{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,5]],"date-time":"2026-05-05T12:32:26Z","timestamp":1777984346259,"version":"3.51.4"},"update-to":[{"DOI":"10.12688\/wellcomeopenres.12613.1","type":"new_version","label":"New version","source":"publisher","updated":{"date-parts":[[2026,3,10]],"date-time":"2026-03-10T00:00:00Z","timestamp":1773100800000}}],"reference-count":35,"publisher":"F1000 Research Ltd","license":[{"start":{"date-parts":[[2026,3,10]],"date-time":"2026-03-10T00:00:00Z","timestamp":1773100800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100000265","name":"Medical Research Council","doi-asserted-by":"publisher","award":["MR\/J008621\/"],"award-info":[{"award-number":["MR\/J008621\/"]}],"id":[{"id":"10.13039\/501100000265","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100004440","name":"Wellcome Trust","doi-asserted-by":"publisher","award":["100715"],"award-info":[{"award-number":["100715"]}],"id":[{"id":"10.13039\/100004440","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100016237","name":"Armauer Hansen Research Institute","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100016237","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":["wellcomeopenresearch.org"],"crossmark-restriction":false},"short-container-title":["Wellcome Open Res"],"abstract":"<ns3:p>Background Obtaining valid informed consent from research participants in low-income settings can be difficult, partly due to participants\u2019 limited familiarity with scientific research concepts. The situation is complicated when conducting genomic research on a stigmatizing familial disease. Methods We used Rapid Ethical Appraisal as a tool to assess local factors that were barriers to obtaining valid informed consent prior to conducting a genetic study of podoconiosis (non-filarial elephantiasis) in two Zones of Ethiopia. The tool included in-depth interviews and focus group discussions with patients, healthy community members, field workers, researchers\/Institutional Review Board (IRB) members, elders, religious leaders, and care coordinators (charity staff who support treatment clinics). Results Most patients and healthy community members did not differentiate research from routine clinical care (therapeutic misconception). Participants felt comfortable when approached in the presence of trusted community members. Regarding the mode of consent, field workers and care coordinators preferred verbal consent, whereas the majority of patients and healthy community members suggested the use of both verbal and written consent to accommodate varying literacy levels. Participants better understood genetic susceptibility concepts when analogies drawn from their day-to-day experience were used. Field workers and care coordinators considered the type of biological sample sought and gender were the most critical factors affecting the recruitment process. Most researchers and IRB members indicated that reporting incidental findings to participants is not a priority in an Ethiopian context. Conclusions Understanding the concerns of local people in areas where research is to be conducted facilitates the design of contextualized consent processes appropriate for all parties and will ultimately result in obtaining valid consent.<\/ns3:p>","DOI":"10.12688\/wellcomeopenres.12613.2","type":"journal-article","created":{"date-parts":[[2026,3,10]],"date-time":"2026-03-10T06:24:07Z","timestamp":1773123847000},"page":"99","update-policy":"https:\/\/doi.org\/10.12688\/wellcomeopenres.crossmark-policy","source":"Crossref","is-referenced-by-count":0,"title":["Rapid Ethical Appraisal: A tool to design a contextualized consent process for a genetic study of podoconiosis in Ethiopia"],"prefix":"10.12688","volume":"2","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-2608-2465","authenticated-orcid":false,"given":"Tewodros Tariku","family":"Gebresilase","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Zebene","family":"Deresse","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Girmay","family":"Tsegay","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4103-5029","authenticated-orcid":false,"given":"Tesfaye Sisay","family":"Tessema","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Abraham","family":"Aseffa","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2796-7468","authenticated-orcid":false,"given":"Gail","family":"Davey","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8946-7525","authenticated-orcid":false,"given":"Melanie","family":"Newport","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Fasil","family":"Tekola-Ayele","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Adamu","family":"Addissie","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"2560","published-online":{"date-parts":[[2026,3,10]]},"reference":[{"key":"ref1","doi-asserted-by":"crossref","first-page":"625-642","DOI":"10.1111\/tmi.12288","article-title":"Informed 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We need to talk!","volume":"15","author":"W Burke","year":"2013","journal-title":"Genet Med."},{"key":"ref36","doi-asserted-by":"crossref","first-page":"21","DOI":"10.1186\/1472-6939-14-21","article-title":"Ethical and legal implications of whole genome and whole exome sequencing in African populations.","volume":"14","author":"G Wright","year":"2013","journal-title":"BMC Med Ethics."},{"key":"ref38","article-title":"Rapid ethical appraisal: a tool to design a contextualized consent process for a genetic study of podoconiosis in ethiopia","author":"T Gebresilase","year":"2017","journal-title":"Open Science Framework."}],"container-title":["Wellcome Open Research"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/wellcomeopenresearch.org\/articles\/2-99\/v2\/xml","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/wellcomeopenresearch.org\/articles\/2-99\/v2\/pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/wellcomeopenresearch.org\/articles\/2-99\/v2\/iparadigms","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,3,23]],"date-time":"2026-03-23T03:14:14Z","timestamp":1774235654000},"score":1,"resource":{"primary":{"URL":"https:\/\/wellcomeopenresearch.org\/articles\/2-99\/v2"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,3,10]]},"references-count":35,"URL":"https:\/\/doi.org\/10.12688\/wellcomeopenres.12613.2","relation":{"has-version":[{"id-type":"doi","id":"10.12688\/wellcomeopenres.12613.1","asserted-by":"subject"}],"has-review":[{"id-type":"doi","id":"10.21956\/wellcomeopenres.28627.r150331","asserted-by":"subject"},{"id-type":"doi","id":"10.21956\/wellcomeopenres.28627.r150331","asserted-by":"object"},{"id-type":"doi","id":"10.21956\/wellcomeopenres.28627.r150536","asserted-by":"object"}]},"ISSN":["2398-502X"],"issn-type":[{"value":"2398-502X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,3,10]]},"assertion":[{"value":"Indexed","URL":"https:\/\/wellcomeopenresearch.org\/articles\/2-99\/v2#article-reports","order":0,"name":"referee-status","label":"Referee status","group":{"name":"current-referee-status","label":"Current Referee Status"}},{"value":"10.21956\/wellcomeopenres.28627.r150331, Jantina de Vries, Syntia Nchangwi Munung, Department of Medicine, Faculty of Health Sciences, University of Cape Town, Cape Town, 23 Mar 2026, version 2, indexed","URL":"https:\/\/wellcomeopenresearch.org\/articles\/2-99\/v2#referee-response-150331","order":0,"name":"referee-response-150331","label":"Referee Report","group":{"name":"article-reports","label":"Article Reports"}},{"value":"10.21956\/wellcomeopenres.13658.r26990, Angela Fenwick, Clinical Ethics and Law, Southampton General Hospital, University of Southampton, Southampton, 22 Nov 2017, version 1, 3 approved with reservations","URL":"https:\/\/wellcomeopenresearch.org\/articles\/2-99\/v1#referee-response-26990","order":1,"name":"referee-response-26990","label":"Referee Report","group":{"name":"article-reports","label":"Article Reports"}},{"value":"<b>Tewodros Gebresilase<\/b>; \n<i>Posted: 19 Feb 2026<\/i>; \n<b>Response to Reviewer 2 (Angela Fenwick)<\/b> \n<b>Abstract:<\/b>  The first sentence of your abstract is unclear and needs clarification: whose knowledge? And what do they need to know about research ethics to consent to a study?  \n<b>Response:<\/b> We accept this suggestion. We have revised the first sentence to clarify that the challenge arises from participants\u2019 &quot;limited familiarity with scientific research concepts,&quot; rather than a lack of knowledge about research ethics per se. Additionally, to address the concern regarding tautology, we have replaced &quot;genuine informed consent&quot; with &quot;valid informed consent&quot; throughout the abstract and manuscript.   In the results section you appear to confuse the process of consent with evidence of consent which is repeated later in the paper.&nbsp;Do you mean that some participants preferred their consent to be recorded on a written consent form?&nbsp;If you clarify this in the body of your text you could come back to rephrase this sentence in the abstract.&nbsp;  \n<b>Response:<\/b> We appreciate this distinction. We have rephrased the Abstract to clarify that the preference refers specifically to the documentation of consent. The text now specifies that while field workers preferred verbal consent to avoid literacy barriers, the majority of patients preferred &quot;written consent (signature or fingerprint)&quot; as tangible evidence of their agreement.   You state that the \u2018type of biological sample sought and gender\u2019 were the two most important factors affecting the recruitment process\u2019 but I assume you mean the two most important factors that the participants identified for the podoconiois study?&nbsp;These two are just listed in a table in your results section which seems inconsistent with the importance you attach to them.  \n<b>Response:<\/b> We accept this suggestion. We have revised the Abstract to explicitly attribute these factors to the field workers and care coordinators (previously referred to as podoconiosis administrators) who identified them as critical logistical barriers. Furthermore, to ensure the text is consistent with the importance placed on these factors in the Abstract, we have significantly expanded the Discussion section to analyze them in depth\u2014specifically addressing the gender norms regarding private interviews and the misconception that saliva is &quot;less significant&quot; data than blood.    \n<b>Introduction<\/b>  This is brief and some complex issues are dealt with rather superficially; for example in relation to therapeutic misconception: might the distinction between clinical care and research be less binary in some contexts, for example participating in research may give you a diagnosis (or a risk prediction) which you could then follow up. Similarly with autonomous decision making: to what extent do people really make decisions on their own?&nbsp;  \n<b>Response<\/b>: We accept this suggestion. We have revised the Introduction to provide a deeper explanation of these nuances. Specifically, we added text acknowledging that in genomic research, the distinction between research and care is often blurred because the research itself provides medically relevant information (e.g., risk prediction), thereby offering potential clinical value. Regarding decision-making, we expanded the text to explicitly describe local practices as 'communal' or 'relational'\u2014involving consultation with family heads and elders\u2014rather than simply framing them as contrary to Western standards of autonomy.   I am not clear about the extent to which the use of REA is faster or cheaper than other \u2018tools\u2019 (which might be?) given the number of interviews and focus groups you conducted etc.&nbsp;Can you clarify?&nbsp;  \n<b>Response<\/b>: We have revised the Introduction to clarify the specific efficiency of the method. We specified that an REA cycle typically requires only 4\u20136 weeks and costs approximately ~2,250 USD per site. We explain that this distinguishes it from traditional, full-scale ethnographies or longitudinal qualitative studies, which often require months of fieldwork and significantly higher budgets.    \n<b>Methods<\/b>  This needs expanding to include: some justification for your sample and sample size; how you decided whether to use a focus group or interview; how you dealt with these two different data sets during analysis.&nbsp;  \n<b>Response<\/b>: We have expanded the \n<i>Methods<\/i> section to address these points:  \n<b>Sampling:<\/b> We clarified that purposive sampling was used to capture diverse stakeholder perspectives and that the final sample size was determined by data saturation. \n<b>Method Choice:<\/b> We added a justification explaining that In-depth Interviews (IDIs) were used with patients to ensure privacy regarding sensitive topics, while Focus Group Discussions (FGDs) were used with community members to elicit shared social norms. \n<b>Analysis:<\/b> We clarified that data from both sources were triangulated during analysis to compare individual narratives against group consensus.     You mention the prevalence of podoconiosis for over 15yrs but you only interviewed people over 18 so you need to give an explanation of why this was the case.&nbsp;  \n<b>Response: <\/b>The prevalence statistics cited (15+ years) refer to established epidemiological data. However, for this study on informed consent, we restricted recruitment to adults (\u226518 years) as this is the legal age of majority for independent consent in Ethiopia. We believe the current description of participants as &quot;adults&quot; (18-68 years) in the \n<i>Results<\/i> sufficiently distinguishes our sample from the general prevalence data.   Under data analysis you say you used a conceptual framework described in previous studies and, whilst I don't think you need to go into lots of detail about this, I should not have to look up other studies to ascertain what you did.&nbsp;A brief summary would suffice.  \n<b>Response: <\/b>We accept this suggestion. We have revised the \n<i>Data Analysis<\/i> section to include a brief summary of the conceptual framework. We clarified that this framework systematically explores local perspectives across the continuum of the consent process, specifically focusing on perceptions of research, recruitment strategies, decision-making dynamics, and consent preferences.    \n<b>&nbsp;Results<\/b>  In para 2 your themes should relate to your following headings more specifically.&nbsp;In general terms, this section would benefit from a re-organization.&nbsp;You move from present to past tense in this section on occasion: a general rule is that when you are reporting what participants felt, said etc you should use the past tense.&nbsp;  \n<b>Response: <\/b>We accept this suggestion. We have revised the introductory paragraph of the Results to ensure the listed themes correspond exactly to the subsequent section headings. Additionally, we have reviewed the entire Results section and standardized the verb tense, ensuring that all reports of participant views are consistent in the past tense (e.g., changing &quot;use&quot; to &quot;used&quot;, &quot;pose&quot; to &quot;posed&quot;).   Perceptions of heath research: I am unclear whether you were asking participants about healthcare research in general or for your particular podoconiosis study: either way this needs clarifying. Without understanding this it is difficult to interpret your narrative (this is the case elsewhere also so a general clarification would help); for eg, the first quote indicates that research gives health status which may well be the case if a participant gets a diagnosis from their participation in research (as I indicated above), but this may not be the case in your particular study.&nbsp; (See also reference to this in the 2\n<sup>nd<\/sup>&nbsp;para of your discussion).&nbsp;The rest of this section reads rather clumsily with some repeated information and could do with streamlining.&nbsp;  \n<b>Response<\/b>: We appreciate this feedback. We have revised the text to clarify that the interviews followed a &quot;funnel&quot; approach: starting with general perceptions of health research to assess baseline understanding, before moving to specific perceptions of the proposed podoconiosis study. To reflect this, we have restructured the section into two distinct subsections: &quot;Perceptions on health research&quot; (covering general views and therapeutic misconception) and &quot;Specific perceptions regarding the podoconiosis study&quot; (covering heredity and study design). We also added an introductory sentence explicitly outlining this structure.   Perceptions on informed consent for health research: this section is rather brief given the complexity of the concept.&nbsp;Could it be joined with another section? Is consent a \u2018tool\u2019?&nbsp;Isn\u2019t it a process? The 2\n<sup>nd<\/sup>&nbsp;quote relates to coercion and you could have highlighted this as it reflects more depth of thinking than your narrative indicates.&nbsp;&nbsp;  \n<b>Response<\/b>: We accept this suggestion. We have merged this section with the subsequent one, now titled &quot;Perceptions of informed consent and preparation of study documents,&quot; to improve the narrative flow. We have also clarified that while participants often referred to the information sheet as a &quot;tool,&quot; they explicitly recognized consent itself as a fundamental ethical &quot;process.&quot; Furthermore, we added an interpretative sentence highlighting the participant\u2019s insight that voluntariness is essential not just for ethics, but for the scientific validity of the data.   Preparation of consent form and information sheet: does the first sentence relate to the forms or to the wider process of consent which includes verbal explanation? This needs clarifying up-front - and in your second para - as from the quotes I would infer it was the latter, but this doesn't match your theme heading.&nbsp;The quote on confidentiality in particular seems to referring to issues inherent in the process rather than what they want included in information sheets.  \n<b>Response<\/b>: We appreciate this insight. We have revised the text to clearly distinguish between the content requirements for written forms (which must be simple) and the challenges inherent in the verbal consent process. Regarding the quote on confidentiality, we agree it relates to the recruitment environment rather than the content of the forms. Accordingly, we have moved this quote to the &quot; Approaching the community and potential study participants&quot; section.   Approaching potential study participants: the title isn\u2019t clear and isn\u2019t clarified in your first sentence where \u2018potential\u2019 is not indicated.&nbsp;Are you referring to whom to approach first about a study (ie people who may not actually be the potential study participants but who act as gatekeepers\/facilitators)?  \n<b>Response:<\/b> We accept this suggestion. We have renamed the section \u201cApproaching the community and potential study participants\u201d to clearly distinguish between the gatekeepers and the study subjects. We also revised the opening sentence to explicitly state that the process involves \u201ctrusted community intermediaries\u201d facilitating access to \u201cpotential study participants.\u201d   Information provision: the distinction between this theme and \u2018Preparation of consent form and information sheet\u2019 isn\u2019t clear to me.&nbsp;Are you referring here to information about podoconiosis?  \n<b>Response:<\/b> We agree that the previous title (&quot;Information provision&quot;) was ambiguous and appeared to overlap with the section on preparing consent forms. The distinction is that the previous section dealt with the \n<i>written<\/i> study documents, whereas this section focuses on \n<i>verbal<\/i> strategies (specifically analogies) used to explain abstract genetic concepts to participants with low literacy. To make this distinction explicit, we have renamed this section &quot;Strategies for explaining complex genetic concepts.&quot; This clarifies that the focus is on pedagogical methods (such as the &quot;improved seed&quot; and &quot;common cold&quot; analogies) rather than general information about podoconiosis.   Decision making, recruitment and consent taking: there is lots wrapped up in this heading and, as I indicated above, I think your table needs an accompanying narrative if it includes the 2 factors you consider most important.&nbsp;The first para is confusing and needs re-writing.&nbsp;In relation to the para that begins: \u2018We found mixed views\u2026\u2019: I don't understand why this wouldn't be under the theme, \u2018Preparation of consent form and information sheet\u2019.&nbsp;  \n<b>Response<\/b>: We have significantly restructured this section to address your concerns. We agree that the findings regarding verbal versus written consent fit more logically under the earlier theme, &quot;Perceptions of informed consent and preparation of study documents,&quot; and have moved that paragraph accordingly to improve the flow. Regarding decision-making, we rewrote the opening paragraph to clearly distinguish between autonomous decision-making (common for low-burden studies) and family-based decision-making (required for studies involving travel, costs, or family planning). Finally, we added a specific narrative preceding Table 2 to explicitly highlight gender and sample type as the most critical recruitment factors identified by field workers   Return of incidental findings: this seems a huge topic to take on in such a brief way in this paper and I would suggest considering removing it as, as it stands, does not really tell me anything useful; and I have no idea whether the podoconiosis study would involve the return of IFs from what you have written. Furthermore I am not clear why you didn't ask patient and community members questions about Ifs.&nbsp;If you removed this theme you could expand on gender and sample type etc., if you are short of words. &nbsp;  \n<b>Response: <\/b>We acknowledge your concern regarding the scope of this section. However, we have chosen to retain it because it documents a critical ethical divergence between international guidelines (&quot;duty to warn&quot;) and local realities (&quot;right not to know&quot; due to limited health infrastructure). regarding the absence of community views, we have revised the text to clarify that this exclusion was deliberate: introducing hypothetical secondary risks to participants who already struggle to distinguish research from clinical care (&quot;therapeutic misconception&quot;) risked confirming their belief that the study was a comprehensive health screening. Finally, to address your suggestion about prioritizing recruitment factors, we have also significantly expanded the narrative regarding gender and sample type in the &quot;Decision-making&quot; section.    \n<b>Discussion<\/b>  In the first sentence you refer to \u2018genuine informed consent\u2019 which is a tautology. If consent is what you outline in your first paragraph then it doesn't need additional adjectives (you use it in your abstract also).&nbsp;In the 4\n<sup>th<\/sup>&nbsp;para you say: \u2018it is important to cover&nbsp;all&nbsp;aspects of the research to help participants reach an informed decision\u2019, and \u2018a&nbsp;thorough&nbsp;explanation\u2019 is necessary but what does this mean in this context?&nbsp;How did your research help you understand what was appropriate?&nbsp;  \n<b>Response:<\/b> We accept the comment regarding the term &quot;genuine informed consent&quot; and have replaced it with &quot;valid informed consent&quot; throughout the manuscript to avoid tautology. Regarding the definition of &quot;appropriate&quot; explanation, we have revised the Discussion to resolve the tension between participants\u2019 preference for simple information and the ethical requirement for disclosure. Our findings indicated that a standard &quot;thorough&quot; explanation\u2014often interpreted as reading a lengthy, legalistic consent form\u2014was counterproductive and signaled a &quot;lack of confidence&quot; to the community. Therefore, we have clarified that &quot;appropriate&quot; explanation in this context refers to a &quot;layered approach&quot;: the consent process should begin with the practical medical information prioritized by the community (disease cause and prevention) as a bridge to explaining abstract genomic concepts using the local analogies identified in our results (e.g., &quot;sowing seeds&quot; for heredity).   If you re-organise your themes and take into account the issues raised above, then your discussion will be strengthened.  \n<b>Response:<\/b> We accept this constructive suggestion. We have extensively revised and reorganized the Discussion section to directly align with the themes identified in our results. Specifically, the discussion now follows the logical progression of the consent process\u2014addressing the tension between simple information and ethical disclosure, the specific role of gatekeepers, and the critical recruitment factors (gender and sample type). These structural changes have significantly strengthened the logical flow and ensured that every claim in the discussion is anchored in the data presented in the results.   Can you say more about what you wouldn't have known for the podoconiosis study if you had just relied on previous REA studies in Ethiopia?&nbsp;I would like a summary of what you took forward into the podoconiosis study.&nbsp;  \n<b>Response:<\/b> We have revised the Discussion to explicitly detail the unique findings of this REA that could not have been extrapolated from previous studies in Ethiopia. Specifically, without this context-specific assessment, we would not have known to implement three critical changes:  \n<b>Linguistic Context:<\/b> We identified a specific barrier where the Amharic term for research (\n<i>Meremer<\/i>) was conflated with criminal investigation (\n<i>Mermera<\/i>), necessitating the use of the term \n<i>Tinat<\/i> (study) to avoid fear of police involvement.           \n<b>Gatekeepers:<\/b> Unlike previous findings where religious leaders were primary gatekeepers, our REA revealed that \n<i>Kebele<\/i> (local government) administrators were the essential authority figures needed to legitimize the study in these specific zones. \n<b>Sample Misconceptions:<\/b> We found that the preference for saliva over blood often stemmed from a misconception that saliva was &quot;less significant&quot; data, prompting us to design a consent process that explicitly clarified that genetic data from saliva carries the same privacy risks as blood.","URL":"https:\/\/wellcomeopenresearch.org\/articles\/2-99\/v2#referee-comment-6436","order":2,"name":"referee-comment-6436","label":"Referee Comment","group":{"name":"article-reports","label":"Article Reports"}},{"value":"10.21956\/wellcomeopenres.13658.r28289, Malcolm de Roubaix, Ciara Staunton, Centre for Medical Ethics and Law, Department of Medicine, Faculty of Medicine and Health Sciences, Stellenbosch University, Cape Town, 07 Dec 2017, version 1, 3 approved with reservations","URL":"https:\/\/wellcomeopenresearch.org\/articles\/2-99\/v1#referee-response-28289","order":3,"name":"referee-response-28289","label":"Referee Report","group":{"name":"article-reports","label":"Article Reports"}},{"value":"<b>Tewodros Gebresilase<\/b>; \n<i>Posted: 19 Feb 2026<\/i>; \n<b>Response to Reviewer 3 (Malcolm de Roubaix &amp; Ciara Staunton)<\/b> \n<b>Introduction<\/b>  Genomic research raises a number of challenges for the informed consent process and it is not clear why there is a focus on incidental findings only, particularly as the results speak to a wider range of issues. A broader discussion of these issues and, in particular, in the African setting would be good.  \n<b>Response: <\/b>We accept this suggestion. We have revised the Introduction to broaden the discussion of ethical challenges in genomic research beyond incidental findings. Specifically, we added text highlighting that data sharing, sample storage, and sample export are critical concerns in the African genomic landscape (citing Tindana et al., 2012). This broader context ensures the Introduction better reflects the wide range of issues covered in our Results, moving beyond a singular focus on incidental findings.    \n<b>Methods<\/b>  The conceptual framework mentioned in the data analysis should be briefly discussed.  \n<b>Response<\/b>: We accept this suggestion. We have revised the &quot;Data Analysis&quot; section to include a brief summary of the conceptual framework so readers do not need to consult external references. We clarified that this framework systematically explores local perspectives across the continuum of the consent process, specifically focusing on: (i) perceptions of health research versus medical care, (ii) preferred channels for approaching and recruiting participants, (iii) individual versus communal decision-making practices, and (iv) preferences for information provision and consent documentation.   Would it not also have been significant to interview traditional healers as well, since many Africans have to rely on their services, they are often trusted and revered members of rural communities and their views may be influential in those societies? Should they be involved in research at all, as has been done with HIV in many settings?  \n<b>Response<\/b>: We agree that a more critical reflection on the methodology is necessary. We have added a &quot;Limitations&quot; paragraph to the end of the Discussion section. In this section, we explicitly acknowledge the inherent limitations of a &quot;rapid&quot; assessment (typically 4\u20136 weeks) compared to long-term ethnography, as well as the potential for selection bias due to our reliance on community gatekeepers. Regarding the exclusion of traditional healers, we explain that while we recognize their influence in rural health-seeking behavior, our sampling was driven by the specific recruitment pathway of the genetic study (facilitated by health extension workers and clinic staff). Therefore, we prioritized stakeholders directly involved in that specific consent interface.    \n<b>Results<\/b>  Under researcher\/IRB members, does this mean that they are both researchers and IRB members?  \n<b>Response: <\/b>We have revised the &quot;Study Design and Participants&quot; section to explicitly clarify this definition. We state that this group consisted of &quot;researchers who also served as Institutional Review Board (IRB) members.&quot; We interviewed these specific experts to capture perspectives that bridge both the scientific conduct of research and the ethical review process.   Throughout the results, it is often not clear whether you are referring to health research generally or podoconiosis. This should be clarified.  \n<b>Response<\/b>: We have reviewed the Results section and added a clarifying statement to the opening paragraph. We explain that due to the research-na&iuml;ve nature of the population, participants\u2019 views on &quot;perceptions of research&quot; and &quot;consent&quot; tended to be about health research in general (often conflated with medical care). However, findings regarding specific logistics\u2014such as recruitment channels, sample types (saliva), and explaining heredity\u2014were directly focused on the context of the planned podoconiosis study.   At times the differences between researcher\/IRB and other groups are discussed, but this is not consistent e.g. perceptions on informed consent for health research contains views of IRB\/researchers only. These divergent views (if any) should be included. If consistent, this should also be made clear. Furthermore, were there any variations according to location?  \n<b>Response<\/b>: We have revised the Results section to address these points. Regarding divergent views, we added text to the &quot;Perceptions of informed consent and preparation of study documents&quot; section explicitly contrasting researcher perspectives (who defined consent as an ethical &quot;process&quot;) with community perspectives (who viewed it as a procedural step for receiving care). Regarding location, we inserted a statement at the beginning of the Results section clarifying that findings were consistent across both zones, with the exception of specific linguistic variations regarding the translation of technical terms.   Under informed consent, you refer to it as a \u2018tool\u2019. Although one respondent does discuss it as a \u2018tool\u2019, it is a process. Moreover, in thinking of it only as a \u2018tool\u2019 presumably to inform\u2019, one downplays the real issue, i.e., obtaining authentic consent. Informing is just one of the ingredients of informed consent. Being a process implies something developing over time, not instantaneously.  \n<b>Response:<\/b> We agree that informed consent is a dynamic process, not merely a tool. The reference to &quot;tool&quot; in the original text reflected the specific language used by interviewees when discussing the \n<i>information sheet<\/i>. We have revised the &quot;Perceptions of informed consent&quot; section to clearly distinguish these concepts. We now state that while stakeholders referred to the documentation as a communication &quot;tool,&quot; they emphasized that consent itself is a fundamental ethical &quot;process&quot; of obtaining permission.   Had the other groups any perceptions on informed consent as they were asked about it?  \n<b>Response: <\/b>In our analysis, we found that distinct perceptions of &quot;informed consent&quot; as a theoretical concept were largely unique to the Researcher\/IRB group. As we have clarified in the revised &quot;Perceptions of informed consent and preparation of study documents&quot; section, patients and community members did not articulate a specific perception of &quot;consent&quot; as an isolated ethical stage. Instead, their views were embedded in their broader understanding of the relationship with health providers. When asked about consent, their responses focused on practical requirements (e.g., wanting to know the cause of the disease) and documentation preferences (e.g., the preference for written versus verbal agreement). We believe the current revision accurately reflects this distinction: professionals discussed the definition of consent, while community members discussed the conditions for agreement.   Considering the questions asked about informed consent, was there any other than it being an important process that was discussed?  \n<b>Response: <\/b>Yes, our analysis extended well beyond defining consent as a process. As detailed in the revised &quot;Perceptions of informed consent and preparation of study documents&quot; and &quot;Decision-making and recruitment factors&quot; sections, the interviews covered:  \n<b>Content Preferences<\/b>: We found a clear divergence where community members prioritized practical information (disease cause and prevention) while researchers emphasized legalistic details (risks and contact info). \n<b>Documentation Format<\/b>: We explored the debate between written consent (viewed by some as &quot;tangible proof&quot;) versus verbal consent (preferred by those fearful of signing legal documents). \n<b>Decision-Making<\/b>: We examined the tension between individual autonomy and family consultation, particularly when logistical burdens were involved.     In \u2018decision making, recruitment and consent taking\u2019, you refer to \u2018most participants\u2019 and \u2018they\u2019.&nbsp; Again, here it is important to indicate which group you are referring to, and if there are differences.  \n<b>Response: <\/b>We have revised this section (now titled &quot;Decision-making and recruitment factors&quot;) to explicitly identify the stakeholder groups for each finding. We clarify that while decision-making was generally viewed as an individual act by patients (unless logistical burdens required family consultation), the specific barriers to recruitment (gender and sample type) were primarily identified by field workers. Additionally, regarding the findings on consent documentation (which we have moved to the earlier &quot;Perceptions of informed consent&quot; section for better flow), we now explicitly contrast the views of care coordinators (previously referred to as podoconiosis administrators), who preferred verbal consent, with those of patients, who preferred written consent. This revision removes all ambiguous references to &quot;most participants&quot;.   Factors affecting recruitment to research: would females not be comfortable being interviewed by any stranger, or just a male stranger? Spelling error on \u2018sample type sought\u2019.  \n<b>Response<\/b>: We have corrected the spelling of &quot;sample type sought&quot; in Table 2. Regarding the query on gender, our data specifically indicates that married women are uncomfortable being interviewed alone by male strangers. We have updated both the narrative text and Table 2 to explicitly specify &quot;male stranger&quot; rather than just &quot;stranger&quot; to ensure precision.   Previous research experience: does this mean that those with no previous exposure to research had sorcery concerns? Are some factors more important than others?  \n<b>Response: <\/b>We have revised the &quot;Decision-making and recruitment factors&quot; section to address these queries. Regarding the relative importance of factors, we explicitly state in the text (and Discussion) that gender and sample type were identified by field workers as the most critical determinants of recruitment success. Regarding previous research experience, we confirm the reviewer\u2019s interpretation: field workers observed that participants with \n<i>no<\/i> prior exposure to research were significantly more susceptible to rumors (such as fear of sorcery or blood-selling), whereas those with previous exposure\u2014particularly through local treatment programs\u2014did not exhibit these specific fears, as noted in Table 2.   The patient groups were not asked about incidental findings. Why is this?  \n<b>Response: <\/b>We intentionally restricted the discussion of &quot;incidental findings&quot; to Researcher\/IRB members because the concept was deemed too abstract for the study population. As our results indicate, many community participants already struggled to distinguish between &quot;research&quot; and &quot;routine medical care&quot; (therapeutic misconception). Introducing the concept of \n<i>secondary<\/i> genomic findings\u2014unexpected information unrelated to the primary study\u2014would have been difficult to translate and likely would have deepened the misconception that the study was a comprehensive health screening. We have updated the &quot;Return of Incidental Findings&quot; section to explicitly state that this topic was excluded from community interviews due to this complexity.    \n<b>Discussion<\/b>  The 4\n<sup>th<\/sup>&nbsp;paragraph is confusing and raises a number of issues. You are stating that participants want to know basic information, but then state that information sheets and consent forms should have much more information. The discussion does not correlate with what has been stated in the results. How does this link to \u2018genuine\u2019 informed consent. Indeed, what is the difference between \u2018genuine informed consent\u2019 and \u2018informed consent\u2019. The term \u2018authentic\u2019 informed consent may be more appropriate.  \n<b>Response: <\/b>We accept the comment regarding the terminology and have replaced &quot;genuine informed consent&quot; with &quot;valid informed consent&quot; throughout the manuscript to avoid tautology. Regarding the discussion on information provision, we have revised the text to resolve the tension between participants' preference for simple information and the ethical requirement for disclosure. We now clarify that our results support a &quot;layered approach&quot; to information provision: consent processes should begin with the practical medical information prioritized by the community (disease cause and benefits) as a bridge to explaining the abstract aims of genomic research. This approach directly links our results\u2014specifically the use of local analogies\u2014to our ethical recommendations.   Incidental findings is a challenging topic and one that needs to be addressed in this context. Some reflection on the views expressed in these findings must be discussed. As the paper stands, the issuer regarding incidental findings is almost an afterthought, despite it appearing to be of importance in the introduction.  \n<b>Response: <\/b>We agree that the issue of Incidental Findings (IFs) required deeper reflection. We have expanded this section to explain that while international guidelines emphasize disclosure, our results suggest a specific context-dependent barrier: the risk of exacerbating therapeutic misconception. We have added an argument that introducing hypothetical secondary risks to participants who already struggle to distinguish research from clinical care may inadvertently confirm their belief that the study is a comprehensive health screening. Therefore, we conclude that in this specific setting, the immediate ethical priority is managing expectations about \n<i>primary<\/i> findings, rather than burdening the initial consent process with complex hypothetical scenarios.   Research should generally benefit the society\/individuals who participate. How would this society benefit? How was feedback provided to the participants, if at all?  \n<b>Response: <\/b>We have revised the Discussion to clarify the benefits provided. While monetary compensation was avoided to prevent undue influence, we followed the recommendation of local field workers to provide soap and sanitary materials to all participants. This provided a direct, tangible health benefit (promoting foot hygiene) relevant to the disease context. Regarding information sharing, we emphasize in the text that community engagement and sensitization were utilized not just for recruitment, but as critical mechanisms to build trust and provide study-related information to the community throughout the process.   Retention of samples\/banking: This may also be an area of great concern. It is troubling that respondents may not have a clear understanding of what this means and the importance be down played, since it is \u2018only\u2019 buccal swabs we\u2019re dealing with. What measures should be taken to improve this misunderstanding?  \n<b>Response: <\/b>We agree that the non-invasive nature of saliva collection risks masking the significance of the biological data. In the revised Discussion, we explicitly address this by noting that the preference for saliva often stemmed from a misconception that it is &quot;less significant&quot; data than blood. To address this, we argue that the consent process must explicitly clarify that genetic data derived from saliva carries the same privacy and sensitivity implications as blood, ensuring participants understand that the resulting genomic data remains identical regardless of the collection method.   Discussion on the REA, its use and limitations should be expanded.  \n<b>Response: <\/b>We have expanded the discussion on the REA methodology as suggested. Regarding its utility, we added text explicitly detailing how the REA allowed us to identify &quot;invisible&quot; barriers\u2014specifically the linguistic conflation of research (\n<i>Meremer<\/i>) with police investigation (\n<i>Mermera<\/i>)\u2014which standard ethical reviews might miss. Regarding limitations, we have added a new paragraph acknowledging that the &quot;rapid&quot; nature of the appraisal (4\u20136 weeks) necessitates a trade-off in anthropological depth compared to long-term ethnography. We also explicitly note the exclusion of traditional healers and the potential selection bias introduced by relying on government gatekeepers (\n<i>Kebele<\/i> leaders), providing a more balanced view of the methodology\u2019s constraints.   Those 20 page hardly comprehensible consent forms are problematic in all settings, more so in this context. Discuss methods of obviating this\u2026e.g., a shortened say 2 page summary dealing with the main issues, in plain language.  \n<b>Response: <\/b>We agree that lengthy, legalistic consent forms are particularly problematic in this context. In the revised Discussion, we address this by advocating for a &quot;layered approach&quot; to information provision. We argue that instead of a standard comprehensive form\u2014which our findings suggest may signal a &quot;lack of confidence&quot; to the community\u2014the process should begin with the practical medical information prioritized by the community (cause and prevention of podoconiosis). This acts as a bridge to explaining abstract genomic concepts using the local analogies identified in our results, effectively serving the function of a simplified summary.","URL":"https:\/\/wellcomeopenresearch.org\/articles\/2-99\/v2#referee-comment-6437","order":4,"name":"referee-comment-6437","label":"Referee Comment","group":{"name":"article-reports","label":"Article Reports"}},{"value":"This work was supported by the Wellcome Trust [100715] (Wellcome Trust Brighton and Sussex Centre for Global Health Research); the UK Medical Research Council [MR\/J008621\/] and from core budget of the Armauer Hansen Research Institute. The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.","order":5,"name":"grant-information","label":"Grant Information"},{"value":"This is an open access article distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.","order":0,"name":"copyright-info","label":"Copyright"}]}}