Piloting a Spanish-Language Web-Based Tool for Hereditary Cancer Genetic Testing
Round 1
Reviewer 1 Report
The authors present a study examining a web-based educational tool for Spanish-language patients to educate them on hereditary cancer. They then examine patients understanding of hereditary cancer for knowledge, attitudes and decisional empowerment.
The study is well-written and clear, and adds to the body of literature surrounding inclusivity in genetic care.
A few minor comments.
1. What is the web-based tool? Is there a URL?
2. There are many Spanish resources for genetics educations in the web (videos, pamphlets), and published upon. The authors should review those resources, and comment on how their study is unique and superior to a video and pamphlet.
3. The authors should highlight the importance of Spanish in a global context, rather than continental US and Puerto Rico. After all, Spanish is the second most spoken language in the world.
4. One of the limitations would be that the web-based tool was designed in English, and then translated to Spanish. To incorporate any culturally nuanced aspects of a non-English person, the design of the web-based tool would have involved the patient population it was geared towards. This could be a future step.
5. The responses of patients are translated into English and having the raw Spanish text may be beneficial for the scientific community (this can be as a supplement).
Author Response
We appreciate the comments from reviewers and have revised the manuscript. The response to reviewer 1 feedback is itemized below and included in the tracked and clean manuscript versions.
Reviewer 1 Comments:
The authors present a study examining a web-based educational tool for Spanish-language patients to educate them on hereditary cancer. They then examine patients understanding of hereditary cancer for knowledge, attitudes and decisional empowerment.
The study is well-written and clear, and adds to the body of literature surrounding inclusivity in genetic care.
RESPONSE: Thank you for the positive feedback.
A few minor comments:
- What is the web-based tool? Is there a URL?
RESPONSE: This is a tool that is still under development and testing in the research setting, thus we are not making it accessible through a public link/URL.
- There are many Spanish resources for genetics educations in the web (videos, pamphlets), and published upon. The authors should review those resources, and comment on how their study is unique and superior to a video and pamphlet.
RESPONSE: Based on our review of the literature, there are no Spanish language audio/visual tools that have been evaluated (and published on) that cover the specific educational elements to be discussed during a genetic counseling session, per the American Society of Clinical Oncology (ASCO) guidelines. Therefore, our tool meets a currently unaddressed resource for Spanish speakers. There are some that are designed to increase genetic counseling and testing uptake and there was one effort to culturally adapt a telephone genetic counseling visual aid booklet (specific to breast cancer survivors). We know people have developed other materials that may be similar but we could not find results published from anyone testing the efficacy of other GC education tools designed for the pre-test education component. We cannot say anything about our tool being superior to a video and pamphlet because we have done no comparisons with other content that is out there. We have clarified this in the discussion.
“While a few prior Spanish language educational tools focused on hereditary cancer have previously been evaluated in published literature [15, 16], none to date have tested the efficacy of a Spanish-language tool that is intended to cover pre-test education as we have.”
- The authors should highlight the importance of Spanish in a global context, rather than continental US and Puerto Rico. After all, Spanish is the second most spoken language in the world.
RESPONSE: This is an excellent point, and we have added the following to the discussion:
“Although this tool was tested in the continental U.S. and Puerto Rico, the potential impact of this tool is much broader given that Spanish is one of the most commonly spoken languages in the world.”
- One of the limitations would be that the web-based tool was designed in English, and then translated to Spanish. To incorporate any culturally nuanced aspects of a non-English person, the design of the web-based tool would have involved the patient population it was geared towards. This could be a future step.
RESPONSE: We agree with the reviewer that this is a limitation. Nevertheless, the key educational elements included those that ASCO recommends being discussed during a genetic counseling session for inherited cancer, and these educational elements would not necessarily be culture dependent. We have added the following to address this limitation:
“While this tool was not originally developed by native Spanish speakers and did not incorporate any cultural nuances, the information contained in this tool is based on American Society of Clinical Oncology guidelines for content to discuss prior to genetic testing for hereditary cancer. Furthermore, the tool was translated by a native Spanish speaker and was acceptable to the Spanish speakers in this study. Future research could further assess cultural acceptability among a broader range of Spanish speakers.”
- The responses of patients are translated into English and having the raw Spanish text may be beneficial for the scientific community (this can be as a supplement).
RESPONSE: We have included versions in an appendix with the Spanish text.
Reviewer 2 Report
Thank you for the opportunity to review this important manuscript that presents adaptation of an online educational tool for genetic testing in Spanish. Given that Spanish is spoken in many parts of the world and the lack of Spanish-speaking genetic counselors, especially in the US, the online tool can be a significant addition to clinical practice.
However, some issues need to be improved/ clarified before the manuscript can be considered for publication.
1) Introduction: Lines 63-71. It is not clear whether the tool is specific for HBOC, Lynch syndrome, or other types of hereditary cancer. The authors need to provide more information about the content/ areas covered by the tool so that the reader would not necessarily have to look for the original publication. Adding a figure presenting some of the content, options about navigation or technical requirements - e.g., is this a YouTube video? would be helpful for the reader.
Participant Recruitment: Lines 73 onward. Participants could have had any type of cancer - this is confusing, since the reader is not clear about the specificity of the tool. In addition, did any of the participants with cancer received a recommendation for genetic testing to confirm diagnosis or guide treatment? This is important information that is currently missing from the manuscript. Table 1 presents that 11 individuals had genetic testing but it is not clearly stated whether they had testing prior to or after viewing the tool.
Pre- and post-intervention measures: Please provide examples of items from each scale. Clarify if they are Likert scales or not and the scoring options (e.g., 1-4 or 1-5, etc.). Is there any information about the Cronbach's alpha of these scales in Spanish? This is information that can easily be added to the manuscript.
Qualitative interviews: The authors provided the 10 participants who agreed to provide narrative data with hypothetical scenarios of receiving a positive/negative/VUS test result. Can the authors please explain the rational for providing these hypothetical scenarios, as this appears to be unrelated to testing the acceptability and usability of the tool.
Results: Approximately 37% of participants completed the tool and both the pre- and post-intervention surveys. This is large attrition rate and the authors should provide some comparisons between those who completed the entire study and those who started the pre-test survey but withdrew (passively or actively). Were there any systematic differences in these two groups?
Lines 170 - 184 present the same information twice - a copy/paste mistake that escaped the attention of the authors.
Discussion: Line 213 - replace "form" with "from"
Author Response
We appreciate the comments from reviewers and have revised the manuscript. The response to reviewer feedback is itemized below and included in the tracked and clean manuscript versions.
Reviewer 2 Comments:
Thank you for the opportunity to review this important manuscript that presents adaptation of an online educational tool for genetic testing in Spanish. Given that Spanish is spoken in many parts of the world and the lack of Spanish-speaking genetic counselors, especially in the US, the online tool can be a significant addition to clinical practice.
However, some issues need to be improved/ clarified before the manuscript can be considered for publication.
1) Introduction: Lines 63-71. It is not clear whether the tool is specific for HBOC, Lynch syndrome, or other types of hereditary cancer. The authors need to provide more information about the content/ areas covered by the tool so that the reader would not necessarily have to look for the original publication. Adding a figure presenting some of the content, options about navigation or technical requirements - e.g., is this a YouTube video? would be helpful for the reader.
RESPONSE: This tool is focused on hereditary cancers in general, and not specific to HBOC, Lynch, etc. We have added additional detail describing the tool within the article (text shown below). This is a tool that is still undergoing testing and refinement in the research setting, thus we are not making it accessible through a public link/URL. Additional details related to the English version of the tool have been published previously (Tezak et al. 2022).
Tezak AL, Zuniga B, Weidner A, Cragun D, Pal T. Qualitative Methods for Refining a Web-Based Educational Tool for Patients Focused on Inherited Cancer Predisposition. J Cancer Educ. 2022 Aug;37(4):1129-1136. PMID: 33400205.
“The tool is focused on hereditary cancers in general and includes 12 core (mandatory) slides with animation and audio focused on inheritance, cancer risks, types of test results, how results may or may not change medical management, testing benefits/limitations, insurance considerations, and the genetic counseling and testing process. There are an additional 22 ‘optional’ slides with detailed content that relates to or expands on core content. Examples of additional content include an explanation of the difference between tumor versus germline testing, the concept of ‘best testable family member’, and gene-specific content’ on 17 inherited cancer-predisposing genes. Individuals can navigate the tool at their own pace using arrows to proceed to the next slides, go back to prior content or click on the ‘learn more’ content.”
- Participant Recruitment: Lines 73 onward. Participants could have had any type of cancer - this is confusing, since the reader is not clear about the specificity of the tool. In addition, did any of the participants with cancer received a recommendation for genetic testing to confirm diagnosis or guide treatment? This is important information that is currently missing from the manuscript. Table 1 presents that 11 individuals had genetic testing but it is not clearly stated whether they had testing prior to or after viewing the tool.
RESPONSE: We clarified above that the educational tool was geared toward genetic testing for hereditary cancers in general per changes described in prior response. Our efforts were focused on evaluating the educational tool, and not focused on collecting comprehensive personal and family histories, which are needed to determine whether someone is eligible for genetic testing. Furthermore, it was out of scope of our efforts to collect comprehensive data about testing, and timing of testing. Given this was beyond the scope of our efforts, we have added the following to the limitations section:
“Given our efforts were focused on evaluating our tool, comprehensive personal and family history information to determine indications for testing was not obtained.”
- Pre- and post-intervention measures: Please provide examples of items from each scale. Clarify if they are Likert scales or not and the scoring options (e.g., 1-4 or 1-5, etc.). Is there any information about the Cronbach's alpha of these scales in Spanish? This is information that can easily be added to the manuscript.
RESPONSE: In the methods we have included more specifics on the content and type of items. We calculate the Cronbach alpha from the pre-survey responses from the current Spanish speaking population in our study. This made sense to include given that it is intended to be a unidimensional scale measure. Given that the SURE and the knowledge measures are really index measures (i.e., they combine different aspects of decisional conflict or different knowledge concepts, respectively), we did not calculate a Cronbach alpha on these as they are not intended to be unidimensional and they are not Likert-type items. See section 2.3 in the manuscript.
- Qualitative interviews: The authors provided the 10 participants who agreed to provide narrative data with hypothetical scenarios of receiving a positive/negative/VUS test result. Can the authors please explain the rational for providing these hypothetical scenarios, as this appears to be unrelated to testing the acceptability and usability of the tool.
RESPONSE: This is a valid point, thus we have added in the following to the manuscript to describe why we asked what they would think if they got the three different types of test results:
“Given that one of the primary goals of the tool was to prepare individuals to receive results of genetic testing, we asked participants how they would feel about the different types of test results for the purpose of assessing participants' understanding of the results and whether they were able to accurately apply that understanding to their own situation.”
- Results: Approximately 37% of participants completed viewing the tool and both the pre- and post-intervention surveys. This is large attrition rate and the authors should provide some comparisons between those who completed the entire study and those who started the pre-test survey but withdrew (passively or actively). Were there any systematic differences in these two groups?
RESPONSE: We have the following text to better describe the attrition and results of statistical comparisons we made with those who did not complete the study.
“The pre-tool survey was accessed 109 times, but only 65 unique individuals completed any questions. Of those 65, 41 (63%) also reviewed the tool and completed the post-tool survey. There were no statistically significant differences between those who did and did not complete the post-tool survey in terms of demographics, clinical information, or baseline scores. Table 1 includes demographic and clinical data for the 41 completers.”
- Lines 170 - 184 present the same information twice - a copy/paste mistake that escaped the attention of the authors.
RESPONSE: We have corrected this error
- Discussion: Line 213 - replace "form" with "from"
RESPONSE: This typo has been corrected.
Round 2
Reviewer 1 Report
the authors have addressed my comments
Reviewer 2 Report
the authors addressed this Reviewer's comments.
