Multiple Sclerosis in the UK

Pharmacoeconomics 2008; 26 (10): 847-860ORIGINAL RESEARCH ARTICLE 1170-7690/08/0010-0847/$48.00/0

© 2008 Adis Data Information BV. All rights reserved.

Multiple Sclerosis in the UKService Use, Costs, Quality of Life and Disability

Paul McCrone,1 Margaret Heslin,1 Martin Knapp,1,2 Paul Bull3 and Alan Thompson4

1 Institute of Psychiatry, King’s College London, London, UK2 London School of Economics, London, UK3 School of Health and Related Research, University of Sheffield, Sheffield, UK4 Institute of Neurology, University College London, London, UK

Background: Multiple sclerosis (MS) is a chronic condition that affects quality ofAbstractlife (QOL), leads to disability and requires ongoing care inputs. Few studies haveexamined the impact of QOL, disability and demographic characteristics ontreatment costs.Objective: To investigate the links between service use, costs, QOL and disabilityfor people with MS.Methods: A cross-sectional study was conducted in people with MS who weremembers of the Multiple Sclerosis Society of Great Britain and Northern Ireland.A questionnaire, consisting of sociodemographic questions, the Client ServiceReceipt Inventory, EQ-5D and Guy’s Neurological Disability Scale (GNDS), wassent to 4000 members of the Society. Service and lost employment costs werecalculated. Regression models were developed to identify the participant charac-teristics that were associated with costs, QOL and disability. All costs arepresented in year 2006–7 values.Results: Questionnaires were returned by 1942 members (48.9%). The mean(SD) EQ-5D QOL score was 0.41 (0.34), whilst the GNDS disability score was20.8 (8.9). The mean (SD) service cost over the preceding 6 months was £8397(£10 404), and most of this (71.6%) was due to informal care. Only a smallamount of cost (8.1%) was due to the use of disease-modifying drugs and drugsfor spasticity. Lost employment costs amounted to a mean of £4240. Higher costswere associated with higher levels of disability and lower levels of health-relatedQOL.Conclusions: This study showed that MS participants had high levels of serviceuse, and that QOL was low in comparison with other conditions. There weresignificant associations between costs, QOL and disability.

Background provision of health and social care services, oftenover a prolonged period of time. There are also often

Multiple sclerosis (MS) can result in high levels substantial personal and social costs to people withof disability and impaired quality of life (QOL), and the condition and their families as a result of earlythe costs associated with MS are potentially substan- retirement and informal care provided by friends ortial.[1-9] Clearly, there are costs associated with the family.

848 McCrone et al.

Various studies have been conducted that have service use data.[10] We adopted a societal perspec-assessed the cost of MS in various countries, includ- tive in that service costs and the indirect costs due toing some in the UK.[2,4,6,7] Common findings are that lost production were included. The objective was to(i) costs increase as individuals progress to higher record service use retrospectively and comprehen-levels of disability; (ii) lost employment costs ex- sively so that the broad economic impact of MSceed those of health and social care services; and could be measured. Participants were asked to give(iii) inpatient care is a key driver of service costs. details of services that they had used during theAnother important finding is that a large proportion previous 6 months because of MS and for otherof the economic burden is borne by informal carers. reasons. Services included hospital care, primary

The aims of this paper are to (i) describe service healthcare, social care, the provision of aids andutilization patterns for a sample of members of the home adaptations, tests and investigations, and med-Multiple Sclerosis Society of Great Britain and ication and informal care provided by family mem-Northern Ireland; (ii) calculate the associated costs bers and/or friends. The length of stay was recordedof these services; (iii) estimate the indirect costs of

for inpatient episodes, whilst the number of contactsMS associated with lost employment and reduced

was recorded for other services. For informal care,productivity; and (iv) identify the characteristics ofinformation was provided on the number of hourspeople with MS associated with variations in costs.per week spent providing personal care, help aroundPrevious UK studies[2,4,6,7] were conducted when usethe home, help outside the home and other tasksof disease-modifying drugs was low; an increase inspecifically because of MS. Some participants re-their use would potentially lead to changes in ser-ported that family members or friends providedvice costs. In addition, this study takes a comprehen-24-hour care often in more than one category. Al-sive approach to costing and includes multivariatethough care when sleeping or multi-tasking by car-analysis of cost variations.ers clearly occurs, we decided to code hours in

Methods excess of 119 per week (17 hours per day) as 119.

Health-related QOL (HR-QOL) was measuredusing the EQ-5D,[11] which consists of five domains:Study Designmobility, self-care, usual activities, pain/discomfort

Questionnaires were mailed to a random sample and anxiety/depression. Each domain can be rated asof 4000 members of the Multiple Sclerosis Society 1 (no problems), 2 (some problems) or 3 (majorof Great Britain and Northern Ireland in 2005. A

problem). This results in a five-figure score for eachcover letter, information sheet and prepaid envelope

respondent that reflects a unique health state. Awere included in the pack. No reminders were sent.selection of these states have been compared byNo information that could identify participants wasusing the ‘time trade-off method’ where people arecollected. Information about the study was alsoasked to state how long they would be willing toplaced in the Multiple Sclerosis Society’s newsletterspend in full health to make it equivalent to 1 yearand members could offer to participate directly.spent in an alternative health state.[12] Such a trade-Approval for the study was obtained from the ethicsoff enables a utility value to be generated anchoredcommittee of the Institute of Psychiatry (King’sby 1 (full health) and 0 (death), with allowance forCollege London), and the study was performed innegative values. Incomplete EQ-5D ratings wouldaccordance with the 1964 Declaration of Helsinki.prevent a utility score being directly estimated. InThe first questionnaire consisted of demographicsuch cases, missing utility values were replaced byquestions covering age, sex, ethnicity, marital sta-imputed scores based on a ‘best subset’ regressiontus, number of children, living arrangements andprocedure, using available EQ-5D ratings. The visu-education. This was followed by the Client Serviceal analogue scale of the EQ-5D was not used.Receipt Inventory (CSRI), which was used to collect

© 2008 Adis Data Information BV. All rights reserved. Pharmacoeconomics 2008; 26 (10)

Cost of Multiple Sclerosis in the UK 849

Disability was measured using the Guy’s Neuro- residential care homes) in order to calculate costs forlogical Disability Scale (GNDS).[13] This was used the entire 6-month period.in preference to the Expanded Disability Status The CSRI asked for information on all medica-Scale (EDSS), which is usually administered by tions taken during the previous 6 months, but in ourclinicians.[14] Although the GNDS is less commonly study only disease-modifying drugs (interferonused, it is suited to self-completion and postal [INF]-β and glatiramer acetate) and drugs to treatsurveys. It is designed to assess a range of disabili- spasticity (tizanidine and baclofen) were costed.ties encountered in MS, covering 12 areas: cogni- Other drugs were not included in the analyses; there-tion, mood, vision, speech, swallowing, lower limb fore, the costs of medication will be somewhat un-function, upper limb function, bladder function, derestimated. Costs were calculated using the unitbowel function, sexual dysfunction, fatigue and costs provided by the British National Formulary‘other’ disabilities. Each of the 12 areas is rated (BNF).[20]

between 0 (normal) and 5 (total loss of function), Lost employment costs were calculated using theand a total score is computed by summing across human capital approach. With this method, sex-scales. Missing data for any of the 12 components of specific national wage rates were used to cost timethe scale were replaced with mean scores from the not in employment during the previous 6 months.[21]

remaining components if at least six of these were Absence could be due to retirement as a conse-available. quence of illness, or employed participants might

Two additional questions were asked to deter- have days off work or reduced working hours (wemine (i) the length of time the participant had had did not distinguish between lost work time due toMS; and (ii) the type of MS if known (relapsing- MS and lost work time due to other health prob-remitting, secondary progressive or primary pro- lems).gressive). As previously stated, costs were initially based

on (or inflated to) 2003–4 values, which were theCalculation of Economic Costs most appropriately available figures as the data were

collected in 2005. However, for the purposes ofService costs were estimated by combining ser- current relevance we have inflated these figures to

vice use information with nationally applicable unit 2006–7 values using indices reported by Curtis.[22]

cost information for (or inflated to) 2003–4.[15-18] Inaddition to services listed in the CSRI, participants Analysiswere able to specify other services that they used.The costs of tests and investigations were calculated Descriptive statistics are provided for sociode-using appropriate NHS reference cost data.[19] mographic variables, service use and cost informa-

Aids and adaptations to the home generally last tion, QOL scores and disability levels. In subsequentfor a longer period of time than the 6 months used to analyses, the associations between costs and soci-cost services. Annuitized costs were available from odemographic variables, QOL and disability werea recognized source, and divided by two to obtain explored. Cost data are usually positively skewed6-month figures.[15] and, therefore, Spearman’s correlations are given

for associations between costs and other continuousInformal carers are not generally paid, but therevariables in addition to Pearson’s correlations.is still a cost to their time if opportunities are for-

gone. We assumed that if the informal carer was not Sensitivity analyses were carried out by weight-available then a paid professional homecare worker ing the mean costs for different types of MS accord-would be needed. Therefore, a unit cost of a home- ing to distributions reported by Richards et al.[23]

care worker was used to value informal carer in- From that review it was deduced that 52% of pa-puts.[15] Informal care hours per week were multi- tients would have relapsing-remitting MS, 30% se-plied by 26 (or less if time was spent in hospital or condary progressive MS and 18% primary progres-


850 McCrone et al.

reduced/increased the base-case cost of £14 per hourby 25% and 50%, respectively.

Regression models, using the ordinary leastsquares method, were used to identify participantcharacteristics that were significantly associatedwith service costs excluding informal care, servicecosts including informal care, and total costs (i.e.including lost employment). All potential variableswere entered into the models and non-significant(p > 0.1) variables were sequentially removed. Amixture of continuous and indicator variables wereincluded. The residuals of the regression modelwere checked for Normality. If residuals wereskewed, the models were repeated using bootstrap-ping methods. Separate regression models were alsoconstructed to identify variables that were asso-ciated with QOL measured by the EQ-5D, and disa-bility measured by the GNDS.

Finally, costs, QOL and disability were com-pared for participants taking the disease-modifyingdrugs INFβ and glatiramer, and all other partici-pants. All analyses were conducted using StatisticalPackage for the Social Sciences (SPSS) andSTATA.

Results

The survey was responded to by 1942 (48.9%) ofthe people approached. The mean (SD) age was 54.5(11.4) years. The majority were female, married orcohabiting, living with a spouse or partner, withoutchildren aged <18 years and were retired because ofillness (table I). Very few were in paid employment.Participants had had MS for a mean (SD) of 179(128) months. Of the participants, 1639 (84.4%)were able to identify the type of MS they had. Ofthese, 651 (39.7%) had relapsing-remitting MS, 618(37.7%) had secondary progressive MS and 358(21.8%) had primary progressive MS. In addition,12 (0.7%) had benign MS.

The sample for which complete data were avail-able for the analysis of service costs (i.e. for use inregression models) consisted of 1401 people; how-

Table I. Sample characteristics (n = 1942)a

Categories Characteristic No. (%)

Sex Male 538 (27.8)

Female 1397 (72.2)

Age group (y) <30 20 (1.1)

30–39 162 (8.5)

40–49 441 (23.2)

50–59 677 (35.6)

60–69 428 (22.5)

70–79 142 (7.5)

80–89 33 (1.7)

Marital status Married/cohabiting 1360 (70.4)

Single 168 (8.7)

Divorced/separated 288 (14.9)

Widowed 116 (6.0)

Children 0 1489 (79.6)

1 188 (10.0)

2 162 (8.7)

>3 32 (1.7)

Accommodation Alone 373 (19.3)

With parents 43 (2.2)

With children, no spouse 88 (4.6)

With spouse/partner and children 465 (24.0)

With spouse/partner 896 (46.3)

Friends 9 (0.5)

Care home 31 (1.6)

Other 29 (1.5)

Education Certificate of Secondary Education 290 (16.7)

General Certificate of Secondary 480 (27.6)Education

A level 200 (11.5)

Diploma 250 (14.4)

Degree 288 (16.5)

Higher degree 104 (6.0)

None of the above 64 (3.7)

No qualifications 65 (3.7)

Employment Employed full time 159 (8.4)

status Employed part time 184 (9.7)

Unemployed 142 (7.5)

Self-employed 55 (2.9)

Retired 180 (9.5)

Retired because of illness 950 (50.1)

Student 6 (0.3)

Housewife/husband 166 (8.8)

Other 54 (2.8)

a The number of participants varies in each category becauseof missing data.

ever, costs could be calculated for 1696 people.sive MS. A further sensitivity analysis was conduct- Those for whom complete data were not availableed around the cost of informal care where we were less likely to be married/cohabiting (65.1% vs



Disability and Health-Related Quality ofLife (QOL)

Total GNDS scores could be calculated for 1925(99.1%) of the participants. Of these, 343 (17.8%)were estimates based on ratings for between 6 and11 of the 12 components. The mean (SD) total scorewas 20.77 (8.88) with a median of 21 and a range of0 to 51. The distribution of scores reveals that nearlythree-quarters of the participants had scores between10 and 29 (table II).

Complete EQ-5D ratings were available for 1843participants (94.9%), and partial ratings from whichscores could be imputed were available for a further

Table II. Distribution of Guy’s Neurological Disability Scale (GNDS)and EQ-5D quality-of-life scoresa

Range No. (%)

GNDS

0–9 192 (10.0)

10–19 691 (35.9)

20–29 734 (38.1)

30–39 265 (13.8)

≥40 43 (2.2)

EQ-5D–0 339 (17.6)

0–0.24 305 (15.9)

0.25–0.49 105 (5.5)

0.50–0.74 967 (50.3)

≥0.75 207 (10.8)

a The number of participants varies in each category becauseof missing data.

80 (4.1%). The frequency of responses to the differ-ent levels for each domain are shown in table III. It71.5%), more likely to have been widowed (9.8% vscan be seen that for each domain most participants5.2%), more likely to have left school with thehad some or major problems. This was particularlylowest level of qualifications (21.7% vs 15.6%), lessthe case with mobility and usual activities. Problemslikely to be in employment (12.1% vs 19.4%) and

less likely to have children (16.3% vs 21.3%). They with anxiety/depression and self-care occurred leastalso were on average older (57.6 vs 53.9 years) and frequently. The mean (SD) utility score was 0.41had a lower level of disability as measured by the (0.34), with a median of 0.52 and a range of –0.59 toGNDS (mean 19.6 vs 21.0). The sample for which 1.0. The difference between the mean and median iscomplete data were available for the analysis of total indicative of a skewed distribution. Nine partici-costs consisted of 1139 people. Those for whom pants (0.5%) had the lowest possible utility score ofcomplete data were not available were less likely to –0.59, representing major problems in each EQ-5Dbe married/cohabiting (62.9% vs 74.0%), more like- domain, whilst 49 (2.5%) had the highest score,ly to be single (11.0% vs 7.6%), more likely to be where there are no problems in any domain. Theredivorced/separated (18.2% vs 13.3%), more likely

was a strong inverse association between the GNDSto have left school with the lowest level of qualifica-

and EQ-5D scores (Pearson correlation –0.561,tions (21.1% vs 15.2%), less likely to be in employ-

p < 0.001).ment (8.1% vs 22.6%) and less likely to have chil-Participants receiving INFβ or glatiramer haddren (17.4% v 21.9%). They also were on average

significantly higher QOL scores (0.56 vs 0.39) andolder (56.1 vs 53.8 years) and had a lower level ofsignificantly lower disability scores (18.6 vs 21.1)QOL as measured by the EQ-5D (mean: 0.37 vsthan those not receiving the drugs.0.43).

Table III. Distribution of EQ-5D ratings: participants [n (%)]a

Level Mobility Self-care Usual activities Pain/discomfort Anxiety/depression

No problems 158 (8.5) 787 (41.1) 174 (9.1) 345 (18.0) 782 (40.9)

Some problems 1492 (80.0) 873 (45.6) 1287 (67.1) 1277 (66.6) 1009 (52.8)

Major problems 216 (11.6) 255 (13.3) 457 (23.8) 294 (15.3) 120 (6.3)

a Complete EQ-5D ratings were available for 1843 participants (94.9%), and partial ratings from which scores could be imputed wereavailable for a further 80 (4.1%).


852 McCrone et al.

Service Use and Costs In addition to the previously mentioned serviceuse, 207 participants (10.9%) had magnetic reso-

Use of specific services is shown in table IV. nance imaging (MRI) scans, 87 (4.6%) had compu-Data are presented for services used for any reason, terised axial tomography (CAT) scans, 80 (4.3%)and those used for MS and non-MS-related reasons had ECGs and 903 (47.7%) had blood tests. The(service use and cost for any reason differs from the mean (SD) number of both MRI and CAT scans forsum of those using services for MS and non-MS those receiving them was 1.1 (0.3). The figures forreasons because some participants did not specify ECGs and blood tests were 1.6 (1.1) and 2.2 (2.6),the reason for service contacts). The average num- respectively. Informal care from family members orber of contacts/inpatient days in table IV is just for friends was received by 1438 participants (75.7%).those using these services. Half of the participants The mean (SD) number of hours per week receivedhad seen a neurologist during the previous 6 months was 19.8 (24.9), ranging from 0.5 to 119.because of MS, and relatively few for other reasons. The number of participants who took medicationAround one-fifth of the participants saw another that the BNF states as being specifically indicatedspecialist for MS and a similar proportion saw a for MS was 726 (37.4%). INFβ was received by 211specialist for non-MS reasons. Over half of the (11.1%) participants, glatiramer acetate by 32sample had seen a GP because of their MS, and (1.7%), baclofen by 432 (22.8%) and tizanidine byapproximately one-third for other reasons. Physio- 117 (6.2%). Surprisingly, the use of INFβ was nottherapy for MS symptoms was used by one-third of widespread by those with relapsing-remitting MSthe participants. Relatively few participants used (24.6%), with few participants with secondary pro-homecare workers, but when they were used, the gressive or primary progressive receiving this med-number of contacts was high (>100 for MS reasons). ication (7.6% and 0.6%, respectively).Few people used complementary healthcare ser-

Finally, a total of 531 participants (29.3%) hadvices. In addition to the services listed in table IV,either received specific aids as a result of their465 participants (24.3%) used other services, in-condition or had adaptations made to their homecluding MS nurses, occupational therapists and chi-during the previous 6 months. A summary of theropodists. A small number specified that they hadcosts of all of these services is provided in table V.night-time or live-in carers.

Total service costs could be calculated for 1696The most expensive service used was ‘other’participants (87.3%). In the remainder of cases thereinpatient care, followed by home helps, neurologywere some missing data for one or more of theinpatient care and neurology outpatient care (tableservice components. The mean total service cost forIV). Of the services used specifically because ofthese 1696 over the 6-month cost period was £8397.MS, home helps had the highest cost followed byInformal care accounted for 71.6% of service costs,‘other’ inpatient care and neurology inpatient care.contacts with professionals 17.2%, medicationIf neurology inpatient and other inpatient costs are8.1%, aids and adaptations 2.6% and tests and inves-combined it can be seen that 58% of these weretigations 0.4%. The total mean (SD) cost of servicesrelated to MS. Services not included in table IV hadused as a result of MS was £8072 (£10 247). Thisa mean (SD) cost of £226 (£2333). The total meanincluded those services specified by the CSRI ascost of inpatient stays and contacts with profession-being used for MS, and three-quarters of the costs ofals was £1445 (table V). Services used specificallytests, investigations, aids, adaptations (based on thebecause of MS had a mean (SD) cost of £933ratio of MS to non-MS costs for contacts with pro-(£2840), whilst the cost for services used for otherfessionals), informal care and medication. The meanreasons was £309 (£1560). As previously stated,(SD) cost of non-MS related services was £354some participants did not state whether the service(£1308). Therefore, the MS-specific costs representuse was MS related or not. This represents a ratio96% of the total.between MS-specific and other costs of 3 : 1.


Cost of M

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Table IV. Inpatient use and contacts with professionals and associated costs (£) in 6 months prior to the surveya [mean (SD) unless otherwise indicated]

Service Service use and costs for any reason MS-related service use and costs Non-MS-related service use and costs

n (%) contacts cost for n (%) contacts cost for n (%) contacts cost forfor users all sample for users all sample for users all sample

Neurology outpatient 1040 (54.0) 1.5 (1.4) 115 (181) 904 (50.0) 1.4 (1.3) 105 (172) 46 (2.5) 1.9 (1.8) 8 (64)

Other outpatient 787 (40.9) 2.8 (4.4) 105 (282) 372 (20.3) 2.6 (5.0) 52 (242) 383 (20.9) 2.4 (2.9) 49 (159)

Day hospital 102 (5.3) 2.1 (1.4) 11 (57) 79 (4.2) 2.3 (1.8) 9 (53) 9 (0.5) 2.7 (3.0) 1 (11)

Nursing/residential homeb 67 (3.5) 41.3 (63.7) 81 (795) 57 (3.0) 41.1 (63.1) 68 (724) 11 (0.6) 38.7 (54.8) 13 (292)

Neurology inpatientb 52 (2.7) 17.5 (25.4) 138 (1477) 41 (2.1) 15.4 (25.2) 109 (1392) 7 (0.4) 15.3 (13.0) 18 (379)

Intensive care unitb 16 (0.8) 6.9 (10.0) 74 (1320) 4 (0.2) 3.5 (2.6) 11 (284) 7 (0.4) 8.9 (11.7) 48 (1157)

Other inpatientb 151 (7.9) 13.4 (25.6) 310 (2176) 55 (2.9) 15.7 (30.8) 134 (1540) 79 (4.2) 11.1 (19.8) 160 (1543)

General practitioner 1529 (79.5) 3.4 (4.2) 58 (78) 936 (56.3) 2.9 (4.1) 35 (72) 646 (38.9) 2.6 (2.3) 21 (40)

Physiotherapist 736 (38.3) 9.0 (14.3) 63 (183) 564 (31.4) 9.1 (14.6) 57 (186) 74 (4.1) 5.0 (5.3) 4 (30)

Social worker 336 (17.5) 2.5 (3.0) 24 (77) 223 (12.2) 2.5 (3.0) 17 (74) 31 (1.7) 2.0 (1.9) 2 (20)

Practice nurse 557 (29.1) 3.0 (7.1) 8 (37) 219 (12.2) 3.6 (9.5) 4 (36) 250 (13.4) 2.0 (2.4) 2 (11)

District nurse 384 (20.0) 18.7 (40.1) 66 (371) 229 (12.6) 18.5 (36.7) 53 (323) 68 (3.7) 14.9 (39.8) 12 (183)

Speech therapist 101 (5.3) 2.1 (2.0) 2 (10) 72 (3.8) 2.1 (2.0) 1 (10) 2 (0.1) 1.0 (0.0) <1 (1)

Home help 283 (14.8) 104.7 (149.7) 183 (899) 171 (9.4) 109.9 (159.2) 162 (914) 15 (0.8) 64.7 (69.8) 9 (137)

Acupuncturist 92 (4.8) 7.5 (7.2) 10 (59) 60 (3.2) 7.4 (7.1) 7 (53) 16 (0.8) 5.3 (4.7) 1 (19)

Homeopath 50 (2.6) 3.4 (4.5) 2 (16) 34 (1.8) 2.9 (2.6) 1 (15) 6 (0.3) 1.2 (0.4) <1 (2)

Herbalist 43 (2.2) 5.7 (15.7) 3 (63) 29 (1.5) 5.7 (16.5) 2 (62) 6 (0.4) 2.4 (1.9) <1 (6)

Aromatherapy 101 (5.3) 5.2 (5.8) 7 (41) 75 (3.9) 5.1 (5.6) 4 (37) 21 (1.1) 3.5 (2.8) 1 (11)

Reflexologist 189 (9.9) 6.7 (7.2) 15 (65) 143 (7.6) 6.5 (6.9) 11 (60) 14 (0.7) 6.4 (8.7) 1 (21)

a Costs inflated to £, year 2006–7 values; n = 1401.

b Contacts = no. of days.

MS = multiple sclerosis.

854 McCrone et al.

Table V. Summary of mean 6-monthly costs by category

Category Mean (SD) Median Range

Inpatient use and contacts with professionals 1 445 (4 229) 394 0–71 691

Tests and investigations 37 (96) 2 0–992

Informal care 6 019 (9 281) 4 838 0–48 514

Medication 682 (1 753) 0 0–16 502

Aids and adaptations 222 (646) 0 0–5 025

Lost employment 4 240 (4 915) 0 0–17 267

Total service costsa 8 397 (10 404) 5 480 0–74 780

Total costs (including lost employment)a 12 655 (12 013) 10 137 0–83 999

a For some participants costs are available for some categories and not others; therefore, the categories do not exactly equate to thetotals.

Productivity and Total Costs £13 214. If the proportions in each type derivedfrom Richards et al.[23] are used, then the mean falls

There were 679 participants aged <65 (men) or to £12 598 (i.e. a difference of 5%).<60 (women) years who reported that they were Reducing informal care costs by 25% and 50%retired because of ill health (35.4% of the sample). results in services costs falling to £6901 and £5407,In addition, 158 participants (8.4%) had days off respectively. Increasing informal care costs by thework because of illness and 144 (7.7%) reduced same percentages results in mean service costs oftheir working hours. The mean cost of lost employ- £9891 and £11 389, respectively.ment through early retirement due to illness, de-creased working hours and sick days for those of Relationship between Costs, QOLusual working age was £4240 for the 6-month and Disabilityperiod. The addition of employment costs to theservice costs resulted in a mean total of £12 655. There was a significant correlation between theLost production costs and informal care accounted GNDS disability score and service costs (0.341,for 33% and 48%, respectively, of the total. p < 0.001) and total costs (i.e. including lost em-

ployment) [0.393, p < 0.001]. Costs were inverselyDrug Therapy and Total Costs related to QOL, with the correlation between the

EQ-5D utility score and service costs being –0.351Participants who had received INFβ or glatiramer

(p < 0.001), and utility and total costs being –0.361during the previous 6 months had mean service costs

(p < 0.001). With disability and QOL scoresof £10 070, compared with £8150 for those not

grouped into bands, the relationship with cost isreceiving these drugs. The total costs for the two

more apparent (figures 1 and 2).groups were £12 346 and £14 722, respectively.These differences were both statistically significant

Multivariate Analysis of Costs, QOLat the p < 0.05 level. However, the differences were

and Disabilitysubstantially less than the cost of the two drugsthemselves (mean of £4953), implying that the costs

Service Costsof other services were being offset.The relationship between service costs, including

informal care, total costs and the participant charac-Sensitivity Analysesteristics is reported in table VI. The coefficients

With benign MS excluded, the mean total costs show the impact on cost of a one-unit difference inwere £9909 for relapsing-remitting MS, £15 602 for each independent variable. The EQ-5D was excep-secondary progressive MS and £15 396 for primary tion where the coefficient shows the impact of aprogressive MS. The average cost for all three is 0.1-point variable. Age was inversely related to ser-



vice costs: each additional year of age was asso-ciated with costs that were £58 lower. Participantswho were single, divorced, separated or widowedhad service costs that on average were lower than forparticipants who were married or cohabiting. Com-pared with participants with relapsing-remittingMS, those with secondary progressive or primaryprogressive MS had significantly higher servicecosts. For every 0.1 additional point on the EQ-5Dscale, service costs were £745 lower whilst servicecosts were higher by an average of £213 for everyadditional unit in disability. Finally, there was a

20181614121086420

EQ-5D utility score

Mea

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£ ×

100

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Service costsTotal costs

<0 0–0.24 0.25–0.49 0.50–0.74 ≥0.75

Fig. 2. Relationship between EQ-5D and costs. Costs inflated to £(year 2006–7 values).

positive relationship between the number of yearswith MS and service costs. This model could explain total costs. As for the previous model, although the17.6% of the variation in service costs. There was distribution of the residuals was slightly skewed,slight skewness in the regression residuals, but all bootstrapping did not cause any of the variables tothe previously mentioned variables remained signif- become non-significant.icant after the model was bootstrapped (this was

QOLexpected given the large sample size).HR-QOL as measured by the EQ-5D was higher

the more children aged <18 years the participant hadTotal Costsand was also higher if they were employed or retiredThe regression model used to explore the varia-because of age than if they were unemployed or ation in total costs contains all the previously men-housewife/husband (table VII). QOL was reduced iftioned variables with the exception of the variable tothe participant was divorced, separated or widowed,indicate widowed participants, which is no longerand was also lower if the MS type was secondarysignificant. The magnitude of most of the effects isprogressive or primary progressive. QOL had in-broadly similar, although age has a larger impactverse relationships with duration of illness and disa-than in the previous model. In addition, women havebility.costs that are on average £2863 lower than for men.

Compared with being retired because of ill health,Disability

unemployed or a student, all of the employmentDisability measured by the GNDS was inversely

variables indicate lower costs. This model couldrelated to age, being female, being economically

explain approximately one-third of the variation inactive (or age retired), and being single, divorced orseparated (table VII). Participants with secondaryprogressive MS had significantly higher levels ofdisability than participants with relapsing-remittingor primary progressive MS.

Discussion

This study collected data from a large sample ofmembers of the Multiple Sclerosis Society of GreatBritain and Northern Ireland, and the response ratewas relatively high for a postal survey (48.9%). Thesample was similar to those used in other surveys interms of the female-to-male ratio (2.6 : 1), and

0

5

10

15

20

25

30

<10 10–19 20–29 30–39 ≥40GNDS score

Mea

n co

st (

£ ×

100

0)

Service costsTotal costs

Fig. 1. Relationship between Guy’s Neurological Disability Scale(GNDS) and costs. Costs inflated to £ (year 2006–7 values).


856 McCrone et al.

Table VI. Regression analysis of relationships between participant characteristics, service costs and total costs in patients with multiplesclerosis (MS)a

Characteristic Service costsb Total costsc

B SE p-value B SE p-value

Age –58 30 0.055 –260 39 <0.001

Femaled –2863 725 <0.001

Marital statuse

single –2482 988 0.012 –2782 1206 0.021

divorced/separated –2781 758 <0.001 –2400 924 0.010

widowed –2762 1308 0.035

Higher degreef –2241 1309 0.087

Economic activityg

full time –8113 1138 <0.001

part time –6010 1065 <0.001

self-employed –6832 1770 <0.001

retired because of age –5246 1284 <0.001

housewife/husband –8297 1136 <0.001

other –5753 1941 0.003

MS typeh

secondary progressive 1584 653 0.015 1604 767 0.037

primary progressive 1774 775 0.022 1949 916 0.034

Years with MS 62 30 0.042 73 36 0.043

EQ-5D utility scorei –745 99 <0.001 –703 119 <0.001

GNDS disability score 213 38 <0.001 287 45 <0.001

Constant 9388 1855 <0.001 31 107 2816 <0.001

a Cost inflated to £, year 2006–7 values.

b R2 = 0.176, adjusted R2 = 0.171, no. of cases = 1401.

c R2 = 0.326, adjusted R2 = 0.316, no. of cases = 1139.

d Reference category = male.

e Reference category = married/cohabiting.

f Reference category = other qualification or none.

g Reference category = unemployed including not working because of illness, and widowed.

h Reference category = relapsing-remitting.

i Regression model shows the impact of a 0.1-point change in the EQ-5D

B = regression coefficient; GNDS = Guy’s Neurological Disability Scale; SE = standard error.

length of time with MS.[23,24] The proportion of utility score was 0.42 (median 0.52), which is simi-participants with relapsing-remitting MS was lower lar to the figures of 0.49 and 0.51 reported by Kobeltthan in other studies, which is probably due to the et al.[7,25] In a study by Brazier et al.,[26] mean (andrelatively low numbers of younger people in the median) EQ-5D scores were obtained for seven dif-sample. However, although the proportion with se-

ferent conditions or populations: chronic obtrusivecondary progressive MS was relatively high, the

airways disease 0.54 (0.62), osteoarthritis 0.44same was not true of primary progressive MS, and(0.59), irritable bowel syndrome 0.66 (0.73), lowerthe sensitivity analyses using data from otherback pain 0.64 (0.69), leg ulcers 0.55 (0.62), postsources did not alter the cost findings substantially.menopausal women 0.73 (0.80) and elderly peopleHR-QOL was measured with the EQ-5D. This is0.61 (0.69). Kind et al.[27] have reported the popula-a generic measure that is especially useful in con-

ducting economic evaluations. The mean EQ-5D tion norm for the EQ-5D to be 0.86. Therefore, the



mean and median EQ-5D utility scores in this sam- elsewhere.[28,29] This possibly reflects the fact thatple of people with MS are relatively low. the sample in our study has a lower proportion of

people with relapsing-remitting MS than in otherThe disability measure used was the GNDS,studies.which is relatively new. It is considered to be appro-

priate for use in postal surveys.[13] The mean score A wide range of services was used by the sample,on the GNDS was 21.3 and the median was 21, both with most having contacts with neurologists andof which are substantially higher than those reported GPs. Inpatient care was the most expensive formal

Table VII. Regression analysis of relationships between participant characteristics, quality of life (QOL) and disability in patients withmultiple sclerosis (MS)a

Characteristic QOL (EQ-5D)b Disability (GNDS)c

B SE p-value B SE p-value

Age –0.059 0.021 0.005

Femaled –1.243 0.433 0.004

Children aged <18 y (n) 0.022 0.010 0.032

Marital statuse

single –1.629 0.748 0.030

divorced/separated –0.043 0.021 0.045 –1.519 0.558 0.007

widowed –0.074 0.034 0.032

Education levelf

Certificate of Secondary Education –0.042 0.021 0.040

higher degree 0.064 0.031 0.035

no qualifications –0.126 0.040 0.002

Economic activityg

full time 0.087 0.026 0.001 –3.679 0.712 <0.001

part time 0.092 0.024 <0.001 –2.472 0.665 <0.001

self-employed 0.106 0.042 0.011 –2.137 1.104 0.053

retired because of age 0.072 0.027 0.009 –2.622 0.752 0.001

housewife/husband –1.759 0.674 0.009

other 0.081 0.045 0.070

MS typeh

secondary progressive –0.080 0.018 <0.001 1.219 0.400 0.002

primary progressive –0.116 0.021 <0.001

Years with MS –0.003 0.001 <0.001

EQ-5D utility score –13.631 0.593 <0.001

GNDS disability score –0.019 0.001 <0.001

Constant 0.903 0.027 <0.001 32.9 1.481 <0.001

a Costs inflated to £, year 2006–7 values.

b R2 = 0.442, adjusted R2 = 0.436, no. of cases = 1284.

c R2 = 0.369, adjusted R2 = 0.364, no. of cases = 1436.

d Reference category = male.

e For EQ-5D model, reference category = married/cohabiting or single; for GNDS model, reference category = married/cohabiting orwidowed.

f Reference category = other qualifications.

g For EQ-5D model, reference category = unemployed including not working because of illness, widowed and housewife/husband; forGNDS model, reference category = unemployed including not working because of illness, widowed and other.

h For EQ-5D model, reference category = relapsing-remitting; for GNDS model, reference category = relapsing-remitting and primaryprogressive.

B = regression coefficient; GNDS = Guy’s Neurological Disability Scale; SE = standard error.


858 McCrone et al.

service. The cost of home helps was also very high, The regression analyses revealed that servicecosts were higher if participants had one of the moreeven though they were used by only 15% of theadvanced types of MS, low QOL scores or highsample. Informal care from family members orlevels of disability. These characteristics were alsofriends accounted for approximately three-quartersrelated to total costs. Age was negatively associatedof the total service cost. Sensitivity analyses re-with service and total costs. Given that disability isvealed that total service costs were very sensitive toalready included in the model, the inverse relation-changes in the cost per hour of informal care. One-ship with age suggests that as the illness progresses,third of the sample was taking medication that wascosts increase but at a diminishing rate. Women hadspecifically indicated for MS, with only 13% receiv-lower total costs than men, which may reflect theing disease-modifying drugs. This finding was un-level of earnings that are potentially lost. Partici-derstandable again because of the age distribution ofpants who were married or cohabiting had signifi-the sample.cantly higher service costs than those who were not,

Kobelt et al.[7] estimated the mean annual direct and this is due to their being more likely to be incosts of MS care in the UK to be £9022, and in receipt of informal care.current values this amounts to approximately Associations between participant characteristics,£12 050, which is 72% of the annual direct costs in QOL and disability generally revealed expected re-this study. The main explanation for the difference lationships. QOL was higher and disability lower ifmay lie in the costing of informal care. Fewer people participants were economically active or age retired.in the Kobelt et al.[7] study received informal care Secondary progressive MS was associated with low-(56% compared with 76% in our study), and the unit er QOL and more disability. However, the reasonscost of informal care was less than that used in our for older participants and women having significant-study. Kobelt et al.[7] estimated annual indirect costs ly less disability than younger participants and mencaused by lost employment to be £7695 (or £10 278 are unclear. Other studies have reported HR-QOL in

patients with MS and have found relationshipsin current values). This is comparable to the annualwith disease severity, disability, sex and employ-costs based on the findings from our study (£4240ment.[31,32]for 6 months, £8480 annually).

In a more recent study, also by Kobelt et al.,[25]

Limitationsthe costs and QOL of patients across Europe weremeasured, with 2048 recruited from members of the There were limitations to the study.Multiple Sclerosis Trust in the UK.[25,30] That study First, and most importantly, the data were col-reported more patients in the UK receiving disease- lected from members of the Multiple Sclerosismodifying drugs (21%) than we found. A similar Society of Great Britain and Northern Ireland and,amount of informal care was received in the Kobelt therefore, are probably not wholly representative ofet al.[25] sample compared with ours. Total mean people with MS in the population. In particular, theservice costs per year reported by Kobelt et al.[25]

proportion of younger people with MS in the sampleamounted to £20 035 in 2006–7 values. This sug- is low and we would expect resource use and disa-gests costs of £10 018 over 6 months, which is 19% bility to be associated to some extent with age. Formore than the costs reported in our study. This is due example, the age distribution is likely to have beento higher medication costs and also higher costs of related to the low use of disease-modifying drugs.specialists, GPs and nurses. Kobelt et al.[25] also Second, data were collected using a postal surveyfound a strong inverse relationship between EQ-5D and although the response rate was reasonable thereutility scores and disability (as measured by the is a potential selection bias. However, it is not clearEDSS), and a positive relationship between costs how this affects the representativeness of the sam-and disability. ple. Those with greater levels of disability may find



it harder to respond to such surveys, but they may ciated with so called ‘presenteeism’ (i.e. reducedalso feel the greater need to do so in order to productivity, but no reduced time spent at work).communicate the impact that MS has on their lives. This is likely to be substantial for some people withA small number of participants responded to an MS and, therefore, the lost employment costs mayinvitation to take part that was published in the be underestimated. In addition, use of the humanMultiple Sclerosis Society’s newsletter. This means capital approach is controversial and has generatedthat the sample selection was not totally random. much debate.[25,37]

The characteristics of this self-selected group may Finally, cost data frequently follow a skewedalso have differed from other people with MS. Char- distribution; therefore, the mean costs reported inacteristics of the Multiple Sclerosis Society mem- our study will not usually be the typical costs for anybers to whom the questionnaires were sent were not individual participant. However, mean costs are therecorded and we were not able to directly check most relevant for the purposes of extrapolation. Toagainst selection bias. However, the characteristics address the problem of skewness we have also pro-of the sample are similar to those reported in a vided medians and ranges when summarizing eachsimilar study by Kobelt et al.[30] If the sample is cost category (table V).biased in any way then this may affect costs. For

Conclusionexample, if the more disabled people with MS wereless likely to participate then the costs may well be

This is one of the largest studies of the economicunderestimated. Conversely, it may be that thosecosts, QOL and disability associated with MS. It haswith more severe problems are more likely to par-been shown that people with MS in the UK have aticipate if they feel that their experiences will helprelatively low level of HR-QOL. People with highothers.levels of disability and low levels of HR-QOL tend

Third, there is a limitation of self-report data, to have higher costs.which is a problem for many studies where thebreadth of service use data required limits the use- Acknowledgementsfulness of case notes or information systems.[33]

The authors thank all the people with multiple sclerosisRecall of services used during the precedingwho responded to the survey. The study was funded by the6 months may be problematic, but other studies haveMultiple Sclerosis Society of Great Britain and Northern

found self-report to be an acceptable method.[34-36]Ireland. The Society distributed the questionnaire pack tomembers but played no role in the analysis of data or theFourth, we used a pragmatic approach to imput-writing of the paper.ing missing values for the GNDS and EQ-5D. The

The authors have no conflicts of interest that are directlyapproach used needs to be validated on otherrelevant to the content of this study.

datasets.

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