The project had an advisory group comprising clinicians specialising in SLE from the fields of rheumatology, dermatology, nephrology, and internal medicine, as well as managers and pharmacists. A representative of SLE patients from the FELUPUS association also participated in the group, as did an expert rheumatologist who served as the project coordinator.

A literature search was conducted in this first phase to identify the existing literature on the different scales and measurement tools used in SLE, as well as the correlation between them. The available evidence was analysed in terms of the validity of each item for use separately or in combination. The search strategy is detailed in Table 1.

The literature search returned 233 records. After removing duplicates, 222 records were evaluated.

Based on the literature found, articles that met the following criteria were selected:

• •

  The objective of the study should be to validate or compare the measurement of disease activity and/or damage using the currently validated different indices and scales.

• •

  The article should provide useful information on some of the scales, comparing or analysing them.

• •

  The conclusions reached by the study provide greater insight into the subject and can serve as a reference for the objectives of the VitaLES workshop for clinicians.

Articles that use disease activity scales in the evaluation of a drug's efficacy, but whose objective is not the measurement or analysis of the validity, comparability and/or usefulness of the scale, were discarded. Studies validating any of the scales in a new language or adapting a scale to specific populations were also discarded.

Four articles that met the search criteria were selected as a result of this synthesis:

• •

  Measuring disease activity and damage with validated metrics: A systematic review on mortality and damage in systemic lupus erythematosus. O Keeling et al. 2018.

• •

  Clinical composite measures of disease activity and damage used to evaluate patients with systemic lupus erythematosus: A systematic literature review.13

• •

  Indices to assess patients with systemic lupus erythematosus in clinical trials, long-term observational studies, and clinical care. Castrejon et al., 2014

• •

  From BILAG to BILAG-based combined lupus assessment-30 years on. Murphy et al., 2016.

The most important conclusions from these articles were shared with group participants prior to the workshop.

In this phase, the advisory group agreed on the profiles of the workshop participants and the practical objectives to be achieved in each workshop, as well as the criteria for participation and agreement in the Delphi phase, and the general framework for action.

Specifically, it was decided to hold four multidisciplinary working meetings including SLE patients, in which the conclusions of each workshop would form the basis for the next one. This would ensure that everyone's contribution added to the final result. The objective of the workshops was for participants to reach a consensus on issues such as what constitutes comprehensive control, which health outcomes should be measured and which validated tools, scales, or indices are most appropriate for the practical management, control, and follow-up of patients with SLE.

Regarding the criteria for the Delphi phase, it was agreed that Trevelyan's recommendations would be followed.8

Four workshops were held. Prior to each workshop, participants were sent a report on the evidence found and the project objectives, to serve as a basis and aid in the development of the workshop. This preliminary report also included the conclusions reached in previous workshops, enabling the final report to draw on contributions from the various working groups.

The first workshop was attended by a group of SLE patients who, using the focus group methodology9 created a 'patient journey map' or SLE patient pathway. This identified unmet needs and set out which health outcomes were important for patients' quality of life (QoL) and should therefore be collected as patient-reported outcomes (PROMs). The focus group,9 methodology was chosen because of its advantages in terms of the quality of information obtained.

During an open and participatory debate, a series of questions were raised to address various issues related to the objectives of the workshop:

• 1

  Definition of the pathway. Key points

• 2

  Identification of unmet needs

• 3

  Health outcomes that matter to patients with SLE

The second workshop comprised a group of clinical experts (Table 2). This working session included a presentation of the analysis of evidence found in the scientific literature, as well as the conclusions of the previous workshop with patient representatives. All of this information formed the basis for reaching a clinical consensus on which aspects of the disease should be measured, and which tools are considered the most useful, reliable and valid for monitoring patients with SLE.

The third workshop was attended by a group of managers, pharmacists, and evaluation and quality experts (Table 2). This working session included a presentation of the conclusions from the patient and clinician workshops, and aimed to reach a consensus from the perspective of evaluators and decision-makers on which aspects of the disease should be measured, and which tools are considered the most useful, reliable, and valid for monitoring patients with SLE.

Finally, the fourth workshop took place. This workshop had a larger group of participants than the previous ones. The reason for holding this workshop with an expanded multidisciplinary group was the need to have a joint vision of all the profiles involved in the pathology. For this reason, the group was expanded, in terms of both number and profiles (Table 2). Based on the conclusions of the workshops with patient representatives, clinicians, and decision-makers presented above, the participants in this multidisciplinary workshop reached a final consensus on which aspects of SLE should be measured, which tools are most appropriate, and the frequency of measurement for better control, management and monitoring of the disease.

The conclusions reached in the workshops, once analysed and reviewed by the advisory group, were brought to consensus by an expanded group of professionals and patient representatives according to the Delphi methodology, following Trevelyan's recommendations.8 The Delphi consensus process was carried out via a survey, with different numbers of questions for professionals and patients. The latter were not sent questions on clinical decisions specifically (Appendix I and II). The survey was designed with a defined number of questions on items assessed and agreed upon in the workshops, asking respondents to indicate whether they agreed or disagreed with each one. Responses were evaluated using a Likert scale with scores from 1 to 5, where 1 indicates total disagreement and 5 indicates total agreement.

The Advisory Group set the minimum required level of participation at ≥60%, stipulating that it should not be less than 50% for each profile. To this end, the participation percentage for each profile and each round was monitored from the outset. The degree of consensus required for a recommendation to be considered relevant was also set at greater than 70%, in accordance with Humphrey-Murto et al.10 In an attempt to improve consensus, the required percentage was set at 80% in the first round and 75% in the second. For this purpose, responses corresponding to 'agree' or 'strongly agree' were considered positive, while the rest were not.

The selected panellists were invited to participate via email. After they accepted, an email was sent with a link to a Google platform website where they could access the questionnaires. Participants generated their responses individually and anonymously.

A sample of 200 people (professionals and patients) was selected. Professionals were selected on the basis that they should have profiles directly related to the care of SLE patients in any of their fields: doctors, nurses, and pharmacists. Managers, quality experts, and patients from the FELUPUS association were also included. The proportion of participation for each profile was intended to match the same proportion as that of the 4th workshop. Multidisciplinary. It was considered that 200 invitations was an ideal number to achieve a minimum participation of 120/130 people.

Once the Delphi methodology had been agreed with the advisory group, a questionnaire with 14 questions was drawn up (Appendix I), which included the following content:

• 1

  Scale: content and measurement.

• 2

  Definition of disease control: what it includes.

• 3

  Usefulness and feasibility of the scale in clinical practice.

• 4

  Domains of health outcome measurement: what it should include.

• 5

  Quality of life: domains, questionnaires, and frequency of measurement.

• 6

  Cumulative harm: when to measure.

The questionnaire was developed based on the working groups' conclusions. If the percentages previously decided by the Advisory Group were not reached in the first round, a second round would be necessary.

Once the responses from the 1st round had been collected and analysed by the advisory group, it was decided that a 2nd round should be carried out to explore those issues that had not achieved the required 80% threshold in greater depth.

The following criteria were established for this round: the number of participants should reach a minimum of 75, and given that the issues pending consensus (Appendix II) affected decisions within the clinical sphere and the patient’s QoL, the advisory group defined that participants should belong to medical specialties directly related to the treatment of SLE and to patients (FELUPUS). The agreed distribution percentages were 20% for patients and 80% for clinicians, as shown in Table 3.

Patients described their 'patient journey map' and, as a result, expressed unmet needs from their perspective as patients. These included the need for coordinated multidisciplinary care with consistent criteria for all professionals treating them, adequate information and training for patients and families, and a voice within the healthcare system. Regarding the PROMs that should be collected and considered when making shared decisions, they concluded that the following should be measured: emotional state, pain, stress, functional capacity, socio-occupational issues, asthenia/fatigue, and sexuality. They considered the Fatigue Severity Scale (FSS)11 and the SF12 and SF36 QoL questionnaires suitable for reporting these items.12

The main conclusion of the SLE clinical expert workshop was that the standardised tools currently available for assessing disease activity are not suitable for use in routine clinical practice and their usefulness is limited to compliance with registries and clinical trials. The experts then agreed on the characteristics that an activity scale should have in order to be useful in therapeutic decision-making and to help achieve the therapeutic goal of remission, and therefore be useful in clinical practice. They concluded that this tool should be straightforward and easy to use, should not take long to complete, and should be easy to interpret. Based on these premises and using the items from the SLEDAI/LAI-P scales13 as a basis, they developed a proposed scale, which is detailed in Table 4. A health-related quality of life (HRQoL) assessment, measured using one of the currently validated scales, should be added to this SLE disease activity scale.

After analysing the results obtained in previous workshops, the participants made the following contributions: in addition to QoL and disease activity, adherence, and adverse effects of medication should also be measured. They also agreed that values indicating remission and exacerbation must be defined in order to make therapeutic decisions.

The working group decided that measuring SLE activity is as important as measuring QoL or cumulative damage. Therefore, an agreement was reached on the domains to be measured in SLE. This set of health outcomes includes the following fields, as described in Fig. 2.

Figure 2.

Health outcomes. Domains to be measured proposed in the multidisciplinary workshop.

It was unanimously agreed that ‘treatment adherence’ should be measured using the Morinsky Green scale.14 However, there was no unanimity on the QoL measurement scale: 65% of participants voted for SF-12 and 35% for EQ-5D.15 Nor was there consensus on establishing the frequency of QoL measurement and, specifically, whether it should be measured when there was a flare-up or one month after a flare-up.

The final results obtained at this multidisciplinary meeting, which also included the partial results of each workshop held, were brought to Delphi consensus.

In the first round, the survey was answered by 19 patients and 154 professionals, thus meeting the minimum participation agreement of 100.

The participation result by profile is shown in Fig. 3

Figure 3.

Number of participants by profile.

The result of the questionnaire consensus (Appendix I) was as follows:

An 80% consensus was not reached on the following responses:

• •

  In the selection of the QoL scale.

• •

  In the following domains to be measured: sexuality, body image, mood and relationships within QoL.

• •

  In the questions referring to the assessment of disease activity and control scale proposed by clinicians, in terms of whether it is suitable for use in patient follow-up and whether it facilitates therapeutic decision-making.

Table 5 shows the percentage of consensus reached on each question in detail:

These questions therefore needed to be taken to a second round (Appendix II), the results of which were as follows:

• •

  The survey was completed by 21 patients and 77 professionals, and the percentage of participation by profile was as shown in Fig. 4.

  
  

  Figure 4.

  Percentage of participants per profile.

Regarding the question about the preferred QoL questionnaire for measurement, none of those presented obtained the 75% required to reach consensus, so it was left to the discretion of each centre, recommending a generalist questionnaire from among those most commonly used in Spain (EuroQoL-5D, SF-12, LES-ESP, and EVA) containing the agreed QoL domains. Taking into account the results of clinicians and patients, the outcome is detailed in Fig. 5.

Figure 5.

Choice of QoL questionnaire.

There were significant differences in the selection of questionnaires to measure QoL between clinicians and patients. Clinicians opted for quicker and easier questionnaires to complete, while patients opted for more comprehensive questionnaires containing a greater number of items, as can be seen in Figs. 6 and 7.

Figure 6.

Choice of QoL questionnaire by patient.

Figure 7.

Choice of QoL questionnaire by clinician.

Consensus was reached on the following issues raised in this round:

• •

  To the question whether the proposed scale is suitable for monitoring patients with SLE, 100% of respondents answered yes, it is suitable.

• •

  To the question whether the proposed scale should preferably be included in the EHR in order to be practical and effective, 97% of participants responded that it should preferably be incorporated into the EHR, and within this percentage, 20% said that they considered it adequate even without including it in the EHR.

• •

  To the question whether they thought the scale was big enough for making therapeutic decisions, 98.5% answered yes

For each domain, a sequence was proposed that describes when the data will be collected or reported during the course of the disease, and who will be responsible for doing so (patient and/or professional). This sequence is described in Fig. 8.

Figure 8.

Summary table of timing and who should collect the data.