Dr Gemma Bryan

Research with young people suffering from a long-term illness has more recently incorporated the use of visual methods to foster engagement of research participants from a wide age range, capture the longitudinal and complex factors involved in young people's experiences of care, and allow young people to express their views in multiple ways. Despite its contributions, these methods are not always easy to implement and there is a possibility that they might not generate the results or engagement initially anticipated by researchers. We hope to expand on the emerging discussion on the use of participatory visual methods by presenting the practical issues we have faced while using this methodology during different stages of research: informed assent/consent, data collection, and the dissemination of findings. We propose a combination of techniques to make sure that the research design is flexible enough to allow research participants to shape the research process according to their needs and interests.

Background Cancer diagnosis in childhood or adolescence impacts significantly on school attendance, experience and educational outcomes. While there is longstanding recognition in clinical practice that these effects span the whole illness trajectory and continue beyond treatment completion, further clarity is required on the specific barriers and facilitators to education during cancer treatment and beyond, as well as on the experiences of children and adolescents across the full range of education settings (hospital, home, virtual, original school of enrolment), in order to determine which interventions are successful in improving access and experience from their perspective. The aim of this review is to identify what is known from the existing literature about access to and experience of education for children and adolescents with cancer during and post treatment. Methods We have planned a scoping literature review searching the following databases from inception onwards: MEDLINE (Ovid), Embase and Embase Classic, Web of Science Core Collection, Education Resources Index, Sociological Abstracts, APA PsycINFO, SCOPUS, CINAHL Plus, Emcare and The Cochrane Library. In addition, DARE, conference abstracts, key journals, and institutional websites will be searched. Arksey and O’Malley’s six-step process will be followed, including a consultation exercise. Studies, reports and policies from any country providing care and treatment for children and adolescents with cancer published in English will be considered eligible for inclusion. Two reviewers will independently screen all citations, full-text articles and abstract data. A narrative summary of findings will be conducted. Data analysis will involve quantitative (e.g., frequencies) and qualitative (e.g., content and thematic analysis) methods. Discussion This is a timely examination given the increased incidence of childhood cancer, more intensive treatment regimens and improved survival rates for childhood cancer. The inclusion of a substantive consultation exercise with families and professionals will provide an important opportunity to examine the scoping review outputs. Findings will assist the childhood cancer community in developing a comprehensive evidence-based understanding of a significant associated bio-psychosocial impact of cancer diagnosis and treatment and will form the first step towards developing effective interventions and policies to mitigate identified detrimental effects.

Rhabdomyosarcoma is the commonest soft tissue sarcoma in children. Around one-third of children with rhabdomyosarcoma experience relapse or have refractory disease, which is associated with a poor prognosis. This systematic review of early phase studies in pediatric relapsed/refractory rhabdomyosarcoma was conducted to inform future research and provide accurate information to families and clinicians making difficult treatment choices. Nine databases and five trial registries were searched in June 2021. Early phase studies of interventions for disease control in patients under 18 years old with relapsed/refractory rhabdomyosarcoma were eligible. No language/geographic restrictions were applied. Studies conducted after 2000 were included. Survival outcomes, response rates, quality of life and adverse event data were extracted. Screening, data extraction and quality assessment (Downs and Black Checklist) were conducted by two researchers. Owing to heterogeneity in the included studies, narrative synthesis was conducted. Of 16,965 records screened, 129 published studies including over 1100 relapsed/refractory rhabdomyosarcoma patients were eligible. Most studies evaluated systemic therapies. Where reported, 70% of studies reported a median progression-free survival ≤6 months. Objective response rate was 21.6%. Adverse events were mostly hematological. One-hundred and seven trial registry records of 99 studies were also eligible, 63 of which report they are currently recruiting. Study quality was limited by poor and inconsistent reporting. Outcomes for children with relapsed/refractory rhabdomyosarcoma who enroll on early phase studies are poor. Improving reporting quality and consistency would facilitate the synthesis of early phase studies in relapsed/refractory rhabdomyosarcoma (PROSPERO registration: CRD42021266254).

Background Xerostomia (the feeling of dry mouth) is a common symptom especially in older adults. Causes of dry mouth include medications, autoimmune disease (Sjögren's Syndrome), radiotherapy or chemotherapy for cancer, hormone disorders and infections. Objectives To determine which topical treatments for dry mouth are effective in reducing this symptom. Search methods We searched the following electronic databases: the Cochrane Oral Health Group Trials Register (28 October 2011), The Cochrane Central Register of Controlled Trials (CENTRAL) (The Cochrane Library, Issue 4 2011), MEDLINE via OVID (1950 to 28 October 2011), EMBASE via OVID (1980 to 28 October 2011), CINAHL via EBSCO (1980 to 28 October 2011), AMED via OVID (1985 to 28 October 2011), CANCERLIT via PubMed (1950 to 28 October 2011). Selection criteria We included randomised controlled trials of topical interventions such as lozenges, sprays, mouthrinses, gels, oils, chewing gum or toothpastes for the treatment of dry mouth symptom. We classified interventions into two broad categories, saliva stimulants and saliva substitutes, and these were compared with either placebo or another intervention. We included both parallel group and crossover trials. Data collection and analysis Two or more review authors independently carried out data extraction and assessed risk of bias. Trial authors were contacted for additional information as required. Main results Thirty‐six randomised controlled trials involving 1597 participants met the inclusion criteria. Two trials compared saliva stimulants to placebo, nine trials compared saliva substitutes to placebo, five trials compared saliva stimulants directly with saliva substitutes, 18 trials directly compared two or more saliva substitutes, and two trials directly compared two or more saliva stimulants. Only one trial was at low risk of bias and 17 were at high risk of bias. Due to the range of interventions, comparisons and outcome measures in the trials, meta‐analysis was possible for only a few comparisons. Oxygenated glycerol triester (OGT) saliva substitute spray shows evidence of effectiveness compared to an electrolyte spray (standardised mean difference (SMD) 0.77, 95% confidence interval (CI) 0.38 to 1.15) which corresponds to approximately a mean difference of 2 points on a 10‐point visual analogue scale (VAS) for mouth dryness. Both integrated mouthcare systems (toothpaste + gel + mouthwash) and oral reservoir devices show promising results but there is insufficient evidence at present to recommend their use. Although chewing gum is associated with increased saliva production in the majority of those with residual capacity, there is no evidence that gum is more or less effective than saliva substitutes. Authors' conclusions There is no strong evidence from this review that any topical therapy is effective for relieving the symptom of dry mouth. OGT spray is more effective than an aqueous electrolyte spray (SMD 0.77, 95% CI 0.38 to 1.15) which is approximately equivalent to a mean difference of 2 points on a 10‐point VAS scale for mouth dryness. Chewing gums appear to increase saliva production in those with residual secretory capacity and may be preferred by patients, but there is no evidence that gum is better or worse than saliva substitutes. Integrated mouthcare systems and oral reservoir devices may be helpful but further research is required to confirm this. Well designed, adequately powered randomised controlled trials of topical interventions for dry mouth, which are designed and reported according to CONSORT guidelines, are required to provide evidence to guide clinical care. For many people the symptom of dry mouth is a chronic problem and trials should evaluate whether treatments are palatable, effective in reducing xerostomia, as well as the long‐term effects of treatments on quality of life of those with chronic dry mouth symptoms.

Interventions for preventing oral mucositis for patients with cancer receiving treatment Treatment for cancer (including bone marrow transplant) can cause oral mucositis (severe ulcers in the mouth). This painful condition can cause difficulties in eating, drinking and swallowing, and may also be associated with infections which may require the patient to stay longer in hospital. Different strategies are used to try and prevent this condition, and the review of trials found that some of these are effective. Two interventions, cryotherapy (ice chips) and keratinocyte growth factor (palifermin®) showed some benefit in preventing mucositis. Sucralfate is effective in reducing the severity of mucositis, and a further seven interventions, aloe vera, amifostine, intravenous glutamine, granulocyte‐colony stimulating factor (G‐CSF), honey, laser and antibiotic lozenges containing polymixin/tobramycin/amphotericin (PTA) showed weaker evidence of benefit. These were evaluated in patients with different types of cancer, undergoing different types of cancer treatment. Benefits may be restricted to the disease and treatment combinations evaluated.

Background Previous studies have found that parents of children with cancer desire more prognostic information than is often given even when prognosis is poor. We explored in audio‐recorded consultations the kinds of information they seek. Methods Ethnographic study including observation and audio recording of consultations at diagnosis. Consultations were transcribed and analyzed using an interactionist perspective including tools drawn from conversation and discourse analysis. Results Enrolled 21 parents and 12 clinicians in 13 cases of children diagnosed with a high‐risk brain tumor (HRBT) over 20 months at a tertiary pediatric oncology center. Clinicians presented prognostic information in all cases. Through their questions, parents revealed what further information they desired. Clinicians made clear that no one could be absolutely certain what the future held for an individual child. Explicit communication about prognosis did not satisfy parents’ desire for information about their own child. Parents tried to personalize prognostic information and to apply it to their own situation. Parents moved beyond prognostic information presented and drew conclusions, which could change over time. Parents who were present in the same consultations could form different views of their child's prognosis. Conclusion Population level prognostic information left parents uncertain about their child's future. The need parents revealed was not for more such information but rather how to use the information given and how to apply it to their child in the face of such uncertainty. Further research is needed on how best to help parents deal with uncertainty and make prognostic information actionable.

The management of cough in adults with respiratory and non-respiratory illnesses is suboptimal and based mostly on clinical opinions rather than evidence. A systematic review was carried out assessing all trials in adult patients with respiratory and non-respiratory diseases (excluding cancer) that had chronic cough as primary or secondary outcome. A total of 1177 trials were retrieved and 75 met the criteria for inclusion in the review. The vast majority were in patients with asthma and chronic obstructive pulmonary disease (COPD). Cough was the primary outcome in less than one-quarter of the studies. The measurement of cough was variable, mostly using unvalidated scales or being part of an overall ‘symptoms’ score. Positive results were overall seen with the use of corticosteroids, leukotriene receptor antagonists, mast cell stabilizers, ipratropium bromide, neltenexine, iodinised glycerol and lidocaine. Speech pathology training and symptom monitoring through SMS messages (accompanied by treatment adjustments) have also shown promise. Evidence for established anti-tussive agents such as codeine was scarce, with positive studies from the 1960s, whilst more recent studies showed no effect in patients with COPD. Many studies had conflicting results. It is imperative that the management of cough and its evidence base be improved, using higher quality research designs and with cough being the primary outcome of trials.

Background A successful academic interview has been reported as the most important factor contributing to ranking of candidates for residency. However, little published guidance exists to help a prospective oncologist or researcher give such an interview. The International Society of Paediatric Oncology (SIOP) Young Investigator (YI) Network and Children's Oncology Group (COG) YI group thus cosponsored a survey of senior investigators seeking their advice. Methods An electronic survey covering aspects of the academic interview of both trainees and faculty were sent to all current/past mentors serving in the COG YI mentorship program and those registered as mentors in the SIOP YI mentorship program. The responses were quantitatively and qualitatively analyzed. Results The response rate was 43.7% (118/270) from 25 countries. Majority of United States (US) interviewers (86.8%) conducted interviews individually, while 74% of non‐US interviewers conducted panel interviews or both types equally (P 

Increased emphasis on the child’s voice and point of view in care and treatment has led to an expansion in the development of methods to access and identify their perspectives. Drawing on our experiences in a study of children with leukemia in hospital, this article explains the challenges and opportunities that arise in the use of five commonly used methods in a study of hospitalized children’s experiences with health care professionals, including: the ‘Draw and Write’ technique; a sticker activity; a paper person exercise; informal interviews; and participant observation. Each of these methods was examined with regard to ease of use, data generation, and utility of data for accessing children’s perspectives and development of initial clinical guidance.

Background: Many parents report a strong desire to take on information-giving roles, and believe they are best positioned to discuss their child’s illness with their child. Healthcare professionals have a supporting role to reduce the burden on parents who feel responsible for conveying information to their child and other family members. Objective: To examine parents’ and healthcare professionals’ perceptions of roles in receiving and communicating information when a child is diagnosed with and treated for acute lymphoblastic leukaemia. Design, setting and participants: We used the principles of a grounded theory approach. This was a single site study, recruiting from a principal children’s cancer treatment centre in the United Kingdom. The sample included parents of children receiving and completed treatment for acute lymphoblastic leukaemia (n=28), and healthcare professionals (n=34). Methods: Methods included individual interviews, face-to-face and telephone, focus groups, and an online forum. Findings: Communication ‘touch points’ are many over the course of a child’s cancer journey. We describe often ‘mismatched’ communication encounters where those seeking information and those providing information have different goals. Healthcare professionals in the encounter have expertise at the outset while parents have less expertise, but this expertise grows over time and this can increase the perceptions of this ‘mismatch’ and create different challenges. Conclusions: Considered in the context of middle range transition theory, we might suggest that parental foreground (seeking information directly) and background (passive actors) roles are the result of differing levels of uncertainty, and depend on the situation and preferences and child and family needs that may present differently over time in different contexts. Our work contributes to the emerging consensus that communication is more than a core set of skills that healthcare professionals just need to learn: clear specifications of mutual roles, responsibilities and a shared understanding of goals is also essential.

Non‐drug treatments for dry mouth symptoms Review question This review, carried out by authors of the Cochrane Oral Health Group, has been produced to assess the effects of non‐drug treatments used to stimulate saliva production for the relief of dry mouth (xerostomia) symptoms. Background Dry mouth is a common problem with an estimated incidence of between 10% and 26% in men and between 10% and 33% in women, which may or may not be due to reduced saliva secretion. Common causes of dry mouth include the side effects of many commonly prescribed medications, diseases (such as Sjögren's syndrome where the immune system destroys tissues in the glands which produce saliva) and radiotherapy treatments for head and neck cancers. Saliva moistens the skin in the mouth and helps to maintain oral health. The presence of saliva facilitates speech, acts to wash away food residue from around the teeth, neutralises potentially damaging food and bacterial acids, enhances a person's ability to taste the food, and generally lubricates the mouth. Saliva also acts to soften food, making it easier to chew and swallow. Enzymes in saliva start the digestion of starch and fats, and other substances in saliva, such as epidermal growth factors, promote tissue growth, differentiation and wound healing. The antibacterial, antifungal and antiviral agents in saliva balance the oral flora and help to prevent oral infections, while the minerals in saliva help to maintain tooth enamel. Non‐drug treatments such as acupuncture, mild electrical stimulation, lasers, tooth brushing and other stimulation techniques are used to improve dry mouth symptoms. Study characteristics The evidence on which this review is based was up‐to‐date as of 16 April 2013. Nine studies were included in this review. A total of 366 adult participants took part in these trials, with an average of 40 participants per trial, and an age range from 12 to 77 years. The causes of dry mouth were radiotherapy for oral cancers in four trials, Sjögren's syndrome in three trials, medication‐related in one trial, and in the remaining trial participants had a range of causes of dry mouth. The included studies were divided into three groups, according the interventions evaluated. 1. Five small studies with a total 153 participants evaluated acupuncture. 2. Three studies evaluated electrostimulation devices. 3. One study evaluated a power toothbrush. Key results The five studies evaluating the effects of acupuncture in people who had dry mouth were generally of poor quality. There was no evidence of a difference in dry mouth symptoms, but there was some evidence of a small increase in saliva production which persisted for a year after the end of the acupuncture treatment. There may not have been enough people included in the trials to show a difference in dry mouth, or it may have been that both the real acupuncture and the 'placebo' acupuncture had some beneficial effect. Acupuncture was associated with more adverse effects (tiny bruises and tiredness which were mild and temporary). The studies evaluating the effects of electrostimulation devices were poorly conducted and reported, and provided insufficient evidence to determine the effects of these devices on either dry mouth or saliva production. The single small study of a powered versus a manual toothbrush also found no difference for either dry mouth or saliva production. None of the included studies reported the outcomes of duration of effectiveness, quality of life, patient satisfaction, or oral health assessment. Quality of the evidence These studies were generally of poor quality (low and very low).

BACKGROUND: Observing and recording the signs and symptoms of oral mucositis are an important part of oral care, essential to the prevention and treatment of mucositis. Structured oral assessment enables a more informed and accurate identification of signs and symptoms and will enable early and individualized interventions. OBJECTIVE: A United Kingdom-based mouth-care group conducted a systematic review of the published literature through to March 2004 and repeated in 2008. The goal of this review was to identify and evaluate the range of instruments used to assess oral mucositis to recommend in evidence-based guidelines the "best" instrument to use in the field of children's and young people's cancer care. METHODS: Search sources included the Cochrane Library, MEDLINE, EMBASE, and CINAHL. Studies were selected using defined criteria and reviewed by 3 pairs of group members. RESULTS: Fifty-four individual oral assessment instruments were identified with only 15 reporting evidence of reliability and validity testing. Only 3 articles reported on oral assessment exclusively in our population. CONCLUSIONS: The guidelines recommend only 1 assessment instrument, the Oral Assessment Guide, or adaptations of this instrument, to be used in clinical practice. Five factors influenced this recommendation: purpose of assessment, population, outcomes assessed, and quality of the instrument and ease of use. IMPLICATIONS FOR PRACTICE: The Oral Assessment Guide has been consistently judged to be user-friendly and appropriate for everyday clinical practice with both adults and children, as well as a useful research tool.

Background Parents of children who are diagnosed with a poor-prognosis cancer want to be involved in making treatment-related decisions for their child. They often make repeated decisions depending on their child’s response to treatment and can experience decisional regret as a consequence. Understanding parent values and preferences when making treatment-related decisions may help enhance discussions with healthcare professionals and identify additional ways of providing support to this parent population. Objectives To explore parent values and preferences underpinning treatment decision-making for children receiving cancer-directed therapy for a poor prognosis cancer. Methods A scoping review of research literature and systematic reviews from qualitative, quantitative, and mixed methods studies was conducted following Joanna Briggs Institute methodology. Articles which included parents of a child who received cancer-directed therapy for a poor-prognosis childhood cancer, under the age of eighteen years were considered. Four electronic databases were searched (CINAHL, Medline, PsychINFO, Web of Science Core Collections). Reference and citation lists of all included full-text articles were also searched. Summative content analysis was used to synthesise findings and develop themes. Results Twelve articles were included. Parent decision-making was affected by underpinning factors: hope for a cure, fear of their child dying and uncertainty. Influencing factors: opinions of others, child’s wishes, and faith and religion had the potential to inform decision-making processes. Parents valued having enough time, being a good parent and being involved in decision-making. Preferences within these values varied resulting in the potential for conflict and ‘trade-offs’ in making decisions. Conclusions Parent decision-making in poor-prognosis childhood cancer is complex and extends beyond values and preferences. Underpinning factors and values are consistent through the decision-making process with influencing factors and preferences varying between parents. Preferences can conflict when parents want to continue cancer-directed therapy whilst maintaining their child’s quality of life or can change depending on a parents’ cognitive state as they realise cure might be unlikely.