A qualitative study to explore the burden of disease in activated phosphoinositide 3-kinase delta syndrome (APDS).

BACKGROUND: Activated phosphoinositide 3-kinase delta syndrome (APDS) is an ultra-rare primary immunodeficiency, with only 256 cases reported globally. This study aimed to explore the disease burden of APDS from the perspective of individuals with APDS and their caregivers. METHODS: Qualitative interviews were conducted with healthcare providers (HCPs), individuals with APDS and caregivers, to explore the symptoms and health-related quality of life (HRQoL) impact of APDS. Some individuals and caregivers also completed a narrative account exercise. All interviews were audio recorded and transcribed. Data were analysed using thematic analysis and saturation was recorded. RESULTS: Semi-structured qualitative interviews were conducted with healthcare providers (HCPs), individuals with APDS and caregivers. Individuals and caregivers had the option of completing a narrative account exercise. Six HCPs participated in an interview. Seven participants completed the narrative account exercise (N = 5 caregivers and N = 2 individuals with APDS) and 12 took part in an interview (N = 4 caregivers and N = 8 individuals with APDS). Themes identified from HCPs interviews included symptoms, clinical manifestations, HRQoL impacts and treatments/management of APDS. The narrative account exercise identified similar themes, but with the addition to the journey to diagnosis. These themes were explored during the individual/caregiver interviews. Reported clinical manifestations and symptoms of APDS included susceptibility to infections, lymphoproliferation, gastrointestinal (GI) disorders, fatigue, bodily pain, and breathing difficulties. HRQoL impacts of living with APDS included negative impacts to daily activities, including work, education and social and leisure activities, physical functioning, as well as emotional well-being, such as concern for the future, and interpersonal relationships. Impacts to caregiver HRQoL included negative impacts to physical health, work, emotional well-being, interpersonal relationships and family life and holidays. The management of APDS included the use of healthcare services and medications including immunoglobulin replacement therapy (IRT), rapamycin, prophylactic antibiotics, leniolisib, as well as medical procedures due to complications. CONCLUSIONS: APDS has a high disease burden and there is an unmet need for licensed, more targeted treatments which modify disease progression. This study was the first to describe the day-to-day experience and HRQoL impact of APDS from the perspective of individuals living with the condition, caregivers and treating physicians.

Caregivers' Perspectives on the Impact of Cannabidiol (CBD) Treatment for Dravet and Lennox-Gastaut Syndromes: A Multinational Qualitative Study.

OBJECTIVE: To increase understanding of the impact of cannabidiol (CBD) on outcomes beyond seizure control among individuals with Dravet syndrome or Lennox-Gastaut syndrome. METHODS: Qualitative interviews were conducted with caregivers of individuals with Dravet syndrome or Lennox-Gastaut syndrome treated with plant-derived, highly purified CBD medicine (Epidiolex in the USA; Epidyolex in Europe; 100 mg/mL oral solution). Symptoms and impacts of Dravet syndrome and Lennox-Gastaut syndrome on individuals were explored, as were the effects of CBD. Data were analyzed using thematic analysis. RESULTS: Twenty-one caregivers of individuals with Dravet syndrome (n = 14) and Lennox-Gastaut syndrome (n = 7) aged 4-22 years participated. Health-related quality of life improvements associated with CBD included cognitive function, communication, behavior, mobility, and participation in daily activities. Seizure frequency reduction was commonly reported (n = 12), resulting in caregivers having greater freedom and family life being less disrupted. Adverse events were reported by 10 caregivers. CONCLUSION: In addition to reduced seizure frequency, CBD may have a wide range of beneficial effects beyond seizure control that warrant further investigation.

Shared decision-making and the caregiver experience in tuberous sclerosis complex: results from a UK survey.

BACKGROUND: Tuberous sclerosis complex (TSC) is a rare genetic condition commonly accompanied by neurological and neuropsychological disorders, resulting in a high burden of illness for individuals and a substantial impact on their caregivers. Due to the diversity and complexity of clinical manifestations, patients with TSC need aligned multidisciplinary healthcare services starting in childhood through to adulthood. However, patients and caregivers are sometimes dissatisfied with the care provided, for which one of the most common reasons is a lack of involvement in clinical decision-making. Shared decision-making, whereby clinicians make clinical management decisions together with patients and their caregivers, is advocated for in the management of epilepsy, but evidence of its benefit in managing TSC is currently lacking. In this cross-sectional, UK-based analysis we used an online survey to capture the experiences of primary caregivers for individuals with TSC, including the impact on work productivity, clinical shared decision-making, satisfaction with care, and the impact of the coronavirus disease 2019 (COVID-19) pandemic. RESULTS: In total, 73 eligible caregivers provided consent (analysis set), with 14 completing the survey partially and 59 completing the full survey. Many caregivers (72%) reported receiving recommendations about new treatments from their doctor and discussing the treatment together, with a high proportion (89%) preferring that treatment was initiated at a low dose. Most caregivers (69%) were satisfied or extremely satisfied with pediatric TSC healthcare services, but only 25% were satisfied or extremely satisfied with the transition to adult TSC healthcare services. Several (n = 30) caregivers specified the impact of caring on their work productivity and career in optional open-ended survey responses. Finally, 80% of caregivers indicated that the COVID-19 pandemic had a "large" or "very large" impact on their caring activities, negatively affecting the emotional wellbeing and behavior of individuals with TSC, and caregivers' ability to work and arrange medical appointments. CONCLUSIONS: Caregivers largely feel involved in treatment decisions, and the majority were satisfied with healthcare services for children with TSC. However, many highlighted the need for an improved transition from pediatric to adult healthcare services. The survey also showed that COVID-19 has considerably affected caregivers and individuals with TSC.

The Burden of Caring for Individuals with Tuberous Sclerosis Complex (TSC) Who Experience Epileptic Seizures: A Descriptive UK Survey.

INTRODUCTION: Tuberous sclerosis complex (TSC) is a rare multisystem genetic condition characterised by benign tumours; prevalent manifestations include epilepsy and neuropsychiatric disorders. This study examined the burden of TSC for primary caregivers and families, exploring the impact of characteristics such as seizures. METHODS: Primary caregivers of individuals with TSC in the United Kingdom participated in an online survey, comprising the Pediatric Quality of Life Inventory™ Family Impact Module, Hospital Anxiety and Depression Scale (HADS), and TSC-specific items. Responses were analysed using descriptive and regression analysis statistics (closed-ended) or qualitative content analysis (open-ended). RESULTS: Seventy-three participants partially completed and 59 fully completed the survey; 95% were female, and 90% were parents of an individual with TSC. A median (range) of 2 (1-11) household members were carers. Primary caregivers spent a mean (standard deviation [SD]) of 104.3 (51.7) hours caring in the previous week, reporting high mean (SD) HADS scores of 11.2 (4.8) (anxiety) and 7.9 (4.4) (depression) and considerable family burden. Increased seizure frequency increased hours spent caring by primary caregivers (p = 0.01) and was associated with a decreased mean (SD) family functioning score of 46.2 (23.0) and parent health-related quality of life (HRQL) score of 45.4 (20.3) (both p = 0.03). Multivariable models predicted intellectual disability increased hours spent caring by primary caregivers (p = 0.01-0.04), and neuropsychiatric comorbidities decreased family functioning (p = 0.02) and caregiver HRQL (p < 0.01). CONCLUSION: These findings highlight the role of epileptic seizures and neuropsychiatric disorders in the considerable burden of TSC on primary caregivers and families.

Demographic and psychological predictors of community pharmacists' cancer-related conversations with patients: a cross-sectional analysis and survey study.

BACKGROUND: There is increasing interest in the role of community pharmacy in the early diagnosis and prevention of cancer. This study set out to examine how often community pharmacists (CPs) encourage patients to spot or respond to potential signs and symptoms of cancer, and how often they help people to make an informed decision about taking part in bowel cancer screening. METHODS: Data from 400 UK CPs, who completed the 2018 Cancer Research UK Healthcare Professional Tracker survey, were analysed. The primary outcomes were: 'how often CPs encourage patients to spot or respond to potential signs and symptoms of cancer' and 'how often CPs encourage eligible people to make an informed decision to participate in bowel cancer screening'. Associations between behaviours and demographic and psychological variables (Capability, Opportunity and Motivation) were assessed using multivariate logistic regression. RESULTS: Most (n = 331, 82.8%) CPs reported occasionally, frequently or always encouraging patients to spot or respond to potential signs and symptoms of cancer, while only half (n = 203, 50.8%) reported occasionally, frequently or always helping people make an informed decision to participate in bowel cancer screening. Female sex (aOR: 3.20, 95%CI: 1.51, 6.81; p < 0.01) and increased Opportunity (aOR: 1.72, 95%CIs: 1.12, 2.64; p < 0.05) and Motivation (aOR: 1.76, 95%CIs: 1.37, 2.27; p < 0.001) were associated with encouraging patients to spot or respond to potential signs and symptoms of cancer; all three psychological variables were associated with helping people to make an informed decision to participate in bowel cancer screening (Capability: aOR: 1.39, 95%CIs: 1.26, 1.52, p < 0.001; Opportunity: aOR: 1.44, 95%CIs: 1.11, 1.87; p < 0.01; Motivation: aOR: 1.45, 95%CIs: 1.05, 2.00; p < 0.05). CONCLUSIONS: Most CPs encourage patients to spot or respond to potential cancer symptoms, while only half help them make an informed decision to participate in bowel cancer screening. A multifaceted approach, targeting multiple COM-B components, is required to change these behaviours.

Symptoms and impacts of aromatic L-amino acid decarboxylase (AADC) deficiency among individuals with different levels of motor function.

BACKGROUND: Aromatic L-amino acid decarboxylase (AADC) deficiency is a rare neurological disorder associated with a range of symptoms and functional impairments. The aim of this study was to describe the experience of AADC deficiency across five different motor milestone health states. METHODS: Qualitative interviews were conducted with caregivers of individuals with AADC deficiency in Italy, Spain, Portugal and the United States. An interview guide was developed with input from clinical experts and caregivers, and explored the symptoms and impacts of AADC deficiency. Interviews were conducted by telephone and were recorded and transcribed. Data were analysed using thematic analysis and the symptoms and impacts were compared across health states. RESULTS: Fourteen caregivers took part, who provided care to 13 individuals with AADC deficiency aged 1-15 years. Six individuals were in the 'no motor function' health state, one in the 'sitting unsupported' health state, one in the 'standing/stepping when fully supported' health state and five in the 'walking with minimal support' health state. The results highlight a substantial impact of AADC deficiency, even among those who were able to walk with minimal support. Overall, those with better motor function also had better functional hand use, communication skills, ability to eat and perform other activities independently, and interact with their peers. The burden of caring was high across all health states, but caregivers of individuals in the walking health state were better able to participate in social and leisure activities. CONCLUSION: Individuals with higher levels of motor function had less severe symptoms and were better able to perform their daily, leisure and social activities. Treatments which improve motor function have the potential to improve other aspects of the lives of individuals with AADC deficiency and their caregivers.

Patient and Caregiver Health State Utilities in Tuberous Sclerosis Complex.

BACKGROUND: Tuberous sclerosis complex (TSC) is a rare multisystem disorder often associated with treatment-resistant epilepsy. Cost-effectiveness analysis for new antiseizure medications typically requires health state utilities (HSUs) that reflect the burden of a given condition. OBJECTIVE: This study aimed to estimate HSUs, with a focus on valuing the impact of seizure type and seizure frequency on health-related quality of life (HRQL) for patients with TSC and their caregivers. METHODS: A targeted literature review and qualitative research with healthcare professionals and caregivers informed the development of health state vignettes describing the experience of living with TSC or caring for a child with TSC. Vignettes were evaluated in interviews with the UK general population using the time trade-off (TTO) method. RESULTS: Sixteen vignettes were developed describing patient HRQL (n = 8) and caregiver HRQL (n = 8). Two hundred interviews were conducted via online video calls due to COVID-19 pandemic restrictions. Two hundred participants evaluated the patient (n = 100) and caregiver (n = 100) health state vignettes. Estimated utility scores varied consistently according to seizure type and seizure frequency. Patient TTO utility scores ranged between -0.234 (highest seizure frequency and multiple seizure types) and 0.725 (seizure-free state). Caregiver TTO utility scores ranged from 0.221 to 0.905. CONCLUSIONS: Findings highlight the substantial burden of living with TSC and caring for a child with TSC. Patient and caregiver burden was greater for generalised versus focal seizures. The burden was greatest for a combination of both seizure types and worsened with increasing seizure frequency.

The impact of caring for an individual with aromatic l-amino acid decarboxylase (AADC) deficiency: a qualitative study and the development of a conceptual model.

BACKGROUND: Aromatic l-amino acid decarboxylase (AADC) deficiency is a rare neurological condition, associated with a wide range of symptoms and functional issues, such as profound motor impairment and learning disability. Most individuals with AADC deficiency are completely dependent on their caregivers. This study explored the impact of caring for an individual with AADC deficiency. METHODS: Qualitative interviews were conducted with caregivers of individuals with AADC deficiency in Italy, Portugal, Spain and the United States. An interview guide was developed with input from clinical experts and caregivers and included questions on the impact of caring for an individual with AADC deficiency. Interviews were conducted by telephone/videoconference and were recorded and transcribed. Data were analysed using thematic analysis. RESULTS: Fourteen caregivers took part who provided care to 13 individuals with AADC deficiency aged 1-15 years. Caregivers reported that their lives centred around the individual with AADC deficiency, due to their need for 24-hour care and regular healthcare appointments. They reported both proximal impacts (impact on time, planning, physical health and emotional wellbeing), and distal impacts (impact on social/leisure activities, relationships, work and finances). These concepts and relationships were illustrated in a conceptual model. CONCLUSIONS: This is the first qualitative study to report on the experience of caring for an individual with AADC deficiency. Caregivers reported that caring had a substantial multifaceted impact on their lives. These findings highlight the importance of considering the caregiver experience when evaluating the burden of AADC deficiency.

Symptoms and impact of aromatic l-amino acid decarboxylase (AADC) deficiency: a qualitative study and the development of a patient-centred conceptual model.

BACKGROUND: Aromatic l-amino acid decarboxylase (AADC) deficiency is a rare neurological condition, with an estimated global prevalence of 1:32,000 to 1:90,000 live births. AADC deficiency is associated with a range of symptoms and functional impairments, but these have not previously been explored qualitatively. This study aimed to understand the symptoms of AADC deficiency and its impact on individuals' health-related quality of life. METHODS: Qualitative interviews were conducted with caregivers of individuals with AADC deficiency in Italy, Spain, Portugal and the United States. An interview guide was developed with input from clinical experts and caregivers, and explored the symptoms and impacts of AADC deficiency. Interviews were conducted by telephone and were recorded and transcribed. Data were analysed using thematic analysis and saturation was recorded. RESULTS: Fourteen caregivers took part, who provided care to 13 individuals with AADC deficiency aged 1-15 years. All individuals had impaired motor function, which was attributed to low muscle tone and muscle weakness. The level of motor function varied considerably, ranging from no motor function (no head control) to being able to take a few steps without support. Other impairments included cognitive impairment, communication difficulties, movement disorders (e.g. oculogyric crises), gastrointestinal symptoms, eating difficulties, fatigue and sleep disruption. Most individuals were completely dependent on their caregivers for all aspects of their lives. This limited function had a negative impact on their ability to socialise with their peers and on their emotional wellbeing. These concepts and relationships are illustrated in a conceptual model, and moderating factors (e.g. physiotherapy and medication) are discussed. CONCLUSION: This is the first qualitative study to report on the experience of living with AADC deficiency. Caregivers report individuals with AADC deficiency experience a wide range of symptoms and functional impairments, which have a substantial impact on their health-related quality of life.

Predictors of intention translation in flexible sigmoidoscopy screening for colorectal cancer.

OBJECTIVE: This prospective study aimed to identify predictors of intention and subsequent attendance of flexible sigmoidoscopy screening using constructs derived from the Health Belief Model (HBM). METHOD: A total of 4,330 people aged 54 years and registered at 1 of 83 participating English general practices were sent a preinvitation questionnaire to assess sociodemographics, HBM variables including perceived benefits, barriers, seriousness, health motivation, and external cues to action as well a range of other constructs and personal characteristics known to relate to cancer screening. RESULTS: Of the 1,578 respondents (36.4%), 1,555 (98.5%) answered the intention question: 52.9% stated definitely yes, 38.1% probably yes, 6.8% probably not, and 2.2% definitely not. Intentions were positively associated with a higher score on a scale of benefits (odds ratio [OR] = 4.62; 95% confidence intervals [CI; 3.24, 6.59]) and health motivation, that is, interest in other ways of preventing colorectal cancer (OR = 2.61; 95% CI [1.62, 4.22]), while a higher score on perceived barriers (OR = 0.19; 95% CI [0.12, 0.31]) and currently following recommended healthy lifestyle behaviors (OR = 0.31; 95% CI [0.16, 0.59]) were negatively associated. Attendance was verified for 922 intenders (65.2%) of whom 737 (79.9%) attended. Attendance was predicted by health motivation (OR = 1.75; 95% CI [1.07, 2.86]), perceived benefits (OR = 1.82; 95% CI [1.37, 2.43]), perceived barriers (OR = 0.47; 95% CI [0.32, 0.69]), individual-level deprivation (OR = 0.26; 95% CI [0.14, 0.50]), and having diabetes (OR = 0.48; 95% CI [0.25, 0.94]). CONCLUSION: This study supported the usefulness of the HBM in predicting cancer screening and was further enhanced by adding non-HBM variables such as individual socioeconomic deprivation and comorbidities. (PsycINFO Database Record (c) 2019 APA, all rights reserved).

Understanding primary care nurses' contribution to cancer early diagnosis: A systematic review.

PURPOSE: Primary care nurses can contribute to cancer early diagnosis. The objective of this systematic review was to identify, appraise and synthesise evidence on primary care nurses' contribution towards cancer early diagnosis in developed countries. METHOD: The following databases were searched in September 2017: MEDLINE, PsychINFO, CINAHL, SCOPUS, and EMBASE. Data were extracted on nurses': knowledge of cancer; frequency of 'cancer early diagnosis-related discussions' with patients; and perceived factors influencing these discussions. Studies were appraised using the Mixed Methods Appraisal Tool. RESULTS: Twenty-one studies were included from: United States, United Kingdom, Ireland, Spain, Turkey, Australia, Brazil and Middle East. Studies were mostly of low quality (one did not meet any appraisal criteria, 15 met one, four met two, and one met three). Nurses' knowledge of cancer, and their frequency of 'cancer early diagnosis-related discussions', varied across countries. This may be due to measurement bias or nurses' divergent roles across healthcare systems. Commonly perceived barriers to having screening discussions included: lack of time, insufficient knowledge and communication skills, and believing that patients react negatively to this topic being raised. CONCLUSIONS: Findings suggest a need for nurses to be adequately informed about, and have the confidence and skills to discuss, the topic of cancer early diagnosis. Further high-quality research is required to understand international variation in primary care nurses' contribution to this field, and to develop and evaluate optimal methods for preparing them for, and supporting them in, this.

Using specialist screening practitioners (SSPs) to increase uptake of bowel scope (flexible sigmoidoscopy) screening: results of a feasibility single-stage phase II randomised trial.

OBJECTIVE: To determine the feasibility of specialist screening practitioners (SSPs) offering patient navigation (PN) to facilitate uptake of bowel scope screening (BSS) among patients who do not confirm or attend their appointment. DESIGN: A single-stage phase II trial. SETTING: South Tyneside District Hospital, Tyne and Wear, England, UK. PARTICIPANTS: Individuals invited for BSS at South Tyneside District Hospital during the 6-month recruitment period were invited to participate in the study. INTERVENTION: Consenting individuals were randomly assigned to either the PN intervention or usual care group in a 4:1 ratio. The intervention involved BSS non-attenders receiving a phone call from an SSP to elicit their reasons for non-attendance and offer educational, practical and emotional support as required. If requested by the patient, another BSS appointment was then scheduled. PRIMARY OUTCOME MEASURE: The number of non-attenders in the intervention group who were navigated and then rebooked and attended their new BSS appointment. SECONDARY OUTCOME MEASURES: Barriers to BSS attendance, patient-reported outcomes including informed choice and satisfaction with BSS and the PN intervention, reasons for study non-participation, SSPs' evaluation of the PN process and a cost analysis. RESULTS: Of those invited to take part (n=1050), 152 (14.5%) were randomised into the study: PN intervention=109; usual care=43. Most participants attended their BSS appointment (PN: 79.8%; control: 79.1%) leaving 22 eligible for PN: only two were successfully contacted. SSPs were confident in delivering PN, but were concerned that low BSS awareness and information overload may have deterred patients from taking part in the study. Difficulty contacting patients was reported as a burden to their workload. CONCLUSIONS: PN, as implemented, was not a feasible intervention to increase BSS uptake in South Tyneside. Interventions to increase BSS awareness may be better suited to this population. TRIAL REGISTRATION NUMBER: ISRCTN13314752; Results.

Faecal immunochemical tests versus colonoscopy for post-polypectomy surveillance: an accuracy, acceptability and economic study.

BACKGROUND: In the UK, patients with one or two adenomas, of which at least one is ≥ 10 mm in size, or three or four small adenomas, are deemed to be at intermediate risk of colorectal cancer (CRC) and referred for surveillance colonoscopy 3 years post polypectomy. However, colonoscopy is costly, can cause discomfort and carries a small risk of complications. OBJECTIVES: To determine whether or not annual faecal immunochemical tests (FITs) are effective, acceptable and cost saving compared with colonoscopy surveillance for detecting CRC and advanced adenomas (AAs). DESIGN: Diagnostic accuracy study with health psychology assessment and economic evaluation. SETTING: Participants were recruited from 30 January 2012 to 30 December 2013 within the Bowel Cancer Screening Programme in England. PARTICIPANTS: Men and women, aged 60-72 years, deemed to be at intermediate risk of CRC following adenoma removal after a positive guaiac faecal occult blood test were invited to participate. Invitees who consented and returned an analysable FIT were included. INTERVENTION: We offered participants quantitative FITs at 1, 2 and 3 years post polypectomy. Participants testing positive with any FIT were referred for colonoscopy and not offered further FITs. Participants testing negative were offered colonoscopy at 3 years post polypectomy. Acceptibility of FIT was assessed using discussion groups, questionnaires and interviews. MAIN OUTCOME MEASURES: The primary outcome was 3-year sensitivity of an annual FIT versus colonoscopy at 3 years for detecting advanced colorectal neoplasia (ACN) (CRC and/or AA). Secondary outcomes included participants' surveillance preferences, and the incremental costs and cost-effectiveness of FIT versus colonoscopy surveillance. RESULTS: Of 8008 invitees, 5946 (74.3%) consented and returned a round 1 FIT. FIT uptake in rounds 2 and 3 was 97.2% and 96.9%, respectively. With a threshold of 40 µg of haemoglobin (Hb)/g faeces (hereafter referred to as µg/g), positivity was 5.8% in round 1, declining to 4.1% in round 3. Over three rounds, 69.2% (18/26) of participants with CRC, 34.3% (152/443) with AAs and 35.6% (165/463) with ACN tested positive at 40 µg/g. Sensitivity for CRC and AAs increased, whereas specificity decreased, with lower thresholds and multiple rounds. At 40 µg/g, sensitivity and specificity of the first FIT for CRC were 30.8% and 93.9%, respectively. The programme sensitivity and specificity of three rounds at 10 µg/g were 84.6% and 70.8%, respectively. Participants' preferred surveillance strategy was 3-yearly colonoscopy plus annual FITs (57.9%), followed by annual FITs with colonoscopy in positive cases (31.5%). FIT with colonoscopy in positive cases was cheaper than 3-yearly colonoscopy (£2,633,382), varying from £485,236 (40 µg/g) to £956,602 (10 µg/g). Over 3 years, FIT surveillance could miss 291 AAs and eight CRCs using a threshold of 40 µg/g, or 189 AAs and four CRCs using a threshold of 10 µg/g. CONCLUSIONS: Annual low-threshold FIT with colonoscopy in positive cases achieved high sensitivity for CRC and would be cost saving compared with 3-yearly colonoscopy. However, at higher thresholds, this strategy could miss 15-30% of CRCs and 40-70% of AAs. Most participants preferred annual FITs plus 3-yearly colonoscopy. Further research is needed to define a clear role for FITs in surveillance. FUTURE WORK: Evaluate the impact of ACN missed by FITs on quality-adjusted life-years. TRIAL REGISTRATION: Current Controlled Trials ISRCTN18040196. FUNDING: National Institute for Health Research (NIHR) Health Technology Assessment programme, NIHR Imperial Biomedical Research Centre and the Bobby Moore Fund for Cancer Research UK. MAST Group Ltd provided FIT kits.

Self-Care Behaviors of Ovarian Cancer Patients Before Their Diagnosis: Proof-of-Concept Study.

BACKGROUND: Longer patient intervals can lead to more late-stage cancer diagnoses and higher mortality rates. Individuals may delay presenting to primary care with red flag symptoms and instead turn to the internet to seek information, purchase over-the-counter medication, and change their diet or exercise habits. With advancements in machine learning, there is the potential to explore this complex relationship between a patient's symptom appraisal and their first consultation at primary care through linkage of existing datasets (eg, health, commercial, and online). OBJECTIVE: Here, we aimed to explore feasibility and acceptability of symptom appraisal using commercial- and health-data linkages for cancer symptom surveillance. METHODS: A proof-of-concept study was developed to assess the general public's acceptability of commercial- and health-data linkages for cancer symptom surveillance using a qualitative focus group study. We also investigated self-care behaviors of ovarian cancer patients using high-street retailer data, pre- and postdiagnosis. RESULTS: Using a high-street retailer's data, 1118 purchases-from April 2013 to July 2017-by 11 ovarian cancer patients and one healthy individual were analyzed. There was a unique presence of purchases for pain and indigestion medication prior to cancer diagnosis, which could signal disease in a larger sample. Qualitative findings suggest that the public are willing to consent to commercial- and health-data linkages as long as their data are safeguarded and users of this data are transparent about their purposes. CONCLUSIONS: Cancer symptom surveillance using commercial data is feasible and was found to be acceptable. To test efficacy of cancer surveillance using commercial data, larger studies are needed with links to individual electronic health records.

Barriers to bowel scope (flexible sigmoidoscopy) screening: a comparison of non-responders, active decliners and non-attenders.

BACKGROUND: Participation in bowel scope screening (BSS) is low (43%), limiting its potential to reduce colorectal cancer (CRC) incidence and mortality. This study aimed to quantify the prevalence of barriers to BSS and examine the extent to which these barriers differed according to non-participant profiles: non-responders to the BSS invitation, active decliners of the invitation, and non-attenders of confirmed appointments. METHODS: Individuals invited for BSS between March 2013 and December 2015, across 28 General Practices in England, were sent a questionnaire. Questions measured initial interest in BSS, engagement with the information booklet, BSS participation, and, where applicable, reasons for BSS non-attendance. Chi-square tests of independence were performed to examine the relationship between barriers, non-participant groups and socio-demographic variables. RESULTS: 1478 (45.8%) questionnaires were returned for analysis: 1230 (83.2%) attended screening, 114 (7.7%) were non-responders to the BSS invitation, 100 (6.8%) were active decliners, and 34 (2.3%) were non-attenders. Non-responders were less likely to have read the whole information booklet than active decliners (x2 (2, N = 157) = 7.00, p = 0.008) and non-attenders (x2 (2, N = 101) = 8.07, p = 0.005). Non-responders also had lower initial interest in having BSS than either active decliners (x2 (2, N = 213) = 6.07, p = 0.014) or non-attenders (x2 (2, N = 146) = 32.93, p < 0.001). Overall, anticipated pain (33%) and embarrassment (30%) were the most commonly cited barriers to BSS participation. For non-attenders, however, practical, appointment-related reasons were most common (27%). CONCLUSIONS: Interventions to improve BSS uptake should be more nuanced and use targeted strategies to address the specific needs of each group.

Text-message Reminders in Colorectal Cancer Screening (TRICCS): a randomised controlled trial.

BACKGROUND: We investigated the effectiveness of a text-message reminder to improve uptake of the English Bowel Cancer Screening programme in London. METHODS: We performed a randomised controlled trial across 141 general practices in London. Eight thousand two hundred sixty-nine screening-eligible adults (aged 60-74 years) were randomised in a 1 : 1 ratio to receive either a text-message reminder (n=4134) or no text-message reminder (n=4135) if they had not returned their faecal occult blood test kit within 8 weeks of initial invitation. The primary outcome was the proportion of adults returning a test kit at the end of an 18-week screening episode (intention-to-treat analysis). A subgroup analysis was conducted for individuals receiving an invitation for the first time. RESULTS: Uptake was 39.9% in the control group and 40.5% in the intervention group. Uptake did not differ significantly between groups for the whole study population of older adults (adjusted odds ratio (OR) 1.03, 95% confidence interval (CI) 0.94-1.12; P=0.56) but did vary between the groups for first-time invitees (uptake was 34.9% in the control and 40.5% in the intervention; adjusted OR 1.29, 95% CI 1.04-1.58; P=0.02). CONCLUSIONS: Although text-message reminders did not significantly increase uptake of the overall population, the improvement among first-time invitees is encouraging.

Using Specialist Screening Practitioners (SSPs) to increase uptake of the Bowel Scope (Flexible Sigmoidoscopy) Screening Programme: a study protocol for a feasibility single-stage phase II trial.

BACKGROUND: The NHS Bowel Scope Screening (BSS) programme offers men and women aged 55 years a once-only flexible sigmoidoscopy (FS), a test that can help reduce colorectal cancer (CRC) incidence and mortality. However, the benefits of BSS are contingent on uptake. This National Institute for Health Research-funded single-stage phase II trial will test the feasibility of using patient navigation (PN), an intervention that offers support to patients to overcome barriers to healthcare, to increase BSS uptake within a socially deprived area of England. METHODS/DESIGN: All individuals invited for BSS at South Tyneside NHS Foundation Trust during the 6-month recruitment period will be invited to take part in the study. Consenting participants will be randomised to receive PN or usual care in a 2:1 ratio. PN involves non-attenders receiving a phone call from a Specialist Screening Practitioner (SSP) who will elicit reasons for non-attendance and offer educational, practical, and emotional support as needed. If requested by the patient, another appointment for BSS will then be arranged. We anticipate 30 % of participants will be non-attenders. Using A'Hern single-stage design, with 20 % significance level and 80 % power, at least 35 participants who receive PN need to subsequently attend for PN to be considered worthy of further investigation in a definitive trial. The primary outcome measure will be the number of participants in the PN group who re-book and attend their BSS appointment. A qualitative analysis of the PN transcripts, and interviews with the SSPs, will also be conducted, alongside a quantitative analysis of completed patient-reported experience questionnaires. An economic analysis will calculate the costs of delivering PN. DISCUSSION: This feasibility study will be instrumental in deciding whether to conduct the first definitive trial of PN in BSS in England. If PN is subsequently shown to be cost-effective at increasing uptake of BSS, NHS policies could be modified to implement PN as a standard service. The results will be disseminated in peer-reviewed journals and at scientific conferences. TRIAL REGISTRATION: International Standard Randomised Controlled Trial Number, ISRCTN13314752.