What matters to you? Engaging with children in the James Lind Alliance Children's Cancer Priority Setting Partnership.

BACKGROUND: Previous priority setting exercises have sought to involve children, but in the final reporting, it is evident that few children had been engaged through the process. A primary aim in the Children's Cancer Priority Setting Partnership was to find out from children what they want research to focus on. We report on our experience to inform methods of engagement with children in future James Lind Alliance Priority Setting Partnerships and similar exercises. METHODS: We followed the James Lind Alliance process, collecting and shortlisting questions via online surveys with adult survivors of childhood cancer, carers, and professionals, and holding a final workshop. Alongside this, a parallel process to collect and prioritise questions from children was undertaken. We created animations for parents/carers to explain the project and surveys to children, gathered questions via online surveys and held a workshop with children to identify their priorities. RESULTS: Sixty-one children and young people with cancer and 10 siblings, aged 3-21 years, submitted 252 potential questions/topics via the surveys. Submissions were refined into 24 summary questions. These questions were discussed at a workshop with eight children; they also added more questions on topics of importance to them. Workshop participants prioritised the Top 5 questions; top priority was, 'How can we make being in hospital a better experience for children and young people? (like having better food, internet, toys, and open visiting so other family members can be more involved in the child's care)'. The Top 5 also included cancer prevention, treatments closer to home, early diagnosis, and emotional support. These questions were taken to the final workshop at which the Top 10 priorities were decided, all five children's priorities were reflected in the final Top 10. CONCLUSIONS: We have demonstrated that it is possible to successfully involve children directly in setting priorities for future research. Future priority setting exercises on topics relevant to children, should seek to include their views. The Children's Cancer Top 10 priorities reflect the voices of children and should inform the funding of future research.

Priority Setting Partnerships find out what areas of research are important to patients, families, and the professionals who care for them. Few Priority Setting Partnerships have involved children, so what matters to them may not have been well-represented. The Children's Cancer Priority Setting Partnership aimed to find out directly from children what research we should do. We collected questions/topics for research from children using online surveys. We made animations to explain the project and surveys to children. Two-hundred and fifty-two questions were sent in by 61 children and young people with cancer and 10 siblings. We grouped similar questions together into 24 summary questions. Summary questions were discussed at a workshop with eight children. Workshop participants added more questions on topics that mattered to them, and decided their Top 5 questions. The top question was, 'How can we make being in hospital a better experience for children and young people? (like having better food, internet, toys, and open visiting so other family members can be more involved in the child's care)'. The Top 5 questions included: preventing cancer, having treatments nearer home, early diagnosis, and emotional support. These questions were taken to the final project workshop, this was with adults, including childhood cancer survivors, where the Top 10 priorities were decided. All five children's priorities were included in the Top 10. We have shown it is possible to successfully involve children in setting research priorities. Future priority setting exercises on topics that affect children should actively seek and include their views.

Research priorities for children's cancer: a James Lind Alliance Priority Setting Partnership in the UK.

OBJECTIVES: To engage children who have experienced cancer, childhood cancer survivors, their families and professionals to systematically identify and prioritise research questions about childhood cancer to inform the future research agenda. DESIGN: James Lind Alliance Priority Setting Partnership. SETTING: UK health service and community. METHODS: A steering group oversaw the initiative. Potential research questions were collected in an online survey, then checked to ensure they were unanswered. Shortlisting via a second online survey identified the highest priority questions. A parallel process with children was undertaken. A final consensus workshop was held to determine the Top 10 priorities. PARTICIPANTS: Children and survivors of childhood cancer, diagnosed before age 16, their families, friends and professionals who work with this population. RESULTS: Four hundred and eighty-eight people submitted 1299 potential questions. These were refined into 108 unique questions; 4 were already answered and 3 were under active study, therefore, removed. Three hundred and twenty-seven respondents completed the shortlisting survey. Seventy-one children submitted questions in the children's surveys, eight children attended a workshop to prioritise these questions. The Top 5 questions from children were taken to the final workshop where 23 questions in total were discussed by 25 participants (young adults, carers and professionals). The top priority was 'can we find effective and kinder (less burdensome, more tolerable, with fewer short and long-term effects) treatments for children with cancer, including relapsed cancer?' CONCLUSIONS: We have identified research priorities for children's cancer from the perspectives of children, survivors, their families and the professionals who care for them. Questions reflect the breadth of the cancer experience, including diagnosis, relapse, hospital experience, support during/after treatment and the long-term impact of cancer. These should inform funding of future research as they are the questions that matter most to the people who could benefit from research.

Experiences of Low Mood in Young People With Cancer: A Qualitative Study.

Young people with cancer are at increased risk of psychological difficulty; yet there is little research on their experiences of low mood or depressive symptoms to help inform treatment interventions. This qualitative study explored experiences and opinions of low mood or depression in young people with cancer, how their mood related to developmental challenges, strategies used to cope, and how services could improve support. Nineteen young people diagnosed and treated for cancer completed semistructured interviews. Transcripts were thematically analyzed using the framework approach, and analysis produced eight themes, organized into three domains: "A Harder Time of Life," "Interpersonal Impact of Cancer," and "Learning to Understand and Describe Low Mood," Participants interviewed experienced low mood during cancer and predominantly felt "low mood" was a helpful term to describe their emotions. There were similarities and variations in their reported mood compared with clinical depression. The developmental challenges of being a young person with cancer negatively affected their mood. Participants used a variety of different coping strategies to manage these challenges. Young people were clear that they would like others to help them understand negative emotions experienced through cancer are normal to feel and support for low mood to be accessible and available.

Neurocognitive, academic and functional outcomes in survivors of infant ependymoma (UKCCSG CNS 9204).

PURPOSE: This is the first UK multi-centre case-controlled study with follow-up in excess of 10 years to report the neurocognitive, academic and psychological outcomes of individuals diagnosed with a brain tumour in early childhood. Children enrolled into the UKCCSG CNS 9204 trial, diagnosed with intracranial ependymoma when aged ≤ 36 months old, who received a primary chemotherapy strategy to defer or avoid radiotherapy, were recruited. METHODS: Outcomes of those who relapsed and subsequently received radiotherapy (n = 13) were compared to those enrolled who did not relapse (n = 16), age-matched controls-diagnosed with solid non-central nervous system (SN-CNS; n = 15) tumours or low-grade posterior fossa pilocytic astrocytoma (PFPA; n = 15), and normative data. Analyses compared nine neurocognitive outcomes as primary measures with quality of survival as secondary measures. RESULTS: Relapsed ependymoma participants performed significantly worse than their non-relapsed counterparts on measures of Full Scale IQ, Perceptual Reasoning, Word Reading and Numerical Operations. The relapsed ependymoma group performed significantly worse than SN-CNS controls on all primary measures, whereas non-relapsing participants only differed significantly from SN-CNS controls on measures of Processing Speed and General Memory. Relapsed ependymoma participants fared worse than all groups on measures of quality of survival. CONCLUSIONS: The relapsed irradiated ependymoma group demonstrated the most significantly impaired neurocognitive outcomes at long-term follow-up. Non-relapsing participants demonstrated better outcomes than those who relapsed. Results tentatively suggest avoiding radiotherapy helped preserve neurocognitive and learning outcomes of individuals diagnosed with ependymoma when aged ≤ 36 months old. Prospective neurocognitive surveillance is required. Recommendations for clinical and research practice are provided.

Cause and outcome of cerebellar mutism: evidence from a systematic review.

PURPOSE: Cerebellar mutism is a serious neurosurgical complication after posterior fossa surgery, but the cause, incidence and outcome remain incompletely defined. The aim of this paper was to identify and review all reports of this phenomenon to better delineate and improve the evidence base. METHODS: A systematic search and retrieval of databases was conducted using advanced search techniques. Review/outcomes criteria were developed, and study quality was determined. RESULTS: The retrieval identified 2,281 papers of which 96 were relevant, identifying 650 children with cerebellar mutism. Causative factors, clinical features and outcomes were reported variably; papers focussed on multiple areas, the majority reporting incidence in single or series of case studies with little or no analysis further than description. CONCLUSIONS: The complexity and variability of data reporting, likely contributing factors and outcomes make cerebellar mutism difficult to predict in incidence and the degree of impact that may ensue. A clear and accepted universal definition would help improve reporting, as would the application of agreed outcome measures. Clear and consistent reporting of surgical technique remains absent. Recommendations for practice are provided.

Effectiveness of melatonin for sleep impairment post paediatric acquired brain injury: evidence from a systematic review.

OBJECTIVE: To retrieve and review all the relevant literature describing the administration of melatonin to treat impaired sleep in children following acquired brain injury (ABI). METHODS: A systematic search and retrieval of the literature was conducted using advanced search techniques. The retrieval identified 589 papers, seven of which were relevant. Review/outcomes criteria were developed and study quality was determined. RESULTS: There is paucity of high-quality evidence to support use of melatonin for sleep impairment post paediatric ABI. Variation in dosage, screening and outcome measures, data reporting and a lack of impairment delineation and treatment stratification were recurrent themes. CONCLUSION: Retrieved evidence for the effectiveness of melatonin for post paediatric ABI sleep impairment appears promising. There is a clear need for further study in this area to inform clinical and research practices. Recommendations are given.