Open access Protocol Protocol to develop a core outcome set in incisional hernia surgery: the HarMoNY Project Deena Harji ‍ ‍,1 Christophe Thomas,2 Stavros Antoniou ‍ ‍,3 Harsha Chandraratan,4,5 Ben Griffiths,1 B Todd Heniford,6 Liam Horgan,7 Ferdinand Koeckerling,8 Manuel Lopez-­Cano,9 Lisa Massey,10 Marc Miserez,11 Agneta Montgomery,12 Filip Muysoms,13 Benjamin Poulose,14 Wolfgang Reinpold,15 Neil Smart16 To cite: Harji D, Thomas C, ABSTRACT Antoniou S, et al. Protocol to Introduction Incisional hernia has an incidence of up STRENGTHS AND LIMITATIONS OF THIS STUDY develop a core outcome set in to 20% following laparotomy and is associated with ⇒ This project will ensure the development of an in- incisional hernia surgery: the ternational, clinically relevant, patient-­oriented core significant morbidity and impairment of quality of life. HarMoNY Project. BMJ Open outcome set (COS) to be used to guide outcome re- A variety of surgical strategies including techniques 2022;12:e059463. doi:10.1136/ and mesh types are available to manage patients with porting in future clinical research. bmjopen-2021-059463 incisional hernia. Previous works have reported significant ⇒ A robust systematic review will identify current out- ► Prepublication history for heterogeneity in outcome reporting for abdominal wall comes in randomised and non-­randomised studies this paper is available online. herniae, including ventral and inguinal hernia. This is reporting outcomes in incisional hernia. To view these files, please visit ⇒ In-­ depth qualitative interviews with key stake- coupled with under-­reporting of important clinical and the journal online (http://dx.doi.​ holders including patients, nurses, radiologists, patient-­reported outcomes. The lack of standardisation in org/10.1136/bmjopen-2021-​ outcome reporting contributes to reporting bias, hinders physiotherapists, members of international hernia 059463). evidence synthesis and adequate data comparison societies and industry partners will identify out- Received 20 November 2021 between studies. This project aims to develop a core comes of importance to all these groups. Accepted 09 February 2022 outcome set (COS) of clinically important, patient-­oriented ⇒ This project will determine which outcomes to mea- outcomes to be used to guide reporting of future research sure, however, further work will be necessary to in incisional hernia. agree and recommend a definition or measurement Methods This project has been designed as an instrument for each of the outcomes in the COS. international, multicentre, mixed-­methods project. Phase I will be a systematic review of current literature to examine the current clinical and patient-­reported outcomes for with a variety of techniques, meshes and incisional hernia and abdominal wall reconstruction. operative strategies available to manage this Phase II will identify the outcomes of importance to all key challenging cohort of patients. Given the stakeholders through in depth qualitative interviews. Phase range of options available there is significant III will achieve consensus on outcomes of most importance complexity involved in the management of and for inclusion into a COS through a Delphi process. Phase IV will achieve consensus on the outcomes that patients with incisional hernia. Alongside this should be included in a final COS. there is considerable variation in manage- Ethics and dissemination The adoption of this COS ment and outcome reporting. Despite an into clinical and academic practice will be endorsed by exponential increase in the number of peer-­ the American, British and European Hernia Societies. Its reviewed publications on the management utilisation in future clinical research will enable appropriate of incisional hernia over the last decade,2 data synthesis and comparison and will enable better the methodological quality of the majority clinical interpretation and application of the current of these studies is poor, with the majority of © Author(s) (or their evidence base. This study has been registered with the studies reporting outcomes on incisional employer(s)) 2022. Re-­use Core Outcome Measures in Effectiveness Trials initiative. hernia being of level 4 quality according to permitted under CC BY-­NC. No PROSPERO registration number CRD42018090084. commercial re-­use. See rights the Oxford Centre for Evidence Based Medi- and permissions. Published by cine.3 A recent systematic review reported BMJ. BACKGROUND over 75% of randomised controlled trials and For numbered affiliations see Incisional hernia following laparotomy has meta-­analyses reporting outcomes on ventral end of article. an incidence of up to 20% and is associated hernias were methodologically flawed, with Correspondence to with significant morbidity and impairment variable adherence to standardised reporting Deena Harji; of quality of life.1 The management of inci- frameworks such as Consolidated Standards ​deena_​harji@​hotmail.​com sional hernia has evolved over recent years, of Reporting Trials checklist or Preferred Harji D, et al. BMJ Open 2022;12:e059463. doi:10.1136/bmjopen-2021-059463 1 Open access Reporting Items for Systematic Reviews and Meta-­Analyses a Delphi process. Phase IV will achieve consensus on the (PRISMA) Checklist PRISMA.3 outcomes that should be included in a final COS. There is little known about standardised outcome reporting in patients with incisional hernia. Previous work Phase I: systematic review of clinical and patient-reported examining outcome reporting for inguinal hernia identi- outcomes fied significant variation in outcomes employed to report A number of detailed systematic reviews of currently clinical outcomes in this group. Significant heterogeneity reported clinical and patient-­reported outcomes in inci- in outcome definitions and assessment instruments exist sional hernia and complex abdominal wall reconstruction in inguinal hernia outcome reporting, alongside under-­ will be conducted. The full protocol including eligibility reporting of a number of important clinical and patient-­ criteria and search strategy is available online via the reported outcomes.4 More recently, work examining PROSPERO database (CRD42018090084). outcome reporting in randomised controlled trials of ventral hernia revealed marked heterogeneity in outcome Phase II: stakeholder qualitative interviews reporting of clinical endpoints related to hernia recur- To ensure all key stakeholders are appropriately repre- rence.5 Subsequently, it may be hypothesised that similar sented and all outcomes are captured within the COS variation and under-­reporting of relevant outcomes exists we will conduct in-­ depth qualitative interviews with within the current literature for incisional hernia repair. patients and other stakeholders that are not adequately Core outcome sets (COS) have been developed to represented within the current literature that is, nurses, overcome heterogeneity in outcome reporting, reduce radiologists, physiotherapists. We will also interview key reporting bias and enable adequate evidence synthesis, members of the international hernia societies including comparison of data between studies and meaningful clin- the American Hernia Society, British Hernia Society and ical interpretation and application of current evidence.6 the European Hernia Society and industry partners in a COS is an agreed set of outcomes, which should be bid to gauge a wider perspective. measured and reported, as a minimum in all studies and trials for a specific clinical area. This work was initiated Recruitment and developed by the Core Outcome Measures in Effec- Healthcare professionals tiveness Trials (COMET) initiative, which aims to facili- All members of the American Hernia Society, the British tate and guide the development of a number of COS.7 Hernia Society and the European Hernia Society will be Currently, there is no COS for incisional hernia. However, contacted and invited to participate. there are guidelines available to guide reporting outcomes Industry partners with regards to mesh properties8 and clinical outcomes9 Industry partners will be identified through key hernia associated with abdominal wall repair. Although, these organisations including the American Hernia Society, the guidelines are useful in trying to standardise reporting British Hernia Society and the European Hernia Society. outcomes, they do not reflect the opinion of all stake- Industry stakeholders will be contacted and invited to holders, in particular patients, when considering which participate. outcomes are of the most importance when reporting outcomes related to incisional hernia repair. To improve Patients the quality of the current evidence base and to improve Members of the American Hernia Society, British Hernia outcome reporting, a COS in incisional hernia is highly Society and the European Hernia Society will be asked desirable. to identify potential patient participants from clinic lists, theatre lists and patient records. Recruitment letters will Aims be sent to the identified patients, either in person during The aim of this project is to develop a COS of clinically routine follow-­up visits or by post. The recruitment letter important, patient-­oriented outcomes to be used to guide will give a full explanation of the qualitative interviews, reporting of future research in incisional hernia. instructions to participate and the contact details of the research team. Methodology METHODS In-­depth face-­to-­face or telephone cognitive interviews An international, mixed-­methods study will be conducted will be undertaken with eligible patients and stakeholders. in accordance with COMET guidelines to develop a COS Interviews will explore patients’ perceptions and experi- for use in incisional hernia. Phase I will examine the ences regarding living with an incisional hernia and will current clinical and patient-­reported outcomes for inci- identify the thoughts and opinions of stakeholders who sional hernia and abdominal wall reconstruction within are not adequately represented within the current liter- the literature. Phase II will identify the outcomes of impor- ature. A standardised, semistructured interview guide tance to all key stakeholders through in depth qualitative will inform the cognitive interviews. All interviews will interviews. Phase III will achieve consensus on outcomes be recorded. Open-­ended questions will be used at the of most importance and for inclusion into a COS through start of the cognitive interview followed by close-­ended 2 Harji D, et al. BMJ Open 2022;12:e059463. doi:10.1136/bmjopen-2021-059463 Open access questions to further explore any relevant themes. To Table 1 Purposive sampling strategy ensure appropriate representation of all stakeholders, we will conduct interviews with patients and other key stake- Patient factors No of patients holders from all participating countries. We will aim to Age conduct between 5 and 10 interviews per country.  18–30 4  31–60 4 Patient eligibility criteria Inclusion criteria as follows:  >60 4 ► Aged >18 years old. Gender ► With an existing incisional hernia.  Male 8-­10 ► A surgically treated incisional hernia in the last 12  Female 8-­10 months. Presentation ► Able to provide written informed consent. Exclusion criteria as follows:  Elective 6-­8 ► An existing other ventral hernia, that is, epigastric,  Emergency 6-­8 umbilical, paraumbilical, inguinal, portsite hernia. Repair ► A surgically treated ventral hernia, that is, epigastric,  Primary 6-­8 umbilical, paraumbilical, inguinal, portsite hernia.  Mesh 6-­8 To ensure our COS is representative of all stakeholders, with particular reference to patients with incisional hernia No of repairs a purposive sampling strategy has been designed to aid  First repair 4 recruitment (table 1). Our sampling strategy will target  Recurrent incisional hernia repair 4 a number of key factors to reflect the range and diversity Hernia size of the target population. There is no minimal sample size for cognitive interviews.  <10 cm in width 8–10  >10 cm in width 8–10 Data analysis Use of adjuncts All interviews will be audio recorded and transcribed  Yes 4 verbatim and transcripts will be imported into NVivo. All  No 4 transcripts will be anonymised. Interviews will be coded using the principles of thematic content analysis.10 Rele- Stakeholder Factors No of participants vant outcomes will be identified and appropriately coded  Specialty from the transcripts using a provisional coding frame-  General Surgery 4-­6 work based on the outcomes extracted from the system- atic review. Coded outcomes that are sufficiently similar  Plastic Surgery 4-­6 will be grouped into similar categories and then themes.  Radiology 4-­6 Analysis will be an iterative process, with data being anal-  Specialist nurses/ 4-­6 ysed after rounds of three consecutive interviews. Data physiotherapists analysis will be continued up until the point of data satu-  Industry partner 4-­6 ration. This is the point on the data analysis process where Country no further information is elicited.  UK 4 Phase III: Delphi study  Europe 4 Consolidation of outcomes  USA 4 The outcomes identified in phase I and II will be  Australia 4 combined, developed into a long-­list of items and catego- rised into broad domains using the principles of thematic content analysis. Appropriate questions will be mapped to these domains and will form the basis of the Delphi The linguistic and translation process should ensure study. Questions will have a lay translation available. We that the translated version of the Delphi are conceptual, will pilot the Delphi study with our steering committee to semantic and pragmatic equivalents of the original ques- ensure it is accessible, comprehensible and content valid. tionnaire, while ensuring it is culturally appropriate, rele- vant and meaningful to the target countries. The original Forward-backward translation Delphi questionnaire (English) will be used as the stan- Given the international nature of this study, we will trans- dard from which all other translations are made. late the Delphi study using forward-­backward translation Forward translation will be undertaken by two health- to ensure accessibility of the study by all international care professionals with an understanding of incisional participants. The aim of translation is to achieve different hernia. The translators will be bilingual with their language versions of the original Delphi questionnaire. primary language being that of the target country. They Harji D, et al. BMJ Open 2022;12:e059463. doi:10.1136/bmjopen-2021-059463 3 Open access will perform a detailed review of the Delphi question- Two sequential rounds of Delphi voting will be held naire and translate the questionnaire appropriately. Two with a feedback round in between. The first Delphi round independent translations will be prepared; these will be will enable participants to suggest outcomes that may not reviewed and compared with achieve a consensus version. have been included or overlooked. The spread of scores Any discrepancies between the translated version and the for each question item should reduced in between rounds original Delphi questionnaire will be discussed with the as consensus is reached. Following the first Delphi round steering committee. participants will be provided with feedback. Participants The final translated version will be translated back will have access to their individual scores from the first into English (backward translation). This will be done round and scores from key stakeholder group. by a native English speaker who is also proficient in the All included outcomes will be scored on a 9-­point Likert target language. The original Delphi questionnaire will scale, with 1 being ‘not essential’ to 9 being ‘absolutely be compared with the backward translation version and essential’ for inclusion into a COS. The 9 point Likert reviewed to ensure consistency. The aim is to ensure scale will be grouped into three categories; 1–3 (limited linguistic and conceptual equivalence between the orig- importance), 4–6 (important but not critical), 7–9 (of inal and translated versions of the Delphi. Any discrep- critical importance). ancies will be discussed and resolved with the steering Consensus will be defined as the following: committee and the bilingual translators who undertook For inclusion: more than 75% of respondents within a the forward translation. If equivalent versions have not stakeholder group rate the outcome as critically important been created further translational work may be required. and less than 15% of respondents rate the outcome as of This may include additional forward translations and/ limited importance. or the addition of further items/questions and will be For exclusion: more than 75% of respondents within a repeated as many times as necessary to achieve a satisfac- stakeholder group the outcome as of limited importance tory translated version. and less than 15% of respondents rate the outcome as of critical importance. Recruitment No consensus. Method of recruitment will be the same as phase II. Healthcare professionals, patients and industry stake- holders will be invited to participate through online web Phase IV: consensus meeting and social media platforms of the participating hernia A consensus meeting of all key stakeholders will be held in societies (American, British and European) and through conjunction with a European Hernia Society meeting to the Northern Surgical Trainees Research Association. discuss the results from the Delphi study. All participants Snowball sampling will be allowed to increase the sample registering to complete the Delphi study will be invited size and reach of the study. to participate in the consensus meeting. The aim of this consensus meeting is to agree on the final COS for inci- Sample size sional hernia. All outcomes will be discussed; a proposal There are no prerequisite criteria for sample size for will be made to include all outcomes in the final COS participation in Delphi studies. We hope by engagement that have been categorised as ‘for inclusion’ by all stake- with the American, British and European hernia societies holders and to exclude all outcomes that have been cate- we will capture the majority of individuals interested in gorised as ‘for exclusion’ by all stakeholders. Participants incisional hernia. will vote electronically to accept or reject these proposals. All other outcomes categories as ‘for inclusion’, ‘for Consent exclusion’ or ‘no consensus’ by one or two stakeholders No explicit consent will be obtained for participation in will be discussed and further rounds of voting will be the Delphi study. Consent will be implied through the used to agree the final COS. If no consensus is achieved a process of participation. The registration page of the further consensus meeting will be held. website hosting the Delphi study will outline that registra- tion to participate in the Delphi process through submis- sion of name and email address will indicate agreement Patient and public involvement to participate. The HarMoNY project has been discussed with patients at national hernia meetings and has been well received. Delphi process A dedicated, international patient and public (PPI) The aim of the Delphi study is to achieve consensus steering group will be appointed to inform the processes among all key stakeholders including patients, surgeons, of phases II–IV of this project. Patients will be approached radiologists and specialist nurses on the importance of by key members of the project team to participate in different outcomes in sequential questionnaires. The this steering group. This PPI steering group will help Delphi questionnaires will be developed using the Delphi- inform recruitment processes, help design and evaluate Manager software developed by the COMET initiative. all patient information sheets to ensure all information is Relevant demographics will be collected for each stake- applicable and understandable and advise on the content holder group. and format of dissemination of the final COS. 4 Harji D, et al. BMJ Open 2022;12:e059463. doi:10.1136/bmjopen-2021-059463 Open access Ethics @AgntMontgomery, Filip Muysoms @FilipMuysoms, Benjamin Poulose @BKP_ Ethical approval has been obtained from the Health Columbus, Wolfgang Reinpold @WolfgangReinpo1 and Neil Smart @Neil_J_Smart Research Authority and Health and Care Research Wales Contributors Study design: DH/CT/NS/BG/LH. Protocol writing: DH/CT/NS/BG. Methodological input: AM/FM/MM/TH/BP/SA/FK/WR. Manuscript review: ML-­C/LM/ (REC 21/WA/0278). Appropriate ethical approval will MM/HC. be sought from all participating countries in accordance Funding This project has been funded by the British Hernia Society and the with local and national guidelines. This study will be European Hernia Society. conducted in keeping with the Declaration of Helsinki. Competing interests None declared. Patient and public involvement Patients and/or the public were involved in the design, or conduct, or reporting, or dissemination plans of this research. Refer to DISCUSSION the Methods section for further details. Defining important outcomes and standardising their Patient consent for publication Not applicable. reporting has been recognised to be of key importance in clinical research, which has subsequently led to the Provenance and peer review Not commissioned; externally peer reviewed. development of a number of COS. There has been a Open access This is an open access article distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-­NC 4.0) license, which steady rise in the adoption and utilisation of COS,11 with permits others to distribute, remix, adapt, build upon this work non-­commercially, a number of key stakeholders, including commissioners and license their derivative works on different terms, provided the original work is and funding bodies recognising the importance and properly cited, appropriate credit is given, any changes made indicated, and the use benefits of COS for improving reporting outcomes.12 is non-­commercial. See: http://creativecommons.org/licenses/by-nc/4.0/. Incisional hernia repair can be complex with significant ORCID iDs variation in clinical management due to the great diver- Deena Harji http://orcid.org/0000-0002-8493-3312 sity of available surgical techniques.13 14 To ensure clinical Stavros Antoniou http://orcid.org/0000-0002-4630-6748 heterogeneity is not reflected in outcome reporting the development of COS in this cohort of patients is essen- tial. Ensuring consistent outcome reporting will reduce reporting bias, improve data synthesis and comparison, REFERENCES and will enable better clinical interpretation and appli- 1 van Ramshorst GH, Eker HH, Hop WCJ, et al. Impact of incisional hernia on health-­related quality of life and body image: a prospective cation of the current evidence base. It is hoped through cohort study. Am J Surg 2012;204:144–50. the development of a COS for incisional hernia, inter- 2 Kulacoglu H, Oztuna D. 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