Top 100 Most Influential Manuscripts in Congenital Abdominal Pediatric Surgery: A Bibliometric Analysis.

Bibliometric analysis identifies the most influential manuscripts that shape our understanding of various congenital gastrointestinal pathologies. This study sets out to analyse the 100 most cited manuscripts on congenital gastrointestinal surgery. The Thomson Reuters Web of Science database was searched for all manuscripts relevant to paediatric congenital gastrointestinal surgery. The number of citations, authorship, publication date, journal of publication, institution and country were recorded for each paper. Eigenfactor and impact factor rankings were obtained for each journal of publication. 971 eligible manuscripts were returned by the search. Within the top 100 manuscripts, the most cited paper (by Metkus et al.) investigated fetal diaphragmatic hernia and was cited 413 times. University of Indianapolis had the most publications (n=5) and Journal of Pediatric Surgery had the most citations (n=3231). The country with the most publications in top 100 were the USA (n=39). The most commonly published topics were diaphragmatic hernia (n=23), abdominal wall defect (n=20) and atresia (n=6). The most cited manuscripts include various topics with a primary focus on the aetiology, diagnosis, management and outcome of diaphragmatic hernia and abdominal wall defects. Additionally, by collecting the most influential works, this study serves to reference what makes a manuscript 'citable'. Copyright:

INTRODUCTION

Congenital disorders have contributed to early deaths and disabilities throughout history and are currently still responsible for approximately 600,000 deaths and 62 million disability-adjusted life years around the globe annually.[12] The advances made in the last several decades have greatly improved outcomes for many previously life-limiting defects, while surgical success rates continue to improve.[3] Congenital defects of the alimentary tract, abdominal wall, and urinary system together affect up to 1000/100,000 of all births and are often managed through abdominal surgeries.[4] These defects reportedly cause up to 1.1% of all neonatal deaths.[5] These disorders are typically caused by defective embryogenesis of the alimentary tract, or its surrounding structures, and encompass pathologies such as malrotation, atresia, stenosis, and congenital absence of the intestines. Hence, it is not unexpected that pediatric abdominal surgery accounts for 31% of all pediatric operations.[6] With the rising prevalence of abdominal malformations, advances in knowledge are crucial to delivering better treatment outcomes and improving morbidity and mortality.[57]

Many pioneering surgeons have introduced new operative techniques and concepts that revolutionized the field. With the vast volume of published material in this field, a method of identifying works that have a significant contribution to the field is bibliometric citation analysis. This involves examining the citation history of an individual publication and providing a quantitative assessment of the relative importance of a research paper. In addition to ranking articles, citation analysis serves to rank journals based on the total number of citations normalized to the number of articles published within the journal over a defined time period (impact factor). Many surgical specialties have employed citation rank analysis to identify the most important published papers, including general surgery, plastic surgery, trauma and orthopedic surgery, and cardiothoracic surgery.[89101112]

To date, no study has been undertaken to determine the most important works on congenital abdominal pediatric surgery. Through extensive search of the available literature, this study aims to provide insight into how our knowledge of abdominal surgery on congenital diseases has developed and how our management has changed over time. In addition, it would serve as a reference for the most cited papers on congenital intestinal diseases.

METHODS

Thomson Reuters Web of Science citation indexing database and research platform was used to conduct the search. All fields were searched and must include all of the following terms: “Congenital,” “Surg,” “Pediatric/Paediatric,” and “Abdominal/Abdomen/Gastro.” Using the method initially developed by Paladugu et al.,[8] and performed again by Wrafter et al.[13] and Powell et al.,[14] the dataset is filtered to only include English language manuscripts published between January 01, 1970, and June 06, 2019, and indexed under the Science Citation Index Expanded, Conference Proceedings Citation Index, or Emerging Sources Citation Index. Manuscripts are screened to exclude those that examine nonabdominal surgical topics or adult populations alone. At Web of Science, the listed citation count across all databases was recorded and includes the following: Web of Science Core Collection, BIOSIS Citation Index, Chinese Science Citation Database, Russian Science Citation Database, and SciELO Citation Index. Articles were then obtained, reviewed, and analyzed using Excel® according to authorship, journal of publication, publication date, topic, institution, and country of origin. In addition, the latest Eigenfactor and impact factor of each journal were recorded.

RESULTS

The Web of Science search returned 971 full-length English language papers, of which 17 were excluded to produce the final list of 100 most cited articles. The list of manuscripts is ordered from most to least cited.[15] This included 87 original articles and proceedings papers and 13 review articles, with a grand total of 5449 citations.

The number of citations for these manuscripts ranged from 29, for “Antenatal diagnosis of choledochal cyst” by Bancroft et al.,[16] to 413, for “Sonographic predictors of survival in fetal diaphragmatic hernia” by Metkus et al.[17] The publication date of the top 100 manuscripts spans from 1971 to 2016, with the oldest manuscript being “Duplications of the alimentary tract in infants and children” by Iyer and Mahour[18] and most recent being “Gastroschisis: A national cohort study to describe contemporary surgical strategies and outcomes” by Owen et al.[19]

The top 100 cited manuscripts were published across 22 journals, with the number of manuscripts per journal ranging from 1 to 56 [Table 1]; 15 of the journals had a single publication, while the Journal of Pediatric Surgery had 56. The Journal of Pediatric Surgery has a total of 3223 citations, while Pediatric Surgery International, with 11 manuscripts, has a total of 550 citations. Intensive Care Medicine has the highest 2017 impact factor (15.008), 5-year impact factor (10.837), and Eigenfactor (4.416) of all the journals, with 1 manuscript and 61 citations.

Table 1

Journals with top 100 cited manuscripts on congenital pediatric abdominal surgeries

Journal	2017 impact factor	5-year impact factor	Normalized Eigenfactor	Manuscripts in top 100	Total citations
Journal of Pediatric Surgery	2.128	1.982	2.160	57	3231
Pediatric Surgery International	1.476	1.394	0.637	11	550
Seminars in Pediatric Surgery	1.645	2.642	0.362	4	171
Surgical Endoscopy and Other Interventional Techniques	3.117	3.299	3.968	3	150
European Journal of Pediatric Surgery	1.494	1.5	0.344	4	147
Pediatric Radiology	1.826	1.765	0.955	2	138
American Journal of Surgical Pathology	5.878	5.723	2.694	2	114
Archives of Disease in Childhood	3.258	3.365	1.848	1	93
The American Surgeon	0.607	0.771	0.834	1	70
Intensive Care Medicine	15.008	10.837	4.416	1	61
International Journal of Obstetrics and Gynaecology	4.876	4.687	2.790	1	56
Plastic and Reconstructive Surgery	3.621	3.798	3.739	1	52
World Journal of Surgery	2.766	3.052	2.804	1	51
Pediatrics	5.515	6.442	13.852	1	40
Annals of Plastic Surgery	1.536	1.675	1.281	1	39
Pediatric Cardiology	1.54	1.565	0.987	1	36
Pediatric Hematology and Oncology	1.154	1.217	0.201	1	35
Pediatric Neurosurgery	0.819	0.841	0.102	1	34
Developmental Medicine and Child Neurology	3.289	4.033	1.598	1	31
European Journal of Pediatrics	2.242	2.106	1.184	1	29
Journal of Pediatric Gastroenterology and Nutrition	2.752	3.03	2.173	1	29

Twenty-five different countries have contributed toward the top 100 cited manuscripts, ranging between 1 and 41 publications [Figure 1]. The United States of America has the most (n = 41; 2682 citations), followed by the United Kingdom (n = 8; 575 citations), Canada (n = 6; 271 citations), and Italy (n = 6; 267 citations). The top 100 manuscripts were published from 77 different institutions with the top 3 all based in the USA: University of Indianapolis (n = 5; 258 citations), Johns Hopkins University (n = 4; 193 citations), and University of California San Francisco (n = 3; 508 citations).

Figure 1

Number of publications by country: An amount of manuscripts are shown in percentages and a total number of citations are shown in parentheses

All first authors had one manuscript in the top 100, with the exception of Professor Piergiorgio Gamba for his work on abdominal wall defects (“Experimental Abdominal Wall Defect Repaired with Acellular Matrix” and “Abdominal Wall Defects: Prenatal Diagnosis, Newborn Management, and Long-Term Outcomes”).[2021] There were two senior authors with multiple manuscripts in the top 100: Professor Nick S. Adzick (n = 3; 627 citations) and Dr. Justin D. Klein (n = 2; 90 citations).[172223]

Within the top 100 manuscripts, various pathologies were studied, including gastrointestinal atresia, cystic lymphangioma, and choledochal cysts. The highest number of manuscripts and citations on congenital diaphragmatic hernia (CDH) (n = 23; 1559 citations), followed by gastroschisis (n = 18; 786 citations) and omphalocele (n = 16; 469 citations), is shown in Table 2. Majority of the manuscripts are either original articles or proceedings paper (n = 88), with a minority being literature reviews (n = 13). Of the original articles, there was one randomized control trial, and the level of evidence of the reviews ranges from III to V. This includes two systematic reviews investigating chylothorax, gastroschisis, and omphalocele.

Table 2

Most frequently studied topics

	Total manuscripts	Total citation count
Pathology
Diaphragmatic hernia	23	1559
Gastroschisis	18	786
Omphalocele	16	469
Gastrointestinal atresia	6	418
Lymphangioma	4	279
Tracheoesophageal fistula	3	274
Neoplasm/tumor	5	256
Choledochal cysts	5	237
Conjoined twins	2	123
Urogenital malformation	2	86
Anorectal malformation	2	85
Manuscript type*
Investigation and diagnosis	44
Management and outcomes	62
Experimental study	7

*Total count exceeds 100 as manuscripts with substantial coverage in multiple topics are counted individually

DISCUSSION

CDH was the most commonly studied condition, followed by gastroschisis and omphalocele. These are closely followed by manuscripts on gastrointestinal atresia, which includes esophageal, duodenal, jejunoileal, and colonic atresia. CDH was addressed by 23 studies out of the top 100 manuscripts and was also the topic of the most highly cited manuscript.[17] CDH results from the failure of diaphragm to fuse, allowing herniation of abdominal contents into the thoracic cavity where it impairs cardiopulmonary function and significantly increases infant mortality.[24] The overwhelming attention CDH received is not surprising as the overall survival rate of infants can range between 50% and 90% even with treatment. Hence, despite the thoracopulmonary nature of CDH, a decision was made to include CDH in our findings due to its clinical significance, involvement of alimentary tract as well as the role abdomen-based surgical approach in its management. Other common topics include lymphangioma, tracheoesophageal fistula, neoplasm and choledochal cysts, and anorectal malformation. Contrasting with CDH, a lower clinical priority of Meckel's diverticulum is reflected by the relatively few cited clinical manuscripts despite being the most common congenital malformation of the gastrointestinal tract.[25] On the other hand, while the abnormalities addressed by the top 3 cited papers are rare, present in 0.04%, 0.05%, and 0.03% of the population, respectively, they often lead to significant respiratory dysfunction and comorbidities.[262728]

The most cited paper in pediatric gastrointestinal tract was the 1996 paper in the Journal of Pediatric Surgery by Metkus et al.,[17] “Sonographic predictors of survival in fetal diaphragmatic hernia,” which used ultrasound to prenatally diagnose CDH and characterize and correlate several anatomical features with outcome and survival of infants. These results aid parent counseling and termination decisions and provide opportunities to conduct in utero surgical intervention shown to improve postpartum pulmonary function. The second most cited paper was a single-author review by Spitz[26] in 2007, presenting detailed etiology, pathophysiology, diagnosis, treatment, outcomes, and complications of esophageal atresia. This manuscript is a Level V evidence review and marks the first comprehensive summary of the available literature on the condition and serves to provide an evidence-based aid to clinical decisions. The third most cited study was by Quinn et al.,[22] “Prenatal magnetic resonance imaging enhances fetal diagnosis,” demonstrated the utility of magnetic resonance imaging (MRI) in diagnosing various anatomical anomalies, such as CDH, while decreasing motion artifacts by moving fetuses. Their use of MRI also allowed improved accuracy, over ultrasound, in determining important factors that affect the outcome of CDH such as residual lung area and liver herniation and thus providing a powerful alternative to standard ultrasound.

Similar to other bibliometric analyses, a potential limitation to this study lies in the differences between publication dates of manuscripts: The scientific community receives longer exposure toward older publications which may amass a larger number of citations irrespective of its scientific significance.[1314] However, this effect may also suggest that the recent manuscripts within the top 100 added significance given their relatively limited exposure to the scientific community. Several biases such as self-citation, institutional bias, language bias, and powerful person bias have not been accounted for and may have affected the distribution of manuscripts within the top 100. As suggested by several studies, manuscripts originating from the USA continue to receive a high number of citations across various specialties and may be reflective of preferential citation of “local research” which can contribute to bias.[142930] Furthermore, Web of Science citation numbers might not be wholly reflective of the true citation count as many available manuscripts are not included, such as those published before 1970. Finally, only first and senior authors were included, and papers that were coauthored may appear under-represented by the study design.

CONCLUSION

The most influential manuscripts highlighted by this study relate to the etiology, diagnosis, and management, advancing our understanding and outcome of congenital diseases. Common congenital anomalies such as Meckel's diverticulum were underrepresented within the top 100 cited manuscripts, while focus was placed on rarer but clinically more significant diseases such as diaphragmatic hernia and abdominal wall defects. In addition to being a reference of what could be considered as influential, this study also provides reference for researchers as to what makes a “citable” paper within the field of congenital abdominal pediatric surgery. With ongoing research continuing in this field, the list of top 100 most cited manuscripts has the potential to change within the coming decade.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.