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to guide treatment decisions, so that more patients can be trial data. Our hypothesis was that ICBT would be cost-
treated effectively and waiting times reduced. Two studies effective compared with IBSM using the criteria of National
have analysed cost effectiveness of CBT for severe health 14
Institute for Health and Care Excellence (NICE), that is,
anxiety delivered in a conventional face-to-face format that an additional case of improve-ment or quality-adjusted
compared with treatment as usual using data from life year (QALY) would be achieved at a cost not exceeding
89 20000.
randomised controlled trials. In the first study, it was found
that CBT but not the control condition reduced consumption
of primary and second-ary healthcare contacts, but total costs
8 METHODS
were unchanged in both conditions. In the second study, a Design of the study
large-scale randomised trial, the health economic analyses This study used a prospective cost-effectiveness analysis
showed that there were no significant differences between the design and a societal perspective was taken. This meant that
9
two treatment conditions. In both of the above studies, CBT direct costs, for example, healthcare consumption, as well as
was superior in reducing health anxiety symptoms compared indirect costs, for example, work cutback, were assessed and
with treatment as usual, which means that as costs were analysed. Data were collected within the context of a
similar across groups, CBT is likely to be the more cost- randomised controlled trial of ICBT (n=79) and IBSM
effective treatment option. (n=79) for adults with severe health anxiety. As outlined by
15
Our research group has developed an internet-delivered Saha and et al, cost-effectiveness analysis is a combined
exposure-based CBT (ICBT), which has been shown to be measure of the incremental costs and gains of a new
effective in reducing health anxiety in two randomised treatment compared with an alternative. The outcome, ICER,
10 11 gives an estimate of the cost for one additional unit of
controlled trials. In terms of format, ICBT can be
improvement when administering ICBT compared with
described as internet-based bibliotherapy with online
IBSM. Information about how the ICER is calculated is
therapist support through a secure asyn-chronous online
11 12 provided below under Data analysis. The trial was pre-
messaging system. ICBT carries several advantages, registered with clinicaltrials.gov (Identifier NCT01673035).
with one of the most essential being that it can increase All participants provided written (through a web portal)
access to psychological treatment for severe health anxiety informed consent. This procedure was approved by the ethics
as each therapist can have up to 80 patients in ongoing committee.
treatment. Only one health eco-nomic evaluation of ICBT for
severe health anxiety has been conducted, and in that study it
Participants and recruitment
was found that the treatment was highly cost-effective in
comparison to a basic attention control condition that did not The total sample comprised 158 adult participants with
13 severe health anxiety, of whom 79 were randomised to ICBT
receive active treatment. The incremental cost-
and 79 to IBSM. The mean age was 41.7 years (SD=13.6) in
effectiveness ratio (ICER) was 1244, meaning that each ICBT and 41.4 years (SD=13.2) in IBSM. There were 64
case of improvement in ICBT relative to the control women (81%) in the ICBT group and the participants had
condition generated a societal net economic gain of 1244. suffered from health anxiety for 13 years on average
That is, the results suggested that when providing ICBT
(SD=13.1). In the IBSM group, there were 61 women (77%)
instead of no treatment, the most likely outcome is that
and the participants had experienced symptoms of health
patients are improved while society reduces its costs for
anxiety for 14 years (SD=13.1). The study was conducted at
health anxiety. No prior study has, however, investigated
the Karolinska Institutet in Stockholm, Sweden, but
whether ICBT is cost-effective when compared with an
recruitment was carried out nationwide. To be included in the
active and comprehensive psychological treatment. In the
study, par-ticipants who applied had to: (1) have a principal
most recently conducted trial of ICBT, we compared it with
diag-nosis of severe health anxiety (hypochondriasis)
internet-delivered behavioural stress management (IBSM),
which contrasts with ICBT in the sense that it is based on according to DSM-IV, (2) be at least 18 years old, (3) have
taking direct control over symptoms through stress no ongoing or prior episode of bipolar disorder or psychosis,
management and applied relaxation. The results showed that (4) have no ongoing substance abuse or addiction, (5) have
both treatments caused large improvements in health anxiety stable dosage since at least 2 months if on antidepressant or
but that participants who received ICBT made significantly anxiolytic medication and agree to keep the dosage constant
12 throughout the study, (6) not have severe depressive
larger reductions of health anxiety than those in IBSM. symptoms or serious suicide ideation as indicated by a total
This trial provides an excellent opportunity for investigating score or 31 or 4 on item 9 of the Montgomery sberg
the cost effective-ness of ICBT compared with an active 16
treatment and adds to the limited body of knowledge on Depression Rating Scale-Self-rated (MADRS-S ), (7)
health eco-nomic aspects of CBT for severe health anxiety. receive no concurrent psychological treatment for severe
health anxiety and have no history of completed CBT for
severe health anxiety during the past 3 years and (8) have no
The aim of this study was to investigate the cost effect- serious somatic disorder to which the health anxiety would
iveness of ICBT versus IBSM for severe health anxiety be
featuring a societal perspective and using randomised
an adequate response. Diagnostic assessments were con- to the behavioural stress management for severe health
ducted using the Mini International Neuropsychiatric 21
anxiety tested in a face-to-face format by Clark et al.
Interview (MINI) and the Anxiety Disorders Interview Applied relaxation did not strictly follow but was inspired by
Schedule. A more detailed description of the recruit-ment 22
12 the treatment developed by Ost and had previously been
procedure is presented in the Hedman et al paper. 23
tested as an internet-based interven-tion. The programme
started with progressive relax-ation, followed by release-only
Treatments relaxation, conditioning a relaxed state to a verbal cue, and
applying rapid relax-ation in distressing situations.
Both treatments comprised extensive self-help texts divided
into 12 modules with associated homework exer-cises and
participants were expected to complete at least one module Clinical outcome assessment
per week during the 12-week treatments. Participants 24
The Health Anxiety Inventory (HAI ) was the primary
accessed the modules through a secure internet-based outcome measure. This instrument has been shown to be a
treatment platform. Throughout the treatment, an online highly reliable health anxiety measure (test-retest r=0.90 and
therapist guided participants and provided feedback on Cronbachs =0.95).
homework exercises through an asynchronous messaging In order to assess health-related quality of life, we used the
system similar to email. As a main principle, the patient is 25
EQ-5D. This is an instrument designed to be a generic
exposed to the same treat-ment components as in face-to- measure of quality of life and suitable for a wide range of
face treatment. patient groups. It assesses mobility, self-care, usual activities,
26
pain/discomfort and anxiety/ depression.
Internet-delivered exposure-based CBT
The main intervention component was systematic expos-ure
to health anxiety-related situations or events in com-bination Cost assessment
with response prevention. As described in the main outcome We used the Trimbos and Institute of Medical Technology
12 27
paper, an example of this could be to trigger feared bodily Assessment Cost Questionnaire for Psychiatry (TIC-P ) to
sensations through physical exer-cise (exposure) while collect economic cost data. The TIC-P is a self-report
refraining from checking that the pulse is normal (response measure that covers three eco-nomic domains: direct medical
prevention). Mindfulness training was used throughout the costs, indirect medical costs and indirect non-medical costs.
treatment as a way to enhance exposure, that is, it was not a Direct medical costs are those associated with healthcare
stand-alone inter-vention but used as a way to increase the consumption, such as emergency care visits or visit to a
possibility that patients would conduct exposure exercises. psychiatrist. Indirect medical costs are costs that are related
This meant that participants were encouraged to use to the clinical problem, but not considered healthcare, such
mindfulness techniques as a means to handle anxiety as participating in self-help groups. Finally, indirect non-
triggered by exposure. The treatment thus differed in this medical costs are those related to loss of productivity, such as
regard from the mindfulness-based cognitive treatment by costs for sick leave or domestic productivity loss. When
17 estimating costs of productivity loss, we used the human
McManus et al, where mindfulness training is the main
treatment component. Treanor
18
has suggested that capital approach, which means that monetary losses for the
duration of the entire period of productiv-ity loss were taken
mindfulness training could facilitate extinction learning 28
during exposure through increasing awareness of condi- into account. The costs were initially assessed in Swedish
tioned triggers of anxiety. The treatment was conducted krona (Kr) and converted into US$ using 2013 as the
within an exposure-extinction paradigm and patients were reference year, yielding a Kr1 equiva-lent of US$0.1535.
encouraged to use mindfulness and acceptance towards Costs of healthcare services and med-ications were, when
aversive internal events. This treatment has pre-viously been available, obtained from official healthcare tariff indexes for
shown to be effective both when given as face-to-face services offered within the publicly funded healthcare
19
therapy and via the internet.
10 12 20 system. The costs of the ICBT and IBSM were modelled as a
function of therapist time using the same healthcare index to
determine ther-apist tariffs, that is, tariffs for licensed
Internet-delivered behavioural stress management psychologists.
The main components of IBSM were applied relaxation and
various stress management strategies including activ-ity
scheduling, structured problem-solving and increas-ing Procedures
recuperating activities. IBSM differed from ICBT in the Participants were randomised to ICBT or IBSM in a 1:1 ratio
important sense that it focused on direct symptom control using no restriction or matching. Randomisation took place
rather than on exposure to health anxiety-related events. That after inclusion assessment, meaning that no allocation bias
is, when feeling anxious, participants were encouraged to use could be present in terms of assessors knowing the status of
applied relaxation and stress reduction techniques to take forthcoming allocations. Clinical and health economic data
direct control over health anxiety symptoms. The treatment of this study were collected at baseline, that is, before
was similar treatment start and at post-
treatment. Self-report assessments were conducted using a proportion of scatter points falling south and east of a range
secure online assessment system. Internet administra-tion has of slopes through the origin of the ICER plane where a slope
been shown to be a reliable format for mea-sures of coefficient of 0 (equivalent to the x-axis) represents that
29
psychiatric symptoms and quality of life. willingness to pay (WTP) is 0 and 1 (equivalent to the y-
34
axis) that WTP is infinite. Within-group cost changes were
Data analysis analysed using sign tests due to non-normality of the cost
data. For the same reason, we used MannWhitney U tests to
STATA IC/V.11.0 (Stata Corp) and SPSS V.22.0 (IBM) were
used to conduct the statistical analyses. Clinical data were analyse between-group cost differences and Spearmans r to
analysed using a mixed-effects model approach for repeated assess association between improvement in health anxiety
data using time and treatment group as independent and gross total changes. Since attrition rates were very low,
30 we imputed missing cost data carrying the last known
variables. Effect sizes were calcu-lated using Cohens d
observation forward.
based on pooled SDs.
As for the health economic analyses, we estimated ICERs
using the formula:
Cost-effectiveness analysis and more costly. Of the ICERs in figure 1, 4340 (87%) are
The cost-effectiveness ICER was estimated to be 310/ located in the northeast quadrant, indicating that the most
0.14=2214, indicating that each additional case of clinic-ally likely outcome is that ICBT generates larger improvements
significant improvement in ICBT (total n improved=38 at additional net societal costs. Of the remaining ICERs, 436
(48%)) compared with IBSM (total n improved=27 (34%)) (9%) are located in the south-eastern quadrant, 203 (4%) in
was associated with a societal cost of $2214. This was driven the northwestern quad-rant and 21 (<1%) in the southwestern
by slightly higher total net costs in ICBT than in IBSM and quadrant.
that participants in ICBT were more likely to be clinically Figure 2 displays a cost-effectiveness acceptability curve
significantly improved. The simulation of scattered ICERs is where estimates vary as a function of societal WTP for an
pre-sented in figure 1 where ICERs located in the south- additional case of improvement. As shown in figure 2, ICBT
eastern quadrant suggest that ICBT is more effective at a has a 9% probability of being cost-effective com-pared with
lower net societal cost, whereas ICERs in the north-western IBSM if WTP for an additional improvement is $0. If WTP is
quadrant indicates that ICBT is less effective increased to $4000, the probability of ICBT being more cost-
effective increases to 72% and if
Figure 2 Cost-effectiveness
acceptability curve comparing
internet-based cognitive behaviour
therapy (ICBT) to internet-based
behavioural stress management
(IBSM).
society were willing to pay $10 000 for an additional case of most likely outcome is that ICBT in comparison to IBSM
clinically significant improvement, the probability of ICBT generates larger improvements in health-related quality of
being cost-effective compared with IBSM is 91%. life at a larger net societal cost.
non-medical costs and costs of domestic work cutback. The as severe health anxiety decreases. The significant associ-
cost-effectiveness ICER indicated that the cost of one ation of health anxiety reduction and cost reduction found in
additional case of clinically significant improvement, when this study and in a previous clinical trial of the same
treating patients with ICBT instead of IBSM, was $2214 19
treatment delivered in a face-to-face format pro-vides
while the cost-utility ICER was $10 000. Taken together, the empirical support for this. In line with this are also the results
findings indicate that ICBT is a cost-effective treatment in 8
from the trial by Seivewright and et al where it was found
comparison to IBSM. that CBT led to reduced healthcare con-sumption. Third,
In comparison to the previously conducted study since each therapist can treat four times as many patients as
investigating cost effectiveness of ICBT for severe health in a face-to-face CBT, this internet-delivered treatment can
13
anxiety, the findings of this study showed a substan-tially be an effective means of making CBT accessible to patients
higher ICER estimate. Since ICBT displayed very similar with severe health anxiety.
outcomes both in terms of treatment pre-to-post effects and
10
costs as in the previous randomised trial, the difference in The central strengths of this study were the rando-mised
ICERs across studies is explained by how the comparator design allowing for control over confounders, the high
performed. IBSM was much more effective in reducing completion rates and the prospective societal per-spective.
health anxiety than the basic control condition in the first As for limitations, this study relied on self-report to obtain
trial and also made signifi-cant cost reductions. Since ICER cost data, but this was regarded as acceptable against the
is a relative measure, this explains the higher ICER of this background that studies have demonstrated high convergence
study. Having said that, it is important to underscore that the between registry data and health economic estimates
37
ICERs of this study were rather small in a broader collected through self-report. A second limitation was that
perspective and clearly below the suggested cost- we did not include a treatment arm of face-to-face CBT,
14 which constitutes a highly important comparison treatment.
effectiveness limit pro-posed by NICE, which is 20 000.
The threshold for what should be considered a cost-effective This is an area for future research, and although available
treatment is of course to some extent arbitrary and varies effect size data suggest that face-to-face CBT and ICBT for
between countries. As described by Mihalopoulos and severe health anxiety produce similar effects, it cannot be
14
Chatterton, the WHO on Macroeconomics and Health has ruled out that the additional direct costs of face-to-face CBT
suggested that a general cost-effectiveness criterion should are balanced through larger health anxiety reductions.
be that if the cost of a disability-adjusted life year (DALY)
does not exceed the average income per capita in a given On the basis of the findings of this study, we conclude that
country, the treatment can be considered very cost- ICBT is a cost-effective treatment compared with IBSM and
35 that the treatment leads to societal cost reduc-tions offsetting
effective. In 2013, the average annual income in Sweden
36 the cost of intervention in a short time frame. ICBT has the
was approximately US$42 500, meaning that also when potential to reduce suffering from a debilitating disorder
using the WHOs criterion, the findings of this study indicate while at the same time reducing strain on limited healthcare
that ICBT is cost-effective. resources. Implementing ICBT could thus potentially be
To summarise, ICBT is extremely cost-effective in com- highly cost-effective not just from a societal perspective but
parison to no treatment and cost-effective compared with also from a health-care provider perspective. ICBT could
IBSM. These results fit well with the cost-effectiveness play an important role of making effective psychological
studies of face-to-face CBT compared with active control treatment access-ible to patients with severe health anxiety.
conditions, which showed that gross total costs were similar
in both treatment conditions but that patients who underwent
89
CBT made larger improvements.
Author affiliations
1
This study has several important implications. First, the Department of Clinical Neuroscience, Division of Psychology,
Karolinska Institutet, Stockholm, Sweden
societal net costs of providing ICBT were estimated to be 2
Department of Clinical Neuroscience, Osher Center for Integrative
offset in a time frame as short as about 3 months. That is, the Medicine, Karolinska Institutet, Stockholm, Sweden
3
societal net cost savings exceed the cost of treatment in a Department of Clinical Neuroscience, Division of Psychiatry,
short time period, which is important for policymakers, as Karolinska Institutet, Stockholm, Sweden
4
Stress Research Institute, Stockholm University, Stockholm, Sweden
ICBT thereby is a win-win treatment option in the sense that
Contributors EH conceived the study, collected data, interpreted the data,
patients are improved at no longer term net cost. Second,
analysed the data and drafted the paper. EA, BL and EAx conceived the
since the main part of cost reductions was in the realm of study, collected data, interpreted the data and drafted the paper. ML
healthcare consump-tion, it could mean that the agent that conceived the study, interpreted the data and drafted the paper.
allocates resources to treat severe health anxiety will benefit
Funding This study was funded by Karolinska Institutet and by
through overall reduced resource use. For example, it may be research grants from Stockholm County Council.
that implementing ICBT for this patient group in primary
Competing interests None declared.
care could lead to reduced strain on general practitioners as
the demand for their services decreases Ethics approval The Regional Ethics Review Board in Stockholm, Sweden.