Experiences of general practice of children with complex and palliative care needs and their families: a qualitative study

Objectives To investigate the views and experiences of general practice of children with life-limiting and life-threatening conditions, and their family members, through secondary analysis of a qualitative serial interview study. Thematic analysis was conducted on all interview data relating to experiences of primary care. Setting West Midlands, UK. Participants A total of 31 participants (10 children with life-limiting and life-threatening conditions and 21 family members) from 14 families. Study design and setting Secondary thematic analysis of qualitative interview data from a study carried out in the West Midlands, UK. Method 41 serial interviews with 31 participants from 14 families: 10 children aged 5–18 years with life-limiting and life-threatening conditions, and 21 of their family members. Results Three key themes emerged: (1) poor experiences of general practice cause children and families to feel isolated, (2) children and families value support from general practice, and (3) there are practical ways through which general practice has the potential to provide important aspects of care. Children and families reported benefits from fostering their relationship with their general practice in order to access important aspects of care, including the assessment and management of acute illness, chronic disease and medication reviews, and holistic support. Conclusion Children with life-limiting and life-threatening conditions and their families value the involvement of general practice in the care, alongside their paediatric specialists. Ways of developing and providing such support as part of an integrated system of care need to be developed.


GENERAL COMMENTS
Thank you so much for the opportunity to read this qualitative study highlighting the often not well-understood experience of families managing life-limiting/life-threatening illnesses. The data collected is richly informative and the significance of the topic is well presented. The in-depth serial interviews are a major methodological strength, a source of immense insight into the care needs of these children.
Background: The background contextualizes the aims of the study well. To strengthen the argument the author may consider why and how GPs provide aspects of palliative care and include more indepth discussion of the types of palliative and other care.
Methods: I recommend that the authors consider specifying their approach. It seems qualitative descriptive may align well, given the aims of the study and secondary nature of the data. It was not immediately clear to me what the authors mean by "explore the views of children and family members in relation to palliative care"did the interviews focus on experiences of palliative care in general? Experiences with GPs who provided palliative care? Experiences with GPs in general? Even though this is a secondary analysis, I would like to see more detail about the recruitment and sampling for the primary study, as well as the interview process (ex. were children and families interviewed together?). It seems to me to be less relevant to include the findings and conclusions of the primary study in the table. I would suggest the authors consider focusing more on increasing the detail in the methods section. The authors might consider further elaborating on how rigor was maintained in this qualitative inquiry, as well as how the PPI team member was found and involved. Was there ethics approval? What was the consent process?
Findings: In the findings, I would suggest referring to the group as a study sample instead of the study population. In the introduction to the findings, when refer to "palliative care" is this the palliative care that GP provides or that others provide? Overall, the quotes and examples are vivid and compelling. However, it struck me that most of the quotes presented are from family membersdid family members speak more to the experience than children? In the first theme especially, I wonder if the authors might think about ways to integrate the quotes and analytic commentary a bit more to build the analysis further. In the second theme, I wonder whether further introduction might be added for some of the quotes. Overall, I'm left wanting to know more about how the themes relate. For a thematic analysis, it seems that most of the themes were more manifest rather than latent content and I wonder if there is potential for further interpretation and expansion.
Discussion: While noted as a strength, the perspectives of children are largely missing from the findings. I encourage the authors to think about other limitations in the research study itself as the limitations noted are more future directions wonder if the comparison of the literature section could be expanded, as it is very similar to the introduction. In the implications, the authors might consider touching on palliative care aspects given the framing of the findings. Are there aspects of palliative care they recommend GPs provide beyond referral to specialist services?
I'm grateful to the authors for this important work and appreciate the opportunity to review this manuscript.

GENERAL COMMENTS
This is a timely secondary analysis given the recognised increased prevalence of children with life limiting or life threatening conditions in England. Findings add to the knowledge base on the care of children with complex and palliative care needs and highlight implications for practice. The manuscript is clearly presented however there are 3 areas that require minor revision: 1.

VERSION 1 -AUTHOR RESPONSE
Reviewer: 1 Briefly, could you define the serial qualitative interview method and explain the aim of its adoption in this study? I have added further detail to the methods section to explain and justify this method Could you review Table 1, please? The number of interviews is thirty-five. The number of children who participated in the interviews is probably less than ten. I am sorry this is a mistake -I have oversimplified the table and in doing so have not provided sufficient detail about individual interviews and group interviews, both of which were conducted and counted in the total of 41. 10 children took part in interviewssome were as part of a group interview, where others were interviewed individually. I have updated the table and clarified in the text Page 5, line 22: Could you provide some contextual information about the Patient and Public involvement (PPI), please? I did not grasp whether PPI is a team explicitly assigned for this study or a team that has already worked in the hospital? I have revised the PPI section on page 5 with further detail Reviewer: 2 Background: The background contextualizes the aims of the study well. To strengthen the argument the author may consider why and how GPs provide aspects of palliative care and include more indepth discussion of the types of palliative and other care. I have revised the introduction to provide more detail about the vital / core aspects of palliative care that GPs may be involve in providing, alongside community teams and specialist colleagues.
Methods: I recommend that the authors consider specifying their approach. It seems qualitative descriptive may align well, given the aims of the study and secondary nature of the data. It was not immediately clear to me what the authors mean by "explore the views of children and family members in relation to palliative care"did the interviews focus on experiences of palliative care in general? Experiences with GPs who provided palliative care? Experiences with GPs in general? Even though this is a secondary analysis, I would like to see more detail about the recruitment and sampling for the primary study, as well as the interview process (ex. were children and families interviewed together?). It seems to me to be less relevant to include the findings and conclusions of the primary study in the table. I would suggest the authors consider focusing more on increasing the detail in the methods section. I have reviewed the methods section and added more detail. Having done so, I do not think there is a need for Box 1 anymore and so I have removed this. The authors might consider further elaborating on how rigor was maintained in this qualitative inquiry. Detail is provided in the final methods paragraph on page 5 as well as how the PPI team member was found and involved. This detail has been added to the PPI section Was there ethics approval? Details of the ethics approval are provided What was the consent process? I have added this detail to the methods.
Findings: In the findings, I would suggest referring to the group as a study sample instead of the study population. I have changed the subtitle In the introduction to the findings, when refer to "palliative care" is this the palliative care that GP provides or that others provide? This detail is provided in the first paragraph of the qualitative findings the children and families associated the term "palliative care" with specialist services rather than a broad approach or something that was provided by the GP. Overall, the quotes and examples are vivid and compelling. However, it struck me that most of the quotes presented are from family membersdid family members speak more to the experience than children? On reflection, the children did not contribute as much as their family members in relation to experiences of GP, so I have discussed this more as a limitation of the study In the first theme especially, I wonder if the authors might think about ways to integrate the quotes and analytic commentary a bit more to build the analysis further. In the second theme, I wonder whether further introduction might be added for some of the quotes. Overall, I'm left wanting to know more about how the themes relate. For a thematic analysis, it seems that most of the themes were more manifest rather than latent content and I wonder if there is potential for further interpretation and expansion. I have reviewed this section and added more detail. Since this was a subanalysis, and experiences of GP were not the only subject of the overarching study, the depth of the analysis is a bit limited by the quality of the data. I have added this as a further limitation.
Discussion: While noted as a strength, the perspectives of children are largely missing from the findings. As above I wonder if the comparison of the literature section could be expanded, as it is very similar to the introduction. I have not changed this as this really reflects the very limited literature in this area In the implications, the authors might consider touching on palliative care aspects given the framing of the findings. Are there aspects of palliative care they recommend GPs provide beyond referral to specialist services? I have slightly edited this section of the discussion to describe the fact that important elements of care provided by GPs may not specifically be considered by GPs as "palliative", but in fact do provide an important foundation the delivery of specialist palliative care and end-of-life care.
Reviewer: 3 1. The enclosed manuscript has been read and approved by all authors. It is not under active consideration for publication elsewhere, has not been accepted for publication, nor has it been published in full or in part.

GENERAL COMMENTS
Well written study. Congratulations!

REVIEWER
Jane Evered University of Wisconsin-Madison, USA REVIEW RETURNED 20-Oct-2020

GENERAL COMMENTS
Thank you for the opportunity to review this important manuscript and for your responses to the comments on the first submission. I appreciate the additions to the background that provide more detail about the aspects of the palliative care GPs are providing. Thank you for adding detail to the methods section about the primary care study. I'm wondering how the study team assured ethical research recruitment when having primary clinical teams approach children and families. The authors may consider adding this to the ethics section. I'd also urge the authors to consider adding slightly more detail about how the analytic process. The added information about PPI is very strong. You mention the quality of the data as a limitation that prevented deeper analysis in the findings. I'm curious in the limitations if you might explain more about what you mean by the quality of the data. The implications of the discussion are compelling and significant. Thank you for the opportunity to read this work.