Experiences of patients and their relatives of postoperative radiological surveillance and surveillance intensity following primary resection of a soft tissue sarcoma and its impact on their quality of life: a systematic review protocol

Abstract

Introduction

Postoperative radiological surveillance following primary resection of a soft tissue sarcoma (sarcoma of the retroperitoneum, abdomen, pelvis, trunk or extremities) is standard of care in all international high-volume sarcoma centres in the world. The intensity of postoperative surveillance imaging is highly varied and knowledge of the impact of surveillance and surveillance intensity on patients’ quality of life is limited. The aim of this systematic review is to summarise the experiences of patients and their relatives/caregivers of postoperative radiological surveillance following resection of a primary soft tissue sarcoma and its impact on their quality of life.

Methods and analysis

We will systematically search MEDLINE, EMBASE, PsycINFO, CINAHL Plus and Epistemonikos. Hand searching of reference lists of included studies will be conducted. Further searches will be performed via Google Scholar, to reveal further studies within unpublished ‘grey’ literature. Two reviewers will independently screen the titles and abstracts following the eligibility criteria. After retrieval of the full text of the selected studies, the methodological quality will be appraised using the Joanna Briggs Institute Critical Appraisal Checklist for Qualitative Research and the Center for Evidence-Based Management checklist for Critical Appraisal of a Cross-Sectional Study. Data on the study population, relevant themes and conclusions will be extracted from the selected papers, and a narrative synthesis will be conducted.

Ethics and dissemination

The systematic review does not require ethics approval. The findings of the proposed work will be published in a peer-reviewed journal and disseminated widely to patients, clinicians and allied health professionals through the Sarcoma UK website, the Sarcoma Patient Advocacy Global Network and the Trans-Atlantic Australasian Retroperitoneal Sarcoma Working Group. In addition, the outcomes of this research will be presented at national and international conferences.

PROSPERO registration number

CRD42022375118.

Strengths and limitations of this study.

• This systematic review will involve a search of multiple databases and the titles and abstracts independently screened by two reviewers.

• We will include articles reporting the findings of surveys as well as qualitative studies on sarcoma of the retroperitoneum, abdomen, pelvis, trunk or extremities.

• To reduce the risk of selection bias, there will be no limits regarding language, geographical setting or publication date.

• The search strategy has been robustly designed with input from information specialists from the Library Research Skills team, University of Birmingham.

• A potential limitation may be difficulty identifying relevant studies. However, this is unlikely as we will be searching five databases, and conducting a search of grey literature.

Introduction

Soft tissue sarcomas (STS) and visceral sarcomas are a heterogenous group of rare, mesenchymal malignancies, including more than 80 histiotypes¹ with distinct clinicopathological and radiological presentations. The estimated incidence of STS is approximately 4–5/100 000/year in Europe.² Retroperitoneal sarcoma (RPS) represent 10%–20% of all soft tissue sarcomas.³ Surgery is the standard treatment for all patients with localised STS.¹ For patients with a high-grade sarcoma in the extremity or trunk, the wide local excision is followed by radiotherapy (RT).⁴ The role of (neo)adjuvant chemotherapy to enhance local control remains a matter of debate,^{5 6} as the benefit of (neo)adjuvant chemotherapy for patients with high-risk resectable STS relies on indirect evidence only.^{6 7} Gaining local control for RPS includes complex, multivisceral resectional surgery, which is critical for cure.1 3 8–10 In contrast to extremity STS, survival for patients with RPS is dictated by local or regional recurrences.¹¹ Despite optimisation of surgical technique, local recurrence (LR) and distant metastatic disease (MD) remain common events,^11–13 with a 5-year overall survival (OS) rate of 60% for RPS^{3 12–14} and long-term OS rates between 66%¹⁵ and 77%⁴—depending on tumour histology—for patients with extremity STS. Patterns and timings of recurrence are subject to histological subtype12 14 16–18 and tumour grade.¹⁸ Low-grade sarcoma tends to present as LR in a linear fashion with time and high-grade sarcoma recurs comparatively earlier with MD.3 12 18–20 Postoperative surveillance is standard of care in all international high-volume sarcoma centres in the world and has rapidly evolved without an evidence base to become highly intensive and prolonged within most centres. The intensity of postoperative surveillance imaging is highly varied across centres throughout the world,1 9 13 18 reflecting a lack of evidence to guide decision-making. Current recommended follow-up protocols1 9 21–24 extrapolate from evidence in sarcoma of the limb^{25 26} and expert opinion.1 7 9 10

Knowledge of the full impact of postoperative surveillance and surveillance intensity on long-term oncological outcomes and quality of life is limited. A Cochrane review across all solid tumour types did not identify a benefit of surveillance in improving OS, but specifically highlighted an absence of data related to health-related quality of life for cancer survivors.²⁷ Postoperative radiological surveillance is time and resource intensive for patients, clinicians and healthcare systems; placing a disproportionate burden on radiological resources.^{28 29} It can also cause significant anxiety for patients while awaiting imaging and subsequent results.^{28 29} However, details of patients’ attitudes towards and acceptability of postoperative surveillance for sarcoma, their lived experiences, its impact on their quality of life and levels of anxiety and psychological stress it might cause patients is scarce and primarily focused on patients with extremity sarcoma.^30–32

Therefore, the aim of this systematic review is to summarise the experiences of patients and their relatives/caregivers of postoperative surveillance following resection of a soft tissue sarcoma, patients’ acceptance and attitudes towards postoperative surveillance and its impact on their quality of life.

Objectives

To summarise:

1. The lived experiences of patients and their relatives/caregivers of postoperative surveillance following resection of a soft tissue sarcoma, patients’ acceptance and attitudes towards postoperative surveillance

2. The impact of postoperative sarcoma surveillance and postoperative surveillance intensity on their quality of life, the levels of anxiety and psychological stress it might cause patients.

Methods and analysis

Design

This protocol has been reported in line with the Preferred Reporting Items for Systematic Review and Meta-Analysis Protocols (PRISMA-P) checklist.³³ It has been registered with PROSPERO. The review will be conducted and reported following the PRISMA guidelines.³⁴ Any protocol amendment will be reported on publication of the systematic review.

Dimensions of interest

This review focuses primarily on the lived experiences of patients and their relatives/caregivers of postoperative surveillance and its impact on their quality of life. The search strategy was developed based on these key dimensions of interest. The search terms included: ‘sarcoma’, ‘surveillance’, ‘quality of life’ and ‘anxiety’. Anxiety was added, as it is often closely related to quality of life. Although not the primary focus of the review, we have included search terms such as ‘fear’, ‘psychological stress’, ‘suicide’ and ‘depression’ within the final search strategy, to keep the search broad and to capture varying degrees of anxiety as well as fear of cancer recurrence. If we find information on surveillance leading to depression or suicide, we will report in the systematic review.

Eligibility criteria

Inclusion criteria

We will include articles:

1. Focused on the experiences of patients (and their relatives/caregivers) under postoperative radiological surveillance, including all different imaging modalities and including both high and lower intensity strategies, following primary resection of a soft tissue sarcoma from the retroperitoneum, abdomen, pelvis, trunk or extremities.

2. In which patients with a soft tissue sarcoma from the retroperitoneum, abdomen, pelvis, trunk or extremities are a subset of a larger patient population if the findings for patients with soft tissue sarcoma are separately provided.

3. That include both paediatric and adult patients with sarcoma if the results for different age groups are presented separately;

4. That include both patients with bone and soft tissue sarcoma if the results for patients with soft tissue sarcoma are presented separately.

5. That report the findings of surveys and qualitative studies (including focus groups, interviews or observations) of patients and/or their relatives/caregivers.

Exclusion criteria

We will exclude articles if:

1. The study population consists of only patients with bone sarcomas, giant cell tumours, tenosynovial giant cell tumours, gastrointestinal stromal tumours, desmoid-type fibromatosis, Kaposi sarcoma, uterine sarcoma, carcinosarcoma or benign soft tissue lesion of mesenchymal origin according to the WHO classification.³⁵

2. Only clinician experiences are reported or if it is impossible to extract the data for patients and/or caregivers.

Search methods

The following electronic databases will be searched: MEDLINE, EMBASE, PsycINFO, CINAHL Plus and Epistemonikos. A comprehensive search strategy has been created with the assistance of information specialists from the Library Skills Team, University of Birmingham. The search will include combinations of keywords and Medical Subjects Headings terms for ‘sarcoma’, ‘surveillance’, ‘quality of life’ and ‘anxiety’. The search strategy developed for MEDLINE is provided in the online supplemental appendix A. In addition, we will review the reference lists of all eligible publications to search for potential eligible studies. Further searches will be performed via Google Scholar, within abstracts and proceedings from conferences and symposia to reveal further studies within unpublished ‘grey’ literature. There will be no restrictions with regards to the year of publication, the geographical setting or language. The aim is to complete the systematic review between November 2022 and June 2023.

Screening process

The titles and abstracts retrieved from the database search will be exported into EndNote and duplicate entries will be removed. Two reviewers will independently screen the titles and abstract following the eligibility criteria and those that do not meet the inclusion criteria will be excluded. The full text for potentially relevant records will be obtained and independently evaluated by the two reviewers. Discrepancies during title and abstract screening or full-text evaluations will be resolved through discussions among the reviewers and a third reviewer will be consulted if required. DM is an orthopaedic surgeon with special interest in sarcoma surgery. This work is part of a PhD project carried out by DM. DM and SJF have extensive clinical experience within the field of sarcoma surgery. SJF is a consultant sarcoma surgeon. OLA and CM are both experienced researchers within the Institute of Applied Health Research who have previously published systematic reviews. CM is highly experienced in the field of qualitative research.

Appraisal of included studies

We will critically appraise the methodological quality of the included studies using the Joanna Briggs Institute Critical Appraisal Checklist for Qualitative Research³⁶ for qualitative research and the Center for Evidence-Based Management checklist for Critical Appraisal of a Cross-Sectional Study for surveys.³⁷ Appraisal of included studies will be done by DM and cross-checked by another reviewer. Based on the appraisal of methodological quality, the strength of evidence as a basis for decision-making will be defined.³⁸

Data extraction and synthesis

Data extraction will be done by DM using a data extraction form. This will be cross-checked by another reviewer for accuracy and completeness.

Data will be extracted on:

1. Characteristics of the study population (including age, gender, ethnicity and type of soft tissue sarcoma); the type of postoperative sarcoma surveillance and, if available, surveillance intensity

2. The findings on the experiences of patients and or relatives/caregivers

3. The impacts of surveillance on the patients and or their relatives/caregivers.

As we anticipate substantial heterogeneity across the included studies, we will not conduct a meta-analysis or a metasynthesis. Instead, a narrative synthesis of the extracted data will be conducted according to the methods described by Popay et al 2006³⁹ and used in the systematic review by Aiyegbusi et al.⁴⁰

The stages include: (1) A preliminary synthesis which involves a descriptive summary of the information extracted on study characteristics and study findings. (2) An exploration of relationships and associations between study characteristics and reported findings within individual studies, as well as across studies will be conducted. The nature of heterogeneity in the studies in terms of variability in study populations, study designs and settings and their influence will be explored during this stage. (3) A discussion of the findings, their implications and the provision of recommendations for future research and clinical practice.

Discussion

Evidence with regards to the full impact of postoperative sarcoma surveillance and postoperative surveillance intensity on long-term oncological outcomes and quality of life is limited. Details of patients’ lived experiences of surveillance for sarcoma, its impact on their quality of life and levels of anxiety is scarce. This systematic review will be the first to summarise results from qualitative evidence and findings of surveys reporting on the experiences of patients and their relatives/caregivers of postoperative surveillance following resection of a soft tissue sarcoma and its impact on their quality of life.

Patient and public involvement statement

This research question has been prioritised by patients and charities with support from Sarcoma UK during a preliminary Patient and Public Involvement meeting. The study protocol was developed in collaboration with members of Sarcoma UK and members of the Centre for Patient-Reported Outcomes Research, University of Birmingham.

Ethics and dissemination

Systematic reviews do not require ethics approval. We will disseminate our project findings widely to patients, clinicians and allied health professionals during the semi-annual Trans-Atlantic Australasian Retroperitoneal Sarcoma Working Group meeting, at appropriate national and international conferences and on the Sarcoma UK website. We will aim to submit our findings for publication in a peer- reviewed journal.

Ethics statements

Patient consent for publication

Not applicable.