Increasing healthcare costs in inflammatory bowel disease 2007-2020 in Sweden.

BACKGROUND: Inflammatory bowel disease has been linked to increasing healthcare costs, but longitudinal data on other societal costs are scarce. AIM: To assess costs, including productivity losses, in patients with prevalent Crohn's disease (CD) or ulcerative colitis (UC) in Sweden between 2007 and 2020. METHODS: We linked data from national registers on all patients with CD or UC and a matched (sex, birthyear, healthcare region and education) reference population. We assessed mean costs/year in Euros, inflation-adjusted to 2020, for hospitalisations, out-patient visits, medications, sick leave and disability pension. We defined excess costs as the mean difference between patients and matched comparators. RESULTS: Between 2007 and 2020, absolute mean annual societal costs in working-age (18-64 years) individuals decreased by 17% in CD (-24% in the comparators) and by 20% in UC (-27% in comparators), due to decreasing costs from sick leave and disability, a consequence of stricter sick leave regulations. Excess costs in 2007 were dominated by productivity losses. In 2020, excess costs were mostly healthcare costs. Absolute and excess costs increased in paediatric and elderly patients. Overall, costs for TNF inhibitors/targeted therapies increased by 274% in CD and 638% in UC, and the proportion treated increased from 5% to 26% in CD, and from 1% to 10% in UC. CONCLUSION: Between 2007 and 2020, excess costs shifted from productivity losses to direct healthcare costs; that is, the patients' compensation for sickness absence decreased, while society increased its spending on medications. Medication costs were driven both by expanding use of TNF inhibitors and by high costs for newer targeted therapies.

Incidence, Management, and Outcomes of Very Early Onset Inflammatory Bowel Diseases and Infantile-Onset Disease: An Epi-IIRN Study.

BACKGROUND & AIMS: In this nationwide study from the Israeli Inflammatory Bowel Disease Research Nucleus, we aimed to describe the incidence of very early onset inflammatory bowel diseases (VEOIBDs) with a focus on infantile-onset disease and to compare management and disease course with older children. METHODS: Data were retrieved from the 4 Israeli Health Maintenance Organizations covering 98% of the population. Pediatric-onset IBD was categorized as follows: adolescent onset (10 to <18 y), early onset (6 to <10 y), VEOIBD (0 to <6 y), toddler onset (2 to <6 y), and infantile onset (<2 y). RESULTS: A total of 5243 children with 35,469 person-years of follow-up evaluation, were diagnosed with IBD during 2005 to 2020: 4444 (85%) with adolescent onset, 548 (10%) with early onset, and 251 (4.8%) with VEOIBD, of whom 81 (1.5%) had infantile onset. The incidence of pediatric-onset IBD increased from 10.8 per 100,000 in 2005 to 15.3 per 100,000 in 2019 (average annual percentage change, 2.8%; 95% CI, 2.2%-3.4%), but that of VEOIBD remained stable (average annual percentage change, 0%; 95% CI, -2.5% to 2.6%). The infantile-onset and toddler-onset groups were treated less often with biologics (36% and 35%, respectively) vs the early onset (57%) and adolescent-onset groups (53%; P < .001). The time to steroid dependency was shorter in infantile-onset (hazard ratio [HR], 2.1; 95% CI, 1.5-2.9) and toddler-onset disease (HR, 1.6; 95% CI, 1.2-2.0) vs early onset and adolescent-onset disease, but time to hospitalizations, time to surgery, and growth delay were worse only in infantile-onset disease. In a multivariable model, infantile-onset patients had a higher risk for surgery (HR, 1.4; 95% CI, 1.1-1.9) and hospitalization (HR, 1.7; 95% CI, 1.2-2.4) than the toddler-onset group. CONCLUSIONS: The incidence of VEOIBD remained stable. Infantile-onset IBD had worse outcomes than older children, while toddler onset had mostly similar outcomes, despite less frequent use of biologics.

Women's Earnings are more Affected by Inflammatory Bowel Disease than Men's: A Register-Based Swedish Cohort Study.

BACKGROUND AND AIMS: Patients with inflammatory bowel disease [IBD] are subject to more work disability than the general population. We aimed to estimate the monetary cost of IBD for the individual through assessment of earnings in relation to diagnosis. METHODS: Through linkage of national registers, we identified patients aged 30-55 years at first IBD diagnosis in Sweden in 2002-2011, and same-sex IBD-free siblings. We estimated taxable earnings and disposable income from 5 years before to 5 years after diagnosis. RESULTS: The 5961 patients [27% Crohn's disease, 68% ulcerative colitis, 4.3% IBD unclassified] had similar taxable earnings to their 7810 siblings until the year of diagnosis, when earnings decreased and remained lower than for siblings during follow-up. The adjusted difference in earnings over the entire 5-year period after diagnosis was -5% [-8212€; 95% confidence interval: -11 458 to -4967€]. The difference was greater in women than in men, and greater in Crohn's disease than in ulcerative colitis. When stratifying for sex and IBD subtype and comparing earnings during each year of follow-up, median annual earnings were lower in women with Crohn's disease and ulcerative colitis than in their sisters during all years of follow-up, whereas the men had similar annual taxable earnings to their brothers. Disposable income was similar between patients and siblings during the investigated time period. CONCLUSION: From the year of diagnosis and at least 5 years onwards, patients with IBD had 5% lower earnings than siblings, mainly explained by differences between women with IBD and their sisters. However, there were no differences in disposable income.

Healthcare use, work loss and total costs in incident and prevalent Crohn's disease and ulcerative colitis: results from a nationwide study in Sweden.

BACKGROUND: There are limited data on population-wide assessment of cost in Crohn's disease (CD) and ulcerative colitis (UC). AIM: To estimate the societal cost of actively treated CD and UC in Sweden. METHODS: We identified 10 117 prevalent CD and 19 762 prevalent UC patients, aged ≥18 years on 1 January 2014 and 4028 adult incident CD cases and 8659 adult incident UC cases (2010-2013) from Swedish Patient Register. Each case was matched to five population comparators. Healthcare costs were calculated from medications, outpatient visits, hospitalisations and surgery. Cost of productivity losses was derived from disability pension and sick leave. RESULTS: The mean annual societal costs per working-age patient (18-64 years) with CD and UC were $22 813 (vs $7533 per comparator) and $14 136 (vs $7351 per comparator), respectively. In patients aged ≥65 years, the mean annual costs of CD and UC were $9726 and $8072 vs $3875 and $4016 per comparator, respectively. The majority of cost for both CD (56%) and UC (59%) patients originated from productivity losses. Higher societal cost of working-age CD patients as compared to UC patients was related to greater utilisation of anti-TNF (22.2% vs 7.4%) and increased annual disability pension (44 days vs 25 days). Among incident CD and UC patients, the mean total cost over the first year per patient was over three times higher than comparators. CONCLUSION: In Sweden, the societal cost of incident and prevalent CD and UC patients was consistently two to three times higher than the general population.

The longitudinal integrated database for health insurance and labour market studies (LISA) and its use in medical research.

Education, income, and occupation are factors known to affect health and disease. In this review we describe the Swedish Longitudinal Integrated Database for Health Insurance and Labour Market Studies (LISA, Longitudinell Integrationsdatabas för Sjukförsäkrings- och Arbetsmarknadsstudier). LISA covers the adult Swedish population aged ≥ 16 years registered on December 31 each year since 1990 (since 2010 individuals aged ≥ 15 years). The database was launched in response to rising levels of sick leave in the country. Participation in Swedish government-administered registers such as LISA is compulsory, and hence selection bias is minimized. The LISA database allows researchers to identify individuals who do not work because of injury, disease, or rehabilitation. It contains data on sick leave and disability pension based on calendar year. LISA also includes information on unemployment benefits, disposable income, social welfare payments, civil status, and migration. During 2000-2017, an average of 97,000 individuals immigrated to Sweden each year. This corresponds to about 1% of the Swedish population (10 million people in 2017). Data on occupation have a completeness of 95%. Income data consist primarily of income from employment, capital, and allowances, including parental allowance. In Sweden, work force participation is around 80% (2017: overall: 79.1%; men 80.3% and women 77.9%). Education data are available in > 98% of all individuals aged 25-64 years, with an estimated accuracy for highest attained level of education of 85%. Some information on civil status, income, education, and employment before 1990 can be obtained through the Population and Housing Census data (FoB, Folk- och bostadsräkningen).

Cost-effectiveness of internet-delivered cognitive-behavioural therapy for adolescents with irritable bowel syndrome.

OBJECTIVE: To assess whether exposure-based internet-delivered cognitive-behavioural therapy (internet-CBT) is a cost-effective treatment for adolescents with irritable bowel syndrome (IBS) compared with a waitlist control, from a societal perspective, based on data from a randomised trial. DESIGN: Within-trial cost-effectiveness analysis. SETTING: Participants were recruited from the whole of Sweden via primary, secondary and tertiary care clinics reached through news media and advertising. PARTICIPANTS: Adolescents (aged 13-17) with a diagnosis of IBS. INTERVENTIONS: Participants were randomised to either an exposure-based internet-CBT, including 10 weekly modules for adolescents and five modules for parents, or a waitlist. OUTCOME MEASURES: The main health outcome was the quality-adjusted life-year (QALY) estimated by mapping Pediatric Quality-of-Life Inventory (PedsQL) scores onto EQ-5D-3L utilities. The secondary outcome was the point improvement on the PedsQL scale. Data on health outcomes and resource use were collected at baseline and 10 weeks post-treatment. Resource use was measured using the Trimbos and Institute of Medical Technology Assessment Cost Questionnaire for Psychiatry (TIC-P) . Incremental cost-effectiveness ratios (ICER) were calculated as the difference in average costs by the difference in average outcomes between groups. RESULTS: The base-case results showed that internet-CBT costs were on average US$170.24 (95% CI 63.14 to 315.04) more per participant than the waitlist. Adolescents in the internet-CBT group showed small QALY gains (0.0031; 95% CI 0.0003 to 0.0061), and an average improvement of 5.647 points (95% CI 1.82 to 9.46) on the PedsQL compared with the waitlist. Internet-CBT yielded an ICER of $54 916/QALY gained and a probability of cost-effectiveness of 74% given the Swedish willingness-to-pay threshold. The ICER for the outcome PedsQL was US$85.29/point improvement. CONCLUSIONS: Offering internet-CBT to adolescents with IBS improves health-related quality of life and generates small QALY gains at a higher cost than a waitlist control. Internet-CBT is thus likely to be cost-effective given the strong efficacy evidence, small QALY gains and low cost. TRIAL REGISTRATION NUMBER: NCT02306369; Results.

Clinical and Cost Effectiveness of Online Cognitive Behavioral Therapy in Children With Functional Abdominal Pain Disorders.

BACKGROUND & AIMS: Scalable and effective treatments are needed for children with functional abdominal pain disorders (FAPDs). We performed a randomized controlled trial of the efficacy and cost effectiveness of cognitive behavioral therapy delivered online (Internet-CBT) compared with usual therapy. METHODS: We studied children (age, 8-12 y) diagnosed with FAPDs, based on the Rome IV criteria, in Sweden from September 2016 through April 2017. The patients were assigned randomly to groups that received 10 weeks of therapist-guided, internet-delivered cognitive behavioral therapy (Internet-CBT, n = 46) or treatment as usual (treatments within the health care and school systems, including medications and visits to doctors and other health care professionals; n = 44). The primary outcome was global child-rated gastrointestinal symptom severity assessed using the Pediatric Quality of Life Gastrointestinal Symptom scale. All outcomes were collected from September 2016 through January 2018. Secondary outcomes included quality of life, gastrointestinal-specific anxiety, avoidance behaviors, and parental responses to children's symptoms. Societal costs and costs for health care consumption were collected during the treatment. RESULTS: Children who received Internet-CBT had a significantly larger improvement in gastrointestinal symptom severity with a medium effect size (Cohen's d = 0.46; 95% CI, 0.05-0.88; number needed to treat, 3.8) compared with children who received the treatment as usual. The children's quality of life, gastrointestinal-specific anxiety, avoidance behaviors, and parental responses to children's symptoms also improved significantly in the Internet-CBT group compared with the treatment as usual group. The effects of Internet-CBT persisted through 36 weeks of follow-up evaluation. Children who received Internet-CBT had significantly less health care use than children who received treatment as usual, with an average cost difference of US $137 (P = .011). We calculated a cost savings of US $1050 for every child treated with Internet-CBT compared with treatment as usual. CONCLUSIONS: In a randomized trial of pediatric patients with FAPDs, we found Internet-CBT to be clinically cost effective compared with treatment as usual. Internet-CBT has the potential to increase the availability of treatment for a number of patients and reduce health care costs. ClinicalTrials.gov: NCT02873078.

Sick Leave and Disability Pension in Prevalent Patients With Crohn's Disease.

BACKGROUND AND AIMS: Crohn's disease may affect the ability to work and lead to permanent disability. We aimed to investigate work loss in prevalent patients. METHODS: We identified patients with Crohn's disease and general population comparators matched by sex, birth year, healthcare region and education. We assessed days of sick leave and disability pension retrieved from the Swedish Social Insurance Agency and estimated the absolute and relative risk of receiving disability pension [minimum 25% work impairment]. RESULTS: In 2014, the 20638 Crohn's disease patients [median age 44 years] had more than twice as many mean lost workdays [disability pension: 44; sick leave: 19] as the 102038 comparators [disability pension: 20; sick leave: 8], mean difference 35 days [95% confidence interval 33-37]. However, the majority had no lost workdays [68% of patients and 85% of comparators]. The proportion of patients receiving disability pension was 15% (6.5% in the comparators, risk ratio 2.34 [2.25-2.43]) and was higher in all subgroups, especially in female patients [28% vs 13% in the comparators], in those with ≤9 years of education [41% vs 23%] and in ages 60-64 years [46% vs 25%]. The relative risk of disability pension within the patient cohort [adjusted for age, sex, region and education] was higher in patients with complicated disease behaviour, extraintestinal manifestations, need of surgery or treatment with biologics. The differences between patients and comparators remained when comparing other calendar years [2006-2013]. CONCLUSION: Work loss was found in approximately one-third of patients. The mean number of lost workdays was twice as high as in the comparators.

Antibodies against deamidated gliadin peptides and tissue transglutaminase for diagnosis of pediatric celiac disease.

OBJECTIVES: The aim of the present study was to evaluate diagnostic performance and actual costs in clinical practice of immumoglobulin (Ig)G/IgA deamidated gliadin peptide antibodies (DGP) as a complement to IgA antibodies against tissue transglutaminase (tTG) for the diagnosis of pediatric celiac disease (CD). METHODS: All of the consecutive patients younger than 18 years tested for tTG and/or DGP, who underwent duodenal biopsy because of suspected CD in Stockholm and Gothenburg, Sweden, from 2008 to 2010, were included. Medical records were reviewed. RESULTS: Of 537 children who underwent duodenal biopsy, 278 (52%) had CD. A total of 71 (13%) were younger than 2 years and 16 (4%) had IgA deficiency. Sensitivity and specificity for tTG were 94% and 86%, respectively. Corresponding values for DGP were 91% and 26%. Positive predictive values (PPV) were 88% for tTG and 51% for DGP. There were 148 children who were tTG-negative and DGP-positive, of which only 5% (8/148) had villous atrophy. Among children younger than 2 years with normal IgA, PPV was 96% (25/26) for tTG and 48% (24/50) for DGP. In 16 IgA-deficient children, 11 were DGP positive, of which 5 had CD (PPV 45%). Eight of 278 cases of CD would possibly have been missed without DGP. The cost of adding DGP and consequently more biopsies to be able to detect 8 extra cases of CD was [Euro sign]399,520 or [Euro sign]49,940 per case. CONCLUSIONS: For diagnosing CD, tTG is superior to DGP, even in children younger than 2 years. Combining tTG and DGP does not provide a better tradeoff between number of missed cases of CD, number of unnecessary duodenal biopsies, and cost than tTG alone.

Socioeconomic position and education in patients with coeliac disease.

BACKGROUND AND AIM: Socioeconomic position and education are strongly associated with several chronic diseases, but their relation to coeliac disease is unclear. We examined educational level and socioeconomic position in patients with coeliac disease. METHODS: We identified 29,096 patients with coeliac disease through biopsy reports (defined as Marsh 3: villous atrophy) from all Swedish pathology departments (n=28). Age- and sex-matched controls were randomly sampled from the Swedish Total Population Register (n=145,090). Data on level of education and socioeconomic position were obtained from the Swedish Education Register and the Occupational Register. We calculated odds ratios for the risk of having coeliac disease based on socioeconomic position according to the European Socioeconomic Classification (9 levels) and education. RESULTS: Compared to individuals with high socioeconomic position (level 1 of 9) coeliac disease was less common in the lowest socioeconomic stratum (routine occupations=level 9 of 9: adjusted odds ratio=0.89; 95% confidence interval=0.84-0.94) but not less common in individuals with moderately low socioeconomic position: (level 7/9: adjusted odds ratio=0.96; 95% confidence interval=0.91-1.02; and level 8/9: adjusted odds ratio=0.99; 95% confidence interval=0.93-1.05). Coeliac disease was not associated with educational level. CONCLUSIONS: In conclusion, diagnosed coeliac disease was slightly less common in individuals with low socioeconomic position but not associated with educational level. Coeliac disease may be unrecognised in individuals of low socioeconomic position.