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Do people with multiple sclerosis want to discuss their long-term prognosis? A nationwide study in Argentina

Published:October 16, 2019DOI:https://doi.org/10.1016/j.msard.2019.101445

      Abstract

      Background

      Demographics, clinical and imaging prognostic factors have been reported in large series of people with multiple sclerosis (PwMS). However, personalized long-term prognosis (LTP) is varied and uncertain in each particular case. Currently, there is limited evidence on how PwMS feel about prognosis communication and their coping strategies. Therefore, we aimed to assess the prognosis communication experiences and preferences of PwMS. In addition, we investigated whether demographic, clinical and neuropsychological factors are associated with prognosis information preferences.

      Methods

      A cross-sectional online survey that included 301 PwMS from Argentina was carried out. Data on self-administered surveys including prognosis in MS questionnaire (PIMS study, evaluating prognosis communication experiences, attitudes and preferences), MS impact scale (MSIS-29), Brief Coping Orientation to Problems Experienced (COPE-28) inventory, Fatigue Severity Scale and Expanded Disability Status Scale (EDSS) were evaluated. A logistic regression model was performed.

      Results

      21.5% of responders never had discussed LTP with their neurologist and 47.1% lacked clarity about their LTP. PwMS had similar preference for LTP information at diagnosis, survey (current) or in the future (72.4%, 71.7%, 73.4%, respectively). Most participants (94.3%) wanted to be informed about LTP tool availability, and 61.7% wanted to know more about conversion to SPMS. Older age (p = 0.03) and lower fatigue (p = 0.04), and COPE denial (p < 0.01), humour (p = 0.03), self-blame (p < 0.01) and venting (p = 0.02) were associated with lower LTP information preference. Trends were observed for higher MS duration (p = 0.06), physical (p = 0.07) and psychological (p = 0.08) impact. Fatigue and COPE denial were predictors of higher LTP information preference in a multivariate model.

      Conclusion

      PwMS from Argentina want more discussion and clarification about their LTP. Several physical and neuropsychological factors predict LTP information preference.

      Keywords

      1. Introduction

      Multiple sclerosis (MS) is a chronic neurodegenerative and inflammatory disease of the central nervous system (CNS) (
      • Reich D.S.
      • Lucchinetti C.F.
      • Calabresi P.A
      Multiple sclerosis.
      ), which mainly presents during the ages of 18–50 years. MS is the most frequent cause of non-traumatic neurological disability in young adults with a universal increase in incidence and prevalence over time in several countries worldwide, including the Argentinean population (
      • Koch-Henriksen N.
      • Sørensen P.S.
      The changing demographic pattern of multiple sclerosis epidemiology.
      ;
      • Cristiano E.
      • Rojas J.I.
      Multiple sclerosis epidemiology in Latin America: an updated survey.
      ;
      • Negrotto L.
      • Correale J.
      Evolution of multiple sclerosis prevalence and phenotype in Latin America.
      ). Approximately 80–85% of people with MS (PwMS) experience a relapsing-remitting (RRMS) course and around 10–15% of PwMS have a progressive decline in disability at disease onset, termed primary progressive MS (PPMS), or after some time of RRMS, termed secondary progressive MS (SPMS) (
      • Lublin F.D.
      • Reingold S.C.
      Defining the clinical course of multiple sclerosis: results of an international survey.
      ;
      • Lublin F.D.
      • Reingold S.C.
      • Cohen J.A.
      • et al.
      Defining the clinical course of multiple sclerosis: the 2013 revisions.
      ). In most PwMS, axon loss leads to the development of persistent neurological deficit over time, with potential impact over many years of life with respect to health, productivity and quality of life (QoL) (
      • Reich D.S.
      • Lucchinetti C.F.
      • Calabresi P.A
      Multiple sclerosis.
      ). Different demographic and environmental features, clinical characteristics, biomarkers and imaging prognostic factors have been reported in large series of PwMS (
      • Rotstein D.
      • Montalban X.
      Reaching an evidence-based prognosis for personalized treatment of multiple sclerosis.
      ). However, personalized long-term prognosis (LTP) is varied and uncertain in each particular case (
      • Galea I.
      • Lederer C.
      • Neuhaus A.
      • Muraro P.A.
      • Scalfari A.
      • Koch-Henriksen N.
      • et al.
      A web-based tool for personalized prediction of long-term disease course in patients with multiple sclerosis.
      ). Prognostication is useful for shared decision-making regarding selection of disease-modifying therapies (DMTs), helping to inform the joint decision of clinicians and patients (
      • Rotstein D.
      • Montalban X.
      Reaching an evidence-based prognosis for personalized treatment of multiple sclerosis.
      ;
      • Dennison L.
      • Brown M.
      • Kirby S.
      • Galea I
      Do people with multiple sclerosis want to know their prognosis? A UK nationwide study..
      ). Nevertheless, there is limited evidence regarding the level of understanding that PwMS have about their LTP and how they feel about LTP communication and their coping strategies. A United Kingdom (UK) qualitative study reported that PwMS have revealed experiences of limited prognosis communication with healthcare professionals and they are often ambivalent about the prospect of receiving more detailed and personalized prognosis information; and consider it may be emotionally deleterious and of limited usefulness (
      • Dennison L.
      • McCloy Smith E.
      • Bradbury K.
      • Galea I
      How do people with multiple sclerosis experience prognostic uncertainty and prognosis communication? A qualitative study.
      ). However, a subsequent quantitative nationwide UK study revealed that half of PwMS had never discussed LTP with healthcare professionals, and three-quarter of them had strong preferences for receiving LTP information, suggesting bias in the preceding study (
      • Dennison L.
      • Brown M.
      • Kirby S.
      • Galea I
      Do people with multiple sclerosis want to know their prognosis? A UK nationwide study..
      ). When PwMS from Germany were asked about their opinion of an online analytical processing tool to help prognostication, this was found to be of modest interest and relevance, though this study was focused around the tool, and its utility in short-term prognostication (
      • Heesen C.
      • Gaissmaier W.
      • Nguyen F.
      • Stellmann J.P.
      • Kasper J.
      • Kopke S.
      • et al.
      Prognostic risk estimates of patients with multiple sclerosis and their physicians: comparison to an online analytical risk counseling tool.
      ). In another German study of LTP, roughly half the participants found doctors' communication about disease progression inadequate, and the majority wanted to more discussion (
      • Buecken R.
      • Galushko M.
      • Golla H.
      • Strupp J.
      • Hahn M.
      • Ernstmann N.
      • et al.
      Patients feeling severely affected by multiple sclerosis: how do patients want to communicate about end-of-life issues?.
      ). In this line, different social support, socioeconomic status, access to DMTs, geography, and socio-cultural factors could influence patient preferences (
      • Rotstein D.
      • Montalban X.
      Reaching an evidence-based prognosis for personalized treatment of multiple sclerosis.
      ). Therefore, we aimed to assess the LTP communication experiences and preferences of PwMS from Argentina, who would be expected to present differences compared with people from the above-mentioned regions. In addition, we investigated whether demographic, clinical and neuropsychological factors are associated with LTP information preferences.

      2. Methods

      A nationwide cross-sectional study from 19 out of 23 Argentinean provinces was carried out between February and April 2019. An anonymous, voluntary, self-administered web-based survey was administered by the coordinating center of the study (Hospital Alemán de Buenos Aires) in collaboration with the University of Southampton, UK (I.G), using the Prognosis in MS (PIMS) questionnaire, originally designed to investigate the prognosis communication experiences, attitudes and preferences of PwMS (
      • Dennison L.
      • Brown M.
      • Kirby S.
      • Galea I
      Do people with multiple sclerosis want to know their prognosis? A UK nationwide study..
      ).
      People diagnosed with MS according to the 2017 McDonald criteria (
      • Thompson A.J.
      • Banwell B.L.
      • Barkhof F.
      • et al.
      Diagnosis of multiple sclerosis: 2017 revisions of the Mcdonald criteria.
      ), who were under follow-up of some of the co-authors (E.C.C, P.A.L, J.P.P, R.A, S.T and V.T), were invited to participate in the survey. In addition, PwMS recruited from the local Asociación de Lucha Contra la Esclerosis Múltiple (ALCEM) database were also invited to answer the survey via an email that explained the investigation and linked to the survey website. All PwMS under follow-up by the co-authors were also ALCEM members. Therefore, all ALCEM members (n = 2051) were invited to participate and 301 completed surveys were identified.

      3. Data collection

      Age at survey, MS duration, gender, MS course (RRMS, PPMS, or SPMS), MS relapses in the last 6 months, education level, current employment and use of MS medications were evaluated. To investigate how much PwMS want to know about their LTP, the PIMS questionnaire was administered, as previously used in a UK nationwide study (
      • Dennison L.
      • Brown M.
      • Kirby S.
      • Galea I
      Do people with multiple sclerosis want to know their prognosis? A UK nationwide study..
      ). This questionnaire, which included 17 questions about LTP communication experiences, attitudes and preferences, was designed and used in PwMS from the UK with a high level of comprehensibility and acceptability (
      • Dennison L.
      • Brown M.
      • Kirby S.
      • Galea I
      Do people with multiple sclerosis want to know their prognosis? A UK nationwide study..
      ). Fatigue was assessed using the Fatigue Severity Score (FSS), a self-administered questionnaire including 9 items. Scores for each item can range from 1 (without fatigue) to 7 (highest fatigue level). Responders with scores ≥45 were diagnosed as having significant fatigue (
      • Krupp L.B.
      • LaRocca N.G.
      • Muir-Nash J.
      • Steinberg A.D
      The Fatigue Severity Scale: application to patients with multiple sclerosis and systemic lupus erythematosus.
      ). The MS impact scale (MSIS-29) is a 29-item self-report measuring the physical (20-items) and psychological (9-items) impact of MS on day-to-day life in the preceding two weeks from the patient's perspective (
      • Hobart J.C.
      • Lamping D.L.
      • Fitzpatrick R.
      • et al.
      The multiple Sclerosis Impact Scale (MSIS-29); a new patient-based outcome measure.
      ). Scores for each item can range from 1 (not at all) to 5 (extremely). Each of the two sub-scales are scored by summing the responses across items, physical impact scoring between 0 and 100 and psychological impact between 0 and 45, with a higher score reflecting a greater degree of disability (
      • Hobart J.C.
      • Lamping D.L.
      • Fitzpatrick R.
      • et al.
      The multiple Sclerosis Impact Scale (MSIS-29); a new patient-based outcome measure.
      ;
      • Riazi A.
      • Hobart J.C.
      • Lamping D.L.
      • Fitzpatrick R.
      • Thompson A.J
      Multiple Sclerosis Impact Scale (MSIS-29): reliability and validity in hospital-based samples.
      ). Coping refers to a variety of cognitive and behavioural strategies individuals use to manage responses to stress (
      • Folkman S.
      • Moskowitz J.T.
      Coping: pitfalls and promise.
      ), including problem-focused and emotion-focused strategies (
      • Folkman S.
      • Lazarus R.S.
      An analysis of coping in a middle-aged community sample.
      ). The Brief Coping Orientation to Problems Experienced (COPE-28) inventory (
      • Carver C.S
      You want to measure coping but your protocol's too long: consider the brief cope.
      ) measures 14 different coping strategies across thought or action that people may adopt under stress or in difficult situations: active coping, planning, acceptance, denial, use of instrumental support, use of emotional support, humour, substance use, behavioural disengagement, self-distraction, positive reframing, self-blame, religion, and venting. For each item, respondents indicate whether they have used the coping response on a four-point Likert scale (0 = I have not been doing this at all, 1 = I have been doing this a little bit, 2 = I have been doing this a medium amount, 3 = I have been doing this a lot). In addition, we used the abbreviated Miller Behavioural Style Scale, during which PwMS were presented with fictitious threatening situations to determine their coping personality (see supplementary data) (
      • Steptoe A.
      An abbreviated version of the Miller Behavioral Style Scale.
      ;
      • Dennison L.
      • Brown M.
      • Kirby S.
      • Galea I
      Do people with multiple sclerosis want to know their prognosis? A UK nationwide study..
      ). Visual analogue scales (0–10) were also used to evaluate perceptions of (i) severity of MS and (ii) seriousness of wheelchair-dependency (
      • Boeije H.R.
      • Janssens A.C
      It might happen or it might not': how patients with multiple sclerosis explain their perception of prognostic risk.
      ). Lastly, disability was evaluated by self-reported Expanded Disability Status Scale (EDSS) (
      • Collins C.D.
      • Ivry B.
      • Bowen J.D.
      • et al.
      A comparative analysis of Patient-Reported Expanded Disability Status Scale tools.
      ;
      • Kobelt G.
      • Eriksson J.
      • Phillips G.
      • Berg J
      The burden of multiple sclerosis 2015: methods of data collection, assessment and analysis of costs, quality of life and symptoms.
      ). Patients-reported EDSS ranges from 0 (without disability) to 9 (confined to bed) and correlates highly (95% of feasibility and reliability) with a clinician-scored EDSS, as previously informed (
      • Collins C.D.
      • Ivry B.
      • Bowen J.D.
      • et al.
      A comparative analysis of Patient-Reported Expanded Disability Status Scale tools.
      ;
      • Kobelt G.
      • Eriksson J.
      • Phillips G.
      • Berg J
      The burden of multiple sclerosis 2015: methods of data collection, assessment and analysis of costs, quality of life and symptoms.
      ).
      This study was approved by the Independent Ethics Committee of the “Hospital Alemán de Buenos Aires”. All the participants signed an electronic informed consent form before data collection.

      4. Statistical analysis

      Data analysis was conducted using SPSS Statistics v22. Results are presented as percentages, mean ± SD, and median values. The Kolmogorov-Smirnov test was used to assess the normal distribution of variables. Continuous data were evaluated using T or Mann-Whitney U tests and χ2 or Fisher exact tests were performed to compare categorical data between groups, as appropriate. Current LTP information preference was dichotomized as follows: higher (want to know a lot, want to know a little) and lower (unsure, don't want to know). We applied multivariate logistic regression analysis to assess the impact of potential variables (demographic, clinical and psychological features) associated with a higher LTP information preference. All variables were included in multivariate regression if univariate analysis showed at least a trend (p < 0.20) towards association with current LTP information preference. For all the analyses, the significance level was established as p < 0.05.

      5. Results

      The survey was sent to 2051 PwMS (with one reminder after 2 weeks) and 301 of them completed the survey (14.6% response rate). General characteristics of the studied Argentinean cohort are summarized in Table 1. Most responders were female (71.4%), were of the RRMS sub-type (89.3%) and had a mean EDSS of 2.3 (±2.1), MS duration of 7.8 years (range: 1–45 years) and a mean age at survey of 39.9 years.
      Table 1General characteristics of the studied Argentinean cohort.
      General characteristics
      N (simple size)301
      Mean age at survey y, (±SD)39.9 (±11.04)
      Female No (%)215 (71.4)
      MS duration, y mean ±SD (range)7.8 ± 7.2 (1–45)
      MS course No (%)
       RRMS269 (89.3)
       PPMS17 (5.6)
       SPMS15 (4.9)
      EDSS, mean (±SD) at survey2.3 (±2.1)
      Last relapse No (%)
      < 6 months56 (18.6)
      Live No (%)
       Alone42 (13.9)
       Spouse or partner256 (85.02)
       Other3 (1)
      Education No (%)
       Primary school44 (14.6)
       High school80 (26.5)
       Tertiary education76 (25.2)
       University101 (33.5)
      Employment No (%)
       Employed (full-time)110 (36.5)
       Employed (part-time)44 (14.6)
       Unemployed28 (9.3)
       Self-employed37 (12.2)
       Housewife25 (8.3)
       Retired12 (3.9)
       Retired by MS24 (7.9)
       Currently studying21 (6.9)
      Fatigue severity scale
       ≥ 45 No (%)131 (43.5)
       Mean (±SD)38.2 (±20)
      MSIS-29 score
       Physical impact mean (±SD)46.4 (±21.6)
       Physiological impact mean (±SD)23.6 (±10.2)
      Use of medications No (%)
       Injectables83 (27.5)
        IFN, Pegylated IFN, GA
       Oral138 (45.8)
        Fingolimod, Teriflunomide, Dimethyl fumarate
       Intravenous37 (12.9)
        Natalizumab, Alemtuzumab, Ocrelizumab
       None43 (14.2)
      As shown in Table 2, 21.5% of responders had never discussed LTP with their neurologist, and when LTP was discussed during the neurology interview, the subject was brought up by the patients in 48.7% of the cases and by the neurologists in 57.2% of them. Interestingly, 47.1% (n = 141) of PwMS lacked clarity about their LTP (100 out of 141 [70.9%] of these had discussions about LTP), but 30.2% and 16.6% of responders thought about their LTP daily and weekly, respectively. In addition, PwMS from Argentina felt that the information communicated was inconsistent among different healthcare professionals, with the most commonly identified source of inconsistency being among different neurologists (63.8%) or different healthcare professionals (21.6%). PwMS had similar preference for LTP information at diagnosis, survey (current) or in the future (72.4%, 71.7%, 73.4%, respectively). Most responders (94.3%) wanted to be informed about LTP tool availability, and 61.7% (166 out of 269) of RRMS patients wanted to know more about conversion to SPMS.
      Table 2Long-term prognosis communication experiences and preferences.
      Prognosis communication experiences and preferences
      Survey ItemAnswer optionsN (%)
      Discussion of LTP during neurology appointmentsYes236 (78.4%)
      No65 (21.5%)
      Who started LTP discussion
      Question applicable only to participants who answered “yes” to discussion of LTP.
      ,
      Response option was “Tick all that apply” therefore% will not add up to 100 and there may be different n responders for each response option.
      Patient115 (48.7%)
      Neurologist136 (57.2%)
      Nurse5 (2.1%)
      General physician7 (2.9%)
      Other healthcare professional11 (4.6%)
      Family member/friend18 (7.6%)
      Do not know/cannot remember22 (9.3%)
      LTP message consistency
      Question applicable only to participants who answered “yes” to discussion of LTP.
      Similar214 (71.1%)
      Different38 (12.6%)
      Did not talk with health professionals49 (16.2%)
      Source of LTP message inconsistency
      Question applicable only to participants who answered “yes” to discussion of LTP.
      ,
      Response option was “Tick all that apply” therefore% will not add up to 100 and there may be different n responders for each response option.
      ,
      Question applicable only to participants who answered “different” to LTP message consistency.
      Neurologists53 (63.8%)
      General physicians12 (14.4%)
      Different healthcare professionals18 (21.6%)
      Clarity about LTPNo idea142 (47.1%)
      Very rough idea68 (22.5%)
      Rough idea46 (15.2%)
      Accurate idea26 (8.6%)
      Very accurate idea19 (6.3%)
      Frequency of thinking about prognosisDaily91 (30.2%)
      Weekly50 (16.6%)
      Monthly41 (13.6%)
      Once a year18 (5.9%)
      Rarely84 (27.9%)
      Never17 (5.6%)
      Who is LTP discussed with (non-healthcare professionals)
      Response option was “Tick all that apply” therefore% will not add up to 100 and there may be different n responders for each response option.
      Partner or spouse107 (35.5%)
      Parents49 (16.2%)
      Children6 (1.9%)
      Other family members1 (0.3%)
      Friends28 (9.3%)
      Colleagues at work7 (2.3%)
      Employer1 (0.3%)
      No-one102 (33.8%)
      Preference for LTP information: current [301]Want to know a lot216 (71.7%)
      Want to know a little30 (9.9%)
      Not sure40 (13.2%)
      Do not want to know15 (4.9%)
      Preference for LTP information: in future [301]Want to know a lot221 (73.4%)
      Want to know a little29 (9.6%)
      Not sure35 (11.6%)
      Do not want to know16 (5.3%)
      Preference for LTP information: at diagnosis [301]Want to know a lot218 (72.4%)
      Want to know a little35 (11.6%)
      Not sure37 (12.2%)
      Do not want to know11 (3.6%)
      Understanding LTP is useful for decisions about
      Response option was “Tick all that apply” therefore% will not add up to 100 and there may be different n responders for each response option.
      [433 responses from 301 patients]
      Treatment131 (43.5%)
      Relationships58 (19.2%)
      Family planning80 (26.5%)
      Job matters60 (19.9%)
      Financial planning52 (17.2%)
      Drawing up a will8 (2.6%)
      End of life medical decisions44 (14.6%)
      Want to be informed about LTP tool availability [301]Yes284 (94.3%)
      No17 (5.6%)
      Acceptable timings of LTP tool use
      Response option was “Tick all that apply” therefore% will not add up to 100 and there may be different n responders for each response option.
      [357 responses from 301 patients]
      At diagnosis133 (44.1%)
      Weeks/months post-diagnosis40 (13.2%)
      At time of treatment decision94 (31.2%)
      At time of life decision52 (17.2%)
      Other times12 (3.9%)
      Never14 (4.6%)
      Acceptable settings for LTP tool use
      Response option was “Tick all that apply” therefore% will not add up to 100 and there may be different n responders for each response option.
      ,
      Applicable only to participants who did not answer “never” to acceptable timings of tool use. eF/FM = friend or family member.
      [411 responses from 301 patients]
      Independently, alone102 (33.8%)
      Independently, with F/FM
      F/FM = friend or family member..
      83 (27.5%)
      With neurologist88 (29.2%)
      With neurologist and F/FM125 (41.5%)
      With MS nurse5 (1.6%)
      With MS nurse and F/FM8 (2.6%)
      Desirable prognostic estimates
      Response option was “Tick all that apply” therefore% will not add up to 100 and there may be different n responders for each response option.
      ,
      Applicable only to participants who did not answer “never” to acceptable timings of tool use. eF/FM = friend or family member.
      [475 responses from 301 patients]
      If/when need stick82 (27.2%)
      If/when need wheelchair87 (28.9%)
      If/when convert to SPMS
      Response option applicable only to participants with RRMS.
      156 (57.9%)
      Life expectancy150 (49.8%)
      Endorsement of public availability of LTP tool e.g. on webYes170 (36.6%)
      No40 (27.0%)
      Not sure83 (36.4%)
      a Question applicable only to participants who answered “yes” to discussion of LTP.
      b Response option was “Tick all that apply” therefore% will not add up to 100 and there may be different n responders for each response option.
      c Question applicable only to participants who answered “different” to LTP message consistency.
      d Applicable only to participants who did not answer “never” to acceptable timings of tool use.eF/FM = friend or family member.
      e F/FM = friend or family member..
      f Response option applicable only to participants with RRMS.
      As shown in Table 3, older age (p = 0.03), lower Fatigue Severity Score (p = 0.01), and lower levels of coping strategies on COPE denial (p < 0.01), humour (p = 0.03), self-blame (p < 0.01) and venting (p = 0.02) were associated with lower LTP information preference. In addition, statistical trends suggesting a lower LTP information preference were observed in PwMS with higher MS duration (p = 0.06), and lower physical (p = 0.07) and psychological (p = 0.08) impact. There were no statistical differences when we compared lower vs. higher LTP information preference (attitudes towards LTP) as regards gender, MS duration, education level, employment status, EDSS score, and MS course. In addition, there were no statistical differences when we evaluated perceptions of severity of MS and seriousness of wheelchair-dependency. As shown in Table 4, univariate analysis showed that age, gender, MS duration, education level, employment status, EDSS score, and MS course were not associated with LTP information preference. However, higher levels of fatigue and COPE denial were predictors of higher LTP information preference in a multivariate model.
      Table 3Comparison of demographics and clinical features among lower and higher current LTP information preference groups.
      Current LTP information preference (Dichotomized: Lower/Higher)
      Potential variables associated with current LTP information preferenceLower (N = 55)Higher (N = 246)P-value (effect size)
      Age at survey42.16 ± 1.75439.47 ± 0.67110.03 (r=0.008)
      Female35 (63.4)180 (73.1)0.21
      MS duration9.182 ± 1.1237.557 ± 0.44130.06 (r=0.007)
      Education (University or not)20 (36.3)104 (42.2)0.51
      Employment (Yes or not)35 (63.6)156 (63.4)0.90
      EDSS2.045 ± 0.27592.427 ± 0.13630.71 (r = 0.004)
      MS course (RR or progressive form)8 (14.5)24 (9.7)0.42
      COPE-28 (scores between 0–3)
      COPE active4.109 ± 0.23884.386 ± 0.10400.26 (r = 0.004)
      COPE planning3.145 ± 0.28243.654 ± 0.11600.06 (r = 0.01)
      COPE acceptance4.927 ± 0.17384.720 ± 0.092500.32 (r = 0.003)
      COPE denial0.5273 ± 0.17181.285 ± 0.11140.002 (r=0.02)
      COPE instrumental support3.255 ± 0.20963.325 ± 0.097080.75 (r = 0.0003)
      COPE emotional support2.927 ± 0.27813.041 ± 0.11860.68 (r = 0.0005)
      COPE humour2.527 ± 0.28413.199 ± 0.13600.03 (r=0.01)
      COPE substance use0.4182 ± 0.13410.5203 ± 0.078290.56 (r = 0.001)
      COPE behavioural disengagement0.9273 ± 0.20240.9675 ± 0.090880.85 (r = 0.0001)
      COPE distraction3.564 ± 0.27683.415 ± 0.11780.59 (r = 0.0009)
      COPE positive reframing3.891 ± 0.24023.736 ± 0.11390.56 (r = 0.001)
      COPE self-blame1.564 ± 0.21522.276 ± 0.11860.008 (r=0.02)
      COPE religion2.364 ± 0.29972.154 ± 0.14000.52 (r = 0.001)
      COPE venting1.891 ± 0.22272.488 ± 0.11300.02 (r=0.01)
      Fatigue severity scale
      Total score32.20 ± 2.63439.56 ± 1.270(r=0.02)
      Fatigue30 (30.9)114 (46.4)0.04
      MSIS-29
      Physical impact41.73 ± 2.54447.52 ± 1.4130.07 (r = 0.01)
      Psychological impact21.45 ± 1.28024.09 ± 0.65110.08 (r = 0.01)
      Perception of severity of MS (range 1–10)6.491 ± 0.37326.415 ± 0.16140.84 (r = 0.0001)
      Perception of severity of MS
      Variable was dichotomized as follows: low-medium perceived severity and higher perceived severity (cut-off used was 6 or higher).
      (higher or not)
      34 (61.8)168 (68.2)0.44
      Perception of severity of wheelchair (range 1–10)8.200 ± 0.28248.081 ± 0.16700.75 (r = 0.003)
      Perception of severity of wheelchair
      Variable was dichotomized as follows: low-medium perceived severity and higher perceived severity (cut-off used was 6 or higher).
      (higher or not)
      46 (83.6)202 (82.1)0.94
      Definition of LTP with patients: Long-term prognosis (LTP) is an estimate of how you will probably be throughout your lifetime as a result of MS.
      Current LTP information preference was dichotomized as follows: higher (want to know a lot, want to know a little) and lower (unsure, don't want to know).
      low asterisklow asterisk Variable was dichotomized as follows: low-medium perceived severity and higher perceived severity (cut-off used was 6 or higher).
      Table 4Results of logistic regression (multivariate) analysis including potential variables associated with current LTP information preference.
      Current LTP information preference (Dichotomized: Lower/Higher)
      Potential variables associated with higher current LTP information preferenceUnivariate model OR (95% Cl)P-valueMultivariate model
      Variables were included in multivariate regression if univariate analysis p < 0.20.
      OR (95% CI)
      P-value
      Age at survey0.97 (0.95–1.04)0.10
      Gender (F or M)0.64 (0.34–1.18)0.15
      MS duration0.97 (0.93–1.00)0.13
      Education (University or not)1.28 (0.70–2.34)0.42
      Employment (Yes or not)0.99 (0.53–1.81)0.97
      EDSS1.09 (0.94–1.26)0.22
      MS course (RR or progressive form)1.63 (0.26–1.50)0.30
      COPE-281.35 (1.01–1.80)0.03
      COPE active1.10 (0.92–1.30)0.26
      COPE planning1.15 (0.98–1.34)0.07
      COPE acceptance0.89 (0.71–1.11)0.32
      COPE denial1.45 (1.12–1.87)0.004
      COPE instrumental support1.03 (0.85–1.24)0.75
      COPE emotional support1.03 (0.88–1.20)0.68
      COPE humour1.15 (1.00–1.33)0.03
      COPE substance use1.08 (0.82–1.41)0.56
      COPE behavioural disengagement1.02 (0.82–1.25)0.85
      COPE distraction0.95 (0.81–1.12)0.59
      COPE positive reframing0.95 (0.80–1.12)0.55
      COPE self-blame1.26 (1.05–1.50)0.01
      COPE religion0.95 (0.84–1.09)0.52
      COPE venting1.22 (1.02–1.45)0.02
      Fatigue severity scale
      Total score1.01 (1.0–1.03)0.011.02 (1.00–1.05)0.03
      MSIS-29
      Physical impact1.01 (0.99–1.02)0.07
      Psychological impact1.02 (0.99–1.05)0.08
      Perception of severity of MS
      Variable was dichotomized to low-medium perceived severity and higher perceived severity (cut-off used was 6 or higher).
      1.33 (0.72–2.44)0.35
      Perception of severity of wheelchair
      Variable was dichotomized to low-medium perceived severity and higher perceived severity (cut-off used was 6 or higher).
      0.89 (0.40–1.96)0.78
      Definition of LTP with patients: Long-term prognosis (LTP) is an estimate of how you will probably be throughout your lifetime as a result of MS.
      low asterisk Variables were included in multivariate regression if univariate analysis p < 0.20.
      low asterisklow asterisk Variable was dichotomized to low-medium perceived severity and higher perceived severity (cut-off used was 6 or higher).

      6. Discussion

      This study is unique since it assesses the experience of PwMS from Argentina regarding LTP communication and preferences, and we find a strikingly similar situation to that in the UK and Germany, suggesting that the issue has worldwide significance.
      The study of prognostic factors in MS is evolving, based on prospective longitudinal cohort studies (
      • Tintore M.
      • Rovira À.
      • Río J.
      • et al.
      Defining high, medium and low impact prognostic factors for developing multiple sclerosis.
      ;
      • Rotstein D.
      • Montalban X.
      Reaching an evidence-based prognosis for personalized treatment of multiple sclerosis.
      ). However, studies on LTP information preferences and communication in PwMS are limited, since the focus tends to be on conveying the diagnosis to the patients (
      • Elian M.
      • Dean G.
      To tell or not to tell the diagnosis of multiple sclerosis.
      ;
      • Solari A.
      Effective communication at the point of multiple sclerosis diagnosis.
      ;
      • Solari A.
      • Acquarone N.
      • Pucci E.
      • Martinelli V.
      • Marrosu M.G.
      • Trojano M.
      • et al.
      Communicating the diagnosis of multiple sclerosis – a qualitative study.
      ). In addition, LTP is a sensitive theme during a medical appointment, and may be difficult to bring up in discussion between the clinician and patient. Our study showed that nearly one-quarter of responders had never discussed LTP with their neurologist, in comparison with 53.1% in the UK (
      • Dennison L.
      • Brown M.
      • Kirby S.
      • Galea I
      Do people with multiple sclerosis want to know their prognosis? A UK nationwide study..
      ). Despite this, 50% of Argentinian PwMS lacked clarity about their LTP, similar to numbers (54.2%) in the UK (
      • Dennison L.
      • Brown M.
      • Kirby S.
      • Galea I
      Do people with multiple sclerosis want to know their prognosis? A UK nationwide study..
      ). This might imply that even though a greater proportion of PwMS receive some amount of information in Argentina, this does not suffice. LTP is of relevance for Argentinian PwMS as almost 50% of them thought about it at least once a week, even when one-third of patients do not discuss the subject with anybody. The fact that relevance and active discussion of LTP seem to be independent from each other, suggests that complex psychological factors are at play. This may explain why several coping strategies were associated with LTP information preference in our data (Tables 3 and 4). We presumed that different coping strategies might be related to different levels of LTP information preferences and found that those participants with lower levels of denial, humour, self-blame and venting coping profiles showed statistically significant lower levels of LTP information preferences. On the other hand, PwMS with higher scores on fatigue showed higher level of LTP information preference. These findings imply that different personality traits and severity of symptoms play an important role in molding LTP information preferences.
      We observed that 61.7% of PwMS (relapsing course) from Argentina wanted to know more about conversion to SPMS. Similar results were observed in a cohort from Germany (
      • Buecken R.
      • Galushko M.
      • Golla H.
      • Strupp J.
      • Hahn M.
      • Ernstmann N.
      • et al.
      Patients feeling severely affected by multiple sclerosis: how do patients want to communicate about end-of-life issues?.
      ), where 76% of PwMS considered it important to discuss progression and 66% wanted end-of-life problems addressed in the medical appointment. However, satisfaction with the degree of communication on these themes was poor in those population (
      • Buecken R.
      • Galushko M.
      • Golla H.
      • Strupp J.
      • Hahn M.
      • Ernstmann N.
      • et al.
      Patients feeling severely affected by multiple sclerosis: how do patients want to communicate about end-of-life issues?.
      ). Thus, it appears that clinicians need to be further trained to communicate about these difficult long-term issues.
      When asked about the proper timing for LTP information, almost three-quarters of PwMS considered the time of the diagnosis, the current moment and the future as possible options in similar proportions: 72.4%, 71.7% and 73.4% respectively. That LTP was not discussed in many cases could be due to several reasons, as follows: simply not being aware of the desire of PwMS to discuss this issue, the lack of precise prediction models for long-term prognostication in MS, a lack of time during medical appointments, the assumption that PwMS already had such a discussion with another clinician, or a lack of clinician training (
      • Dennison L.
      • McCloy Smith E.
      • Bradbury K.
      • Galea I
      How do people with multiple sclerosis experience prognostic uncertainty and prognosis communication? A qualitative study.
      ;
      • Heesen C.
      • Gaissmaier W.
      • Nguyen F.
      • Stellmann J.P.
      • Kasper J.
      • Kopke S.
      • et al.
      Prognostic risk estimates of patients with multiple sclerosis and their physicians: comparison to an online analytical risk counseling tool.
      ;
      • Buecken R.
      • Galushko M.
      • Golla H.
      • Strupp J.
      • Hahn M.
      • Ernstmann N.
      • et al.
      Patients feeling severely affected by multiple sclerosis: how do patients want to communicate about end-of-life issues?.
      ;
      • Janssen D.J.
      • Spruit M.A.
      • Schols J.M.
      • Wouters E.F
      A call for high-quality advance care planning in outpatients with severe COPD or chronic heart failure.
      ;

      Reinke L.F., Slatore C.G., Udris E.M., Moss B.R., Johnson E.A., Au D.H. The association of depression and preferences for life-sustaining treatments in veterans with chronic obstructive pulmonary disease. J. Pain Symptom Manage.2011;41: 402–11.

      ).
      The idea of a tool that might provide individualized LTP estimates was demonstrated to be of interest to the vast majority of study participants (94.3%). Only prototypes of such tools exist and the PwMS in our study were not shown any such tool, so this was a hypothetical question. However, it is important to know, to help inform the need for future research into such tools. A LTP tool was perceived to be useful at various time-points and helpful for decision-making, similar to other studies (
      • Dennison L.
      • Brown M.
      • Kirby S.
      • Galea I
      Do people with multiple sclerosis want to know their prognosis? A UK nationwide study..
      ,
      • Dennison L.
      • McCloy Smith E.
      • Bradbury K.
      • Galea I
      How do people with multiple sclerosis experience prognostic uncertainty and prognosis communication? A qualitative study.
      ;
      • Heesen C.
      • Gaissmaier W.
      • Nguyen F.
      • Stellmann J.P.
      • Kasper J.
      • Kopke S.
      • et al.
      Prognostic risk estimates of patients with multiple sclerosis and their physicians: comparison to an online analytical risk counseling tool.
      ). Similar proportions of PwMS in this cohort as in the UK, about one-third, considered that this tool could be used in different settings: alone, with friends or family member, or with their neurologist. Interestingly, 41.5% thought that the best setting to use this tool was with the neurologist and friends or family members. This suggests concerns about accessing and interpreting important and emotive information without the input of a healthcare professional and the support of their loved ones. This aligns with qualitative findings where PwMS emphasized the simultaneous need for expertise and emotional support when receiving prognosis predictions (
      • Dennison L.
      • McCloy Smith E.
      • Bradbury K.
      • Galea I
      How do people with multiple sclerosis experience prognostic uncertainty and prognosis communication? A qualitative study.
      ).
      Several limitations of this study should be mentioned. Firstly, this was a cross-sectional study and changes over time to explore causality could not be evaluated. Secondly, we used a self-reported EDSS, although there is growing evidence that patient-reported outcomes are valid (
      • Collins C.D.
      • Ivry B.
      • Bowen J.D.
      • et al.
      A comparative analysis of Patient-Reported Expanded Disability Status Scale tools.
      ;
      • Kobelt G.
      • Eriksson J.
      • Phillips G.
      • Berg J
      The burden of multiple sclerosis 2015: methods of data collection, assessment and analysis of costs, quality of life and symptoms.
      ). Thirdly, this was a study with relatively small population size compared with other cohorts from Europe. Fourthly, we had a relatively lower response rate, since only around 15% (n = 301) of PwMS invited to participate answered the survey. One possible explanation is related to survey deployment method: it cannot be known with certainty how many patients received the invitation as it was an online survey sent by email through a patient association (ALCEM members). Another possible reason is that PwMS might be reluctant to talk about LTP, either because of lack of interest or due to anxiety or stress related to this theme. Hence, PwMS who answered this questionnaire might have been biased towards wanting to know more about their LTP. Fourthly, we did not evaluate cognitive impairment and other mood disorders such as anxiety and depression. Despite these limitations, we consider that this is a valuable first study into LTP communication and information preferences amongst Latin American PwMS.

      7. Conclusions

      There is an apparent unmet need amongst Argentinian PwMS to discuss LTP, which is strikingly similar to the UK and Germany. Many think of their LTP at least on a weekly basis, and those who have received information find it inconsistent among health care professionals or other sources. A great majority of responders were interested in being informed about the availability of a LTP tool to help the discussion. Such a tool may convey to PwMS the reasons for information consistency by appreciation of confidence intervals. Also, its use could function as a trigger for LTP discussion.
      As in other countries, a small proportion of patients do not want to know about their LTP. It is probably unlikely to be able to predict which patient does or does not want to receive this information. One possible strategy could be asking individual PwMS at the time of diagnosis and on a regular basis about their LTP communication preferences, in case they change their opinion, and proceed accordingly.
      Further research is needed into the preferences of PwMS on LTP communication and the best strategies to deploy in clinical practice, to ensure the patient´s emotional wellbeing and optimize their coping abilities.

      Funding/support

      This research received no specific grant from any funding agency in public, commercial, or non-profit sectors.

      Declaration of Competing Interest

      None of the authors has any financial conflict of interest relating to this manuscript.

      Acknowledgments

      The authors thank Laura Dennison, Martina Brown and Sarah Kirby from the University of Southampton (United Kingdom), since this project was based on their published experience. In addition, we thank the participants for responding to the survey and Asociación de Lucha Contra la Esclerosis Múltiple for promoting the survey.

      Appendix. Supplementary materials

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