Editorial
So Very Much to Learn
D
Nancy C. Chescheir, MD
See related article on page 506.
Dr. Chescheir is the Editor-in-Chief of Obstetrics & Gynecology; e-mail: [email protected]. Financial Disclosure The author did not report any potential conflicts of interest. © 2014 by The American College of Obstetricians and Gynecologists. Published by Lippincott Williams & Wilkins. ISSN: 0029-7844/14
VOL. 123, NO. 3, MARCH 2014
uring our weekly editorial meeting, Dwight Rouse, Bill Hurd, Tom Riggs, the editorial staff, and I discuss the peer reviews and revisions for papers being evaluated for acceptance in Obstetrics & Gynecology. I look forward to these meetings, as I always learn something from my partners and from the work we are considering for publication. Conversations focus on whether we believe the topic is appropriate for the journal and of interest to you, research methodology and analysis, ways to help the authors focus their papers for increased clarity, and research and publication ethical issues. An article in this month’s issue by Hurst et al (see page 506) evoked a lot of conversation because of its relatively novel (for us) research methodology.1 I wanted to write an editorial about this article, because web-based surveys are interesting and challenge some truths about surveys as research tools and because this topic illustrates how research can help us take better care of patients. Depending on how one views the world, research questions can be divided into different types. Sue and Ritter, in their book Conducting Online Surveys,2 divide research into three broad areas: exploratory, descriptive, and explanatory. In exploratory research, the investigator seeks to clarify concepts and form hypotheses by seeking answers from nonrandom samples of individuals knowledgeable about the topic. In contrast, descriptive research is generally not hypothesis-driven but does tend to have at least one guiding research question. Results from studying samples of populations describe characteristics of populations either qualitatively or quantitatively. Lastly, explanatory research is hypothesis-driven, and investigators of these types of studies want to explain why things happen, whether they are likely to occur in the future, or whether there are treatments to alter the course of a disease. Techniques include probability sampling, and results can be more or less generalized from sampled populations. Our journal publishes all three types of research studies. Hypothesisdriven or explanatory research seems to be the most highly valued, oftentimes using the holy grail of randomized clinical trial methodology to answer the question. There is a role, however, for descriptive and exploratory research, especially when trying to understand problems of individuals with rare or unusual diseases. Ehlers-Danlos is considered by the National Institutes of Health as a rare disease, occurring in about 1 of every 5,000 individuals.3 Very few obstetrician–gynecologists will care for a large number of women with this disorder. Given its rarity, what is the best methodology to understand the unique concerns of women with this disorder? Hurst et al opted to perform a descriptive study and recruited visitors to the Ehlers-Danlos National Foundation web site (http://www.ednf.org) to complete a web-based survey regarding their reproductive health. Now what you—the reader—need to consider is whether you think the data these authors report are valid.
OBSTETRICS & GYNECOLOGY
497
The history of surveys is interesting and suggests that web-based surveys are a logical progression in methodology.4 Census results commonly informed nation states when populations were small. As an example, Figure 1 is a picture of a quipus, a decimal-based colored knot system that recorded information at the Incan tribal level such as numbers of people, houses, llamas, and men eligible to be conscripted into the army. A quipus then was carried to the capital for national compilation until the Incan empire collapsed in about 1500 AD. Fast-forward to 1895, when a Norwegian statistician, Anders Kiaer, published a game-changing book on sampling methodology5 and then surveys were started. A century later, the web allowed for fast, cheap surveys of large numbers of people who may be geographically dispersed. For descriptive research on rare disorders, such surveys can provide useful information. On our editorial conference call, we discussed the validity of a web survey. We rarely publish survey data with less than a 60–70% response rate. How does one measure the response rate to this survey: is it the number of people who visited the web site, opened the link to the survey, or started the survey even if they did not complete it? The survey respondents selfreported their condition and type of Ehlers-Danlos, and we believed that is no different from concerns for survey respondents in face-to-face, mail, or telephone contact. What about duplicate respondents? At the end of the day, we believed it likely that individuals with Ehlers-Danlos who completed the survey were motivated to provide information that might help themselves and others with this disease and that
Fig. 1. Quipus—a decimal-based colored knot system that recorded information at the Incan tribal level such as numbers of people, houses, llamas, and men eligible to be conscripted into the army. Image courtesy of Museo Larco, Lima – Peru´. Used with permission. Chescheir. So Very Much to Learn. Obstet Gynecol 2014.
498
Chescheir
So Very Much to Learn
there would be few people who would choose to “vote early and often.” Were the respondents representative of people with Ehlers-Danlos? Perhaps not; respondents had to be aware of the Ehlers-Danlos National Foundation and its web site, had to have Internet connectivity, and had to be motivated to respond to the survey, so perhaps they had more significant problems than did nonrespondents. After the decision was made to accept the paper, believing that the authors had well-utilized a tool to provide descriptive information about a rare disease that seems to have significant effects on the reproductive health of women, I went to the EQUATOR Network web site (www.equator-net.org) and unexpectedly found the Checklist for Reporting Results of Internet E-Surveys (CHERRIES) statement on web-based surveys, published in the Journal of Medical Internet Research.6 Obstetrics & Gynecology does not require the use of this checklist as it does for others such as CONSORT (Consolidated Standards of Reporting Trials), PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses), and MOOSE (Meta-analysis Of Observational Studies in Epidemiology). To minimize the risks of bias in Internet surveys, the CHERRIES statement and checklist suggest reasonable requirements from the authors. Although there are checklist items not addressed by Hurst et al (in part because I was unaware of the existence of this checklist), I believe that this descriptive study does provide useful information to help clinicians ask the right questions and to plan appropriate counseling and preventive strategies for the care of pregnant and nonpregnant women with Ehlers-Danlos. As Editor-in-Chief of Obstetrics & Gynecology, I always learn something. The Hurst et al article1 taught me a lot about Ehlers-Danlos. I also learned that there are publication guidelines about reporting web-based survey studies and that sometimes there are no other reasonable ways to get useful information than a potentially flawed one, which must be done to high standards. I thank the authors of this article for their persistence in the publication process and the respondents to the survey for helping our community of women’s health providers to be better informed, and I pledge to continue to learn in this work. Cheers. REFERENCES 1. Hurst BS, Lange SS, Kullstam SM, Usadi RS, Matthews ML, Marshburn PB, et al. Obstetric and gynecologic challenges in women with Ehlers-Danlos syndrome. Obstet Gynecol 2014: 123:506–13. 2. Sue VM, Ritter LA. Conducting online surveys. 2nd ed. Thousand Oaks (CA): Sage Publications; 2012.
OBSTETRICS & GYNECOLOGY
3. National Library of Medicine. Genetics home reference: your guide to understanding genetic conditions. Ehlers-Danlos syndrome. Available at: http://ghr.nlm.nih.gov/condition/ehlersdanlos-syndrome. Retrieved December 13, 2013. 4. Bethlehem J, Biffignandi S. Handbook of web surveys. Hoboken (NJ): John Wiley and Sons; 2012.
5. Kiaer AN. Observations et Expériences Concernant des Dénombrements Représentatives. Bulletin of the International Statistical Institute, IX, Book 2. 1895. pp. 176–83. 6. Eysenbach G. Improving quality of Web surveys: the Checklist for reporting results of internet E-surveys (CHERRIES). J Med Internet Res 2004;6:e34.
Submitting a Clinical Trial? Register Your Trial in a Public Trials Registry Obstetrics & Gynecology complies with the International Committee of Medical Journal Editors (ICMJE) requirement that clinical trials be registered in a public trials registry at or before the time of first patient enrollment in order to be considered for publication.1–3 Registries approved by the ICMJE are ClinicalTrials.gov or any registry that is a primary register of the WHO International Clinical Trials Registry Platform (http://www.who.int/ictrp/network/primary/en/index.html). Provide the trial registry name and URL and the registration number at the end of the abstract. In the cover letter, the corresponding author must attest to registering the trial and that the protocol they are reporting to Obstetrics & Gynecology is identical to the posted trial and, if not, precisely where and why it varies. Any changes in protocol should also be discussed in the manuscript itself as well as documented on the trials registry web site. If you cannot provide this information and wish to be considered for an exception, please contact the editor directly via e-mail prior to submitting your manuscript ([email protected]). References 1. Schulz KF, Altman DG, Moher D, for the CONSORT Group. CONSORT 2010 statement: updated guidelines for reporting parallel group randomized trials. Obstet Gynecol 2010;115:1063–70. 2. DeAngelis CD, Drazen JM, Frizelle FA, Haug C, Hoey J, Horton R, et al. Clinical trial registration: a statement from the International Committee of Medical Journal Editors. JAMA 2004;292:1363–4. 3. International Committee of Medical Journal Editors. Frequently asked questions: questions about clinical trial registration. Available at: http://www.icmje.org/faq_clinical.html. Retrieved November 6, 2013. rev 3/2014
VOL. 123, NO. 3, MARCH 2014
Chescheir
So Very Much to Learn
499
So Very Much to Learn
D
Nancy C. Chescheir, MD
See related article on page 506.
Dr. Chescheir is the Editor-in-Chief of Obstetrics & Gynecology; e-mail: [email protected]. Financial Disclosure The author did not report any potential conflicts of interest. © 2014 by The American College of Obstetricians and Gynecologists. Published by Lippincott Williams & Wilkins. ISSN: 0029-7844/14
VOL. 123, NO. 3, MARCH 2014
uring our weekly editorial meeting, Dwight Rouse, Bill Hurd, Tom Riggs, the editorial staff, and I discuss the peer reviews and revisions for papers being evaluated for acceptance in Obstetrics & Gynecology. I look forward to these meetings, as I always learn something from my partners and from the work we are considering for publication. Conversations focus on whether we believe the topic is appropriate for the journal and of interest to you, research methodology and analysis, ways to help the authors focus their papers for increased clarity, and research and publication ethical issues. An article in this month’s issue by Hurst et al (see page 506) evoked a lot of conversation because of its relatively novel (for us) research methodology.1 I wanted to write an editorial about this article, because web-based surveys are interesting and challenge some truths about surveys as research tools and because this topic illustrates how research can help us take better care of patients. Depending on how one views the world, research questions can be divided into different types. Sue and Ritter, in their book Conducting Online Surveys,2 divide research into three broad areas: exploratory, descriptive, and explanatory. In exploratory research, the investigator seeks to clarify concepts and form hypotheses by seeking answers from nonrandom samples of individuals knowledgeable about the topic. In contrast, descriptive research is generally not hypothesis-driven but does tend to have at least one guiding research question. Results from studying samples of populations describe characteristics of populations either qualitatively or quantitatively. Lastly, explanatory research is hypothesis-driven, and investigators of these types of studies want to explain why things happen, whether they are likely to occur in the future, or whether there are treatments to alter the course of a disease. Techniques include probability sampling, and results can be more or less generalized from sampled populations. Our journal publishes all three types of research studies. Hypothesisdriven or explanatory research seems to be the most highly valued, oftentimes using the holy grail of randomized clinical trial methodology to answer the question. There is a role, however, for descriptive and exploratory research, especially when trying to understand problems of individuals with rare or unusual diseases. Ehlers-Danlos is considered by the National Institutes of Health as a rare disease, occurring in about 1 of every 5,000 individuals.3 Very few obstetrician–gynecologists will care for a large number of women with this disorder. Given its rarity, what is the best methodology to understand the unique concerns of women with this disorder? Hurst et al opted to perform a descriptive study and recruited visitors to the Ehlers-Danlos National Foundation web site (http://www.ednf.org) to complete a web-based survey regarding their reproductive health. Now what you—the reader—need to consider is whether you think the data these authors report are valid.
OBSTETRICS & GYNECOLOGY
497
The history of surveys is interesting and suggests that web-based surveys are a logical progression in methodology.4 Census results commonly informed nation states when populations were small. As an example, Figure 1 is a picture of a quipus, a decimal-based colored knot system that recorded information at the Incan tribal level such as numbers of people, houses, llamas, and men eligible to be conscripted into the army. A quipus then was carried to the capital for national compilation until the Incan empire collapsed in about 1500 AD. Fast-forward to 1895, when a Norwegian statistician, Anders Kiaer, published a game-changing book on sampling methodology5 and then surveys were started. A century later, the web allowed for fast, cheap surveys of large numbers of people who may be geographically dispersed. For descriptive research on rare disorders, such surveys can provide useful information. On our editorial conference call, we discussed the validity of a web survey. We rarely publish survey data with less than a 60–70% response rate. How does one measure the response rate to this survey: is it the number of people who visited the web site, opened the link to the survey, or started the survey even if they did not complete it? The survey respondents selfreported their condition and type of Ehlers-Danlos, and we believed that is no different from concerns for survey respondents in face-to-face, mail, or telephone contact. What about duplicate respondents? At the end of the day, we believed it likely that individuals with Ehlers-Danlos who completed the survey were motivated to provide information that might help themselves and others with this disease and that
Fig. 1. Quipus—a decimal-based colored knot system that recorded information at the Incan tribal level such as numbers of people, houses, llamas, and men eligible to be conscripted into the army. Image courtesy of Museo Larco, Lima – Peru´. Used with permission. Chescheir. So Very Much to Learn. Obstet Gynecol 2014.
498
Chescheir
So Very Much to Learn
there would be few people who would choose to “vote early and often.” Were the respondents representative of people with Ehlers-Danlos? Perhaps not; respondents had to be aware of the Ehlers-Danlos National Foundation and its web site, had to have Internet connectivity, and had to be motivated to respond to the survey, so perhaps they had more significant problems than did nonrespondents. After the decision was made to accept the paper, believing that the authors had well-utilized a tool to provide descriptive information about a rare disease that seems to have significant effects on the reproductive health of women, I went to the EQUATOR Network web site (www.equator-net.org) and unexpectedly found the Checklist for Reporting Results of Internet E-Surveys (CHERRIES) statement on web-based surveys, published in the Journal of Medical Internet Research.6 Obstetrics & Gynecology does not require the use of this checklist as it does for others such as CONSORT (Consolidated Standards of Reporting Trials), PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses), and MOOSE (Meta-analysis Of Observational Studies in Epidemiology). To minimize the risks of bias in Internet surveys, the CHERRIES statement and checklist suggest reasonable requirements from the authors. Although there are checklist items not addressed by Hurst et al (in part because I was unaware of the existence of this checklist), I believe that this descriptive study does provide useful information to help clinicians ask the right questions and to plan appropriate counseling and preventive strategies for the care of pregnant and nonpregnant women with Ehlers-Danlos. As Editor-in-Chief of Obstetrics & Gynecology, I always learn something. The Hurst et al article1 taught me a lot about Ehlers-Danlos. I also learned that there are publication guidelines about reporting web-based survey studies and that sometimes there are no other reasonable ways to get useful information than a potentially flawed one, which must be done to high standards. I thank the authors of this article for their persistence in the publication process and the respondents to the survey for helping our community of women’s health providers to be better informed, and I pledge to continue to learn in this work. Cheers. REFERENCES 1. Hurst BS, Lange SS, Kullstam SM, Usadi RS, Matthews ML, Marshburn PB, et al. Obstetric and gynecologic challenges in women with Ehlers-Danlos syndrome. Obstet Gynecol 2014: 123:506–13. 2. Sue VM, Ritter LA. Conducting online surveys. 2nd ed. Thousand Oaks (CA): Sage Publications; 2012.
OBSTETRICS & GYNECOLOGY
3. National Library of Medicine. Genetics home reference: your guide to understanding genetic conditions. Ehlers-Danlos syndrome. Available at: http://ghr.nlm.nih.gov/condition/ehlersdanlos-syndrome. Retrieved December 13, 2013. 4. Bethlehem J, Biffignandi S. Handbook of web surveys. Hoboken (NJ): John Wiley and Sons; 2012.
5. Kiaer AN. Observations et Expériences Concernant des Dénombrements Représentatives. Bulletin of the International Statistical Institute, IX, Book 2. 1895. pp. 176–83. 6. Eysenbach G. Improving quality of Web surveys: the Checklist for reporting results of internet E-surveys (CHERRIES). J Med Internet Res 2004;6:e34.
Submitting a Clinical Trial? Register Your Trial in a Public Trials Registry Obstetrics & Gynecology complies with the International Committee of Medical Journal Editors (ICMJE) requirement that clinical trials be registered in a public trials registry at or before the time of first patient enrollment in order to be considered for publication.1–3 Registries approved by the ICMJE are ClinicalTrials.gov or any registry that is a primary register of the WHO International Clinical Trials Registry Platform (http://www.who.int/ictrp/network/primary/en/index.html). Provide the trial registry name and URL and the registration number at the end of the abstract. In the cover letter, the corresponding author must attest to registering the trial and that the protocol they are reporting to Obstetrics & Gynecology is identical to the posted trial and, if not, precisely where and why it varies. Any changes in protocol should also be discussed in the manuscript itself as well as documented on the trials registry web site. If you cannot provide this information and wish to be considered for an exception, please contact the editor directly via e-mail prior to submitting your manuscript ([email protected]). References 1. Schulz KF, Altman DG, Moher D, for the CONSORT Group. CONSORT 2010 statement: updated guidelines for reporting parallel group randomized trials. Obstet Gynecol 2010;115:1063–70. 2. DeAngelis CD, Drazen JM, Frizelle FA, Haug C, Hoey J, Horton R, et al. Clinical trial registration: a statement from the International Committee of Medical Journal Editors. JAMA 2004;292:1363–4. 3. International Committee of Medical Journal Editors. Frequently asked questions: questions about clinical trial registration. Available at: http://www.icmje.org/faq_clinical.html. Retrieved November 6, 2013. rev 3/2014
VOL. 123, NO. 3, MARCH 2014
Chescheir
So Very Much to Learn
499
