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Cost-effectiveness of chronic fatigue self-management versus usual care: a pilot randomized controlled trial

Cost-effectiveness of chronic fatigue self-management versus usual care: a pilot randomized... Background: Fatigue is a common yet difficult to treat condition in primary care. The objective of this study is to evaluate the cost-effectiveness of a brief cognitive behavioral therapy (CBT) based fatigue self-management (FSM) intervention as compared to usual care among patients with chronic fatigue in primary care. Methods: An economic evaluation alongside of a parallel randomized controlled study design was used. Computer-generated variable-sized block randomization plan was used to assign patients into treatment groups and data collection staff were blinded to group assignments. Patients aged between 18 and 65 years with at least six months of persistent fatigue and no medical or psychiatric exclusions were enrolled from a large primary care practice in Stony Brook, New York. The FSM group (n = 37) received two sessions of a nurse-delivered, fatigue self-management protocol and a self-help book and the usual care group (n = 36) received regular medical care. The effectiveness measure was the Fatigue Severity Scale and the cost measure was total health care expenditures derived from monthly health services use diaries during follow-up. A societal perspective was adopted and bootstrapped incremental cost-effectiveness ratios (ICERs) and net monetary benefit (NMB) were calculated as measures of cost-effectiveness. Results: The ICER for FSM was -$2358, indicating that FSM dominates UC and it may generate societal cost savings as compared to usual care. Complete case analysis yielded smaller ICER (−$1199) with greater uncertainties. Net monetary benefit analysis showed that FSM has a probability of 0.833 (95% CI: 0.819, 0.847) to achieve positive NMB and the favorable results were not sensitive to assumptions about informal care or treatment costs. Conclusion: This economic evaluation found initial evidence that a two-session brief CBT-based FSM may be cost-effective as compared to usual care over 12 months. The FSM intervention is potentially a promising intervention for chronic fatigue patients in primary care. Additional research is needed to examine the reproducibility and generalizability of these findings. Trial registration: ClinicalTrials.gov (NCT00997451, March 28, 2009). Keywords: Cognitive behavioral therapy, Cost-effectiveness, Fatigue Background burden of disease was especially high among those who Chronic fatigue is characterized by persistent and recur- access specialist services [11]. ring fatigue that cannot be alleviated by rest [1,2]. It has Cognitive behavioral therapy (CBT) has been shown to been associated with lower quality of life [3-5] and higher be effective in reducing fatigue symptoms as compared health care utilization [6-10]. The cost of lost productivity to adaptive pacing therapy or usual care [12], while associated with chronic fatigue has been estimated to be graded exercise therapy (GET) has seen mixed results between £75-£129 million annually in the U.K. and the [13-16]. CBT combines elements of both behavioral ther- apy and cognitive therapy to facilitate the identification and reduction of negative thoughts and to build activity * Correspondence: meng@usf.edu tolerance and positive coping skills among chronic fatigue School of Aging Studies, University of South Florida, 13301 Bruce B Downs Blvd., MHC 1300, Tampa, FL 33620, USA patients. However, economic evaluations for the treatment Full list of author information is available at the end of the article © 2014 Meng et al.; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Meng et al. BMC Family Practice 2014, 15:184 Page 2 of 9 http://www.biomedcentral.com/1471-2296/15/184 of chronic fatigue in primary care have been limited FSM group received two individual face-to-face fatigue and the findings are generally inconclusive [17-20]. In self-management training sessions with a nurse (for up to addition, no economic evaluation study has been re- 60 minutes) and a 61-page self-management booklet ported in the US. The assessment of the relative value of containing material assigned and discussed in the two CBT will be important for programmatic and policy deci- sessions. This protocol was adapted from an efficacious sions that must balance costs and outcomes of care [21,22]. 12-session CBT program for Chronic Fatigue Syndrome The purpose of the present study is to compare the (CFS) [26]. Patients in the AC group received two sessions cost-effectiveness of a brief CBT-based Fatigue Self- with a nurse therapist regarding emotional support and Management (FSM) intervention and usual care (UC) home-based self-monitoring of symptoms, affect, and conditions in a sample of primary care patients with stress. The AC group was designed to control for therapist chronic fatigue. attention and homework assignments so that potential placebo effects can be isolated from the FSM treatment ef- Methods fect. The UC group received no treatment beyond usual Chronic fatigue self-management study medical care. All three groups were assessed at baseline The Chronic Fatigue Self-Management Study is a ran- and 12-month follow-up [23]. For the purpose of this domized controlled trial involving 111 primary care pa- study, patients in the AC group were excluded because tients with chronic fatigue in New York between 2009 the attention control would be dominated by the control and 2011. Details of the study and results of the primary group as it requires higher costs (due to therapists’ atten- end point were reported elsewhere [23]. While the study tion) with no commensurate benefit. was powered for the primary outcome of fatigue impact on functioning, the economic evaluation was designed Outcome measures as a pilot and feasibility study. All patients were re- The primary outcome was the Fatigue Severity Scale cruited from a family medicine/primary care practice (FSS). The FSS was designed to measure the effect of fa- with 14 attending physicians and 21 family practice resi- tigue on functioning. It is comprised of nine items rated dents. The inclusion criteria for participants were (a) be- on a 7-point Likert scale, where one indicates no impair- tween 18 and 65 years of age; (b) at least six months of ment and seven indicates severe impairment. A one-point persistent fatigue with no medical or psychiatric exclu- decrease on the FSS is considered clinically significant im- sions (as determined by primary care physicians and a provement. It is a validated scale for use in CFS with high psychiatric nurse). Exclusion criteria were: (a) Medical: internal consistency [27] and has been shown to be sensi- fatigue due to identifiable medical conditions (such as tive to treatment change [28]. autoimmune diseases) or to medications (such as beta blockers); (b) Psychiatric: psychosis or dementia, alcohol Service use and costs or substance abuse, depression with melancholic or Health resource use and costs were identified and valued psychotic features, and anorexia nervosa or bulimia ner- from the societal perspective for the Reference Case ana- vosa. These Axis I psychiatric diagnoses were identified lysis following the “Panel Recommendations” [29]. Health from a nurse-conducted Structured Clinical Interview for care resource use was measured with a modified version DSM-IV (SCID) [24]. The study protocol received ethical of the Client Service Receipt Inventory (CSRI), a validated approval from the Stony Brook University Institutional health care utilization diary [30], to record health service Review Board (IRB) and the drafting of this manuscript use as well as informal care for the 3 month period prior adheres to the CONSORT statement [25]. to baseline and on a monthly basis by trained staff via a After written informed consent forms and baseline telephone interview during the post-treatment follow-up assessments were obtained, patients were randomly assigned period. to one of three groups as follows: CBT-based FSM (n = 37), To evaluate the economic effects of the prescribed attention control (AC) (n = 38), and usual care (UC) treatments, we identified relevant cost categories of re- (n = 36). A variable-sized block randomization proced- source use by measuring utilization in each resource cat- ure was used to minimize potential selection bias. The egory (direct and indirect) and identifying the unit costs study statistician generated the random allocation se- (prices) of the corresponding category. As economic quence, the principal investigator conducted the initial endpoints, direct health care costs, direct non-health telephone interview, and a graduate student assigned care costs and indirect costs were included [31]. The dir- participants to interventions. Data collection staff were ect study-based health care costs included costs of the blinded to the group assignment and sample size was behavioral interventions and the economic consequences chosen to ensure adequate power to detect treatment of the programs in terms of health services utilization effect on the primary outcome. Additional details of the before and after the intervention (direct health care costs). study have been reported elsewhere [23]. The CBT-based Intervention costs include costs of personnel (clinical Meng et al. BMC Family Practice 2014, 15:184 Page 3 of 9 http://www.biomedcentral.com/1471-2296/15/184 Table 1 Unit prices used to value the different types of psychologist, nurse interventionists, and staff), training, services in the analysis (in 2010 $) material (self-help booklet), time spent by study personnel Service Unit Unit cost ($) and patients (intervention sessions and travel), facility costs (space, maintenance, and utilities), and other costs Primary care physician visit 116 (advertising and telephone services). Costs were allo- Nurse practitioner visit 87 cated to individual patients based on the number of Specialist visit 147 sessions they attended. Direct health care costs in- Physical/Occupational therapist visit 87 cluded the costs of hospitalizations and visits to health Social worker visit 73 care providers (e.g. general practitioner, specialist, physical Homeopath/Acupuncturist visit 59 therapist, alternative medicine providers) and the use of prescription and over-the-counter medications. The direct Dentist visit 147 non-health care costs include out-of-pocket expenses, Emergency room visit 638 costs of paid and unpaid help, and travel costs of attend- Hospital visit 1916 ing medical appointments. As part of the modified CSRI, Prescription medication count 31 information on the frequency of paid help, travel time for MRI count 401 medical appointments, and the number of illness-related CT count 220 absences from paid or unpaid work were collected. Indir- ect costs include the value of production lost to society Ultrasound count 50 due to illness-related absence from work (paid or unpaid). X-ray count 76 For each category of health care resources, we used Blood test count 34 standard approaches to estimate costs [32,33]. Unit costs Child/personal care hour 10 for major health care services (e.g. provider office visits) Hourly wage hour 21 and prescription medications were based on national average of Medicare payment rates, estimated from the 2010 Medical Expenditure Panel Survey (MEPS). Medi- Analysis care payment rates are widely used as approximate mea- Outcomes sures of the opportunity costs associated with health Statistical analyses were performed using STATA (Version services use in economic evaluations. Unit costs of vari- 11, College Station, TX). We first compared patients’ ous diagnostic tests were based on 2010 Medicare Phys- baseline characteristics in the FSM and UC groups using ician Fee Schedule Payment Schedule published by the appropriate tests of statistical significance (i.e. Chi-square Centers for Medicare and Medicaid Services (Table 1). test for binary variables, t-test for continuous variables). Although informal caregivers are not paid for their in- Last observation carried forward (LOCF) method was used puts, there is still a cost involved from the societal perspec- to impute the 12-month outcome data for 26 individuals tive when other opportunities are forgone. It is assumed who did not complete the 12-month assessment and no that the work provided by informal caregivers will be simi- cost data was imputed. For the effectiveness measure, lar to that of home care workers. Therefore, we used the we used the difference-in-difference approach in multi- national average hourly wage of home health and personal variate regression analysis to identify the effects of the care aides from the 2010 Occupational Employment and intervention by controlling for baseline effectiveness or Wage Estimates produced by the Bureau of Labor Statistics cost measures, as well as baseline patient characteris- (BLS) to approximate the unit cost of informal caregivers tics (age, gender, education, marital status, employ- as well as unpaid help by family and friends. ment status, number of chronic conditions, and The days of lost work were valued using average number of symptoms). wages obtained from the U.S. Census Bureau (U.S. For the cost measure, our primary interest was to Census Bureau, Statistical Abstract of the United States, examine the between-group differences in total health 2012). We calculated daily wages from annual wages and care expenditures among participants in the FSM as then estimated the total lost income for each patient as a compared to those in the UC group. Therefore, we esti- product of the total number of days missed work and daily mated total expenditures using a generalized linear model wages. For participants who did not work, we used ½ wage (GLM) with a gamma distribution and log link function to rates as estimates of lost productivity [29]. Because the account for the distributional characteristics of expend- treatment phase for all patients began in 2009 and ended iture data. We chose GLM over ordinary least squares in 2011, we used 2010 prices and did not adjust for infla- models (with log-transformed dependent variables) based tion [32]. For each patient, total health care expenditures on the modified Park Test examining the distributional were calculated as the sum of the volume of various ser- characteristics of residuals from both approaches as sug- vices multiplied by the corresponding unit costs. gested by Manning and Mullahy [22,34]. Meng et al. BMC Family Practice 2014, 15:184 Page 4 of 9 http://www.biomedcentral.com/1471-2296/15/184 Cost-effectiveness analysis estimates. Results from this analysis will show whether ICERs were estimated using the standard formula: the main findings are sensitive to changes in interven- ICER =(ΔC − ΔC )/(ΔE − ΔE ), where ΔC − ΔC is the tion costs. 1 2 1 2 1 2 difference in the average cost change from baseline to 1- year follow-up between two groups and (ΔE − ΔE )isthe Results 1 2 difference in the average effectiveness change between the Sample characteristics two groups [35]. We plotted a cost-effectiveness plane Although 75 individuals were randomized into the FSM (with a cost dimension and a FSS dimension) to show the (n = 37) and UC (n = 36) groups, the complete-case incremental change in FSS scores and in costs for FSM cost-effectiveness analysis excluded 26 individuals due to versus UC. The plane is divided into four quadrants: missing both effectiveness and costs data (Figure 1). Pa- northeast (more effective, more costly), northwest (less ef- tients in FSM and UC groups did not differ significantly fective, more costly), southwest (less effective, less costly), in baseline patient characteristics, nor are those in- and southeast (more effective, less costly). To account for cluded in the cost-effectiveness analysis differ signifi- uncertainty involved in the statistical inference, 3000 in- cantly from those excluded (data not shown). cremental cost-effectiveness values were obtained through Table 2 presents average use of services and average bootstrapping, a non-parametric method of statistical in- costs by resource use categories for both groups during ference in which the empirical sampling distribution is the study period. Overall, the FSM group had lower un- estimated by repeated re-sampling from the observed adjusted average annual total cost as compared to the distribution [36]. To evaluate the potential impact of UC group before intervention ($3026 vs. $4862) and imputation on ICER, plots from both the imputed after the intervention ($4039 vs. $6903). As a result, the sample and the complete case analysis were generated. FSM group had smaller increase in average annual total Because negative ICER may result in ambiguity as to costs over the study period ($1012 vs. $2041) even after which group is dominated, we used the net-benefit ap- the intervention costs were factored in. In terms of proach to evaluate the cost-effectiveness of the treatment effectiveness, the FSM group had bigger reduction in group as suggested in the literature [37-40]. The net bene- FSS score as compared to the UC group (0.99 vs. 0.26). fit approach can be defined as: NMB = R ΔE − ΔC, It appears that patients in the FSM group had smaller where NMB = Net Monetary Benefit, R =Threshold increases in provider visits, larger decreases in ER/ of Willingness-to-pay per unit of benefit, ΔE =differ- hospital visits and absence from work. In summary, the ence in effectiveness (net reduction in FSS score), and unadjusted analysis showed that on average, the FSM group ΔC =difference in cost. Given a certain level of willingness- had better outcome and smaller increase in costs as to-pay (often unknown from the societal perspective), compared to the UC group. NMB measures the net benefit the decision-maker is will- Table 3 summarizes the regression-adjusted incremen- ing to pay per unit of increased effectiveness (R ), less the tal cost, incremental effectiveness, and incremental cost- increase in cost (ΔC). As a result, a program is deemed effectiveness ratios (ICERs) for FSM versus UC. The cost-effective if NMB > 0 [32]. In the present study, net FSM appeared to be more effective in improving FSS benefits were calculated for each patient in the sample score and was associated with somewhat lower total using a range of values ($0 to $10000 in $50 increments) costs as compared to UC. Over the 12 months study for R to reflect the uncertainty regarding the societal period, the FSM group had an ICER of −2358 (FSM willingness-to-pay per unit of effectiveness. We then com- dominant). This means that compared to UC, FSM inter- pared differences in net benefits between FSM and UC vention generated a societal saving of $2358 for each point using bootstrapped multiple regression models controlling reduction in the FSS score. When the bootstrapped 95% for patient characteristics and pre-treatment FSS and costs. confidence interval for the ICER is considered, results suggest that the favorable ICER results for FSM should be Sensitivity analysis interpreted as preliminary evidence because zero was To test the robustness of the results, we conducted sen- included in the confidence interval. The same analysis sitivity analyses under two conservative scenarios. First, using only the complete cases yielded similar results because the cost of informal care is likely to be excluded with somewhat smaller savings with wider confidence from the total cost in the employer’s decision-making intervals, as expected from a smaller sample. process of whether to adopt the intervention, we calcu- Figure 2 shows the incremental cost-effectiveness for lated the alternative total costs by assuming that the unit FSM and AC as compared to UC in 3000 bootstrapped cost of informal care equals to zero. Second, as there is samples. Consistent with results from bivariate analysis some uncertainty regarding the cost of the FSM inter- presented in Table 2, the great majority of the ICER for vention, we also calculated total costs assuming the FSM vs. UC fell in the southeast quadrant of the ICER intervention costs are 100 percent higher than our plane, indicating than FSM is likely to be more effective Meng et al. BMC Family Practice 2014, 15:184 Page 5 of 9 http://www.biomedcentral.com/1471-2296/15/184 Assessed for eligibility Enrollment (n=289) Excluded (n=178) Due to exclusion criteria (n=102) Declined to participate (n=76) Randomized (n=111) Allocation Allocated to FSM (n=37) Allocated to AC (n=38) Allocated to UC (n=36) Completed baseline Completed baseline Completed baseline assessment (n=37) assessment (n=38) assessment (n=36) Withdrawal (n=5) Withdrawal (n=4) Withdrawal (n=2) Lost to follow-up (n=13) Lost to follow-up (n=12) Lost to follow-up (n=16) 12-mo Follow-up Excluded from cost- 7 had missing outcome 3 had missing outcome effectiveness analysis and cost data and cost data because AC is dominated by UC due to higher costs with no expected effectiveness Cost-effectiveness analysis Cost-effectiveness analysis (n=30) (n=33) 19 with complete data 18 with complete data 11 with imputed effectiveness 15 with imputed effectiveness data data Figure 1 CONSORT flow diagram. with lower costs as compared to UC. The analysis care equals to zero and scenario 2 assumes that the using complete cases yielded similar results with greater intervention costs are 100 percent higher than the costs uncertainty. calculated in the study. For the base case, even if society Figure 3 presents the cost-effectiveness acceptability values each point reduction in FSS score at $0, the prob- curve for FSM as compared to UC, as well as acceptabil- ability that the FSM would generate a positive NMB is ity curves under the two scenarios of the sensitivity ana- 0.833 (95% CI: 0.819, 0.847). Reducing the value of infor- lysis. Scenario 1 assumes that the unit cost of informal mal care had virtually no impact on NMB and doubling Meng et al. BMC Family Practice 2014, 15:184 Page 6 of 9 http://www.biomedcentral.com/1471-2296/15/184 Table 2 Costs and changes in costs for UC and FSM, by category and period Pre (3 months) Post (12 months) † † † † Variables n (%) users # of Contacts ± SD Cost ($) n (%) users # of Contacts ± SD Cost ($) Changes in Costs ($) Usual Care (UC), n = 33 1. GP visit 21 (64) 1 ± .3 70 21 (76) 0 ± .2 47 −23 2. Specialist visit 15 (45) 1 ± .5 26 15 (67) 1 ± 1.5 60 34 3. Other provider visit 11 (33) 1 ± 1.1 58 11 (67) 1 ± 1.6 93 35 4. Provider (1 + 2 + 3) 26 (79) 1 ± 1.2 97 26 (85) 3 ± 2.4 162 65 5. ER/hospital visit 2 (6) 1 ± .2 319 2 (24) 0 ± .2 260 −59 6. Rx medications 26 (79) 1 ± .6 32 26 (85) 1 ± .8 29 −3 7. Laboratory test 21 (64) 1 ± .5 44 21 (79) 1 ± .9 47 3 8. Informal care, hours 16 (48) 43 ± 38.3 427 16 (79) 33 ± 32.3 334 −93 9. Missed work, hours 13 (39) 8 ± 8.1 125 13 (55) 6 ± 4.5 91 −34 10. Total cost (4 + 5 + 6 + 7 + 8 + 9)* 1216 1726 11. Annualized average cost 4862 6903 2041 12. Intervention cost 0 0 13. Grand total (11 + 12) 4862 6903 2041 Fatigue Self-Management (FSM), n=30 1. GP visit 15 (50) 1 ± .4 70 15 (73) 0 ± .7 49 −21 2. Specialist visit 8 (27) 1 ± 1.7 37 8 (80) 1 ± .8 43 6 3. Other provider visit 8 (27) 1 ± .8 77 8 (63) 1 ± 1.8 83 6 4. Provider (1 + 2 + 3) 21 (70) 1 ± 1.3 93 21 (97) 2 ± 2 127 34 5. ER/hospital visit 3 (10) 0 ± 0 213 3 (20) 0 ± .1 102 −111 6. Rx medications 21 (70) 1 ± .6 30 21 (87) 1 ± .5 21 −9 7. Laboratory test 14 (47) 1 ± .4 38 14 (77) 1 ± 1.1 29 −9 8. Informal care, hours 8 (27) 27 ± 29.5 269 8 (40) 22 ± 29.5 220 −49 9. Missed work, hours 10 (33) 10 ± 7.5 166 10 (50) 5 ± 4.7 83 −83 10. Total cost (4 + 5 + 6 + 7 + 8 + 9)* 757 939 11. Annualized average cost 3026 3754 728 12. Intervention cost 0 285 13. Grand total (11 + 12) 3026 4039 1012 GP = General Practitioner; ER = Emergency Room; Rx = Prescription; contacts/costs were calculated among users; *Total costs during the post- period were standardized to 3-months so that the results are comparable to the pre- period. Intervention costs included: Personnel ($69), booklet ($10), time spent ($154), and facility and other ($52). the intervention costs reduced the probability of posi- our knowledge, this study appears to be the first eco- tive NMB to 0.735 (95% CI: 0.710, 0.760) assuming $0 nomic evaluation conducted alongside of a randomized willingness-to-pay. controlled trial to examine the cost-effectiveness of a brief CBT-based FSM intervention in the U.S. The ana- lysis of the outcome results at 12-month showed signifi- Discussion cant differences between the FSM group and the UC While CBT has an effect on fatigue symptoms compar- group after adjusting for baseline characteristics. The able to graded exercise therapy and counseling in pri- FSM intervention appears to be cost-effective in that it mary care, the longer term cost-effectiveness of CBT was associated with reduced fatigue impact on function- remains unclear [17-20]. This analysis tested the cost- ing and lower total costs. effectiveness of a brief two-session CBT intervention Previous studies have shown that CBT interventions with a self-management education component in a pilot conducted in primary care by trained professionals were study of primary care patients with chronic fatigue. To Meng et al. BMC Family Practice 2014, 15:184 Page 7 of 9 http://www.biomedcentral.com/1471-2296/15/184 Table 3 Adjusted incremental costs, effectiveness, and cost-effectiveness ratios Intervention Incremental cost (95% CI) Incremental effectiveness (95% CI) ICER Imputed effectiveness data, 12 mo UC Reference Reference Reference FSM -$1729 (−5125,1095) 0.73 (0.15, 1.42) FSM dominant Complete cases, 12 mo UC Reference Reference Reference FSM -$1464 (−6670,3350) 1.22 (0.16,2.55) FSM dominant UC = Usual Care; FSM = Fatigue Self-Management; CI = Confidence Interval. ICER = Incremental Cost-Effectiveness Ratio, in 2010 US dollars; ICER = −2358 for imputed data, and −1199 for complete cases. Because the magnitude of negative ICER do not convey the same information as positive ICER do, “FSM dominant” is reported to indicate that FSM is more effective at lower costs as compared to UC. Effectiveness and costs were obtained from multivariate regression models adjusting for the following baseline characteristics: age gender, education, marital status, employment status, number of chronic conditions, and number of symptoms. effective in reducing fatigue symptoms [18,19] whereas the FSM group may due to chance. Future studies of group CBT was not [15]. This study provides the first U.S. CBT interventions should focus on better data collection economic evidence that a brief FSM intervention may techniques such as a dedicated staff person for follow-up offer a promising alternative to traditional multi-session and implementing oversight processes to reduce the CBT delivered by experienced therapists, as it only in- amount of missing data [42]. However, given the self- volves two individual training sessions plus a self-help management nature of this intervention, it mimics adop- booklet, as compared to 6–16 visits in CBT trials for CFS tion rates for other behavioral interventions (such as diet patients [9]. This is consistent with the finding from a re- and exercise) in real world settings. In addition, to the cent study of non-traditional face-to-face CBT, which extent that the rates of attrition were similar across all showed that an internet-based CBT was cost-effective for three groups and that no statistical differences were de- severe health anxiety at 1-year follow-up [41]. tected between completers and non-completers, the at- A number of limitations should be considered in inter- trition is unlikely to bias our findings. Nevertheless, preting the findings. First, the attrition rate of this pilot future studies should examine what factors contribute to study was high, suggesting that the compliance for the attrition and explore whether attrition can be reduced FSM intervention is not optimal or that the burden of by modifying the intervention protocols. A second limi- monthly follow-up may be high, or both. As a result, tation is the imputation method used, which is based on findings such as a larger decrease in ER/hospital costs in the assumption that those who did not complete the Legend Complete Cases Imputed Sample -1.5 -1 -.5 0 .5 1 1.5 2 2.5 3 3.5 4 Incremental Effects Figure 2 Plots of incremental cost-effectiveness ratios for fatigue self-management and attention control from bootstrapped samples. Note: Four quadrants: northeast (more effective, more costly), northwest (less effective, more costly), southwest (less effective, less costly), and southeast (more effective, less costly). Imputed sample included 26 individuals with imputed fatigue assessment data. Incremental Costs ($) -13000 -8000 -3000 2000 7000 12000 17000 Meng et al. BMC Family Practice 2014, 15:184 Page 8 of 9 http://www.biomedcentral.com/1471-2296/15/184 Legend Base Case Scenario 1 Scenario 2 0 2000 4000 6000 8000 10000 Value of one point improvement on the FSS ($) Figure 3 Cost-effectiveness acceptability curve comparing fatigue self-management versus usual care, base case and sensitivity analysis. Note: Scenario 1: informal help was valued at $0; Scenario 2: intervention cost was valued at 2 times of the base case rate. study on average had no change in fatigue impact which findings should be cautious. Nevertheless, less labor- may not be the case. However, the complete case ana- intensive modalities of CBT such as the brief nurse-led lysis suggests that the findings were not driven by the self-management approach reported here should be imputation method used. Third, because patients were tested in future studies in primary care. recruited from one geographic location, findings may Abbreviations not be generalizable to patients in other locations. A UC: Usual care; FSM: Fatigue self-management; CI: Confidence interval; multi-center randomized controlled study is needed to FSS: Fatigue severity scale; ICER: Incremental cost-effectiveness ratio; NMB: Net monetary benefit. test whether the FSM intervention is cost-effective in more diverse chronic fatigue patient populations. Finally, Competing interests it is unclear to what extent the beneficial treatment ef- The authors declare that they have no competing interests. fect for fatigue impact on functioning lasts beyond the 1-year follow-up period. Authors’ contributions HM performed the data analysis, participated in the research design, and Despite these limitations, the present study provides im- revised the manuscript. FF originated the idea of the study, developed the portant initial evidence of cost-effectiveness for a new research proposal and led the study. FF supervised the overall study, data brief two-session CBT-based FSM intervention in primary collection, reviewed the analysis, and participated in the interpretation of the data and preparation of the manuscript. MCB participated in the care. It has the strength of incorporating the economic interpretation of the data and preparation of the manuscript. All authors data collection into the clinical outcomes measures by have read and approved the final manuscript. design and as a result, both cost and effectiveness data were collected from the same individuals [43]. Never- Acknowledgements Conflict of Interest and Source of Funding: theless, additional research is needed to examine how The study was supported by NIH grant R01NR010229 (National Institute of to improve treatment compliance and whether similar Nursing Research). cost-effectiveness can be achieved in a broader patient Author details population across multiple primary care practices and/ School of Aging Studies, University of South Florida, 13301 Bruce B Downs or regions. 2 Blvd., MHC 1300, Tampa, FL 33620, USA. Department of Psychiatry, Stony Brook University Medical Center, Department of Psychiatry and Behavioral Science, Putnam Hall/South Campus, Stony Brook, NY 11794, USA. Conclusion The brief two-session CBT-based fatigue self-management Received: 21 May 2014 Accepted: 24 October 2014 intervention appeared to be cost-effective in this pilot study inthat theinterventioncosts wasmorethanoffset bycost References savings generated from reduced health services utilization 1. 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Sabes-Figuera R, McCrone P, Hurley M, King M, Donaldson AN, Ridsdale L: Cite this article as: Meng et al.: Cost-effectiveness of chronic fatigue Cost-effectiveness of counselling, graded-exercise and usual care for self-management versus usual care: a pilot randomized controlled chronic fatigue: evidence from a randomised trial in primary care. trial. BMC Family Practice 2014 15:184. BMC Health Serv Res 2012, 12:264. 21. Yates BT: Toward the incorporation of costs, cost-effectiveness analysis, and cost-benefit analysis into clinical research. J Consult Clin Psychol 1994, 62(4):729–736. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png BMC Family Practice Springer Journals

Cost-effectiveness of chronic fatigue self-management versus usual care: a pilot randomized controlled trial

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Springer Journals
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Copyright © 2014 by Meng et al.; licensee BioMed Central Ltd.
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Medicine & Public Health; General Practice / Family Medicine; Primary Care Medicine
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1471-2296
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10.1186/s12875-014-0184-7
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25421363
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Abstract

Background: Fatigue is a common yet difficult to treat condition in primary care. The objective of this study is to evaluate the cost-effectiveness of a brief cognitive behavioral therapy (CBT) based fatigue self-management (FSM) intervention as compared to usual care among patients with chronic fatigue in primary care. Methods: An economic evaluation alongside of a parallel randomized controlled study design was used. Computer-generated variable-sized block randomization plan was used to assign patients into treatment groups and data collection staff were blinded to group assignments. Patients aged between 18 and 65 years with at least six months of persistent fatigue and no medical or psychiatric exclusions were enrolled from a large primary care practice in Stony Brook, New York. The FSM group (n = 37) received two sessions of a nurse-delivered, fatigue self-management protocol and a self-help book and the usual care group (n = 36) received regular medical care. The effectiveness measure was the Fatigue Severity Scale and the cost measure was total health care expenditures derived from monthly health services use diaries during follow-up. A societal perspective was adopted and bootstrapped incremental cost-effectiveness ratios (ICERs) and net monetary benefit (NMB) were calculated as measures of cost-effectiveness. Results: The ICER for FSM was -$2358, indicating that FSM dominates UC and it may generate societal cost savings as compared to usual care. Complete case analysis yielded smaller ICER (−$1199) with greater uncertainties. Net monetary benefit analysis showed that FSM has a probability of 0.833 (95% CI: 0.819, 0.847) to achieve positive NMB and the favorable results were not sensitive to assumptions about informal care or treatment costs. Conclusion: This economic evaluation found initial evidence that a two-session brief CBT-based FSM may be cost-effective as compared to usual care over 12 months. The FSM intervention is potentially a promising intervention for chronic fatigue patients in primary care. Additional research is needed to examine the reproducibility and generalizability of these findings. Trial registration: ClinicalTrials.gov (NCT00997451, March 28, 2009). Keywords: Cognitive behavioral therapy, Cost-effectiveness, Fatigue Background burden of disease was especially high among those who Chronic fatigue is characterized by persistent and recur- access specialist services [11]. ring fatigue that cannot be alleviated by rest [1,2]. It has Cognitive behavioral therapy (CBT) has been shown to been associated with lower quality of life [3-5] and higher be effective in reducing fatigue symptoms as compared health care utilization [6-10]. The cost of lost productivity to adaptive pacing therapy or usual care [12], while associated with chronic fatigue has been estimated to be graded exercise therapy (GET) has seen mixed results between £75-£129 million annually in the U.K. and the [13-16]. CBT combines elements of both behavioral ther- apy and cognitive therapy to facilitate the identification and reduction of negative thoughts and to build activity * Correspondence: meng@usf.edu tolerance and positive coping skills among chronic fatigue School of Aging Studies, University of South Florida, 13301 Bruce B Downs Blvd., MHC 1300, Tampa, FL 33620, USA patients. However, economic evaluations for the treatment Full list of author information is available at the end of the article © 2014 Meng et al.; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Meng et al. BMC Family Practice 2014, 15:184 Page 2 of 9 http://www.biomedcentral.com/1471-2296/15/184 of chronic fatigue in primary care have been limited FSM group received two individual face-to-face fatigue and the findings are generally inconclusive [17-20]. In self-management training sessions with a nurse (for up to addition, no economic evaluation study has been re- 60 minutes) and a 61-page self-management booklet ported in the US. The assessment of the relative value of containing material assigned and discussed in the two CBT will be important for programmatic and policy deci- sessions. This protocol was adapted from an efficacious sions that must balance costs and outcomes of care [21,22]. 12-session CBT program for Chronic Fatigue Syndrome The purpose of the present study is to compare the (CFS) [26]. Patients in the AC group received two sessions cost-effectiveness of a brief CBT-based Fatigue Self- with a nurse therapist regarding emotional support and Management (FSM) intervention and usual care (UC) home-based self-monitoring of symptoms, affect, and conditions in a sample of primary care patients with stress. The AC group was designed to control for therapist chronic fatigue. attention and homework assignments so that potential placebo effects can be isolated from the FSM treatment ef- Methods fect. The UC group received no treatment beyond usual Chronic fatigue self-management study medical care. All three groups were assessed at baseline The Chronic Fatigue Self-Management Study is a ran- and 12-month follow-up [23]. For the purpose of this domized controlled trial involving 111 primary care pa- study, patients in the AC group were excluded because tients with chronic fatigue in New York between 2009 the attention control would be dominated by the control and 2011. Details of the study and results of the primary group as it requires higher costs (due to therapists’ atten- end point were reported elsewhere [23]. While the study tion) with no commensurate benefit. was powered for the primary outcome of fatigue impact on functioning, the economic evaluation was designed Outcome measures as a pilot and feasibility study. All patients were re- The primary outcome was the Fatigue Severity Scale cruited from a family medicine/primary care practice (FSS). The FSS was designed to measure the effect of fa- with 14 attending physicians and 21 family practice resi- tigue on functioning. It is comprised of nine items rated dents. The inclusion criteria for participants were (a) be- on a 7-point Likert scale, where one indicates no impair- tween 18 and 65 years of age; (b) at least six months of ment and seven indicates severe impairment. A one-point persistent fatigue with no medical or psychiatric exclu- decrease on the FSS is considered clinically significant im- sions (as determined by primary care physicians and a provement. It is a validated scale for use in CFS with high psychiatric nurse). Exclusion criteria were: (a) Medical: internal consistency [27] and has been shown to be sensi- fatigue due to identifiable medical conditions (such as tive to treatment change [28]. autoimmune diseases) or to medications (such as beta blockers); (b) Psychiatric: psychosis or dementia, alcohol Service use and costs or substance abuse, depression with melancholic or Health resource use and costs were identified and valued psychotic features, and anorexia nervosa or bulimia ner- from the societal perspective for the Reference Case ana- vosa. These Axis I psychiatric diagnoses were identified lysis following the “Panel Recommendations” [29]. Health from a nurse-conducted Structured Clinical Interview for care resource use was measured with a modified version DSM-IV (SCID) [24]. The study protocol received ethical of the Client Service Receipt Inventory (CSRI), a validated approval from the Stony Brook University Institutional health care utilization diary [30], to record health service Review Board (IRB) and the drafting of this manuscript use as well as informal care for the 3 month period prior adheres to the CONSORT statement [25]. to baseline and on a monthly basis by trained staff via a After written informed consent forms and baseline telephone interview during the post-treatment follow-up assessments were obtained, patients were randomly assigned period. to one of three groups as follows: CBT-based FSM (n = 37), To evaluate the economic effects of the prescribed attention control (AC) (n = 38), and usual care (UC) treatments, we identified relevant cost categories of re- (n = 36). A variable-sized block randomization proced- source use by measuring utilization in each resource cat- ure was used to minimize potential selection bias. The egory (direct and indirect) and identifying the unit costs study statistician generated the random allocation se- (prices) of the corresponding category. As economic quence, the principal investigator conducted the initial endpoints, direct health care costs, direct non-health telephone interview, and a graduate student assigned care costs and indirect costs were included [31]. The dir- participants to interventions. Data collection staff were ect study-based health care costs included costs of the blinded to the group assignment and sample size was behavioral interventions and the economic consequences chosen to ensure adequate power to detect treatment of the programs in terms of health services utilization effect on the primary outcome. Additional details of the before and after the intervention (direct health care costs). study have been reported elsewhere [23]. The CBT-based Intervention costs include costs of personnel (clinical Meng et al. BMC Family Practice 2014, 15:184 Page 3 of 9 http://www.biomedcentral.com/1471-2296/15/184 Table 1 Unit prices used to value the different types of psychologist, nurse interventionists, and staff), training, services in the analysis (in 2010 $) material (self-help booklet), time spent by study personnel Service Unit Unit cost ($) and patients (intervention sessions and travel), facility costs (space, maintenance, and utilities), and other costs Primary care physician visit 116 (advertising and telephone services). Costs were allo- Nurse practitioner visit 87 cated to individual patients based on the number of Specialist visit 147 sessions they attended. Direct health care costs in- Physical/Occupational therapist visit 87 cluded the costs of hospitalizations and visits to health Social worker visit 73 care providers (e.g. general practitioner, specialist, physical Homeopath/Acupuncturist visit 59 therapist, alternative medicine providers) and the use of prescription and over-the-counter medications. The direct Dentist visit 147 non-health care costs include out-of-pocket expenses, Emergency room visit 638 costs of paid and unpaid help, and travel costs of attend- Hospital visit 1916 ing medical appointments. As part of the modified CSRI, Prescription medication count 31 information on the frequency of paid help, travel time for MRI count 401 medical appointments, and the number of illness-related CT count 220 absences from paid or unpaid work were collected. Indir- ect costs include the value of production lost to society Ultrasound count 50 due to illness-related absence from work (paid or unpaid). X-ray count 76 For each category of health care resources, we used Blood test count 34 standard approaches to estimate costs [32,33]. Unit costs Child/personal care hour 10 for major health care services (e.g. provider office visits) Hourly wage hour 21 and prescription medications were based on national average of Medicare payment rates, estimated from the 2010 Medical Expenditure Panel Survey (MEPS). Medi- Analysis care payment rates are widely used as approximate mea- Outcomes sures of the opportunity costs associated with health Statistical analyses were performed using STATA (Version services use in economic evaluations. Unit costs of vari- 11, College Station, TX). We first compared patients’ ous diagnostic tests were based on 2010 Medicare Phys- baseline characteristics in the FSM and UC groups using ician Fee Schedule Payment Schedule published by the appropriate tests of statistical significance (i.e. Chi-square Centers for Medicare and Medicaid Services (Table 1). test for binary variables, t-test for continuous variables). Although informal caregivers are not paid for their in- Last observation carried forward (LOCF) method was used puts, there is still a cost involved from the societal perspec- to impute the 12-month outcome data for 26 individuals tive when other opportunities are forgone. It is assumed who did not complete the 12-month assessment and no that the work provided by informal caregivers will be simi- cost data was imputed. For the effectiveness measure, lar to that of home care workers. Therefore, we used the we used the difference-in-difference approach in multi- national average hourly wage of home health and personal variate regression analysis to identify the effects of the care aides from the 2010 Occupational Employment and intervention by controlling for baseline effectiveness or Wage Estimates produced by the Bureau of Labor Statistics cost measures, as well as baseline patient characteris- (BLS) to approximate the unit cost of informal caregivers tics (age, gender, education, marital status, employ- as well as unpaid help by family and friends. ment status, number of chronic conditions, and The days of lost work were valued using average number of symptoms). wages obtained from the U.S. Census Bureau (U.S. For the cost measure, our primary interest was to Census Bureau, Statistical Abstract of the United States, examine the between-group differences in total health 2012). We calculated daily wages from annual wages and care expenditures among participants in the FSM as then estimated the total lost income for each patient as a compared to those in the UC group. Therefore, we esti- product of the total number of days missed work and daily mated total expenditures using a generalized linear model wages. For participants who did not work, we used ½ wage (GLM) with a gamma distribution and log link function to rates as estimates of lost productivity [29]. Because the account for the distributional characteristics of expend- treatment phase for all patients began in 2009 and ended iture data. We chose GLM over ordinary least squares in 2011, we used 2010 prices and did not adjust for infla- models (with log-transformed dependent variables) based tion [32]. For each patient, total health care expenditures on the modified Park Test examining the distributional were calculated as the sum of the volume of various ser- characteristics of residuals from both approaches as sug- vices multiplied by the corresponding unit costs. gested by Manning and Mullahy [22,34]. Meng et al. BMC Family Practice 2014, 15:184 Page 4 of 9 http://www.biomedcentral.com/1471-2296/15/184 Cost-effectiveness analysis estimates. Results from this analysis will show whether ICERs were estimated using the standard formula: the main findings are sensitive to changes in interven- ICER =(ΔC − ΔC )/(ΔE − ΔE ), where ΔC − ΔC is the tion costs. 1 2 1 2 1 2 difference in the average cost change from baseline to 1- year follow-up between two groups and (ΔE − ΔE )isthe Results 1 2 difference in the average effectiveness change between the Sample characteristics two groups [35]. We plotted a cost-effectiveness plane Although 75 individuals were randomized into the FSM (with a cost dimension and a FSS dimension) to show the (n = 37) and UC (n = 36) groups, the complete-case incremental change in FSS scores and in costs for FSM cost-effectiveness analysis excluded 26 individuals due to versus UC. The plane is divided into four quadrants: missing both effectiveness and costs data (Figure 1). Pa- northeast (more effective, more costly), northwest (less ef- tients in FSM and UC groups did not differ significantly fective, more costly), southwest (less effective, less costly), in baseline patient characteristics, nor are those in- and southeast (more effective, less costly). To account for cluded in the cost-effectiveness analysis differ signifi- uncertainty involved in the statistical inference, 3000 in- cantly from those excluded (data not shown). cremental cost-effectiveness values were obtained through Table 2 presents average use of services and average bootstrapping, a non-parametric method of statistical in- costs by resource use categories for both groups during ference in which the empirical sampling distribution is the study period. Overall, the FSM group had lower un- estimated by repeated re-sampling from the observed adjusted average annual total cost as compared to the distribution [36]. To evaluate the potential impact of UC group before intervention ($3026 vs. $4862) and imputation on ICER, plots from both the imputed after the intervention ($4039 vs. $6903). As a result, the sample and the complete case analysis were generated. FSM group had smaller increase in average annual total Because negative ICER may result in ambiguity as to costs over the study period ($1012 vs. $2041) even after which group is dominated, we used the net-benefit ap- the intervention costs were factored in. In terms of proach to evaluate the cost-effectiveness of the treatment effectiveness, the FSM group had bigger reduction in group as suggested in the literature [37-40]. The net bene- FSS score as compared to the UC group (0.99 vs. 0.26). fit approach can be defined as: NMB = R ΔE − ΔC, It appears that patients in the FSM group had smaller where NMB = Net Monetary Benefit, R =Threshold increases in provider visits, larger decreases in ER/ of Willingness-to-pay per unit of benefit, ΔE =differ- hospital visits and absence from work. In summary, the ence in effectiveness (net reduction in FSS score), and unadjusted analysis showed that on average, the FSM group ΔC =difference in cost. Given a certain level of willingness- had better outcome and smaller increase in costs as to-pay (often unknown from the societal perspective), compared to the UC group. NMB measures the net benefit the decision-maker is will- Table 3 summarizes the regression-adjusted incremen- ing to pay per unit of increased effectiveness (R ), less the tal cost, incremental effectiveness, and incremental cost- increase in cost (ΔC). As a result, a program is deemed effectiveness ratios (ICERs) for FSM versus UC. The cost-effective if NMB > 0 [32]. In the present study, net FSM appeared to be more effective in improving FSS benefits were calculated for each patient in the sample score and was associated with somewhat lower total using a range of values ($0 to $10000 in $50 increments) costs as compared to UC. Over the 12 months study for R to reflect the uncertainty regarding the societal period, the FSM group had an ICER of −2358 (FSM willingness-to-pay per unit of effectiveness. We then com- dominant). This means that compared to UC, FSM inter- pared differences in net benefits between FSM and UC vention generated a societal saving of $2358 for each point using bootstrapped multiple regression models controlling reduction in the FSS score. When the bootstrapped 95% for patient characteristics and pre-treatment FSS and costs. confidence interval for the ICER is considered, results suggest that the favorable ICER results for FSM should be Sensitivity analysis interpreted as preliminary evidence because zero was To test the robustness of the results, we conducted sen- included in the confidence interval. The same analysis sitivity analyses under two conservative scenarios. First, using only the complete cases yielded similar results because the cost of informal care is likely to be excluded with somewhat smaller savings with wider confidence from the total cost in the employer’s decision-making intervals, as expected from a smaller sample. process of whether to adopt the intervention, we calcu- Figure 2 shows the incremental cost-effectiveness for lated the alternative total costs by assuming that the unit FSM and AC as compared to UC in 3000 bootstrapped cost of informal care equals to zero. Second, as there is samples. Consistent with results from bivariate analysis some uncertainty regarding the cost of the FSM inter- presented in Table 2, the great majority of the ICER for vention, we also calculated total costs assuming the FSM vs. UC fell in the southeast quadrant of the ICER intervention costs are 100 percent higher than our plane, indicating than FSM is likely to be more effective Meng et al. BMC Family Practice 2014, 15:184 Page 5 of 9 http://www.biomedcentral.com/1471-2296/15/184 Assessed for eligibility Enrollment (n=289) Excluded (n=178) Due to exclusion criteria (n=102) Declined to participate (n=76) Randomized (n=111) Allocation Allocated to FSM (n=37) Allocated to AC (n=38) Allocated to UC (n=36) Completed baseline Completed baseline Completed baseline assessment (n=37) assessment (n=38) assessment (n=36) Withdrawal (n=5) Withdrawal (n=4) Withdrawal (n=2) Lost to follow-up (n=13) Lost to follow-up (n=12) Lost to follow-up (n=16) 12-mo Follow-up Excluded from cost- 7 had missing outcome 3 had missing outcome effectiveness analysis and cost data and cost data because AC is dominated by UC due to higher costs with no expected effectiveness Cost-effectiveness analysis Cost-effectiveness analysis (n=30) (n=33) 19 with complete data 18 with complete data 11 with imputed effectiveness 15 with imputed effectiveness data data Figure 1 CONSORT flow diagram. with lower costs as compared to UC. The analysis care equals to zero and scenario 2 assumes that the using complete cases yielded similar results with greater intervention costs are 100 percent higher than the costs uncertainty. calculated in the study. For the base case, even if society Figure 3 presents the cost-effectiveness acceptability values each point reduction in FSS score at $0, the prob- curve for FSM as compared to UC, as well as acceptabil- ability that the FSM would generate a positive NMB is ity curves under the two scenarios of the sensitivity ana- 0.833 (95% CI: 0.819, 0.847). Reducing the value of infor- lysis. Scenario 1 assumes that the unit cost of informal mal care had virtually no impact on NMB and doubling Meng et al. BMC Family Practice 2014, 15:184 Page 6 of 9 http://www.biomedcentral.com/1471-2296/15/184 Table 2 Costs and changes in costs for UC and FSM, by category and period Pre (3 months) Post (12 months) † † † † Variables n (%) users # of Contacts ± SD Cost ($) n (%) users # of Contacts ± SD Cost ($) Changes in Costs ($) Usual Care (UC), n = 33 1. GP visit 21 (64) 1 ± .3 70 21 (76) 0 ± .2 47 −23 2. Specialist visit 15 (45) 1 ± .5 26 15 (67) 1 ± 1.5 60 34 3. Other provider visit 11 (33) 1 ± 1.1 58 11 (67) 1 ± 1.6 93 35 4. Provider (1 + 2 + 3) 26 (79) 1 ± 1.2 97 26 (85) 3 ± 2.4 162 65 5. ER/hospital visit 2 (6) 1 ± .2 319 2 (24) 0 ± .2 260 −59 6. Rx medications 26 (79) 1 ± .6 32 26 (85) 1 ± .8 29 −3 7. Laboratory test 21 (64) 1 ± .5 44 21 (79) 1 ± .9 47 3 8. Informal care, hours 16 (48) 43 ± 38.3 427 16 (79) 33 ± 32.3 334 −93 9. Missed work, hours 13 (39) 8 ± 8.1 125 13 (55) 6 ± 4.5 91 −34 10. Total cost (4 + 5 + 6 + 7 + 8 + 9)* 1216 1726 11. Annualized average cost 4862 6903 2041 12. Intervention cost 0 0 13. Grand total (11 + 12) 4862 6903 2041 Fatigue Self-Management (FSM), n=30 1. GP visit 15 (50) 1 ± .4 70 15 (73) 0 ± .7 49 −21 2. Specialist visit 8 (27) 1 ± 1.7 37 8 (80) 1 ± .8 43 6 3. Other provider visit 8 (27) 1 ± .8 77 8 (63) 1 ± 1.8 83 6 4. Provider (1 + 2 + 3) 21 (70) 1 ± 1.3 93 21 (97) 2 ± 2 127 34 5. ER/hospital visit 3 (10) 0 ± 0 213 3 (20) 0 ± .1 102 −111 6. Rx medications 21 (70) 1 ± .6 30 21 (87) 1 ± .5 21 −9 7. Laboratory test 14 (47) 1 ± .4 38 14 (77) 1 ± 1.1 29 −9 8. Informal care, hours 8 (27) 27 ± 29.5 269 8 (40) 22 ± 29.5 220 −49 9. Missed work, hours 10 (33) 10 ± 7.5 166 10 (50) 5 ± 4.7 83 −83 10. Total cost (4 + 5 + 6 + 7 + 8 + 9)* 757 939 11. Annualized average cost 3026 3754 728 12. Intervention cost 0 285 13. Grand total (11 + 12) 3026 4039 1012 GP = General Practitioner; ER = Emergency Room; Rx = Prescription; contacts/costs were calculated among users; *Total costs during the post- period were standardized to 3-months so that the results are comparable to the pre- period. Intervention costs included: Personnel ($69), booklet ($10), time spent ($154), and facility and other ($52). the intervention costs reduced the probability of posi- our knowledge, this study appears to be the first eco- tive NMB to 0.735 (95% CI: 0.710, 0.760) assuming $0 nomic evaluation conducted alongside of a randomized willingness-to-pay. controlled trial to examine the cost-effectiveness of a brief CBT-based FSM intervention in the U.S. The ana- lysis of the outcome results at 12-month showed signifi- Discussion cant differences between the FSM group and the UC While CBT has an effect on fatigue symptoms compar- group after adjusting for baseline characteristics. The able to graded exercise therapy and counseling in pri- FSM intervention appears to be cost-effective in that it mary care, the longer term cost-effectiveness of CBT was associated with reduced fatigue impact on function- remains unclear [17-20]. This analysis tested the cost- ing and lower total costs. effectiveness of a brief two-session CBT intervention Previous studies have shown that CBT interventions with a self-management education component in a pilot conducted in primary care by trained professionals were study of primary care patients with chronic fatigue. To Meng et al. BMC Family Practice 2014, 15:184 Page 7 of 9 http://www.biomedcentral.com/1471-2296/15/184 Table 3 Adjusted incremental costs, effectiveness, and cost-effectiveness ratios Intervention Incremental cost (95% CI) Incremental effectiveness (95% CI) ICER Imputed effectiveness data, 12 mo UC Reference Reference Reference FSM -$1729 (−5125,1095) 0.73 (0.15, 1.42) FSM dominant Complete cases, 12 mo UC Reference Reference Reference FSM -$1464 (−6670,3350) 1.22 (0.16,2.55) FSM dominant UC = Usual Care; FSM = Fatigue Self-Management; CI = Confidence Interval. ICER = Incremental Cost-Effectiveness Ratio, in 2010 US dollars; ICER = −2358 for imputed data, and −1199 for complete cases. Because the magnitude of negative ICER do not convey the same information as positive ICER do, “FSM dominant” is reported to indicate that FSM is more effective at lower costs as compared to UC. Effectiveness and costs were obtained from multivariate regression models adjusting for the following baseline characteristics: age gender, education, marital status, employment status, number of chronic conditions, and number of symptoms. effective in reducing fatigue symptoms [18,19] whereas the FSM group may due to chance. Future studies of group CBT was not [15]. This study provides the first U.S. CBT interventions should focus on better data collection economic evidence that a brief FSM intervention may techniques such as a dedicated staff person for follow-up offer a promising alternative to traditional multi-session and implementing oversight processes to reduce the CBT delivered by experienced therapists, as it only in- amount of missing data [42]. However, given the self- volves two individual training sessions plus a self-help management nature of this intervention, it mimics adop- booklet, as compared to 6–16 visits in CBT trials for CFS tion rates for other behavioral interventions (such as diet patients [9]. This is consistent with the finding from a re- and exercise) in real world settings. In addition, to the cent study of non-traditional face-to-face CBT, which extent that the rates of attrition were similar across all showed that an internet-based CBT was cost-effective for three groups and that no statistical differences were de- severe health anxiety at 1-year follow-up [41]. tected between completers and non-completers, the at- A number of limitations should be considered in inter- trition is unlikely to bias our findings. Nevertheless, preting the findings. First, the attrition rate of this pilot future studies should examine what factors contribute to study was high, suggesting that the compliance for the attrition and explore whether attrition can be reduced FSM intervention is not optimal or that the burden of by modifying the intervention protocols. A second limi- monthly follow-up may be high, or both. As a result, tation is the imputation method used, which is based on findings such as a larger decrease in ER/hospital costs in the assumption that those who did not complete the Legend Complete Cases Imputed Sample -1.5 -1 -.5 0 .5 1 1.5 2 2.5 3 3.5 4 Incremental Effects Figure 2 Plots of incremental cost-effectiveness ratios for fatigue self-management and attention control from bootstrapped samples. Note: Four quadrants: northeast (more effective, more costly), northwest (less effective, more costly), southwest (less effective, less costly), and southeast (more effective, less costly). Imputed sample included 26 individuals with imputed fatigue assessment data. Incremental Costs ($) -13000 -8000 -3000 2000 7000 12000 17000 Meng et al. BMC Family Practice 2014, 15:184 Page 8 of 9 http://www.biomedcentral.com/1471-2296/15/184 Legend Base Case Scenario 1 Scenario 2 0 2000 4000 6000 8000 10000 Value of one point improvement on the FSS ($) Figure 3 Cost-effectiveness acceptability curve comparing fatigue self-management versus usual care, base case and sensitivity analysis. Note: Scenario 1: informal help was valued at $0; Scenario 2: intervention cost was valued at 2 times of the base case rate. study on average had no change in fatigue impact which findings should be cautious. Nevertheless, less labor- may not be the case. However, the complete case ana- intensive modalities of CBT such as the brief nurse-led lysis suggests that the findings were not driven by the self-management approach reported here should be imputation method used. Third, because patients were tested in future studies in primary care. recruited from one geographic location, findings may Abbreviations not be generalizable to patients in other locations. A UC: Usual care; FSM: Fatigue self-management; CI: Confidence interval; multi-center randomized controlled study is needed to FSS: Fatigue severity scale; ICER: Incremental cost-effectiveness ratio; NMB: Net monetary benefit. test whether the FSM intervention is cost-effective in more diverse chronic fatigue patient populations. Finally, Competing interests it is unclear to what extent the beneficial treatment ef- The authors declare that they have no competing interests. fect for fatigue impact on functioning lasts beyond the 1-year follow-up period. Authors’ contributions HM performed the data analysis, participated in the research design, and Despite these limitations, the present study provides im- revised the manuscript. FF originated the idea of the study, developed the portant initial evidence of cost-effectiveness for a new research proposal and led the study. FF supervised the overall study, data brief two-session CBT-based FSM intervention in primary collection, reviewed the analysis, and participated in the interpretation of the data and preparation of the manuscript. MCB participated in the care. It has the strength of incorporating the economic interpretation of the data and preparation of the manuscript. All authors data collection into the clinical outcomes measures by have read and approved the final manuscript. design and as a result, both cost and effectiveness data were collected from the same individuals [43]. Never- Acknowledgements Conflict of Interest and Source of Funding: theless, additional research is needed to examine how The study was supported by NIH grant R01NR010229 (National Institute of to improve treatment compliance and whether similar Nursing Research). cost-effectiveness can be achieved in a broader patient Author details population across multiple primary care practices and/ School of Aging Studies, University of South Florida, 13301 Bruce B Downs or regions. 2 Blvd., MHC 1300, Tampa, FL 33620, USA. Department of Psychiatry, Stony Brook University Medical Center, Department of Psychiatry and Behavioral Science, Putnam Hall/South Campus, Stony Brook, NY 11794, USA. Conclusion The brief two-session CBT-based fatigue self-management Received: 21 May 2014 Accepted: 24 October 2014 intervention appeared to be cost-effective in this pilot study inthat theinterventioncosts wasmorethanoffset bycost References savings generated from reduced health services utilization 1. 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Published: Nov 25, 2014

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