3. STUDY DESIGN
SSPOTS will be an exploratory sequential three phase study design. The overarching aim will be to feasibility test a co-produced supportive self-management intervention based on the experience of POTS. Phase one will be exploratory interviews; phase two exploratory workshops to develop core components of a POTS supportive self-management intervention; and phase three intervention feasibility testing. Figure one is the overall study design.
3.1 STUDY SETTING
All phases of the SSPOTS study will take place in a virtual setting using a secure, end-to-end encrypted, online platform approved by the study sponsor. Telephone interviewing may be used if required by interviewees. These methods allow for a range of participants, from a wide geographical area to participate and will include those with the most disabling POTS, those unable to travel or having limited access for face-to-face participating. People who express an interest will be contacted to discuss the study, be sent a participant information sheet and consent will be gained prior to participation in each of the study phases. Each phase will be digitally audio recorded, transcribed and pseudonymised.
Collection, storage and processing of data will comply with good data protection principes (39) and sponsorship regulations. For online group activities / communications in phase two and phase three the “BCC” (“blind carbon copy”) or the hide list option for group meetings will be used. Using these options maintains participants’ data protection security as email details will not be accessible to participants in meetings or in correspondences.
The fellowship grant provides funding, in keeping with national guidance (40), as follows; in phase one vouchers of £20.00 will be given as a token of appreciation for participants’ contributions and time; in the phase two workshops payment for workshops’ participation and document reviewing; and in phase three payment for the facilitators training and their time for delivering the intervention.
3.2 SAMPLING & RECRUITMENT
In phases one and three there will be purposive sampling of potential participants with PoTS. Eligibility screened will take place at three NHS PoTS participant identification sites by PoTS clinicians or a member of their team. Table one below outlines the study inclusion and exclusion criteria for phases one and three. General practitioners will be notified of NHS participant’s recruitment. For the health care practitioners in phase one, snowball sampling will drive recruitment, and be initially via the PoTS UK charity network and the Arrhythmia Nurses Network. Figure two is a flow chart of NHS study participants. For the Phase two workshops, purposive sampling of a diverse range of primarily people with POTS, health care practitioner/s and the charity will be recruited via the POTS UK charity.
Inclusion Criteria | Exclusion Criteria |
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1) | Able to provide informed written consent. | 1) | Under the age of 18 |
2) | English speaker | 2) | Underlying serious psychological disorder with ongoing symptoms that would significantly interfere with study participation (screened by local gatekeeper clinician) |
3) | 18 years or over, | 3) | Those unable to give informed consent. |
4) | For Patients: A recorded diagnosis of POTS made from a Tilt table test, or an active stand test, screened by the NHS gatekeeper clinician | 4) | Those who are involved in current research or have recently (within the past year) been involved in any research prior to recruitment to reduce participant burden |
5) | OR Health Care Practitioner inclusion: more than six months experience with POTS. | | |
TABLE ONE: SSPOTS PHASE ONE AND PHASE 3 ELGIBILGITY CRITERIA |
3.3 INTERVENTION DEVELOPMENT METHODS
3.31 INTERVIEWS PHASE ONE
Qualitative methods such as interviews allow for an exploration of the diverse experience of POTS (41). The rationale for interviews in phase one is to gather in depth personal accounts and exploring issues in depth (42). Phase one’s aim will be to gain an insight into the diverse experience POTS. The two objectives will be to interview 1) up to 30 people with POTS and 2) up to 30 health care professionals.
3.311 OUTCOMES
There will be four outcomes for phase one:
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An understanding of the experiences of POTS.
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An understanding of the experiences of health care professionals working with people with POTS.
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A developed conceptual framework based on the experiences of POTS.
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Identification of components of POTS supportive self-management.
3.312 QUALITATIVE DATA COLLECTION
An interview topic guide will be informed by the PPI advisory committee and previous focus group work. Interviews will explore the experience of POTS, self-management and support. This will likely to be up to 30 interviews in each group. Determining the final interview data set will be driven by a combination of theoretical saturation, whereby no new themes emerge from the data, and theoretical sampling, whereby a diverse range of perspectives are sought (43). The data collection process will ensure a diverse range of perspectives are included.
3.313 DATA ANALYSIS
Thematic analysis (42) will be used to analyse the interviews. This method is helpful in answering questions of the form and nature of what exists (the experience of POTS); appraising what exists (how people with POTS have self-managed) and identifying new theories (the conceptual model of PoTS experiences and requirements of a self-management support intervention in POTS) (44). A key analytical technique will be constant comparative analysis whereby data will be analysed for constant differences and similarities (43). NVivo 12 software will manage the data.
The findings where applicable will then be conceptualised using a Critical Realist ontological perspective. The data on potential components of supportive self-management will be extracted and taxonomized according to the PRISMS taxonomy (21). The components and their activities will then be presented at the phase two workshops.
3.32 METHODS: PHASE TWO CO-CREATION WORKSHOPS
The workshops aim will be to co-define and co-refine a SSPOTS intervention with people with POTS and key stakeholders. The outcome will be a co-created POTS self-management support intervention that can be feasibility tested in phase three. The workshops will begin with introductions and an agreed framework for incorporating a genuine shared space for equal voices. The workshops will address key problems and explored solution formulation in a shared space with equal voices.
3.321 WORKSHOP ONE: CO-DEFINING THE SSPOTS INTERVENTION
A member of the PPI advisory board team and the researcher will present the overall workshops purpose with the aim being to decide and develop SSPOTS intervention priorities. The phase one taxonomized components of supportive self-management will be visually displayed. The nominal group four step technique (45, 46) will structure the running of workshop one and inform reaching consensus.
1) Step one the participants will silently generate their priorities to be included in a SSPOTS supportive self-management intervention.
2) Step two participants will share their first priority in a “round robin” technique that will continue until all ideas had been presented (47).
3) Step three the group with explore, with equal voices and avoiding judgement, underlying reasons for people’s specific priorities.
4) Step four will be reaching final consensus with anonymous voting on ranking each person’s top priorities (47).
The exact number of components for ranking will be determined by step two and three of the nominal group technique. The results will then be tallied to reach the workshops consensus of the SSPOTS intervention priorities (45). The final priorities will be summarised in an initial intervention framework (section 3.33).
3.322 CO-REFINING THE SSPOTS INTERVENTION: WORKSHOP TWO
Following workshop one an initial intervention will be developed. This will be circulated to workshop one attendees through email correspondence. This will allow participants time to reflect and formulate their ideas on the intervention. The second workshop will be undertaken approximately eight weeks later for feedback on areas for intervention refinement.
3.33 THE FINAL SSPOTS INTERVENTION
An intervention manual will be formulated as a result of the final workshops analysis. The final SSPOTS intervention design, delivery method and prospective outcome measures will be explored with the SSPOTS PPI advisory group with the research team making the final decision.
3.331 INTERVENTION OUTCOME MEASURES
The outcome measures will be determined by exploratory work with the SSPOTS PPI advisory group and a discussion with a statistician. This will consider a wide range of outcome measures, including a POTS specific outcome measure such as Malmo POTS Symptom Score (48), a self-efficacy outcome measure, and standard quality of life measures such as the SF-36 (49) and the EQ-5D-5L (50).
3.4 METHODS: PHASE THREE SSPOTS INTERVENTION FEASIBILITY TESTING
The feasibility testing will be preliminary work to assess important parameters needed to be considered for conducting a future trial, with a focus on process (23, 51). Testing the feasibility of an intervention is an essential step in complex intervention development (52) and recommended by the MRC (22). The is no hypothesis as the feasibility study is a hypothesis generating step prior to planning a further randomised control trial. The aim of the SSPOTS intervention feasibility study will be to determine if SSPOTS is feasible as designed, feasible with adjustment, or not feasible.
3.41 OUTCOME MEASURES
The primary outcome measure of interest for SSPOTS will be to assess number of eligible potential participants who convert into participants. The secondary outcome measures of interest will be
1) adherence, attendance, at programme sessions (n = 4)
2) intervention fidelity of the facilitator to the SSPOTS manual
3) outcome measures calculations of mean, medium and standard deviation to inform sample size calculation in a potential future trial
4) a focused process evaluation of intervention acceptability.
Outcome measures will be administered twice: Baseline measures will be taken within 1 month prior to the intervention delivery and then repeated within six months of initial baseline assessment.
3.42 FEASIBILITY SAMPLING & RECRUITMENT
Feasibility studies randomise to determine the acceptability of randomisation for potentially future trials (53). Acceptability of randomisation has been determined by firstly pre-grant focus groups and secondly a POTS exercise feasibility study (54). Preliminary data suggests randomisation is acceptable for people with POTS. In an audit of feasibility sample sizes, 30 was the most cited sample size (55). A minimum sample of 12 is statistically advised for the purpose of power calculations to inform a future potential randomised control trial (56). For the SSPOTS study, a sample will be needed with a minimum number of 12 and a maximum number of 30 participants.
3.43 DATA COLLECTION
Delivery of the intervention will be delivered by a SSPOTS facilitator in accordance with the SSPOTS manual developed from the exploratory phases one and two. The SSPOTS research fellow will be present to evaluate delivery of the intervention and support the facilitator should this be necessary.
The feasibility study will collect quantitative data on rates of eligibility, recruitment, and completion; validated quality of life measures; simple rating scales of intervention adherence by the person delivering the intervention; and a limited health care utilisation record will be collected. An embedded qualitative study of post intervention interviews will provide for a focused process evaluation.
3.431 PRIMARY OUTCOME DATA COLLECTION
The primary outcome measure will be the number of people screened eligible, recruited and number of intervention sessions completed. Adherence will be measured on a scale of 1–4 as follows: no sessions attended, non-adherent (n = 1); mild adherence, 1 session attended (n = 2); moderate adherence, 2 or 3 sessions attended (n = 3); and maximum adherence, all 4 sessions attended (n = 4).
3.432 SECONDARY OUTCOME DATA COLLECTION
The secondary outcome measures data will be:
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Data on percentage of those providing follow up outcome measure completion.
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Intervention fidelity is defined as “the methodological strategies to monitor and enhance the reliability (consistency) and validity (appropriateness) of programmes”(57).
Fidelity assessment will be informed by facilitator manual adherence, on a scale of 1–3 as follows: adhered to manual (n = 3), partially (n = 2) and no (n = 1); the facilitators competency in delivery; the group’s impression of the intervention component; and the delivery environment. Fidelity will be determined by observation of delivery session and analyse of audio recordings (57). The PPI advisory group will advise on interpretation of results.
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Study acceptability will be determined with a focused process evaluation. A selection of participants, lay trainees and recruiting clinicians (up to potentially ~ 30 in total) (58) will be interviewed for the following: SSPOTS material including manual and videos, the outcome measurements, burden of participant assessment, intervention accessibility and usability, and the SSPOTS intervention facilitators or barriers (23).
3.44 ANALYSIS
Quantitative and qualitative data integration will utilise a mixed methods matrix on individual cases (59) thereby bringing all results together. Meta-Themes will be developed to cut across different findings. Convergence coding matrix will display findings and meta-themes (59). The PPI advisory group will enhance and facilitate understanding of data convergence, divergence or silence.
The feasibility study results will be reported using the CONSORT guidelines, incorporating PPI items which should be available and published during the feasibility study (23) with a view to the practicality of a definitive SSPOTS intervention randomized controlled trial (23).
Results of the feasibility study will be discussed with the SSPOTS PPI advisory board and the research team to determine future applications for a SSPOTS randomised control trial.
4.0 STUDY RISKS
One potential risk will be the possibility of causing distress when discussing experiences in either the interviews or during the intervention delivery sessions. This will be addressed by discussions with any distressed person which may require actions such as contacting the person’s general practitioner. Risks associated with data protection will be mitigated as outlined earlier in sections 3.1 and 3.2.
5.0 STUDY REPORTING & FINAL DATASET
Result reporting will follow the Standards for reporting of qualitative research, GRIPP standard for reporting of PPI in research and CONSORT Standard for reporting randomised feasibility studies (60). The CORES co-creating reporting tool, registered on EQUATOE, is currently being developed and will be integrated in SSPOST reporting (61) Results will be shared via conference presentations and journal publications. Access to the data will be determined on a case-by-case basis, by applying to the chief investigator.
3.432 SECONDARY OUTCOME DATA COLLECTION
The secondary outcome measures data will be:
-
Data on percentage of those providing follow up outcome measure completion.
-
Intervention fidelity is defined as “the methodological strategies to monitor and enhance the reliability (consistency) and validity (appropriateness) of programmes”(57).
Fidelity assessment will be informed by facilitator manual adherence, on a scale of 1–3 as follows: adhered to manual (n = 3), partially (n = 2) and no (n = 1); the facilitators competency in delivery; the group’s impression of the intervention component; and the delivery environment. Fidelity will be determined by observation of delivery session and analyse of audio recordings (57). The PPI advisory group will advise on interpretation of results.
-
Study acceptability will be determined with a focused process evaluation. A selection of participants, lay trainees and recruiting clinicians (up to potentially ~ 30 in total) (58) will be interviewed for the following: SSPOTS material including manual and videos, the outcome measurements, burden of participant assessment, intervention accessibility and usability, and the SSPOTS intervention facilitators or barriers (23).
3.44 ANALYSIS
Quantitative and qualitative data integration will utilise a mixed methods matrix on individual cases (59) thereby bringing all results together. Meta-Themes will be developed to cut across different findings. Convergence coding matrix will display findings and meta-themes (59). The PPI advisory group will enhance and facilitate understanding of data convergence, divergence or silence.
The feasibility study results will be reported using the CONSORT guidelines, incorporating PPI items which should be available and published during the feasibility study (23) with a view to the practicality of a definitive SSPOTS intervention randomized controlled trial (23).
Results of the feasibility study will be discussed with the SSPOTS PPI advisory board and the research team to determine future applications for a SSPOTS randomised control trial.