scholarly journals Direct and indirect costs and cost-driving factors in adults with tuberous sclerosis complex: a multicenter cohort study and a review of the literature

2021 ◽  
Vol 16 (1) ◽  
Author(s):  
Johann Philipp Zöllner ◽  
Janina Grau ◽  
Felix Rosenow ◽  
Matthias Sauter ◽  
Markus Knuf ◽  
...  
Keyword(s):  
Tuberous Sclerosis ◽  
Direct Cost ◽  
Nursing Care ◽  
Tuberous Sclerosis Complex ◽  
Indirect Costs ◽  
Direct Costs ◽  
Driving Factors ◽  
Medication Costs ◽  
Organ Manifestations ◽  
Care Costs

Abstract Background Tuberous sclerosis complex (TSC) is a monogenetic, multisystem disorder characterized by benign growths due to TSC1 or TSC2 mutations. This German multicenter study estimated the costs and related cost drivers associated with organ manifestations in adults with TSC. Methods A validated, three-month, retrospective questionnaire assessed the sociodemographic and clinical characteristics, organ manifestations, direct, indirect, out-of-pocket (OOP), and nursing care-level costs among adult individuals with TSC throughout Germany from a societal perspective (costing year: 2019). Results We enrolled 192 adults with TSC (mean age: 33.4 ± 12.7 years; range: 18–78 years, 51.6% [n = 99] women). Reported TSC disease manifestations included skin (94.8%) and kidney and urinary tract (74%) disorders, epilepsy (72.9%), structural brain defects (67.2%), psychiatric disorders (50.5%), heart and circulatory system disorders (50.5%), and lymphangioleiomyomatosis (11.5%). TSC1 and TSC2 mutations were reported in 16.7% and 25% of respondents, respectively. Mean direct health care costs totaled EUR 6452 (median EUR 1920; 95% confidence interval [CI] EUR 5533–7422) per patient over three months. Medication costs represented the major direct cost category (77% of total direct costs; mean EUR 4953), and mechanistic target of rapamycin (mTOR) inhibitors represented the largest share (68%, EUR 4358). Mean antiseizure drug (ASD) costs were only EUR 415 (6%). Inpatient costs (8%, EUR 518) and outpatient treatment costs (7%; EUR 467) were important further direct cost components. The mean care grade allowance as an approximator of informal nursing care costs was EUR 929 (median EUR 0; 95% CI EUR 780–1083) over three months. Mean indirect costs totaled EUR 3174 (median EUR 0; 95% CI EUR 2503–3840) among working-age individuals (< 67 years in Germany). Multiple regression analyses revealed mTOR inhibitor use and persistent seizures as independent cost-driving factors for total direct costs. Older age and disability were independent cost-driving factors for total indirect costs, whereas epilepsy, psychiatric disease, and disability were independent cost-driving factors for nursing care costs. Conclusions This three-month study revealed substantial direct healthcare, indirect healthcare, and medication costs associated with TSC in Germany. This study highlights the spectrum of organ manifestations and their associated treatment needs in the German healthcare setting. Trial registration: DRKS, DRKS00016045. Registered 01 March 2019, http://www.drks.de/DRKS00016045.

scholarly journals Direct and indirect costs and cost-driving factors of Tuberous sclerosis complex in children, adolescents, and caregivers: a multicenter cohort study

2021 ◽  
Vol 16 (1) ◽  
Author(s):  
Janina Grau ◽  
Johann Philipp Zöllner ◽  
Susanne Schubert-Bast ◽  
Gerhard Kurlemann ◽  
Christoph Hertzberg ◽  
...  
Keyword(s):  
Tuberous Sclerosis ◽  
Direct Cost ◽  
Nursing Care ◽  
Tuberous Sclerosis Complex ◽  
Indirect Costs ◽  
Mtor Inhibitors ◽  
Direct Costs ◽  
Driving Factors ◽  
Care Level ◽  
Organ Manifestations

Abstract Background Tuberous sclerosis complex (TSC), a multisystem genetic disorder, affects many organs and systems, characterized by benign growths. This German multicenter study estimated the disease-specific costs and cost-driving factors associated with various organ manifestations in TSC patients. Methods A validated, three-month, retrospective questionnaire was administered to assess the sociodemographic and clinical characteristics, organ manifestations, direct, indirect, out-of-pocket, and nursing care-level costs, completed by caregivers of patients with TSC throughout Germany. Results The caregivers of 184 patients (mean age 9.8 ± 5.3 years, range 0.7–21.8 years) submitted questionnaires. The reported TSC disease manifestations included epilepsy (92%), skin disorders (86%), structural brain disorders (83%), heart and circulatory system disorders (67%), kidney and urinary tract disorders (53%), and psychiatric disorders (51%). Genetic variations in TSC2 were reported in 46% of patients, whereas 14% were reported in TSC1. Mean total direct health care costs were EUR 4949 [95% confidence interval (95% CI) EUR 4088–5863, median EUR 2062] per patient over three months. Medication costs represented the largest direct cost category (54% of total direct costs, mean EUR 2658), with mechanistic target of rapamycin (mTOR) inhibitors representing the largest share (47%, EUR 2309). The cost of anti-seizure drugs (ASDs) accounted for a mean of only EUR 260 (5%). Inpatient costs (21%, EUR 1027) and ancillary therapy costs (8%, EUR 407) were also important direct cost components. The mean nursing care-level costs were EUR 1163 (95% CI EUR 1027–1314, median EUR 1635) over three months. Total indirect costs totaled a mean of EUR 2813 (95% CI EUR 2221–3394, median EUR 215) for mothers and EUR 372 (95% CI EUR 193–586, median EUR 0) for fathers. Multiple regression analyses revealed polytherapy with two or more ASDs and the use of mTOR inhibitors as independent cost-driving factors of total direct costs. Disability and psychiatric disease were independent cost-driving factors for total indirect costs as well as for nursing care-level costs. Conclusions This study revealed substantial direct (including medication), nursing care-level, and indirect costs associated with TSC over three months, highlighting the spectrum of organ manifestations and their treatment needs in the German healthcare setting. Trial registration: DRKS, DRKS00016045. Registered 01 March 2019, http://www.drks.de/DRKS00016045.

scholarly journals Measuring the Total Cost of Ankle Fractures: A Prospective Analysis

2020 ◽  
Vol 5 (4) ◽  
pp. 2473011420S0006
Author(s):  
Peter C. Noback ◽  
Tess Dougherty ◽  
Christina Freibott ◽  
Eric F. Swart ◽  
Melvin P. Rosenwasser ◽  
...  
Keyword(s):  
Return To Work ◽  
Indirect Cost ◽  
Direct Cost ◽  
Indirect Costs ◽  
The United States ◽  
Direct Costs ◽  
Cost Data ◽  
Ankle Fractures ◽  
Total Cost ◽  
True Value

Category: Trauma; Ankle Introduction/Purpose: Ankle fractures (AFx) are the most common foot and ankle fracture seen at hospitals in the United States, and are undoubtedly costly to patients. Quantification of the costs of fractures and their associated treatments has garnered increased attention in orthopedics in recent years through cost-effectiveness analysis. However, literature pertaining to AFx’s almost never reports on the indirect costs of AFx’s, and thus fails to accurately assess the true value of treatments. The purpose of this study was to prospectively assess the direct and indirect costs of AFx’s in operatively and nonoperatively treated patients. Secondary analysis included evaluation of the composition of indirect cost, the duration these costs are endured, and the factors that influence their magnitude. Methods: A prospective observational single-center study was performed. Adult patients presenting for initial consult for an AFx that could speak English or Spanish were enrolled. Polytrauma patients and those unable to provide complete indirect cost data were excluded. Patients completed a cost form that asked the money they had spent in the last week on transportation, household chores, and self-care due to their AFx. Patients were considered to have complete indirect cost data if they returned for follow-up visits until they reported no recurring indirect costs and had returned to work. Direct cost data was obtained directly from the hospital billing department. Amount collected was utilized. Direct costs included any costs incurred from staff treating the patient, supplies required for treatment, and the use of healthcare facilities. A descriptive analysis of the entire cohort and stratification by operative status was performed for the primary comparative analysis. Results: 60 patients were ultimately analyzed. Average age was 46.5 years. 55% were female. 10% of patients were diabetic. 17% smoked cigarettes actively. Weber A, B, and C fractures composed 12%, 72%, and 18% of fractures, respectively. Operatively treated patients (n=37) had a significantly higher total and direct cost than non-operative patients (P<0.01). Average salary of the 39 employed patients was $61,416 and return to work period was 11.2 weeks. In all patients, lost income accounted for the largest portion of total and indirect cost, averaging 38% of total cost. Longer periods of return to work were significantly associated with undergoing surgery and having less than a college-level education (P<0.05). Average number of weeks for indirect costs to amount to zero was 19.1. Conclusion: In patients treated operatively and nonoperatively, the largest cost component was an indirect cost: missed wages at 28.6% and 63.3%, respectively. While the majority of the direct costs of AFx’s are accrued in the period immediately following the injury, indirect cost components will regularly be incurred for nearly 5 months and often longer. The degree and duration to which these indirect costs accumulate are novel findings. Future research should no longer neglect reporting on an intervention’s impact on the indirect costs of AFx’s. [Table: see text]

Two Nosocomial Pertussis Outbreaks and Their Associated Costs—King County, Washington, 2004

2007 ◽  
Vol 28 (05) ◽  
pp. 537-543 ◽  
Author(s):  
Henry C. Baggett ◽  
Jeffrey S. Duchin ◽  
William Shelton ◽  
Danielle M. Zerr ◽  
Joan Heath ◽  
...  
Keyword(s):  
Tertiary Care ◽  
Indirect Costs ◽  
Hospital Staff ◽  
Direct Costs ◽  
Contact Tracing ◽  
King County ◽  
Care Hospital ◽  
Total Cost ◽  
Personnel Costs ◽  
Medication Costs

Objective.Pertussis outbreaks in healthcare settings result in resource-intensive control activities, but studies have rarely evaluated the associated costs. We describe and estimate costs associated with 2 nosocomial pertussis outbreaks in King County, Washington, during the period from July 25 to September 15, 2004. One outbreak occurred at a 500-bed tertiary care hospital (hospital A), and the other occurred at a 250-bed pediatric hospital (hospital B).Methods.We estimated the costs of each outbreak from the hospitals' perspective through standardized interviews with hospital staff and review of contact tracing logs. Direct costs included personnel time and laboratory and medication costs, whereas indirect costs were those resulting from hospital staff furloughs.Results.Hospital A incurred direct costs of $195,342 and indirect costs of $68,015; hospital B incurred direct costs of $71,130 and indirect costs of $50,000. Cost differences resulted primarily from higher personnel costs at hospital A ($134,536), compared with hospital B ($21,645). Total cost per pertussis case was $43,893 for hospital A (6 cases) and $30,282 for hospital B (4 cases). Total cost per person exposed to a pertussis patient were $357 for hospital A (738 exposures) and $164 for hospital B (737 exposures).Conclusions.Nosocomial pertussis outbreaks result in substantial costs to hospitals, even when the number of pertussis cases is low. The cost-effectiveness of strategies to prevent nosocomial pertussis outbreaks, including vaccination of healthcare workers, should be evaluated.

scholarly journals Quality of life and its predictors in adults with tuberous sclerosis complex (TSC): a multicentre cohort study from Germany

2021 ◽  
Vol 3 (1) ◽  
Author(s):  
Johann Philipp Zöllner ◽  
Nadine Conradi ◽  
Matthias Sauter ◽  
Markus Knuf ◽  
Susanne Knake ◽  
...  
Keyword(s):  
Quality Of Life ◽  
Regression Analysis ◽  
Adverse Events ◽  
Tuberous Sclerosis ◽  
Tuberous Sclerosis Complex ◽  
Neurological Disorders ◽  
Organ Manifestations ◽  
Neuropsychiatric Manifestations ◽  
Active Epilepsy

Abstract Background Tuberous sclerosis complex (TSC) is a monogenetic, multisystemic disease characterised by the formation of benign tumours that can affect almost all organs, caused by pathogenic variations in TSC1 or TSC2. In this multicentre study from Germany, we investigated the influence of sociodemographic, clinical, and therapeutic factors on quality of life (QoL) among individuals with TSC. Methods We assessed sociodemographic and clinical characteristics and QoL among adults with TSC throughout Germany using a validated, three-month, retrospective questionnaire. We examined predictors of health-related QoL (HRQoL) using multiple linear regression analysis and compared the QoL among patients with TSC with QoL among patients with other chronic neurological disorders. Results We enrolled 121 adults with TSC (mean age: 31.0 ± 10.5 years; range: 18–61 years, 45.5% [n = 55] women). Unemployment, a higher grade of disability, a higher number of organ manifestations, the presence of neuropsychiatric manifestations or active epilepsy, and a higher burden of therapy-related adverse events were associated with worse QoL, as measured by two QoL instruments (EuroQoL-5 dimensions [EQ-5D] and Quality of Life in Epilepsy Patients [QOLIE-31]). Neuropsychiatric and structural nervous system manifestations, the number of affected organs, and therapy-related adverse events were also associated with higher depression, as measured by the Neurological Disorders Depression Inventory for Epilepsy (NDDI-E). In multiple regression analysis, more severe therapy-related adverse events (large effect, p < 0.001), active epilepsy (large effect, p < 0.001), and neuropsychiatric manifestations (medium effect, p = 0.003) were independently associated with worse HRQoL, explaining 65% of the variance (p < 0.001). The HRQoL among patients with active TSC-associated epilepsy was worse than that among patients with drug-refractory mesial temporal lobe epilepsy (p < 0.001), and the generic QoL among patients with more than three TSC organ manifestations was similar to those of patients with severe migraine and uncontrolled asthma. Conclusions Active epilepsy, neuropsychiatric manifestations (such as anxiety and depression), and therapy-related adverse events are important independent predictors of worse quality of life among adults with TSC. Generic quality of life in TSC with several manifestations is similar to uncontrolled severe chronic diseases and significantly negatively correlates with TSC severity. Trial registration DRKS, DRKS00016045. Registered 01 March 2019.

scholarly journals Sequential everolimus for angiomyolipoma associated with tuberous sclerosis complex: a prospective cohort study

2021 ◽  
Vol 16 (1) ◽  
Author(s):  
Liangyou Gu ◽  
Cheng Peng ◽  
Fan Zhang ◽  
Cunjin Fang ◽  
Gang Guo
Keyword(s):  
Cohort Study ◽  
Treatment Group ◽  
Tuberous Sclerosis ◽  
Direct Cost ◽  
Prospective Cohort Study ◽  
Tuberous Sclerosis Complex ◽  
Prospective Cohort ◽  
Standard Treatment ◽  
Skin Lesions ◽  
Sequential Treatment

Abstract Background To evaluate the efficacy, safety and health economics of sequential everolimus in treating angiomyolipoma (AML) associated with tuberous sclerosis complex (TSC). Methods In this prospective cohort study, patients met the inclusion criteria received standard or sequential treatment according to their willingness. All patients received an initial dose of everolimus (10 mg oral, once a day) for 3 months. The standard treatment group maintained 10 mg QD for 12 months, while the sequential treatment group reduced the dose to 5 mg QD from the 4th month. The efficacy, serum everolimus concentration and safety were evaluated at 1, 3, 6, 9 and 12 months after treatment. The primary efficacy endpoint was the proportion of patients with confirmed angiomyolipoma response of at least a 50% reduction in the total volume of target AML relative to baseline. Results Between June 1, 2016 and June 1, 2017, a total of 53 patients were included. Twenty-three patients received standard treatment, 30 patients received sequential treatment. At 1, 3, 6, 9 and 12 months after treatment, the proportion of patients whose total target tumor volume decreased by ≥ 50% from baseline was 39.1% versus 36.7%, 43.5% versus 56.7%, 47.8% versus 50%, 47.8% versus 60% and 47.8% versus 23.3% respectively (P > 0.05 for all). The overall response rate of skin lesions in the two groups was 40.4%, and the response rates of skin lesions at different times were similar for two groups (P > 0.05 for all). Major adverse effects (AEs) included mouth ulceration, hypertriglyceridemia, hypercholesterolemia, menstrual disorders. There was no significant difference between the two groups in the incidence of AEs at 3 months after treatment. The incidence of overall and grade 3/4 AEs at 12 months after treatment were significantly lower in the sequential treatment group. The average direct cost of the two groups in 12 months was $15,466 and $11,120, respectively. Conclusions Compared to standard treatment, sequential treatment was equally effective, with a lower incidence of adverse events and a lower direct cost, suggesting that it may be an alternative treatment for AML associated with TSC.

The Economic Costs Associated With Physical Inactivity and Obesity in Canada: An Update

2004 ◽  
Vol 29 (1) ◽  
pp. 90-115 ◽  
Author(s):  
Peter T. Katzmarzyk ◽  
Ian Janssen
Keyword(s):  
Health Care ◽  
Health Care Costs ◽  
Physical Inactivity ◽  
Meta Analysis ◽  
Indirect Costs ◽  
Health Care Expenditures ◽  
Direct Costs ◽  
Economic Costs ◽  
Economic Output ◽  
Care Costs

The purpose of this analytical review was to estimate the direct and indirect economic costs of physical inactivity and obesity in Canada in 2001. The relative risks of diseases associated with physical inactivity and obesity were determined from a meta-analysis of existing prospective studies and applied to the health care costs of these diseases in Canada. Estimates were derived for both the direct health care expenditures and the indirect costs, which included the value of economic output lost because of illness, injury-related work disability, or premature death. The economic burden of physical inactivity was $5.3 billion ($1.6 billion in direct costs and $3.7 billion in indirect costs) while the cost associated with obesity was $4.3 billion ($1.6 billion of direct costs and $2.7 billion of indirect costs). The total economic costs of physical inactivity and obesity represented 2.6% and 2.2%, respectively, of the total health care costs in Canada. The results underscore the importance of public health efforts aimed at combating the current epidemics of physical inactivity and obesity in Canada. Key words: overweight, lifestyle, meta analysis, population attributable risk, cost-of-illness

SKIN DISEASES CAUSED BY ULTRAVIOLET RADIATION: THE COST OF ILLNESS

2003 ◽  
Vol 19 (4) ◽  
pp. 724-730 ◽  
Author(s):  
Gunnar H. Nilsson ◽  
Lennart Carlsson ◽  
Henrik Dal ◽  
Henrik Ullén
Keyword(s):  
Health Care ◽  
Ultraviolet Radiation ◽  
Cost Of Illness ◽  
Hospital Care ◽  
Skin Diseases ◽  
Indirect Costs ◽  
Direct Costs ◽  
Economic Losses ◽  
Discounted Cost ◽  
Care Costs

Objectives: To assess the annual direct and indirect costs of skin diseases caused by ultraviolet radiation.Methods: A model for cost-of-illness, including costs for hospital care, primary care, pharmaceuticals, mortality, and morbidity, for approximately 1.8 million inhabitants in Stockholm.Results: The total annual discounted cost-of-illness in Stockholm was approximately 162 million SEK (MSEK; approximate 2002 exchange rate: 1 U.S. dollar=10 SEK). The indirect costs were predominant and constituted approximately 91 MSEK (56% of total costs), mainly due to an estimated cost of mortality for cutaneous malignant melanoma of 84 MSEK. The direct costs of these diseases, approximately 71 MSEK, were predominated by hospital ambulatory care costs of approximately 33 MSEK. The direct costs constituted approximately 0.4% of the overall health-care costs for hospital care and primary health care in the area.Conclusions: Skin diseases caused by ultraviolet radiation result in moderate economic losses in the community. Therefore, it may not be easy to make successful prevention of these diseases economically beneficial.

scholarly journals THU0545 THE ASSOCIATION OF RHEUMATOID ARTHRITIS WITH HEALTH CARE COSTS FROM THE PAYERS PERSPECTIVE FROM 2001-2015

2020 ◽  
Vol 79 (Suppl 1) ◽  
pp. 512-513
Author(s):  
M. Tatangelo ◽  
G. Tomlinson ◽  
J. M. Paterson ◽  
N. Bansback ◽  
E. Keystone ◽  
...  
Keyword(s):  
Rheumatoid Arthritis ◽  
Health Care ◽  
Medical History ◽  
Health Care Costs ◽  
Indirect Costs ◽  
Direct Costs ◽  
Control Group ◽  
Research Support ◽  
Care Costs ◽  
Over Time

Background:Costing studies require long-term follow-up data ideally within a homogenous payment structure without multiple potential insurers. We examined trends in associated costs of rheumatoid arthritis from the public payer’s perspective over time.Objectives:The purpose of this study was to measure the population RA associated direct and indirect health care costs using statistical attribution methods among a population with identical health care coverage.Methods:The study was matched longitudinal study of patients with algorithm defined RA residing in Ontario Canada with an RA diagnosis from 2001-2016 (n=104,933). Cases were matched 1:1 during the year of first diagnosis to 2 distinct control groups without RA. Control group 1 were matched on year of birth, and sex (underlying costs). Control group 2 were matched on year of birth, sex, and medical history. Medical history was measured using The John Hopkins Expanded Diagnostic Clusters, a tool that categorizes people with similar conditions based upon their use of healthcare services (physician visits, hospitalizations, emergency department visits, ambulatory care). After matching, healthcare costs were reported in 2015 Canadian Dollars (CAD) overall and by cost sub-category by exposure group and calendar year before versus after RA diagnosis. RA-associated costs were measured as the difference in costs among RA patients and their corresponding matched comparators.Results:At the year of diagnosis, RA cases were predominantly female (70%, n=72,873), mean year of birth 1950, (SD=17.7, Range=1910-2000), with mean year of diagnosis 2006, (SD=6.3, Range=2001-2016). RA patients experienced inceased indirect cost acquisition compared to the direct costs of RA alone. The health care costs of RA patients were identical to age/sex/medical history matched controls yet rising up until 2 years before RA diagnosis where they diverged from age/sex/disease matched controls $3,585/ptyr compared to $3,352/ptyr. The costs of RA patients were highest $8,468 CAD/ptyr ($888,572,644 total) in the year of diagnosis declining to a steady state by 2 years post diagnosis $7,867/ptyr with a slight increase in costs by 8 years post diagnosis $8,035/ptyr in (Figure 1). Differences age/sex matched controls were detectable throughout the study time period even up to 19-years pre-diagnosis. In the year of diagnosis, the direct costs of RA were $4046/ptyr (8468-4422), while the indirect costs of RA were $1254/ptyr ($4422-$3168) and the underlying age-sex specific costs were $3,168/ptyr. Our analysis suggest that both the direct and inflammatory costs of RA are growing over time compared to matched controls (Figure 1) by year 8 the direct joint-related costs of RA were 2528 (8035-5507) the indirect costs were 1,144 (5507-4363) and the age/sex underlying costs were 4,363.Figure 1.Annual Per-Patient Total Health Care Costs Over Time Grouped by Exposure Before and After Rheumatoid Arthritis DiagnosisConclusion:The costs of RA have grown over time with medications representing a smaller than expected portion of total costs but a large increase in costs relative to matched controls. These results indicate that the direct costs of RA are decreasing over time compared to matched controls at 8 years post diagnosis and that over time, the indirect associated portion of costs remains almost constant, showing that over time the costs of indirectly related issues are becoming a larger proportion of total costs. Methodological advancements in costing attribution over time could contribute to understanding the patterns in health care resource usage among populations with chronic diseases.Disclosure of Interests:Mark Tatangelo: None declared, George Tomlinson: None declared, J Michael Paterson: None declared, Nick Bansback: None declared, Edward Keystone Grant/research support from: AbbVie; Amgen; Gilead Sciences, Inc; Lilly Pharmaceuticals; Merck; Pfizer Pharmaceuticals; PuraPharm; Sanofi, Consultant of: AbbVie; Amgen; AstraZeneca Pharma; Bristol-Myers Squibb Company; Celltrion; F. Hoffman-La Roche Ltd.; Genentech, Inc; Gilead Sciences, Inc.; Janssen, Inc; Lilly Pharmaceuticals; Merck; Myriad Autoimmune; Pfizer Pharmaceuticals, Sandoz, Sanofi-Genzyme, Samsung Bioepsis., Speakers bureau: AbbVie; Amgen; Bristol-Myers Squibb; Celltrion; F. Hoffman-La Roche Ltd, Janssen, Inc; Merck; Pfizer Pharmaceuticals; Sanofi-Genzyme; UCB, Claire Bombardier Grant/research support from: Dr Bombardier reports sources of funding for Ontario Best Practice Research Initiative Research grants from Abbvie, Janssen, Amgen, Medexus, Merck, Pfizer, and Novartis outside of the submitted work. Consulting Agreements: Abbvie, Covance, Janssen, Merck, Pfizer, Sanofi and Novartis outside of the submitted work. Advisory Board Membership: Hospira, Sandoz, Merck, Pfizer and Novartis outside of the submitted work.

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