Author s response to reviews

Transcription

1 Author s response to reviews Title: Evaluation of the Effectiveness of Music Therapy in Improving the Quality of Life of Palliative Care Patients: a Randomised Controlled Pilot and Feasibility Study Authors: Tracey McConnell (t.mcconnell@qub.ac.uk) Lisa Graham-Wisener (lisa.graham@mariecurie.org.uk) Joan Regan (joan.regan@mariecurie.org.uk) Miriam Mckeown (miriam.mckeown@mariecurie.org.uk) Jenny Kirkwood (jenny@everydayharmony.org) Naomi Hughes (naomi@everydayharmony.org) Mike Clarke (m.clarke@qub.ac.uk) Janet Leitch (janet.leitch@mariecurie.org.uk) Kerry McGrillen (kerry.mcgrillen@mariecurie.org.uk) Sam Porter (s.porter@qub.ac.uk) Version: 1 Date: 27 Sep 2016 Author s response to reviews: Response to Reviewers Editor s comments: Further to your answer about my funding and ethics approval queries, please update the funding section with your declaration that the funder had no part in the design of the study, and please update the ethics approval section with your ethics approval reference number and statement that the study will be performed in accordance with the declaration of Helsinki. We have now updated the funding section with our declaration that the funder had no part in the design of the study, and the ethics approval section with our ethics approval reference number and statement that the study will be performed in accordance with the declaration of Helsinki.

2 Editor s comment - Major concerns about your primary and secondary outcomes, which appear to be for the future definitive trial rather than the pilot. You state under your aims and objectives section that the study aims to establish the feasibility and the trial will test procedures; outcomes and validated tools; estimate recruitment and attrition rates; and calculate the sample size required for a phase III randomised trial, which is good, but then you state under primary outcome that the main aim of the study is to evaluate the effect. It needs to be clear that the main aims/objective of the pilot study are to look at feasibility, but that you will also look at potential effectiveness as a secondary outcome but with a caveat that you are not powered for this. We have now made it clear that the main aims of the pilot study are to look at feasibility, but that we will also look at potential effectiveness as a secondary outcome, with the caveat that we are not powered for this. Editor s comment - Your data analysis/assessment section should outline how you will assess each of your feasibility objectives first, and then how you will assess your secondary objective of potential effectiveness. For the latter, as your pilot study does not include a formal power calculation to test a hypothesis, confidence intervals must be clearly stated with any estimates. Any forms of hypothesis testing should be treated as secondary to the main feasibility results and should come with a clear reason and a caveat of caution that no power calculations have been performed. The data analysis/assessment section now outlines how we will assess each of our feasibility objectives first, and how we will assess our secondary objective of potential effectiveness. We have stated that confidence intervals will be clearly stated with any estimates, and we have clearly stipulated that findings should be interpreted with caution as no power calculations have been performed. Line As this pilot study does not include a formal power calculation, confidence intervals will be clearly stated with any estimates, and findings should be interpreted with caution. Editor s comment - Structure needs to be rewritten to list the objectives of the pilot trial, and then the methods used to address each pilot trial objective, whether descriptive, qualitative or quantitative.

3 We have restructured the manuscript in light of all review comments to make it easier for the reader. Editor s comment - Line 147: Should read It is not possible to *blind patients - Line 183: The sentence In line with the intention-to-treat principle, patients who attend fewer sessions will not be excluded from data analysis seems a bit out of place in the Intervention section and should instead be under data assessment section. Line 147 and 183 have been revised as advised. Editor s comment - Line 234: Sample size could do with some more justification. We have now included more justification for the sample size. Line The calculation of the sample size was informed both by good practice for feasibility trials, and by our expectations on our ability to recruit eligible patients with the time and funding available for this feasibility trial. As noted, we are hoping to exceed the recommended number for a feasibility trial in order to maximise the information we can gather with the resources available. Editor s comment - Do you have any progression criteria to judge whether, or how, to proceed with the main trial? We have now included progression criteria to help judge whether, or how, to proceed with the main trial. Line Progression criteria will be based on those that we have used from other feasibility studies. These consider the proportion of the target sample size that is achieved during the feasibility study: % recruitment: progress to application for main trial with little or no change to relevant aspects of the protocol.

4 50-74% recruitment: progress to application for main trial following a review of details on patients who were not deemed eligible or decline to join the trial, an assessment of any barriers to recruitment and possible changes to relevant aspects of the protocol % recruitment: progress to application for main trial if a "rescue plan" can be developed by the trial team and other potential co-applicants. This plan might include the need to identify additional sites or changes to the relevant aspects of the protocol. Less than 25% recruitment: the trial will probably not progress. Reviewer #1: I think it is well written overall. However, I would consider re-organizing the methods section as follows: 1. Trial aims, objective, and study setting (i.e., move lines in page 7 to here.) 2. Trial design 3. Inclusion/exclusion criteria and sample size. Also include allocation and randomization here. 4. Measures - primary and secondary outcomes 5. Quantitative data collection 6. Qualitative data collection 7. Data management & data monitoring and interim analyses 8. Others - ethics, potential harms, etc. in order We have restructured the manuscript in light of all review comments to make it easier for the reader. Reviewer #2: A well written paper suitable for publication. Just a few comments: Abstract and Methods: Is life expectancy going to be an inclusion/exclusion criteria or is it covered by the other criteria (patients are expected to have 3 weeks of therapy)? We did not include life expectancy as an inclusion/exclusion criteria as this is covered by the other criteria; namely ECOG performance status which provides an indication of the patient s fitness to take part in the study. As indicated within the manuscript, we will be assessing the viability of a 3 week intervention for this patient population. Background: Can you choose another word instead of institutions - the whole idea is that they are not institutions!

5 Thank you for highlighting this important point. We have removed reference to institutions and have just stipulated the time frame instead Music therapy is among the two most common forms of complementary therapy provided in hospices [4], with inclusion of music therapy in palliative care settings having a long tradition stemming back to the 1970 s [5]. Could you include the date of the Cochrane review in the text Page 5 line 103. We have now included the date of the Cochrane review in the text. Line 147. What is "mi" This was a typing error on our part which should have read It is not possible to blind patients to treatment allocation This has now been amended. Line 145. Will patients be stratified according to inpatient/outpatient. We have now revised the manuscript in light of approved substantial amendments, which have included focusing on an inpatient population only. Therefore, we have not included stratification according to inpatient/outpatient. See below for further clarification of this amendment. When we first drafted the protocol for our Music Therapy Project, the plan was to recruit from mainly outpatients, so a 3 week time frame for the intervention seemed feasible. However, we have had to focus our recruitment efforts on inpatients only due to other studies being conducted with outpatients, to avoid overburdening them. Line 171. Is music therapy part of routine care in the hospice - if so will control patients receive therapy whilst in the trial? Will it be withdrawn if they are having it? Line 180. We have now clarified within the manuscript that music therapy is not part of routine care within this hospice.

6 Line 359. The hospice is identified here - should it not be at the first opportunity? Line 41. The hospice is now identified at the first opportunity. Line 382. What happens if music therapy is found to be ineffective? Will you recommend it be stopped as a therapy? Will not the likely conclusion of a any large RCT be that music therapy is good for some people but not for others? As noted in the manuscript, if music therapy is found to be effective in the main randomised trial, this would provide support for commissioners and service providers to make an evidencebased decision on whether to incorporate music therapy in palliative care services, with patients as beneficiaries. We would not necessarily recommend as the decision may need to take account of factors other than effectiveness. Likewise, if the main trial shows that music therapy is ineffective on an outcome such as quality of life of the patient, we will make this information available to decision makers, rather than making recommendations about its withdrawal. As with many interventions of this type, we agree with the reviewer that the effects of music therapy may differ between different types of patient and we will explore this through subgroup analyses of both quantitative and qualitative analyses in the main trial; with a view to providing the evidence of effects that is needed by (but currently lacking for) decision makers. Reviewer #3: Background It would be helpful to have more information on current palliative care service provision, for example how are these set-up across the country, numbers of hospices etc. Also it would be helpful to understand more about existing provision of music therapy in palliative care services (are there any figures). Is it currently regularly provided across the country or is it patchy. How is it funded? How do services currently decide whether to offer music therapy? Would the outcome be to implement a new service or replace an existing service? Also are there any current guidelines on use of music therapy in palliative care? We would like to thank the reviewer for suggesting the inclusion of this useful information. We have now included this information within the background section of the manuscript. Line In terms of palliative care provision, figures for 2006 show there were 193 specialist in-patient units, 295 home care services, 314 hospital based services, 234 day care

7 services, and 314 bereavement support services across England, Wales and Northern Ireland [7]. While there are no figures available for the existing provision of music therapy in palliative care services as a whole, a survey by Daykin et al. [8] showed that music therapy is still an emerging therapy within cancer care across the UK compared to more established interventions such as art therapy. This survey showed that the majority of music therapy provision was funded externally by the NHS, or in a small number of cases from internal funding. Music therapy was also more likely to be used in settings were complementary and alternative therapies were already in place, suggesting a trend toward the use of music therapy as an additional supportive service rather than replacing existing services. There are currently no guidelines in place for the use of music therapy in palliative care. This highlights the need for a stronger evidence base that takes due cognisance of benefits and risks to help inform future music therapy provision [9]. In the discussion section it is mentioned about the planned RCT providing evidence for commissioners and service providers - need to be clearer on who they are. We have made it clearer in the manuscript that the planned RCT will provide evidence for NHS and third sector Specialist Palliative Care commissioners and service providers. Part of the discussion in para 2 is outlining benefit to patients but there is no information provided on benefits to service providers and commissioners. Information on benefits to service providers and commissioners is included as below. Line If music therapy is found to be effective for improving the quality of life of palliative care patients in a subsequent phase III RCT, this finding will provide support for NHS and third sector Specialist Palliative Care commissioners and service providers to make an evidence-based decision on whether to incorporate music therapy in palliative care services, with patients as beneficiaries. Trial Design Include details of how you are going to maintain blinding to the intervention for the researcher since they will be administering study measures to participants.

8 We have had to amend our original plans for maintaining blinding to the intervention for the researcher as we have found this was not feasible for the duration of the study. This amendment has been approved and details included in the manuscript. Line It is not possible to blind patients to treatment allocation, but the researcher administering the study measures will be blinded up until completion of the first follow-up visit,, after which time the researcher will invite patients and Family members/carers to talk freely about their experience of music therapy if they have been in the intervention arm of the trial. In order to maintain a level of blinding, an outcome adjudicator will conduct a data check of data entry, and a statistician will conduct data analysis, both blinded to treatment allocation. Intervention It is not clear what the process is for identifying patient needs, and what/how is equipment provided for the music therapy i.e. in hospices and in people's homes. The process for identifying patient needs, and what/how equipment will be provided is now clearly outlined. Line Patient s needs will be identified by the music therapist during the first session, guided by preliminary discussions with the hospice clinical team, and by the patient s own assessment of what needs are most important to them. The musical equipment used will include accessible musical instruments provided by the music therapist. It is a little unclear what the actual intervention looks like, especially the 'intervention manual'. Is it for the therapist or is for the participant. Is it a workbook? What is the content? What is the manual for? We have now made clear that the intervention manual is for the therapist, and have described its content and purpose within the manuscript. Line While the content of the sessions will be influenced by each patient s needs, interests, preferences and energy level and adapted accordingly in the moment, the music therapist will complete an intervention manual [23] at the end of each session, which will include details on who chose the music, what strategy was used (i.e. music listening, active musicmaking, improvising, legacy work, ), along with detailing strategies used to enhance treatment fidelity (how the intervention was monitored for consistency). This will aid transparent reporting,

9 along with guiding the development of a standardized protocol for use across sites in the main trial. Within the 'carer interview' section it refers to carers sitting in on music therapy sessions. This is not described as part of the intervention earlier in the manuscript. It would be helpful to be clear on what the intervention consists of i.e. whether carers can be present or not and how participants consent to this. We have clarified in the manuscript that this is not part of the intervention, but rather guided by the patient s wishes as recommended by our PPI representatives. We have also clarified how participants consent to this. Line As recommended by our PPI (Patient and Public Involvement) collaborators on this protocol, patients can choose to have their Carer present during the intervention as time together is precious. Patient s written informed consent to have their Carer present will be obtained by the hospice clinician during recruitment. Carers will only be observing the music therapy sessions, and will not be directly involved. Consent (carer interviews) More information on the consent process would be helpful, particularly how this works with those who do not wish to have a carer involved in the trial either attending therapy sessions or to be interviewed. Are participants being asked whether they want a carer involved? We have provided more information on the consent process for carer interviews, particularly for those carers who are not sitting in on sessions, or who do not wish to be interviewed. See previous response which clarifies that participants will be asked, and consent obtained if they wish a carer to be present. Line For those Carers who chose not to sit in on music therapy sessions, an invitation letter and information sheet will be left with their loved one to give to them on their next visit. Carers who return a Notification of Interest Form to the Music Therapist or Member of Hospice Staff will be contacted by the researcher and given the opportunity to ask questions about the study, at which point they can decide to opt out or provide written informed consent.

10 Discussion Para 2 in this section "Music therapy approaches " this would be better placed in the 'background' section as it describes music therapy. This paragraph has been moved to the background section. General comment The flow of the paper jumps around a little. Consider presenting this how the study will flow e.g. intervention description, study setting, eligibility, recruitment, consent, randomisation, data collection etc. Will be easier for the reader. We have restructured the manuscript in light of all review comments to make it easier for the reader.