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Abstract

Objective

We strive to analyze Cleft Lip and Palate repair (CLP) from low-and-middle income countries (LMICs) and understand authorship patterns between different countries.

Design

Five high-impact journals were selected for analysis from 2008 to 2024. Articles were initially screened to determine if the study focused on a LMIC according to the World Health Organization and if the study's subject was CLP.

Setting

Our study involved reviewing papers from multiple levels of clinical care.

Patients, Participants

We categorized the selected studies by their stated diagnoses (cleft lip, cleft palate, CLP, velopharyngeal insufficiency, and other).

Interventions

Specific interventions were not recorded due to the nature of the study.

Main Outcome Measure(s)

Nationality and gender of first author, nationality and gender of senior author, and country income status (based on World Health Organization classification).

Results

Most first authors were male (54%) and from upper-middle-income countries (69%). Kappa scores showed significant agreement for country income classification and the income classification of the country of the first author (K = 0.779, P < .01). There was minimal change in the representation of LMICs over the study's time period (b = −0.0017, SE = 0.008, P = .8429). There were 9 low-income country articles but only 1 (11%) had first author from the represented country.

Conclusions

Overall, authors from middle-income countries are well-represented in CLP articles from their countries, but not the case for low-income country authors. Continued scrutiny of equitable publication should be conducted as low-income countries continue to be underrepresented in the literature.

Keywords

cleft lip and palate global health authorship

Introduction

Nine of 10 children with a serious congenital disorder are born in low- and middle-income countries (LMIC).¹ Orofacial clefts are among the most common congenital disorders worldwide, especially in LMIC where approximately 1 in every 730 children are born with a cleft lip and/or palate (CLP).² Cleft lip and palate significantly impacts health, development, and quality of life of affected children and their families. For example, children with CLP are at increased risk of malnutrition and middle ear dysfunction.^3,4 Children with CLPs are also at increased risk for learning difficulties, discrimination, emotional adjustment problems, and self-esteem due to facial disfigurement and speech difficulties.^5,6

Children with CLP can have CLP repairs, which are relatively safe surgeries with short postoperative recovery periods, making them a common surgical procedure completed globally. Surgeons from other countries may travel to underresourced areas to complete surgeries and mentor other physicians through the procedure. Theoretically, given the mixing of physicians from different countries, there are many opportunities for cleft and lip palate research to occur. However, authorship trends in CLP research occurring in LMIC are not well understood.

In general, authors from LMIC produce less research compared to authors from high-income countries.^7,8 Low-and-middle income countries authorship represented only 35% of articles published in the Lancet Global Health between 2013 and 2017.⁹ Thus, our study sought to understand authorship and gender patterns in CLP articles from LMICs and compare our findings to what is known about global research as a whole. A better understanding of the publishing trends of authors from LMICs studying CLP can highlight existing disparities and recognize differences between LMIC countries.

Methods

This study was approved for Institutional Review Board Review by the Vanderbilt Institutional Review Board.

Data Collection

Five high-impact journals were selected for analysis. They included Facial Plastics & Aesthetic Medicine journal, The Journal of Craniofacial Surgery, The Cleft Palate and Craniofacial Journal, Facial Plastic Surgery Journal, and Plastic and Reconstructive Surgery. All published articles between 2008 and 2024 within these issues were reviewed for inclusion through PubMed database. All original research articles, reviews, and case reports were included. Editorials and letters were excluded from the study.

Articles were initially screened to determine if the study focused on an LMIC according to the World Health Organization (WHO) and if the study's subject was CLP. Articles from HIC or articles that were not focused on CLP were excluded, which is depicted in Figure 1.

Figure 1.

Preferred Reporting Items for Systematic reviews and Meta-Analyses (PRISMA) flow diagram depicting the number of records identified, included, and excluded, and the reasons for exclusions.

Variables of Interest

For each article that met the inclusion criteria, data were collected on: title of the paper; DOI; year published; type of study (prospective, retrospective, case series, device, cross-sectional, other); subject of study (cleft lip, cleft palate, CLP, velopharyngeal insufficiency, other); country that the paper was about; the continent of the country; WHO classification of the country (low-income, lower middle-income, upper middle-income); total number of authors; number of authors from the country; the country that the first author and its WHO classification; the gender of the first author (female, male, other, unsure); the country that the last author and its WHO classification; the gender of the last author; and if there were organizations involved in the study (Smile Train, Operation Smile, Others).

An author was determined to be from a country based on the affiliation listed in the paper. If their affiliation was unclear, reviewers investigated internet search engines, institutional websites, and social media. The gender of the authors was determined by inspection of first name. If this was unclear, internet search engines, institutional websites, and social media were scanned to find photos or biographies.

Reproducibility

Articles were independently screened by 1 of 3 reviewers. All authors used a standardized data collection tool (RedCap) to collect data. Any discrepancies were resolved via group discussion with the lead author and principal investigator. The country's economic classification was checked using the WHO website after data collection. To ensure reproducibility, data collection was duplicated by a second reviewer at random.

Statistical Analysis

Univariate linear regression analysis was conducted to understand changes in the representation of LMICs and female first authors by year of publication. Additionally, the kappa statistic was calculated to understand the agreement between the country income classification and the income classification of the (1) first author and (2) senior author. Statistical analysis was performed using R (R Studio, V.2023.06.2). 132 Statistical significance was set at P < .05.

Results

A total of 1824 articles were found using our PubMed search strategy. These articles underwent screening of the titles and abstracts, and those from HIC or with topics unrelated to CLP were excluded. Of those studies, 576 were determined to meet the inclusion criteria and underwent full-text studies assessment for eligibility. Ultimately, data collection and analysis was completed for 390 studies. This breakdown is depicted in Figure 1.

Out of the journals selected, 1247 articles were published from HIC, compared to only 390 from LMIC. Studies were published between 2008 and 2024, with most articles published in 2021 (14%). The change in Otolaryngology papers over time is shown in Figure 2. Studies were classified as retrospective (N = 148, 37.9%), prospective (N = 141, 36.2%), case series (N = 61, 15.6%), or cross-sectional (N = 21, 5.38%), depicted in Figure 3A. Articles that did not fall under those categories were classified as “other” and included technique papers, commentaries, and technical notes. The primary subject of studies was CLP (N = 211, 54.1%) followed by cleft lip (N = 85, 21.8%), cleft palate (N = 74, 19.0%), other (N = 10, 2.56%), and velopharyngeal insufficiency (N = 9, 2.31%), depicted in Figure 3B.

Figure 2.

Annual number of indexed cleft lip and palate (CLP) publications from low-and-middle income countries (LMICs) (2008-2024).

Figure 3.

(A) Pie chart displaying number of articles per category of study. (B) Additional pie chart displaying number of article per subject.

Articles included in the study were from 6 continents and 37 countries. Most publications were from Asia (244, 62.6%) followed by South America (87, 22.3%) and Africa (45, 11.5%), shown in Figure 4A. China published the most articles, producing 110 articles (28.2%) between 2008 and 2024. Brazil published the second most, producing 57 articles (14.6%). India followed Brazil, which published 53 articles (13.6%). The breakdown of these countries is shown in Table 1.

Figure 4.

Pie charts displaying number of (A) articles from each continent. Additional pie charts classify (B) publication country, (C) first author, and (D) senior author in low-and-middle income countries (LMICs).

Table 1.

The Breakdown of the 39 Countries That Were Involved in our Study, the Number of Articles, and WHO Classification for Each Country (Low-Income, Lower Middle-Income, Upper Middle-Income).

Country	# of articles	WHO classification
Argentina	4	Upper Middle
Bangladesh	1	Lower middle
Bolivia, Madagascar, Vietnam, and Morocco	1	Lower middle
Brazil	57	Upper Middle
China	108	Upper Middle
China, Yemen	1	Upper Middle
Colombia	7	Upper Middle
Democratic Republic of the Congo	1	Low Income
Ecuador	2	Upper Middle
Ecuador/China	1	Upper Middle
Egypt	16	Lower middle
El Salvador	2	Upper Middle
Ethiopia	1	Low Income
Ghana	1	Lower Middle
Guatemala	1	Upper Middle
Haiti	1	Lower Middle
India	53	Lower Middle
Indonesia	9	Upper Middle
Iran	18	Upper Middle
Iraq	1	Upper Middle
Kenya	1	Lower Middle
Lebanon	4	Lower Middle
Malawi	1	Low Income
Malaysia	3	Upper Middle
Mali	1	Low Income
Mexico	3	Upper Middle
Morocco	1	Lower middle
Morocco, Bolivia, Vietnam, and Madagascar	2	Lower middle
Nepal	2	Lower middle
Nicaragua	1	Lower middle
Nigeria	11	Lower middle
Pakistan	2	Lower middle
Peru	13	Upper Middle
Peru, Equador	1	Upper Middle
Philippines	1	Lower middle
South Africa	5	Upper Middle
Sudan	2	Low Income
Syria	1	Low Income
Tanzania	1	Lower middle
Thailand	7	Upper Middle
Thailand	1	Upper Middle
Turkey	33	Upper Middle
Uganda	2	Low Income
Vietnam	5	Lower middle

Abbreviation: WHO, World Health Organization.

Regarding the WHO classifications, 278 (71.3%) of the papers came from upper-middle countries such as China, Brazil, and Turkey; 102 (26.2%) papers came from low-middle-income countries such as Egypt, India, and Nigeria; 10 (2.56%) articles were published in low-income countries such as Ethiopia, Sudan, and Uganda shown in Figure 4B.

Most first authors were from upper-middle countries (N = 266, 68.2%), depicted in Figure 4C. The first authors for 80 articles came from lower-middle-countries (20.5%). The first authors for 43 articles came from high-income countries (11.0%). Out of the 11 articles from low-income countries, only 1 article (9.09%) had a first author from a low-income country. Most articles from low-income countries had first authors from high-income countries (10, 90.9%).

The senior authors came from upper-middle-income countries 67.2% (N = 262) of the time, depicted in Figure 4D. The senior authors for 75 articles came from lower-middle-countries (19.2%). The senior authors for 50 articles came from high-income countries (12.8%). There were 3 articles out of 11 (27.3%) from a low-income country that had a senior author from a low-income country—the other 8 (72.7%) senior authors came from high-income countries. Kappa scores showed significant agreement for country income classification and the income classification of the country of the first author (K = 0.779, P < .01) and senior author (K = 0.747, P < .01). There was minimal change in the representation of LMIC over the study's period (b = −0.0017, SE = 0.008, P = .8429), shown in Figure 5A. Most first authors were male (62.6%). Most senior authors were also male (63.2%). However, there was an increase in the percentage of female first authors from 33% in 2008 to 50% in 2024 (b = 0.016, SE = 0.007, P = .0392), shown in Figure 5B.

Figure 5.

Trends in authorship representation over the study period. (A) Representation of authors from low-and-middle income countries (LMICs) remained relatively stable from 2008 to 2024 (B) Percentage of female first authors increased over the same period.

Forty-three articles were involved with an organization—13 (32.5%) of which were associated with Smile Train, 17 (42.5%) of which were associated with Operation Smile, 2 (5%) of which were associated with Global Smile, and the other 11 (26.8%) with other organizations. Articles were focused on upper-middle-income countries 224 (71.3%) followed by low-middle-income countries 103 (26.4%) and low-income countries 11 (2.82%). In articles involving an organization, most first authors came from high-income countries (23 articles, 56.1%), especially the United States (20 articles, 48.8%). The same was true for senior authors, where 25 (61.0%) articles had senior authors from high-income countries. In 10 (24.4%) studies, the first author came from an LMIC. 8 (19.5%) studies had the principal author come from an LMIC.

Discussion

Our systematic review examines authorship trends in CLP publications originating from LMICs. Our findings reveal that most CLP research originates from a few upper-middle-income countries, with China alone accounting for 100 articles (28%). Notably, we demonstrated that most CLP publications and first and senior authors are concentrated in upper-middle-income countries.¹⁰ Representation from low-income countries in CLP research is minimal, reflecting a broader trend in global research.^8,11 Among the 9 articles from low-income countries in our study, 7 last authors were researchers from high-income countries. Our study in CLP showed no significant changes in the proportion of articles from LMICs or the rate of LMIC authorship over the study period.

Investigations into otolaryngology research, regardless of subspecialty, in LMICs have also shown no authorship trends over a similar period, though studies solely focusing on facial plastic reconstructive surgery subspecialty of otolaryngology did show an increase in the past several decades.^7,12 A number of factors may cause disparities seen in our research and global research overall. The number of biomedical and clinical researchers in LMIC is disproportionately low, especially since there are many scientists who emigrate out of LMICs for education and employment opportunities.^13,14 Low-and-middle income countries scientists who remain have less institutional support, fewer training grants focused on building research mentorship, and fewer senior mentors to mentees.^15,16 Other factors may include the marginalization of underrepresented patient communities, language barriers, limited research training and infrastructure, competing clinical demands, and funding limitations.^7,17 These factors restrict the scope and number of academic projects, contributing to a negative cycle where authors from LMIC publish fewer articles, receive less recognition in academic medicine, and therefore are less funded.^18,19

Several studies have proposed solutions to the disparities seen in global research. Since articles written in “non-native” English face issues related to grammar or syntax, journals can provide and advertise low-cost services in proofreading or translating submitted manuscripts with authors from non-English-speaking countries, including artificial intelligence programs (with the proper disclosures).^20,21 When done correctly, cross-national or intercountry collaborations can further be useful.^22,23 Other solutions include increasing LMIC representation in the editorial board and reviewer pool, advocating for an alternative to authorship guidelines from the International Committee of Medical Journal Editors when research involves LMIC, and reducing costs to open-access (OA) articles.^24–26

Open-access articles are a key area of improvement since one of the main barriers to research activities is the lack of information resources.^27–30 Half of the publications in OA are not legally accessible to be read by users in LMIC without institutional license or publishers’ fees.³¹ Some journals, including JAMA-Otolaryngology Head and Neck Surgery and The Laryngoscope, have waived the article publication charges for authors from LMIC.³⁰ Of the journals we investigated in our study, most did not offer a waiver. This effort could be strengthened by requiring funding recipients to share their research findings in some form within low-income countries.

In addition to support from journals, other organizations involved in research can promote the growth and development of local providers and surgeons. Smile Train, for example, provides training, funding, and resources to empower local medical professionals.³¹ Operation Smile works with local healthcare providers, hospitals, and governments to create sustainable change. Our study showed that in articles involved with an organization, 65% of first authors and 74% of senior authors came from HIC. Organizations such as Smile Train and Operation Smile represent an opportunity to support local providers not only in healthcare training but also in research. Since these organizations have established foundations and offices in LMIC and are providing innovative care that relies on relationships with local community members, there are many opportunities for CLP research to occur and local physicians to participate in authorship, partly due to the credibility and established publication track record of their institutions. Establishing a positive cycle—where research is shared with the rest of the world, used to inform evidence-based policies and plans, and then effectively implemented—has the potential to improve global health outcomes.

In addition to authors from LMIC, female authors have faced similar barriers to publishing in high-impact journals, including less mentorship, less research funding, fewer submissions and acceptances, fewer citations, and overall, less recognition in academic settings.^32–34 One study that investigated female trends in high-impact otolaryngology journals found that female first authorship increased to 31.5% and female senior first authorship remained at 18.4% between 2011 and 2020.⁸ Our study found a greater percentage of female first authors (46%) and female senior authors (46%) in CLP research from LMIC, with an increase in the number of female authors. In HIC, the rise in first authorship corresponds with an increase in the number of female otolaryngologists, including trainees, residents, and attendings.^35,36 Female otolaryngologists match their male colleagues more closely in research output in certain subspecialties, which may lead to more recognition in academic settings and a greater number of invitations to be peer reviewers or editors.³⁷ These positive steps that have increased female authorship representation demonstrate how authors from LMIC can be supported—by supporting the training of more otolaryngologists from LMIC and removing barriers to receiving mentorship, funding, and journal article acceptances.

Authorship carries significant academic, social, and financial implications, especially for individuals listed as the first, second, and last authors.³⁸ Inclusion of local authors adds to fairness, context, and implications of research, especially when the authors come from LMICs.³⁹ If most articles about LMIC are led by authors from HIC, how can we be sure that the papers really answer the priorities for local partners?^40,41 The hope is that if local perspectives are better represented in the global health literature, there will be an increase in local funding and transfer of power to local structures.⁴² There are success stories when collaborations between authors from HIC and LMICs improve patient care. For example, a study in Nigeria highlighted the need to evaluate hearing-impaired children and suggested a comprehensive hearing screening program.⁴³ All authors were from Nigeria and acknowledged contributors from Edinburgh for their assistance while preparing the manuscript. Another example is studies examining the increased incidence of specific genetic mutations in patients with breast cancer in the Caribbean allowed for new screening protocols for oncogenic gene mutations to be put into place, allowing patients with said mutations to benefit from updated screening protocols and preventative measures.^44,45 Research trends in CLP care are shifting from trip reports to outcome-based studies, emphasizing sustainable, long-term care—an area where local authors can play a key role in future research.⁴⁶

Our study is subject to several limitations. We limited our search to top 5 journal articles, excluding lower-impact journals and potentially missing certain author trends in those journals. As data collection was performed manually by multiple collaborators, there is room for error in the application of our criteria, regarding the detection of studies, the inclusion criteria, and the data recording. In particular, the methodology used to determine the nationality of authors is based on either listing from personal webpages or profiles found on the webpages of their institutions. Although a great deal of care was taken to ensure the accuracy of the nationalities, there may be some variance due to external factors—such as authors having a profile from an institution in a higher-middle-income country, despite originating from a lower-income country. We also excluded articles that were written in languages other than English. There could be other articles not included in this study from LMICs that could affect our analysis and findings.

Conclusion

In conclusion, our paper is among the first to examine trends in LMIC and female authorship within CLP research. Our findings reveal a scarcity of articles focused on low-income countries and a frequent involvement of authors from high-income countries in research originating from these regions. Ongoing work is needed to address these inequities, whether by reducing or waiving costs to OA or asking organizations such as Smile Train and Operation Smile to prioritize local authorship in research collaborations.

Footnotes

Acknowledgments

The authors would like to thank Camille Ivey and Rachel Walden from the Annette and Irwin Eskind Family Biomedical Library and Learning Center, Vanderbilt University, Nashville, TN, for their expertise and assistance with the development of our search strategies.

ORCID iDs

Ina Lee

Ryan Belcher

Ethical Approval and Informed Consent Statements

This study received ethical approval from the 240499 IRB on April 25, 2024. This is an IRB-approved retrospective study, all patient information was de-identified and patient consent was not required.

Funding

The author(s) received no financial support for the research, authorship, and/or publication of this article.

Declaration of Conflicting Interests

The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

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Cleft Lip and Palate Publishing Trends From Authors in Low- and Middle-Income Countries in Cleft and Craniofacial Journals

Abstract

Objective

Design

Setting

Patients, Participants

Interventions

Main Outcome Measure(s)

Results

Conclusions

Keywords

Introduction

Methods

Data Collection

Variables of Interest

Reproducibility

Statistical Analysis

Results

Discussion

Conclusion

Footnotes

Acknowledgments

ORCID iDs

Ethical Approval and Informed Consent Statements

Funding

Declaration of Conflicting Interests

References