How can trial designs better serve the needs of children and young people with juvenile idiopathic arthritis?

In juvenile idiopathic arthritis we have seen remarkable progress in the number of available licensed biological and small molecule treatments in the past two decades, leading to improved outcomes for patients. Designing clinical trials for these therapeutics is fraught with ethical, legislative, and practical challenges. However, many aspects of current clinical trial design in juvenile idiopathic arthritis do not meet the needs of patients and clinicians. Commonly used withdrawal trial designs raise substantial ethical concerns for patients and families who believe that they do not enable evidence-based and patient-centred decisions around medication choices. In this Viewpoint, we present the personal views of a patient and parent network that is of the opinion that current trial design in juvenile idiopathic arthritis is failing children and young people with juvenile idiopathic arthritis and set out the need for change informed by lived experience.

Children's behaviour following diagnosis of acute lymphoblastic leukaemia: a qualitative longitudinal study.

Survival rates for children with cancer have improved significantly, but lengthy and painful treatments compromise the quality of life of children and their families. The aim of this article is to describe children's behaviour and coping over the 2- or 3-year-course of treatment. We interviewed 32 mothers of children newly diagnosed with Acute Lymphoblastic Leukaemia (ALL) shortly following diagnosis, 1 and 2 years later. Thematic analysis was used to extract themes related to adjustment and behaviour. The results were organized separately for children in three age groups: 0-4 years, 5-9 years and 10-14 years. Mothers described children in the 0-4-year age group as adjusting well, and attributed this to their limited understanding and ability to integrate treatments into normal life. Children in the 5-9-year group were adjusting less well, experiencing social problems and worries about appearance. Older children (10-14 years) adjusted least well. Many withdrew socially and were concerned about the need to look and feel normal. These findings will be useful for parents and clinicians in identifying typical behaviours of children coping with ALL. They also have implications for the development of child-centred age-specific measures of quality of life in children treated for ALL.

Follow-up care for childhood cancer survivors: a focus group analysis.

Follow-up of survivors of childhood cancer is recommended to improve detection of late-effects, and provide individuals with information and advice. This study aimed to follow-up survivors of childhood cancer and report on their attitudes to current follow-up methods. Twenty-six survivors (13-25 years) of childhood cancer and their parent(s) attended focus groups (n = 7) to discuss views about follow-up care. Transcripts were analysed using interpretative phenomenological analysis (IPA). Three themes were identified: strategies to achieve a normal life (through playing down possibility of late-effects or careful monitoring of health); expectations about follow-up (facts and information, advice about self-care, everyday living, and psychosocial consequences) and preferences for different models of care. Given that some families had reservations about the benefits of follow-up, it is important that services address survivors' interests and meet their expectations. Changes to service delivery must take account of individual needs and expectations. Possible limitations of focus group methods (recruitment, bias reduction, methods of analysis and influence of other participants' views) are discussed.

'He Never Liked Sport Anyway' - Mother's Views of Young People Coping With a Bone Tumour in the Lower Limb.

PURPOSE: Treatment for a bone tumour can compromise quality of life (QOL), especially for young patients. We used qualitative methods to assess mothers' views of patients' experiences and their coping strategies at approximately 6 months after diagnosis (T1: n=12) and 12-18 months later (T2: n=11). SUBJECTS: Mothers of young people (aged 6-22 years) who were undergoing treatment for either osteosarcoma or Ewing's sarcoma in the lower limb took part. METHODS: A semi-structured interview was devised to assess participation in sport, social life, schooling and general mobility. Interviews were transcribed and analysed using content analysis. RESULTS: Mothers reported a number of situations in which QOL was compromised, and these remained relatively constant over time (mean=4.25 at T1 and 4.27 at T2). However, strategies to manage these difficulties changed from Problem (constructive attempts to deal with challenges) to Emotion (managing the situation through use of emotions) focused coping from T1 to T2. DISCUSSION: Although the sample size is small, our results suggest that patients adopt a variety of coping strategies to deal with the physical and social restrictions associated with disease and treatment. The findings suggest that young people continue to experience many stresses up to 18 months after diagnosis. The shift from Problem to Emotion focussed coping over time may suggest a degree of acceptance of practical problems.