Disentangling trust of patients with rare cancer in their healthcare professionals and the healthcare system: a qualitative interview study.

PURPOSE: Patients with a rare cancer face challenges, e.g., delayed diagnosis, that may affect trust in the healthcare system and the healthcare professionals (HCPs) involved. This study aimed to explore trust of patients with a rare cancer in their HCPs and the healthcare system. METHODS: Semi-structured interviews were conducted with 20 purposively sampled patients with a rare cancer. The interview guide included topics related to trust, including level, development, barriers and facilitators, importance, and trust dimensions. Thematic analysis was conducted with use of Atlas.ti. RESULTS: The mean age of patients was 50 years, 60% were female, and 70% were highly educated. Three themes were constructed: (1) "Confirmed expertise is a prerequisite of trust." Patients need confirmation of their HCPs' expertise, as it could not be assumed due to the rarity of their cancer; (2) "Trust depends on the adequacy of information and how it is provided." Limited information about rare cancer reduced patients' trust in health care, whereas interpersonal trust was mainly affected by how HCPs provided information; and (3) "Trust is built on properly coordinated and supportive care." Proper organization and cooperation within and between hospitals, and integration of supportive care, enhanced trust. CONCLUSION: Patients with a rare cancer experience challenges that influence trust in HCPs and the healthcare system. Further research should examine trust among subgroups of patients with a rare cancer, to enable development of tailored interventions. IMPLICATIONS FOR CANCER SURVIVORS: HCPs may improve trust by focusing on expertise, effective information provision, proper coordination of care, and provision of adequate supportive care.

The solitary versus supported experience: Care inequality between rare and common cancer patients.

OBJECTIVE: Patients with a rare cancer (RC) often have a more complex disease trajectory than patients with a common cancer. Research involving both patient groups is needed to identify differences and resemblances. In this study, we aimed to explore and compare experiences, needs and quality of life of patients with rare and common cancer throughout the disease trajectory. METHODS: A qualitative focus group study was conducted, including patients with rare and common cancer (n = 25). Participants were purposively selected to reflect heterogeneity of cancer types. A semi-structured topic list was used. Focus groups (n = 4) were recorded, transcribed verbatim and analysed, using thematic analysis. RESULTS: Three themes were identified emphasizing care inequality between patients with rare and common cancer: (1) The solitary experience: lack of information and support impact the RC patient, (2) Sudden impact, but recognition reduces the common cancer burden, and (3) Absence of psychosocial care requires being empowered as a cancer patient. CONCLUSIONS: Patients with RC are faced with enormous challenges due to the high impact of their solitary experience on their quality of life, while patients with common cancer generally experience social support and recognition alleviating their burden. Centralisation of care for patients with RC is needed and tailored psychosocial care should be provided to overcome inequalities.

Quality of life of patients with rare cancer: a comparison with patients with colorectal cancer and the association with disease trajectory-related factors.

PURPOSE: Differences in quality of life (QoL) between patients with rare and common cancer might be explained by the specific challenges patients with rare cancer face during their disease trajectory, but research is scarce. This study aimed to (1) assess the difference in QoL between patients with rare and common cancer (i.e. colorectal cancer (CRC)) and (2) examine the association between disease trajectory-related factors and QoL in patients with rare cancer. METHODS: Cross-sectional data were collected among adults with rare cancer by a nationwide online survey in the Netherlands. For comparison with patients with CRC, data from the Prospective Dutch Colorectal Cancer (PLCRC) cohort were used. Associations were assessed by linear regression analyses. RESULTS: Data from 1525 patients with rare cancer and 1047 patients with CRC were analysed. Having a rare cancer was significantly associated with a lower QoL compared to having CRC (p < 0.001). Disease trajectory-related factors significantly associated with QoL in patients with rare cancer were time until diagnosis, misdiagnoses, information on best treatment options, information on late and/or long-term effects, and both satisfaction with physician and specialized nurse care (all: p < 0.05). CONCLUSION: Patients with rare cancers have a lower self-reported QoL than patients with CRC, and several disease trajectory-related factors are associated with QoL in patients with rare cancer. IMPLICATIONS FOR CANCER SURVIVORS: To improve QoL of patients with rare cancer, appropriate guidance and support by healthcare professionals throughout the disease trajectory are needed, as well as early diagnosis and proper referral to centres of expertise.

Rare cancer and return to work: experiences and needs of patients and (health care) professionals.

PURPOSE: Patients with a rare cancer often face delays in their diagnostic and treatment trajectory, which may affect their work. In this study, we explored experiences and needs of: 1) patients with a rare cancer regarding return to work (RTW) and work retention, and 2) (health care) professionals (HCPs) regarding work-related support of patients with a rare cancer, throughout their disease trajectory. METHODS: Semi-structured, in-depth interviews with working-age patients with a rare cancer (n = 16), and HCPs (n = 9) were conducted. During the interviews, a predefined topic list was used. Interviews were transcribed verbatim and analysed by means of thematic analysis. RESULTS: In total, three themes emerged from the interviews: 1) Awareness in patients and HCPs as a first step towards work, 2) Being/becoming an expert is a tough job, and 3) Enhancing employability through early personalized guidance. CONCLUSIONS: Patients with a rare cancer are confronted with uncertainties regarding work, due to an overall lack of knowledge and experience with these types of cancer. Raising awareness among patients, HCPs and employers about rare cancer and its implications, and providing timely individualized, supportive occupational care are required to improve rare cancer patients' ability to work.Implications for rehabilitationRecognizing and paying attention to the work-related consequences of a rare cancer diagnosis and its treatment is essential to facilitate rare cancer patients' occupational rehabilitation process.Multidisciplinary collaboration and involvement of HCPs specialized in rare cancer are required to provide suitable work-related support.A personalized approach is necessary to adequately address rare cancer patients' RTW and work retention needs.

Challenges and controversies patients and (health care) professionals experience in managing vaginal, vulvar, penile or anal cancer: The SILENCE study.

OBJECTIVE: Patients with vaginal, vulvar, penile or anal cancer experience deteriorated psychosocial functioning and decreased Quality of Life (QoL). The aims of this study were to explore (1) the challenges and controversies patients experience in managing vaginal, vulvar, penile or anal cancer; their unmet needs; and how this affects their psychosocial functioning and (2) the gaps health care professionals (HCPs) experience in providing psychosocial support and potential improvements in care. METHODS: Semi-structured interviews with patients with vaginal, vulvar, penile or anal cancer and with HCPs were conducted. All interviews were transcribed verbatim and thematically analysed. RESULTS: Fourteen patients (86% female; mean age 55.5) and 12 HCPs (75% female; mean age 46.4) participated. Four themes were identified: (1) recognisable symptoms but unfamiliar diagnosis, (2) 'double hit' has severe impact on psychosocial functioning, (3) personal and tailored information is important but not guaranteed and (4) all-encompassing care to improve psychosocial functioning and QoL. CONCLUSION: Patients with vaginal, vulvar, penile or anal cancer encounter a lack of awareness and knowledge about their rare cancer type, difficulties regarding communication and long-term changes in body image and sexuality. Awareness of symptoms should be raised and psychosocial care should be offered on a structural basis.

The gap between rare and common cancers still exists: Results from a population-based study in the Netherlands.

INTRODUCTION: Epidemiological discrepancies exist between rare and common cancers. The aim of this population-based study was to compare rare versus common adult solid cancers in the Netherlands, by providing incidence, prevalence and survival rates, evaluating trends in survival and comparing individual entities within domains and families. METHODS: All adult patients with malignant solid cancers in the Netherlands between 1995 and 2019 were identified from the Netherlands Cancer Registry. Data on patient, tumour and treatment characteristics were collected, and relative survival and survival trends were analysed. RESULTS: A total of 170,628 patients with rare adult solid cancers and 806,023 patients with common adult solid cancers were included. Rare cancers accounted for 18% of all cancer diagnoses (mean incidence), and 15% of the total ten-year cancer prevalence during 2010-2019. Overall 5-year survival was worse for rare cancers than for common cancers (52.0% versus 68.7%). Between 1995-1999 and 2015-2019, 5-year survival rates for rare cancers increased to a lesser extent (from 46.2% to 52.6%, i.e. 6.4%) than for common cancers (56.9%-70.1%, i.e. 13.2%), and for most rare cancer domains compared to common cancer domains. The majority of rare cancer entities did not show an improvement in 5-year survival. Differences for individual entities between domains and families were found. CONCLUSION: Differences in survival between rare and common cancers indicate major challenges for rare cancer care and emphasise that improvement is highly needed. Observed inequalities need to be overcome by investing in early diagnosis, novel therapies, scientific research and in establishing centres of expertise.

Unmet supportive care needs of patients with rare cancer: A systematic review.

OBJECTIVE: Patients with rare cancers may experience different unmet needs than those with common cancer. The objective of this systematic review was to (1) investigate unmet supportive care needs of rare cancer patients throughout the disease trajectory and (2) identify predictive factors for these unmet needs. METHODS: PubMed, PsycINFO and CINAHL were searched for publications (January 2011 to March 2021) focusing on unmet needs of patients with rare cancer. Two reviewers independently selected studies, extracted data and performed quality assessment. Findings were synthesised. RESULTS: The search yielded 4,598 articles, of which 59 articles met eligibility criteria and 57 were of medium or high quality. Rare cancer patients most frequently reported unmet needs in the healthcare system and information domain (up to 95%), followed by the psychological domain (up to 93%) and the physical and daily living domain (up to 80%). Unmet needs were mainly reported in the posttreatment phase. The most frequently identified predictors were higher anxiety, younger age and higher neuroticism. CONCLUSION: Patients with rare cancer have unmet needs throughout their disease trajectory. Supportive care needs of rare cancer patients should be addressed individually, depending on the rare cancer subdomain and phase of disease and from diagnosis onwards.

Differences in health care experiences between rare cancer and common cancer patients: results from a national cross-sectional survey.

BACKGROUND: Patients with rare cancers face challenges in the diagnostic and treatment phase, and in access to clinical expertise. Since studies on health care experiences of these patients in comparison to patients with more common cancers are scarce, we aimed to explore these differences. METHODS: Data were cross-sectionally collected among (former) adult cancer patients through a national online survey in the Netherlands (October 2019). Descriptive statistics were reported and subgroups (rare vs. common patients) were compared. RESULTS: In total, 7343 patients (i.e., 1856 rare and 5487 common cancer patients) participated. Rare cancer patients were more often diagnosed and treated in different hospitals compared to common cancer patients (67% vs. 59%, p < 0.001). Rare cancer patients received treatment more often in a single hospital (60% vs. 57%, p = 0.014), but reported more negative experiences when treated in multiple hospitals than common cancer patients (14% vs. 9%, p < 0.001). They also more often received advise from their physician about the hospital to go to for a second opinion (50% vs. 36%, p < 0.001), were more likely to choose a hospital specialized in their cancer type (33% vs. 22%, p < 0.001), and were more willing to travel as long as necessary to receive specialized care than common cancer patients (55% vs. 47%, p < 0.001). CONCLUSIONS: Rare and common cancer patients differ in their health care experiences. Health care for rare cancer patients can be further improved by proper referral to centers of expertise and building a clinical network specifically for rare cancers.