treatment; aged 11-17 years), their parent/carer and healthcare professionals (HCPs) from one specialist UK paediatric CFS/ME service. We conducted semi-structured interviews to explore barriers to recovery; views on current treatments; acceptability of ACT; and feasibility of an effectiveness RCT. Thematic analysis was used to identify patterns in data. Results: Twelve adolescents, eleven parents and seven HCPs were interviewed. All participants thought ACT was acceptable. Participants identified reasons why ACT might be efficacious: pragmatism, acceptance and compassion are valued in chronic illness; values-focussed treatment provides motivation and direction; psychological and physical needs are addressed; normalising difficulties is a useful life-skill. Some adolescents preferred ACT to cognitive behavioural therapy as it encouraged accepting (rather than challenging) thoughts. Most adolescents would consent to an RCT of ACT but a barrier to recruitment was reluctance to randomisation. All HCPs deemed ACT feasible to deliver. Some were concerned patients might confuse 'acceptance' with 'giving up' and called for clear explanations. All participants thought the timing of ACT should be individualised. Conclusions: All adolescents with CFS/ME, parents and HCPs thought ACT was acceptable, and most adolescents were willing to try ACT. An RCT needs to solve issues around randomisation and timing of the intervention.

Same but different? A thematic analysis on adalimumab biosimilar switching among patients with juvenile idiopathic arthritis.

BACKGROUND: Biologic medications have dramatically enhanced the treatment of many chronic paediatric inflammatory conditions. Their high cost is a factor that prohibits their broader use. Cheaper generic versions, or biosimilars, are increasingly being used. Healthcare services are switching some patients over to biosimilars for economic reasons, known as 'non-medical switching'. Some patients unsuccessfully switch due to perceived decreases in efficacy or non-specific drug effects. The implications of failed switching include exhaustion of therapeutic options, unnecessary exposure to other medications, increased healthcare utilisation, worse patient outcomes and higher overall healthcare costs. Patient perceptions almost certainly play a role in these 'failed switches'. METHODS: A thematic analysis was performed to better understand patient and parent perceptions on non-medical biosimilar switching. The study was conducted in accordance with the Consolidated Criteria for Reporting Qualitative Research recommendations. Patients with juvenile idiopathic arthritis currently taking adalimumab were included. RESULTS: Nine families were interviewed just prior to a hospital trust-wide non-medical switch to an adalimumab biosimilar. Several common themes were identified. The most frequent concerns were regarding practical aspects of the switch including the medication administration device type; the colour of the medication and administration device; and whether the injections would sting more. The relative safety and efficacy of the biosimilar was raised although most families felt that there would be no significant difference. Anxieties about the switch were largely placated by reassurances from the medical team. CONCLUSIONS: We derived recommendations based on existing adult literature and the observations from our study to optimise the benefits from non-medical biosimilar switching.