Peer support for adults with type 2 diabetes starting continuous glucose monitoring-An exploratory randomised controlled trial.

AIMS: To explore the feasibility and potential benefits of a peer support programme for adults with insulin-treated type 2 diabetes (T2D) starting continuous glucose monitoring (CGM). METHODS: This part of the Steno2tech study is an exploratory, single-centre, open-labelled, prospective, randomised controlled trial (RCT). A total of 60 participants were randomised 2:1 to 12 months of CGM with or without peer support. All participants received a 3-h diabetes self-management education course including a CGM part on how to use the CGM and interpret the CGM-derived data. Peer support consisted of three 3-h peer support meetings over the first 6 months of the study period with groups of three to six people. The exploratory outcomes included the acceptability and feasibility of the peer support intervention, and the between-group difference in change in several glycaemic, metabolic and participant-reported outcomes measured at baseline, 6 and 12 months. RESULTS: The peer support intervention was found acceptable and feasible. Participants shared their experiences of using and interpreting CGM data and its association with health behaviour. While both groups had improvements in glycaemic, metabolic and participant-reported outcomes, there were no significant between-group differences. CONCLUSIONS: Although feasible, we found no measured additional benefits when adding a peer support programme after starting CGM in this exploratory RCT including adults with insulin-treated T2D. Understanding the perceived effect of and preferences for a peer support intervention from the participants' points of view, including why individuals declined to participate, would be of value for future research.

Do physicians' attitudes toward prioritization predict poor-health patients' access to care?

Physicians often face tight resource constraints, meaning they have to make trade-offs between which patients they care for and the amount of care received. Studies show that patients requiring many resources disproportionately suffer a loss of care when resources are constrained. This study uncovers whether physicians' attitudes toward prioritization of healthcare predicts poor-health patients' access to care. We combine unique survey data on Danish GPs' preferred prioritization principle with register data on their patients' contacts in general practice. We consider different types of contacts as the required effort could impact the need for prioritization. Our results show variation in GPs' prioritization principles, where a majority prefers a principle that may lead to an unequal distribution of services. We further find that GPs' attitudes toward prioritization predict some poor-health patients' access to general practice. GPs who state they prefer the principle of prioritizing patients in the poorest health state when resources tightened provide more contacts to poor-health patients. The additional contacts are typically high-effort contacts such as annual status meetings and home visits, but also low-effort contacts such as emails. Our findings indicate inequity in poor-health patients' access to care across general practices.

Going the extra mile? General practitioners' upcoding of fees for home visits.

General practitioners' (GPs') income often relies on self-reported activities and performances. They can therefore 'game the system' to maximize their remuneration. We investigate whether Danish GPs game their travel fees for home visits. Combining administrative and geographical data, we measure the difference between GPs' traveled and billed distances. We exploit a rise in the fees for home visits. If there is a link between the rise in fees and upcoding, we interpret this finding as indicative of gaming behavior. We find that upcoding occurs slightly more often than downcoding (16% vs. 13% of visits) for visits that can be both upcoded and downcoded. Using linear probability models with GP fixed effects, we find that the fee rise is associated with a reduction in upcoding of 0.6% of home visits (2.8% for visits where upcoding is feasible) and no change in downcoding. Importantly, we find no statistically significant differences in the reduction in upcoding across distance bands despite large differences in their fee rises. We therefore conclude that there is no causal evidence of GPs gaming their fees.

Girls with idiopathic central precocious puberty did not display substatial changes in body mass index after treatment with gonadotropin-releasing hormone analogues.

AIM: To evaluate changes in body mass index (BMI) in girls during and after treatment for idiopathic central precocious puberty (iCPP). METHODS: We studied 123 girls receiving gonadotropin-releasing hormone analogue (GnRHa)treatment for iCPP from 2009 to 2019. Pubertal and anthropometric measurements were monitored at routine clinical visits. BMI standard deviation scores (SDS) were estimated at baseline and followed in two stages from baseline to end of treatment (median 18.9 months) and from end of treatment to end of follow-up (median 18.2 months). The influence of baseline BMI SDS and the frequency and dose of treatment was evaluated using BMI trajectories and latent class mixed models. RESULTS: The median age at treatment initiation was 8.5 years. The median BMI SDS at baseline was 0.7, corresponding to a median BMI of 17.4 kg/m2. Overall, no changes in BMI SDS were observed during treatment. According to baseline BMI subgroups, an increasing trend in BMI trajectories during treatment was observed for girls in the lowest BMI group. After treatment, most girls maintained stable BMI levels. CONCLUSION: Our retrospective study did not provide evidence that GnRHa treatment for iCPP had a significant impact on BMI trajectories in girls.

Patient-initiated follow-up in women with early-stage endometrial cancer: A long-term follow-up of the OPAL trial.

OBJECTIVE: A long-term follow-up of the OPAL trial to compare the effect of patient-initiated (PIFU) versus hospital-based (HBFU) follow-up on fear of cancer recurrence (FCR), quality of life (QoL) and healthcare use after 34 months of follow-up. DESIGN: Pragmatic, multicentre randomised trial. SETTING: Four Danish departments of gynaecology between May 2013 and May 2016. POPULATION: 212 women diagnosed with stage I low-intermediate risk endometrial carcinoma. METHODS: The control group attended HBFU with regular outpatient visits (i.e., 8) for 3 years after primary treatment. The intervention group underwent PIFU with no prescheduled visits but with instructions about alarm symptoms and options of self-referral. MAIN OUTCOME MEASURES: The endpoints were FCR as measured by the Fear of Cancer Recurrence Inventory (FCRI) and QoL as measured by the European Organisation for Research and Treatment of Cancer Quality of Life Core Questionnaire C-30 (EORTC QLQ C-30), and healthcare use as measured by questionnaires and chart reviews after 34 months of follow-up. RESULTS: FCR decreased from baseline to 34 months in both groups and no difference was found between allocations (difference -6.31 [95% confidence interval -14.24 to 1.63]). QoL remained stable with no difference in any domains between the two arms at 34 months using a linear mixed model analysis. The use of healthcare was significantly lower in the PIFU group (P < 0.01). CONCLUSION: Patient-initiated follow-up is a valid alternative to hospital-based follow-up for people who have been treated for endometrial cancer and have low risk of recurrence.

Age, morbidity, or something else? A residual approach using microdata to measure the impact of technological progress on health care expenditure.

This study measures the increment of health care expenditure (HCE) that can be attributed to technological progress and change in medical practice by using a residual approach and microdata. We examine repeated cross-sections of individuals experiencing an initial health shock at different point in time over a 10-year window and capture the impact of unobservable technology and medical practice to which they are exposed after allowing for differences in health and socioeconomic characteristics. We decompose the residual increment in the part that is due to the effect of delaying time to death, that is, individuals surviving longer after a health shock and thus contributing longer to the demand of care, and the part that is due to increasing intensity of resource use, that is, the basket of services becoming more expensive to allow for the cost of innovation. We use data from the Danish National Health System that offers universal coverage and is free of charge at the point of access. We find that technological progress and change in medical practice can explain about 60% of the increment of HCE, in line with macroeconomic studies that traditionally investigate this subject.

Cost-effectiveness of a transdiagnostic psychotherapy program for youth with common mental health problems.

OBJECTIVES: Our objective was to evaluate the cost-effectiveness of the transdiagnostic psychotherapy program Mind My Mind (MMM) for youth with common mental health problems using a cost-utility analysis (CUA) framework and data from a randomized controlled trial. Furthermore, we analyzed the impact of the choice of informant for both quality-of-life reporting and preference weights on the Incremental Cost-Effectiveness Ratio (ICER). METHODS: A total of 396 school-aged (6-16 years) youth took part in the 6-month trial carried out in Denmark. CUAs were carried out for the trial period and four one-year extrapolation scenarios. Costs were based on a combination of budget and self-reported costs. Youths and parents were asked to report on the youth's quality-of-life three times during the trial using the Child Health Utility 9D (CHU9D). Parental-reported CHU9D was used in the base case together with preference weights of a youth population. Analyses using self-reported CHU9D and preference weights of an adult population were also carried out. RESULTS: The analysis of the trial period resulted in an ICER of €170,465. The analyses of the one-year scenarios resulted in ICERs between €23,653 and €50,480. The ICER increased by 24% and 71% compared to the base case when using self-reported CHU9D and adult preference weights, respectively. CONCLUSION: The MMM intervention has the potential to be cost-effective, but the ICER is dependent on the duration of the treatment effects. Results varied significantly with the choice of informant and the choice of preference weights indicating that both factors should be considered when assessing CUA involving youth.

Healthcare practices that increase the quality of care in cancer trajectories from a general practice perspective: a scoping review.

OBJECTIVE: General practice plays an important role in cancer trajectories, and cancer patients request the continuous involvement of general practice. The objective of this scoping review was to identify healthcare practices that increase the quality of care in cancer trajectories from a general practice perspective. DESIGN, SETTING, AND SUBJECTS: A scoping review of the literature published in Danish or English from 2010 to 2020 was conducted. Data was collected using identified keywords and indexed terms in several databases (PubMed, MEDLINE, EBSCO CINAHL, Scopus, and ProQuest), contacting key experts, searching through reference lists, and reports from selected health political, research- and interest organizations' websites. MAIN OUTCOME MEASURES: We identified healthcare practices in cancer trajectories that increase quality care. Identified healthcare practices were grouped into four contextual domains and allocated to defined phases in the cancer trajectory. The results are presented according to the Preferred Reporting Items for Systematic Reviews and Meta-analysis extension for scoping reviews (PRISMA-ScR). RESULTS: A total of 45 peer-reviewed and six non-peer-reviewed articles and reports were included. Quality of care increases in all phases of the cancer trajectory when GPs listen carefully to the full story and use action plans. After diagnosis, quality of care increases when GPs and practice staff have a proactive care approach, act as interpreters of diagnosis, treatment options, and its consequences, and engage in care coordination with specialists in secondary care involving the patient. CONCLUSION: This scoping review identified healthcare practices that increase the quality of care in cancer trajectories from a general practice perspective. The results support general practice in investigating own healthcare practices and identifying possibilities for quality improvement.KEY POINTSIdentified healthcare practices in general practice that increase the quality of care in cancer trajectories:Listen carefully to the full storyUse action plans and time-out-consultationsPlan and provide proactive careAct as an interpreter of diagnosis, treatment options, and its consequences for the patientCoordinate care with specialists, patients, and caregivers with mutual respectIdentified barriers for quality of care in cancer trajectories are:Time constraints in consultationsLimited accessibility for patients and caregiversHealth practices to increase the quality of care should be effective, safe, people-centered, timely, equitable, integrated, and efficient. These distinctions of quality of care, support general practice in investigating and improving quality of care in cancer trajectories.

Systematic identification and stratification of help-seeking school-aged youth with mental health problems: a novel approach to stage-based stepped-care.

We investigated whether a novel visitation model for school-aged youth with mental health problems based on a stage-based stepped-care approach facilitated a systematic identification and stratification process without problems with equity in access. The visitation model was developed within the context of evaluating a new transdiagnostic early treatment for youth with anxiety, depressive symptoms, and/or behavioural problems. The model aimed to identify youth with mental health problems requiring an intervention, and to stratify the youth into three groups with increasing severity of problems. This was accomplished using a two-phase stratification process involving a web-based assessment and a semi-structured psychopathological interview of the youth and parents. To assess problems with inequity in access, individual-level socioeconomic data were obtained from national registers with data on both the youth participating in the visitation and the background population. Altogether, 573 youth and their parents took part in the visitation process. Seventy-five (13%) youth had mental health problems below the intervention threshold, 396 (69%) were deemed eligible for the early treatment, and 52 (9%) had symptoms of severe mental health problems. Fifty (9%) youth were excluded for other reasons. Eighty percent of the 396 youth eligible for early treatment fulfilled criteria of a mental disorder. The severity of mental health problems highlights the urgent need for a systematic approach. Potential problems in reaching youth of less resourceful parents, and older youth were identified. These findings can help ensure that actions are taken to avoid equity problems in future mental health care implementations.

Physical activity, return to work self-efficacy, and work status among employees undergoing chemotherapy for cancer - a prospective study with 12 months follow-up.

BACKGROUND: Numerous studies emphasize the positive effects of physical activity on health and well-being in cancer patients. The effects of physical activity on the working lives of cancer patients have received less attention. The aim of the present study was to examine the association between physical activity and work status in employees with cancer, and the mediating role of return to work self-efficacy (RTWSE) in this association. METHODS: Data from questionnaires (physical activity, RTWSE, performance status, sociodemographic), patient records, and Danish national registries (work status, education) were collected for 217 employees initiating chemotherapy for cancer. The associations of physical activity at baseline with work status at baseline and at twelve months follow-up, respectively, were estimated with logistic regression. The mediating role of RTWSE was investigated using the Sobel Goodmann test. RESULTS: Employees with moderate (> 30 min/day) or high (> 150 min/day) levels of current daily activity at baseline had significantly increased odds for working at baseline (OR = 2.83, 95%CI = 0.73-10.96 and OR = 6.13, 95%CI = 1.68-22.40, respectively) and at twelve months (OR = 3.90, 95%CI = 1.19-12.77 and OR = 3.43, 95%CI = 1.12-10.51, respectively), compared to sedentary employees. Likewise, employees, physically active in their leisure time (light or vigorous psychical activity) for 2-4 h/week or > 4 h/week of light activity at baseline, had increased odds for working at twelve months (OR range = 1.20 (95%CI = 0.40-3.61)-5.39(95%CI = 0.78-37.32)), compared to sedentary employees. RTWSE was not found to mediate the observed associations. CONCLUSIONS: Physical activity appears positively associated with work status in employees undergoing treatment for cancer in the twelve months period after initiating chemotherapy.

The PROMova study comparing active and passive use of patient-reported outcome measures in ovarian cancer follow-up: effect on patient-perceived involvement, satisfaction with care, and usefulness.

BACKGROUND: Patients with ovarian cancer often experience substantial health problems and side effects resulting in reduced quality of life (QoL). Different models of using patient-reported outcome measures (PROMs) during follow-up may improve the quality of care. This national, multicenter observational study investigated the effect of active use of PROMs on patient-perceived involvement, satisfaction with care, unmet needs, and QoL during follow-up of ovarian cancer. MATERIAL AND METHODS: Ovarian cancer patients were recruited at the end of primary treatment at eight centers in Denmark. During 18 months of follow-up patients repeatedly completed European Organization for Research and Treatment of Cancer (EORTC) questionnaires covering health related QoL and symptoms. At the sites using PROMs actively (ACT), the clinician had access to an overview of the patient's scores during the clinical encounter. Clinicians using PROMs passively were alerted in case of severe development of symptoms. Following each encounter, patients evaluated their health service experience by completing the CollaboRATE scale of involvement in decision making, the Patient Experience Questionnaire, and ad hoc questions covering patient-perceived usefulness of the PROMs. RESULTS: A total of 223 patients were enrolled, i.e., 168 (75.3%) at five sites using ACT and 53 (23.8%) at three sites using them passively. We found no statistically significant difference in involvement in the decision making, satisfaction with care, unmet needs, and QoL between the two groups. The majority of patients found it useful to complete the PROMs, although it did not seem to significantly support them in raising issues with the oncologist. CONCLUSION: Active use of PROMs did not improve patients' experience of involvement in follow-up care as compared to passive use.

Identification of endometrial cancer recurrence - a validated algorithm based on nationwide Danish registries.

INTRODUCTION: Recurrence of endometrial cancer is not routinely registered in the Danish national health registers. The aim of this study was to develop and validate a register-based algorithm to identify women diagnosed with endometrial cancer recurrence in Denmark to facilitate register-based research in this field. MATERIAL AND METHODS: We conducted a cohort study based on data from Danish health registers. The algorithm was designed to identify women with recurrence and estimate the accompanying diagnosis date, which was based on information from the Danish National Patient Registry and the Danish National Pathology Registry. Indicators of recurrence were pathology registrations and procedure or diagnosis codes suggesting recurrence and related treatment. The gold standard for endometrial cancer recurrence originated from a Danish nationwide study of 2612 women diagnosed with endometrial cancer, FIGO stage I-II during 2005-2009. Recurrence was suspected in 308 women based on pathology reports, and recurrence suspicion was confirmed or rejected in the 308 women based on reviews of the medical records. The algorithm was validated by comparing the recurrence status identified by the algorithm and the recurrence status in the gold standard. RESULTS: After relevant exclusions, the final study population consisted of 268 women, hereof 160 (60%) with recurrence according to the gold standard. The algorithm displayed a sensitivity of 91.3% (95% confidence interval (CI): 85.8-95.1), a specificity of 91.7% (95% CI: 84.8-96.1) and a positive predictive value of 94.2% (95% CI: 89.3-97.3). The algorithm estimated the recurrence date within 30 days of the gold standard in 86% and within 60 days of the gold standard in 94% of the identified patients. DISCUSSION: The algorithm demonstrated good performance; it could be a valuable tool for future research in endometrial cancer recurrence and may facilitate studies with potential impact on clinical practice.

Sample restrictions and the elicitation of a constant willingness to pay per quality adjusted life year.

It is well established that the underlying theoretical assumptions needed to obtain a constant proportional trade-off between a quality adjusted life year (QALY) and willingness to pay (WTP) are restrictive and often empirically violated. In this paper, we set out to investigate whether the proportionality conditions (in terms of scope insensitivity and severity independence) can be satisfied when data is restricted to include only respondents who pass certain consistency criteria. We hypothesize that the more we restrict the data, the better the compliance with the requirement of constant proportional trade-off between WTP and QALY. We revisit the Danish data from the European Value of a QALY survey eliciting individual WTP for a QALY (WTP-Q). Using a "chained approach" respondents were first asked to value a specified health state using the standard gamble (SG) or the time-trade-off (TTO) approach and subsequently asked their WTP for QALY gains of 0.05 and 0.1 (tailored according to the respondent's SG/TTO valuation). Analyzing the impact of the different exclusion criteria on the two proportionality conditions, we find strong evidence against a constant WTP-Q. Restricting our data to include only respondents who pass the most stringent consistency criteria does not impact on the performance of the proportionality conditions for WTP-Q.

Cross-sectoral video consultations in cancer care: perspectives of cancer patients, oncologists and general practitioners.

PURPOSE: Multidisciplinary video consultations are one method of improving coherence and coordination of care in cancer patients, but knowledge of user perspectives is lacking. Continuity of care is expected to have a significant impact on the quality of cancer care. Enhanced task clarification and shared responsibility between the patient, oncologist and general practitioner through video consultations might provide enhanced continuity in cancer care. METHOD: We used descriptive survey data from patients and doctors in the intervention group based on a randomised controlled trial to evaluate the user perspectives and fidelity of the intervention. RESULTS: Patients expressed that they were able to present their concerns in 95% of the consultations, and believed it was beneficial to have both their doctors present in 84%. The general practitioner and oncologist found that tripartite video consultation would lead to better coordination of care in almost 90% of the consultations. However, the benefits of handling social issues and comorbidity were sparser. Consultations were not accomplished in 11% due to technical problems and sound and video quality were non-satisfactory in 20%. CONCLUSION: Overall, multidisciplinary video consultations between cancer patient, general practitioner and oncologist were feasible in daily clinics. Initial barriers to address were technical issues and seamless planning. Patients reported high satisfaction, patient centredness and clarity of roles. General practitioners and oncologists were overall positive regarding role clarification and continuity, although less pronounced than patients. TRIAL REGISTRATION: www.clincialtrials.gov , NCT02716168.

Association of patient-reported outcomes and ovarian cancer recurrence.

BACKGROUND: The vast majority of patients with advanced ovarian cancer experience disease recurrence after primary treatment. OBJECTIVE: To explore the diagnostic accuracy of repeated measurement of patient-reported outcomes and quality-of-life scores in relation to ovarian cancer recurrence. METHODS: Patients with ovarian cancer were recruited to the PROMova study by the end of their primary treatment at eight centers in Denmark. The purpose of the PROMova study was to explore the applicability of repeated use of patient-reported outcomes, which consisted of the European Organization for Research and Treatment of Cancer generic questionnaire and the ovarian specific questionnaire. The patient-reported outcomes were completed 3, 6, 9, 12, and 15 months after enrollment or until recurrence. The 3-month interval between completions was the period in which recurrence was assessed. Imaging and the biomarker CA125 were used as reference modality for recurrence. Mixed effects logistic regression was used to investigate the association between mean patient-reported outcome scores and recurrence. Receiver operating curves were used to establish cut-off scores. The diagnostic accuracy of patient-reported outcomes, including sensitivity, specificity, and positive and negative predictive values was estimated based on the Youden index. For combined scales, diagnostic accuracy was investigated based on multivariate analysis. RESULTS: The analysis included 196 patients with an overall recurrence rate of 50.5% and an overall mean time to recurrence of 302 days. With imaging as reference, patients with recurrence reported significantly lower global health, worse physical functioning, and more abdominal symptoms preceding recurrence. With CA125 as reference, global health, physical and emotional functioning were impaired. Despite the worsening of a number of symptoms prior to recurrence whichever reference modality was applied, the patient-reported outcome scores did not provide adequate diagnostic accuracy. CONCLUSION: Repeated use of patient-reported outcomes during surveillance of ovarian cancer was not of diagnostic value. Future efforts should be directed at improving the administration of patient-reported outcomes as well as exploring the potential of using these outcomes as an indicator of clinical relevance.

Post robotic investment: Cost consequences and impact on length of stay for obese women with endometrial cancer.

INTRODUCTION: The aim of the study was to investigate whether robotic-assisted surgery is associated with lower incremental resource use among obese patients relative to non-obese patients after a Danish nationwide adoption of robotic-assisted surgery in women with early-stage endometrial cancer. This is a population-based cohort study based on registers and clinical data. MATERIAL AND METHODS: All women who underwent surgery (robotic, laparoscopic and laparotomy) from 2008 to 2015 were included and divided according to body mass index (<30 and ≥30). Robotic-assisted surgery was gradually introduced in Denmark (2008-2013). We compared resource use post-surgery in obese vs non-obese women who underwent surgery before and after a nationwide adoption of robotic-assisted surgery. The key exposure variable was exposure to robotic-assisted surgery. Clinical and sociodemographic data were linked with national register data to determine costs and bed days 12 months before and after surgery applying difference-in-difference analyses. RESULTS: In total, 3934 women were included. The adoption of robotic-assisted surgery did not demonstrate statistically significant implications for total costs among obese women (€3,417; 95% confidence interval [CI] -€854 to €7,688, p = 0.117). Further, for obese women, a statistically significant reduction in bed days related to the index hospitalization was demonstrated (-1.9 bed days; 95% CI -3.6 to -0.2, p = 0.025). However, for non-obese women, the adoption of robotic-assisted surgery was associated with statistically significant total costs increments of €9,333 (95% CI €3,729-€1,4936, p = 0.001) and no reduction in bed days related to the index hospitalization was observed (+0.9 bed days; 95% CI -0.6 to 2.3, p = 0.242). CONCLUSIONS: The national investment in robotic-assisted surgery for endometrial cancer seems to have more modest cost implications post-surgery for obese women. This may be partly driven by a significant reduction in bed days related to the index hospitalization among obese women, as well as reductions in subsequent hospitalizations.

Validation of the Return To Work Self-Efficacy questionnaire in a population of employees undergoing treatment for cancer.

OBJECTIVE: Several Return To Work Self-Efficacy (RTWSE) questionnaires have been developed and found relevant to understanding the return to work (RTW) process of employees with various health problems, including employees with cancer. The aim of the present study was to examine the reliability and validity of the Danish 19-item RTWSE questionnaire (RTWSE-19DK ) in a sample of employees with cancer. METHODS: Employees undergoing treatment for cancer completed the RTWSE-19DK at baseline (n = 68) and at 1 week (n = 49). Additional questionnaires measured work ability, cancer-related self-efficacy and psychological distress. Internal consistency, test-retest reliability and construct validity were examined. RESULTS: The total and the three subscales of the RTWSE-19DK showed good internal consistencies with Cronbach's alphas between 0.90 and 0.97 and high test-retest reliability with Intraclass Correlation Coefficients between 0.84 and 0.90. Examining construct validity, the RTWSE-19DK showed medium and large correlations with cancer-related self-efficacy (r = 0.54), mental work ability (r = 0.51), and general work ability (r = 0.35), small correlations with physical work ability (r = 0.26) and anxiety (r = -0.10), and no correlations with depression (r = -0.08) and test date (r = 0.03). CONCLUSION: The RTWSE-19DK showed good reliability and adequate validity in employees undergoing cancer treatment. In clinical practice, the RTWSE-19 questionnaire may help practitioners identify areas of concern in the RTW process of cancer patients.

Novel phenotype of syndromic premature ovarian insufficiency associated with TP63 molecular defect.

There is growing evidence that TP63 is associated with isolated as well as syndromic premature ovarian insufficiency (POI). We report two adolescent sisters diagnosed with undetectable ovaries, uterine hypoplasia, and mammary gland hypoplasia. A novel paternally inherited nonsense variant in TP63 [NM_003722.4 c.1927C > T,p.(Arg643*)] in exon 14 was identified by exome sequencing. One of the syndromes linked to TP63 is limb mammary syndrome (LMS), an autosomal dominant inherited disorder characterized by ectrodactyly, hypoplasia of mammary-gland and nipple, lacrimal duct stenosis, nail dysplasia, dental anomalies, cleft palate and/or cleft lip and absence of skin and hair defects. The TP63 variant segregated with symptoms of LMS in the family, however, no affected individual had limb defects. The phenotype reported here represents a novel syndromic phenotype associated with TP63. Reported cases with TP63 associated POI are reviewed.

Age-stratified validation of the functional assessment of chronic illness therapy-spiritual well-being based on a large cohort of Danish cancer survivors.

OBJECTIVE: Previous research indicates that the FACIT-Sp instrument is susceptible to bias when measuring spiritual well-being in older patients. Our first focus was to evaluate the two-factor vs the three-factor model of the FACIT-Sp and our second focus was to explore how these models behave for different age groups. METHODS: We used a large national cohort of Danish cancer patients (N = 3439) which included a significant number of patients aged at least 70 years (N = 1033). Item-test correlations and factor analyses were conducted on complete cases (N = 2820). Additionally, a reliability analysis was performed using Cronbach's alpha and Guttman's lower-bound estimate. RESULTS: Factor analysis revealed a loading pattern for the oldest age group (70+) showing items on peace and meaning loading into a single factor, as originally proposed in the two-factor model. The loading estimates for the patients younger than 70 matched the three-factor model. Furthermore, item-test correlations changed as age increased. Based on Cronbach's alpha and Guttman's estimate of .83 and .89, respectively, total scores proved reliable. Items 4, 8, and 12 are discussed separately concerning their problematic influence on instrument validity in their current formulation. CONCLUSION: Overall, the three-factor model had a good fit; however, for the eldest patients a two-factor solution proved even better. Interpretation of FACIT-Sp subscale scores of older cancer patients must therefore be done with caution.

Associations between lifestyle, erectile dysfunction and healthcare seeking: a population-based study.

Objective: To investigate associations between age, lifestyle and erectile dysfunction (ED) in the general population and to explore associations between age, lifestyle and contact with a general practitioner (GP) regarding ED.Design: Cross-sectional web-based questionnaire study.Setting: The general Danish population.Subjects: A randomly selected sample of 48,910 men aged 20 years and older.Main outcome measures: Prevalence of ED and probability of contacting a GP regarding ED. In logistic regression models we analysed associations between age, smoking status, alcohol consumption, body mass index (BMI), and self-rated physical fitness on both ED and GP contact.Results: A total of 22,198 men (47.6%) completed the question regarding ED. The overall prevalence of ED was 19.3%, varying from 2.3% among men aged 20-29 years to 55.3% among men aged 80 years and above. 31.8% of men reporting ED had contacted a GP regarding ED. Increasing age, current or former smoking, complete alcohol abstinence or alcohol consumption above seven units per week, high BMI, and poor self-rated physical fitness were significantly associated with reporting ED. The proportion of GP contacts was significantly associated with age. Overall, no significant associations between lifestyle and healthcare seeking were observed, although lower odds of GP contact were found when physical fitness was rated as poor.Conclusion: Reporting ED and GP contact were significantly associated with age. Furthermore, lifestyle was significantly associated with reporting ED, but largely not associated with healthcare seeking. These findings are important for future interventions aiming to improve diagnosis and treatment of ED.Key pointsExperiencing erectile dysfunction is frequent in the general population, especially among older men. • In this large-scale national survey, age and lifestyle were significantly associated with reporting erectile dysfunction. • Healthcare seeking with erectile dysfunction was significantly associated with age, but not with lifestyle. • Diagnosis and treatment of erectile dysfunction might be challenged when erectile dysfunction does not lead to healthcare seeking.