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Chronic thromboembolic pulmonary hypertension (CTEPH) is a rare but serious complication after a pulmonary embolism. Healthcare resource utilization (HCRU; hospitalization, outpatient visits, and drug utilization) as well as productivity loss (sick leave and disability pension) before and after the CTEPH diagnosis is sparsely studied. By linking several Swedish national databases, this study estimated the societal costs in a national CTEPH cohort (n = 369, diagnosed with CTEPH in 2008-2019) 5 years before and 5 years after diagnosis (index date) and compared to an age, sex, and geographically matched control group (n = 1845, 1:5 match). HCRU and productivity loss were estimated per patient per year. Patients were stratified as operated with pulmonary endarterectomy (PEA group) or not operated (non-PEA group). Direct and indirect societal costs were 2.1 times higher before, and 8.1 times higher after the index date for patients with CTEPH compared to the matched control groups. The higher costs were evident already several years preceding the index date. The main cost driver before the index date in both the PEA and the non-PEA groups was productivity loss. The productivity loss remained high for both groups in the 5-year period following the index date, but the main cost drivers were prescribed drugs and hospitalizations for patients that underwent PEA and prescribed drugs in the non-PEA group. In conclusion, CTEPH was associated with large societal costs related to healthcare consumption and productivity loss, both before and after diagnosis., Funding Agencies|Swedish Ethical Review Authority [Dnr 2020-02573]

Pulmonary arterial hypertension (PAH) is a progressive disease with no cure. Healthcare resource utilization (HCRU; hospitalization, outpatient visits, and drug utilization) before diagnosis and productivity loss (sick leave and disability pension) before and after PAH diagnosis are not well known. By linking several Swedish national databases, this study have estimated the societal costs in a national PAH cohort (n = 749, diagnosed with PAH in 2008-2019) 5 years before and 5 years after diagnosis and compared to an age, sex, and geographically matched control group (n = 3745, 1:5 match). HCRU and productivity loss were estimated per patient per year.The PAH group had significantly higher HCRU and productivity loss compared to the control group starting already 3 and 5 years before diagnosis, respectively. HCRU peaked the year after diagnosis in the PAH group with hospitalizations (mean +/- standard deviation; 2.0 +/- 0.1 vs. 0.2 +/- 0.0), outpatient visits (5.3 +/- 0.3 vs. 0.9 +/- 0.1), and days on sick leave (130 +/- 10 vs. 13 +/- 1) significantly higher compared to controls. Total costs during the entire 10-year period were six times higher for the PAH group than the control group. In the 5 years before diagnosis the higher costs were driven by productivity loss (76%) and hospitalizations (15%), while the 5 years after diagnosis the main cost drivers were drugs (63%), hospitalizations (16%), and productivity loss (16%). In conclusion, PAH was associated with large societal costs due to high HCRU and productivity loss, starting several years before diagnosis. The economic and clinical burden of PAH suggests that strategies for earlier diagnosis and more effective treatments are warranted., Funding Agencies|Svensk Forening for Pulmonell Hypertension; Actelion Pharmaceuticals

I denna studie undersöks värdet av diagnostik vid sällsynta sjukdomar hos unga individer. Då området är mångfacetterat studeras två fall med olika karaktär. Det första fallet undersöker värdet av screening för den sällsynta sjukdomen fenylketonuri (PKU) bland nyfödda, denna screening har utförts sedan 1960-talet. Det andra fallet fokuserar på en mer modern teknologisk utveckling och utvärderar värdet av införandet av helgenomsekvensering (WGS) som genetiskt test vid sökandet efter sällsynta sjukdomar. Båda fallen använder sig av kostnadseffektivitetsanalys som metod där kostnader respektive hälsoeffekter estimeras för de utvärderade insatserna. Fallen skiljer sig åt med avseende på tillgängliga dataunderlag vilket innebär att tillvägagångssättet för att skatta kostnaderna och hälsoeffekterna är olika i de båda fallen. I fallet med PKU-screening används Markovmodellering där data från olika källor syntetiseras i en simuleringsmodell. I fallet med WGS-testning används i större utsträckning ett insamlat empiriskt datamaterial som utgörs av faktiskt uppmätta sjukvårdskostnader. Resultaten i båda fallen indikerar att de diagnostiska metoderna har en rimlig kostnad i förhållande till hälsoeffekterna. Fall ett åskådliggör att dagens screening för PKU genererar ökade hälsoeffekter till lägre kostnader i jämförelse med att inte screena för PKU. För en kohort på 100 000 nyfödda barn blir den sammanlagda hälsoeffekten en ökning med 73 QALYs och screeningen medför samtidigt en besparing på 53 376 602 kr, sett över ett livstidsperspektiv. Fall två visar att WGS som första genetiskt test i genomsnitt minskar sjukvårdskostnaderna med 15 903 kr per individ jämfört med nuvarande vård och ökar samtidigt chansen till diagnos med 9,5 procentenheter (45,7%). Resultaten bör tolkas med viss försiktighet då de är förknippade med osäkerheter, men kan samtidigt användas som en del av det underlag beslutsfa, This study examines the value of diagnostics in rare diseases in young individuals. As the field is varied, two cases with different character are studied. The first case examines the value of screening for the rare disease phenylketonuria (PKU) among newborns, this screening has been performed since the 1960s. The second case focuses on a more modern technological development and evaluates the value of the introduction of whole genome sequencing (WGS) as a genetic test in the search for rare diseases. Both cases utilize the method of cost-effectiveness analysis where costs and health effects are estimated for the evaluated measures. The cases differ regarding available data, which means that the approach to estimating costs and health effects is different in the two cases. In the case of PKU- screening, Markov modeling is used where data from different sources are synthesized in a simulation model. In the case of WGS-testing, an empirical data material is used to a greater extent, which is based on actually measured healthcare costs. The results in both cases indicate that the diagnostic methods have a reasonable cost in relation to the health effects. Case one illustrates that today's screening for PKU generates increased health effects at lower costs compared to not screening for PKU. For a cohort of 100 000 newborns, the total health effect will be an increase of 73 QALYs and the screening will also result in cost- savings of SEK 53 376 602, seen from a lifetime perspective. Case two shows that WGS used as an initial genetic test on average reduces healthcare costs by SEK 15 903 per individual compared with current care and at the same time increases the chance of diagnosis by 9.5 percentage points (45.7%). The results should be interpreted with some caution as they are associated with some uncertainties, but can still be used as part of the basis on which decision-makers need to make decisions on how health care resources should be prioritized.

Medborgarskapsenheten på Migrationsverket har i regleringsbrev från regeringen fått i uppdrag att digitalisera verksamheten, där ett av målen för digitalisering är ökad effektivitet. Med anledning av den långa kötiden för beslut i svenskt medborgarskap, har medborgarskapsenheten implementerat automatisering inom två ärendetyper. Detta för att förenkla handläggningsprocessen och på så vis minska kötiden. Syftet med denna studie är att utvärdera om implementeringen av automatiseringarna är lönsam. För att undersöka detta genomförs en kostnads-nyttoanalys som bygger på data från Migrationsverket. Kostnads-nyttoanalysen bygger även på två enkätundersökningar som tillämpar metoden contingent valuation method (CVM). Värderingen av effekterna av automatiseringen sker med hänsyn till två värderingsgrunder. Dels en värdering grundad på förändrad medborgarnytta, dels en alternativ värdering grundad på lönekostnaden som de frigjorda resurserna motsvarar. Effekterna av automatiseringen undersöks under tidsintervallen 1-4 år samt 1-10 år och innefattar kvantitativa kostnads- och nyttoposter samt kvalitativa nyttoposter. Kvantitativt sett visar båda värderingsalternativen en olönsamhet efter fyra år och en lönsamhet efter tio år. Om de kvalitativa posterna överstiger underskotten vid fyra år uppstår en lönsamhet redan då. Känslighetsanalysen visar på viss osäkerhet i beräkningarna då flera underliggande poster kan variera i storlek., The Citizenship Department at the Swedish Migration Agency has in letters of regulation been instructed by the government to digitalize the department. One of the objectives for the digitalization is to increase efficiency. Due to a long processing time regarding citizenship applications, the department has implemented automatization in two types of applications. This was done in order to simplify the case process and thereby decrease the processing time. The purpose of this thesis is to evaluate if the implementation of the automatization is profitable. A cost-benefit analysis based on data from the Swedish Migration Agency was conducted to investigate the effects on efficiency. The cost-benefit analysis also conducts two surveys applying the contingent valuation method (CVM). The valuation of the effects of the automatization is executed with regards to two different bases of valuation. One valuation is based on the change in the benefits of the citizen, and an alternative valuation is based on the salary cost that freed resources correspond to. The effects of automatization are investigated during the time intervals 1-4 years and 1-10 years, and include quantitative cost and benefit entries as well as qualitative benefit entries. Quantitatively, both valuation options are unprofitable after four years and are profitable after ten years. Profitability would arise already after four years if the qualitative entries exceeds the deficits. The sensitivity analysis shows some uncertainty in the calculations, as several underlying entries may vary in size.