Global trends in research of Hirschsprung disease over the last decade: a bibliometric analysis

Introduction

Hirschsprung disease (HSCR), which is also called congenital megacolon or congenital aganglionosis, is clinically characterized by delayed meconium in infants, and by a long constipation history form birth in older children (1). As one of the most common neonatal gastrointestinal malformations, this disease occurs once in 5,000 newborns. The affected intestinal segments were found to be devoid of enteric ganglia cells on histopathology, resulting in persistent spasm of the affected segments and dilatation of the proximal intestinal segments, which is a characteristic feature of the disease (2). The defects in enteric neuron development caused by abnormal proliferation, differentiation and/or migration of enteric neural crest cells (ENCCs) lead to an aberrant enteric nervous system (ENS) (2-4).

Although there are a few methods, such as barium enema and abdominal X-ray, that can help pediatricians make a diagnosis, rectal suction biopsy (RSB) is still the gold standard to find out whether the enteric neurons are survive (5,6). However, RSB is an invasive procedure, and while surgical resection is the only definitive treatment, it is not without risks, and long-term complications, such as postoperative bowel dysfunction, remain an issue. Therefore, there is an ongoing need for better diagnostic tools and treatment methods that minimize invasiveness and improve long-term outcomes.

While traditional literature reviews are a valuable tool for summarizing specific research findings, they often prove inadequate for keeping pace with the rapidly growing volume of scientific publications. Furthermore, they are limited in their capacity to systematically quantify and compare trends across a vast array of publications. In contrast, bibliometric analysis enables a more objective and comprehensive evaluation of the literature through the utilization of quantitative methods to identify trends, research outputs, key contributors, and collaboration networks. This method is particularly well-suited for emerging or rapidly developing fields, such as HSCR research, where a substantial body of literature has accumulated in recent years.

In recent years, the research hotspots and trends of HSCR have shifted due to the rapid development of science and technology. Many scholars and teams have put much effort and published a series of papers so far. However, there is a lack of comprehensive articles that could summarize the old and new literatures. As an emerging interdisciplinary approach, bibliometric analysis is able to quantitatively evaluate the literature with mathematical and statistical methods. This method not only provides insights into the current state of research but also highlights future clinical directions, potentially offering novel approaches to diagnosis and treatment.

By employing bibliometric analysis, this study not only offers a broad overview of global HSCR research trends between 2014 and 2023 but also provides unique insights that traditional literature reviews may overlook. Specifically, it identifies the most influential publications, key research institutions, and emerging topics, offering clinicians and researchers a data-driven foundation for future investigations. By identifying these factors, we aim to offer a roadmap for future research that directly addresses current clinical challenges and unmet needs, guiding clinicians and researchers toward more effective solutions for diagnosing and managing HSCR.

Methods

Date resources and search strategies

Web of Science (WoS) is one of the most widely accessed academic databases, providing a standard and comprehensive citation records for export (7). Therefore, all the bibliometric data were obtained from the Web of Science Core Collection (WoSCC) database. The literature search was conducted in one day (11 January, 2024) to avoid deviations caused by daily database renewal. The timespan for retrieval was limited to 2014 to 2023 to capture the most recent decade of research and reflect the current advancements and directions in HSCR studies, which are essential for identifying recent clinical needs and future research trends. Search strategies were as follows: search terms were TS = (hirschsprung*) OR TS = ((congenital* OR aganglionic*) AND megacolon*) OR TS = (aganglion* AND (sigmoid* OR rectal* OR rectum* OR rectosigmoid* OR anus* OR anal OR colon* OR intestin*)), the literature types were limited to “article” and “review” by excluding all other types, and the language was “English” only. Finally, a total of 1,770 articles and 281 reviews were exported for the sequent analysis. The overall working flow can be found in Figure 1.

Figure 1 Flowchart of the work.

Bibliometric analysis and data visualization

VOSviewer (version 1.6.20) was used to perform the networks of organizations, authors, countries, keywords and reference co-citations (8). Citespace (version 6.2.R6) was used to carry on keywords and citation bursts, cluster analysis and timeline graphs, which enables us to determine the research status and hotspots at the moment, and sheds light on the new trends in the future (9).

To measure the bibliometric data quantitatively, the number of publications (NP) and the number of citations (NC) without self-citations are often used as the two significant bibliometric indicators, because NP is often employed to qualify the productivity and NC can show the impact.

The H-index has been increasingly used to evaluate the influence of a scholar or a region in a given field. By the definition proposed by Hirsch (10), an author’s H-index means that he has published at least H articles and each article has been cited at least H times (excluding self-citations). This innovative, simple and convenient method comprehensively combines NP and NC to show the achievements of researchers, and could also be used to predict the future trend (10).

Statistical analysis

Microsoft Office Excel 2021 is employed for the management of dates and the analysis of the NPs per year. Each document was subjected to a rigorous scientific analysis, resulting in data that included key elements such as the type of document, the country or region of primary focus, the institution, the journal, the author, the most frequently cited references, and the co-occurrence of keywords.

Results

An overview of HSCR publications

Based on the search strategy, a total of 2,051 publications were retrieved from 2014 to 2023, of which the total NC was 10,351, the average NC per article was 10.81, and the H-factor for all publications was 53.

Figure 2A depicts the geographical distribution of the total number of papers on HSCR research from all nations and regions. The top five nations accounted for over half of the 2,051 articles. The USA was the country with the most papers published, followed by China, Japan, England, and the Netherland. Figure 2B depicts the top 12 countries in terms of annual publications in HSCR research from 2014 to 2023 (because Australia, India and Sweden published the same number of articles, all of them were included, resulting in 12 countries on the list). The USA has the highest NPs in 2022 [77], followed by China [62] and Japan [20]. This indicates that the USA remains the country with the highest number of HSCR studies.

Figure 2 The overview of HSCR publications. (A) Geographical distribution of publications on HSCR research from 2014 to 2023. (B) Top 12 countries in terms of annual publications on HSCR research, from 2014 to 2023. The circle’s size and colors show the number of papers. The larger the circle, the color from light blue to red, the higher the number of articles issued in that country. (C) Curve fitting of the total annual growth trend of publications (R²=0.7121). (D) The number of publications by year over the past 10 years. NP, number of publications; HSCR, Hirschsprung disease.

The annual trend of publications

Figure 2C shows the annual NP polynomial curve for HSCR with R²=0.7121, suggesting that no significant correlation was found between publication year and annual NP. Figure 2D presents the NP of each year associated with HSCR. Overall, although the NPs fluctuates from year to year, the overall trend remains upward, from 186 in 2014 to 251 in 2023, peaking at 310 in 2021, which indicates that the study of HSCR has become a hot research trend.

Analysis of countries/regions

We listed the top 12 countries in NP order and summarized them in Table 1. The USA published the most articles (574/27.99%), followed by China (453/22.09%) and Japan (152/7.41%). The USA had the highest NCs [7,709], followed by Germany [3,117] and France [2,734]. The USA had the highest H-index [45], followed by China [29] and England [29]. In comparison with Japan and England, Germany and France had a significantly higher NC and H-index than both countries, although the NP was lower. Despite Australia, India, and Sweden published the same number of articles, India had lower NC and H-index than the other two countries. As the visualization network graph shows, there is close cooperation between the different countries/regions (Figure 3A). France, Germany, and Australia conducted research in this field earlier than other countries/regions. Although China started later, it has more research outputs (Figure 3B).

Figure 3 The visualization analyses in HSCR research. (A) Visual cluster analysis of cooperation among countries/regions. (B) Timeline visualization of cooperation among countries/regions. (C) Visual cluster analysis of cooperation among affiliations. (D) Timeline visualization of cooperation among affiliations. (E) Visual cluster analysis of cooperation among authors. (F) Timeline visualization of cooperation among authors. HSCR, Hirschsprung disease.

Analysis of journals

As shown in Table 2, Journal of Pediatric Surgery [196 publications, Impact Factor (IF) =2.4] published the most papers related to HSCR. Journal of Pediatric Surgery aims to offer the latest and most impactful clinical and scientific knowledge and evidence in the field of pediatric surgery, with the goal of improving surgical care for infants and children. Followed by Pediatric Surgery International (182 publications, IF =1.8) and European Journal of Pediatric Surgery (53 publications, IF =1.8). The top ten journals published about 32% of the papers (664/2,051, 32.37%). In addition, Journal of Pediatric Surgery had the highest NC and H-index.

Analysis of affiliations

Table 3 illustrates the ten affiliations with the highest NPs related to HSCR. University System of Ohio had the highest number of NP [81], followed by Harvard University [76] and Ohio State University [70]. Notably, the Institut national de la santé et de la recherche médicale (INSERM) had a lower NP but a much higher NC than other productive affiliations. In addition, five of the top ten affiliations were based in the USA. Huazhong University of Science Technology, Johns Hopkins University and Harvard Medical School stand at the core position (Figure 3C). Nationwide Children’s Hospital, University of Amsterdam and University of Cincinnati started research late (Figure 3D).

Analysis of authors

Table 4 provides an overview of the top ten authors with the highest NP. They published 311 papers, accounting for 15.16% of the total NP [2,051]. Their NC was 4,280, accounting for 31.56% of the total NC [13,561]. Weibing Tang from China is the most prolific author (NP =43), followed by Prem Puri (NP =39) and Allan M. Goldstein (NP =39). Furthermore, more than half of the top ten authors were from China and the USA.

To show how the different authors collaborated, VOSviewer was utilized to create co-authorship networks. In the last decade, over 8,700 authors have published papers on HSCR. One hundred and thirty-eight authors have contributed to the record with at least 8 publications, while only 131 authors were found to have connections with other authors in the network. Allan M. Goldstein and Weibing Tang were located at the center of the network (Figure 3E), and Marc A. Levitt, Rebecca M. Rentea, and Andrea Bischoff started relevant research after 2020 (Figure 3F). The data indicated that cooperation among the various authors remains robust.

Analysis of highly cited articles

Table 5 presents the total citations of articles in descending order. The top ten most cited articles mainly were published between 2014 and 2018. The writing in Nature Cell Biology ranked first (IF: 21.3, title: Oncogenic roles of EMT-inducing transcription factors, type of study: review, 773 citations). This review emphasizes the importance of epithelial-mesenchymal transition (EMT) in embryonic development. Follow by Genetics (IF: 3.3, title: The “Domestication syndrome” in mammals: a unified explanation based on neural crest cell behavior and genetics, type of study: article, 382 citations) and Nature Medicine (IF: 82.9, title: Engineered human pluripotent-stem-cell-derived intestinal tissues with a functional enteric nervous system, type of study: article, 381 citations). Focusing on pressing issues in contemporary clinical medicine, Nature Medicine covers a lot of original research, which ranges from new concepts in human biology and disease pathogenesis to new therapeutic modalities and drug development. Research on HSCR was published in Nature Medicine, indicating the significance of studying the pathogenesis of HSCR.

Analysis of paper Global Citation Score (GCS)

Figure 4A depicts the annual status of articles with high GCSs. The paper written by Puisieux et al. have achieved the amazing GCSs in the last decade, ranking the first. In this paper, the authors noted that ZEB2 (SIP1) is involved in cell fate decisions between neuroectoderm and mesendoderm differentiation in human embryonic stem cells (hESCs). It has been shown that Zeb2 deletion in mice leads to neural crest cell defects, and heterozygous mutations in ZEB2 have been observed in Mowat-Wilson syndrome, a syndrome of mental retardation characterized by a variety of congenital malformations, including HSCR (11,12). In addition, Fattahi et al. successfully derived and isolated ENS progenitors from human pluripotent stem cells (hPSCs) and differentiated them into functional enteric neurons. ENS precursors derived in vitro are capable of targeted migration in the developing chick embryo and extensive colonization of the adult mouse colon, and those derived in vivo rescue disease-related mortality in HSCR mice (Ednrb^s-l/s-l), which sheds light on the study of human ENS development (13). Most of the above studies focus on the pathogenesis of HSCR and the development of ENS. Figure 4B displays the 97 articles (other publications are cited no more than 40 times) with high citation frequency, corresponding to Figure 4A, where the nodes of articles with high GCS are relatively large and are at the core of the mesh.

Figure 4 Citation map in HSCR. (A) The yearly number of global citations for papers having a high GCS. The circle’s size and colors show the GCS of papers. The larger the circle and the color from blue to red, the higher the GCS of the article and the more influential it is in the field. (B) Network of document citation. Given a large number of references available, the minimum number of citations for a reference was placed at 40. Ninety-seven of the 2,051 papers were chosen for citation analysis. The nodes in different colors represent various documents, with larger nodes indicating more frequently cited articles. GCS, global citation score; HSCR, Hirschsprung disease.

Analysis of co-cited references

Co-citation networks accentuate research topics that are closely related to specific fields, which is different from global citation analysis. Due to the large NC, the minimum NC for a reference was set as 35. Of the 40,900 references cited by retrieved papers, 133 met the threshold, and the co-citation networks had been made (Figure 5A). The size of the node indicates the total link strength, representing the NC of the document. The line between two nodes indicates that they were both cited in a single paper, and a shorter line denotes a closer connection. In addition, the different colors of nodes were used to divide them into different clusters. Cluster 1 (in red) comprised 69 references that concentrated on the bowel function and quality of life after surgery, as well as the diagnosis and management of Hirschsprung-associated enterocolitis (HAEC), an inflammatory syndrome characterized by fever, sepsis, diarrhea and abdominal distension (14). Cluster 2 (in green) focused on the development of the ENS, investigating key mechanisms related to neural stem cell migration, proliferation and genetic variation associated with HSCR. Cluster 3 paid special attention to the etiology, mechanisms and possible gene mutations in the development of HSCR. Based on the clustering, it was found that the majority of studies concentrated on the pathogenesis of HSCR. Figure 5B showed the top 25 references with the strongest citations bursts. The study of Lake and Heuckeroth had the highest burst strength (15.48). Their article summarizes the time course of NCCs’ migration from the neural tube to the gut and how they differentiate to from the ENS. It also discusses the critical molecular mediators and signaling pathways required for this process (15). Figure 5C illustrates the most typical references in terms of burst length, burst strength, and burst time. As shown in it, the top six clusters of co-cited references were “calretinin”, “enteric nervous system”, “enterocolitis”, “quality of life”, “transanal surgery” and “rectal suction biopsy”.

Figure 5 Mapping based on co-cited references from HSCR-related research. (A) A network diagram of co-cited references. Of the 40,900 references, 133 (divided into three clusters) were cited at least 35 times. (B) The top 25 co-cited references with the most citation burstiness. The years between “Begin” and “End” represent the period when the reference was more influential. Years in light green mean that the reference has not yet appeared, years in dark green mean that the reference is less influential, and years in red mean that the reference is more influential. (C) The top 6 clusters’ timeline distribution. HSCR, Hirschsprung disease.

Keywords analysis

In addition to search terms, VOSviewer and Citespace analyzed keywords extracted from the titles and abstracts of 2,051 publications. According to Figure 6A, cluster 1 (in red) was mainly about postoperative outcomes, complications, long-term follow-up, and quality of life in children with HSCR. Cluster 2 (in green) primarily revolved around the genetic and molecular mechanisms, mutations, and pathways associated with neural crest development, neural system disorders, cancer susceptibility, and the role of specific genes such as Phox2b, Ednrb, Ret, and Sox10 in mediating processes like migration, proliferation, and differentiation. Cluster 3 (in blue) focused on research topics related to the ENS, gastrointestinal disorders, and therapeutic approaches such as cell therapy and transplantation, emphasizing factors such as neurogenesis, neural crest cells, and smooth muscle stem cells. Cluster 4 (in yellow) was mainly about the accuracy and diagnosis of aganglionosis pathology, specifically through techniques such as rectal biopsy, acetylcholinesterase histochemistry and calretinin immunohistochemistry. Cluster 5 (in purple) focused on Mowat-Wilson syndrome, including associated phenotypes, molecular features, and genes such as SIP1 (ZEB2, ZFHX1B).

Figure 6 HSCR’s keywords map. (A) Using different colors, the 196 terms that appeared more than 15 times were separated into five clusters. (B) Keyword visualization according to the APY. The different colors indicate the relevant year of publication. Yellow keywords came later than purple keywords. (C) Timeline distribution of keyword cluster analysis. (D) The top 25 keywords with the most bursts. The years between “Begin” and “End” represent the period when the keyword was more influential. Years in light green mean that the keyword has not yet appeared, years in dark green mean that the keyword is less influential, and years in red mean that the keyword is more influential. HSCR, Hirschsprung disease; APY, average publication year.

As seen in Figure 6B, VOSviewer divided the colors of all keywords into categories based on their average publication year (APY). The latest keyword was “botulinum toxin” (cluster 1, APY: 2020.90), followed by “care” (cluster 1, APY: 2020.41) and “health” (cluster 1, APY: 2020.32). Besides, “bowel management” (cluster 1, APY: 2020.00) and “postoperative obstructive symptom” (cluster 1, APY: 2020.00), as well as “outcomes” (cluster 1, APY: 2019.93) and “complications” (cluster 1, APY: 2019.91), were the most recent hot topics in this field. Additionally, “enteric nervous system”, “quality of life”, “Mowat-Wilson syndrome”, “rectal biopsy”, and “pediatric surgery” have been the research focus on HSCR for a long time (Figure 6C). Meanwhile, the terms “pediatric surgery”, “care”, “health”, and “case report” were the most recently hotspots during the last 3 years (Figure 6D).

The common theme in Figure 6A-6D is to investigate the pathogenesis of HSCR, improve postoperative management, and enhance children’s quality of life. There will be more studies on development of ENS, and more researchers will be increasingly inclined to explore the migration and differentiation mechanisms of NCCs through animal model experiments, paving the way for additional treatment possibilities for HSCR.

Discussion

This study performed bibliometric analysis to explore research hotspots and trends in HSCR utilizing the WoSCC database, VOSviewer, and Citespace. We retrieved 2,051 articles and reviews published from 2014 to 2023. Although the number of published papers has some fluctuation during the decade, there is an overall trend toward increasing the number of articles published, as indicated by the polynomial fit curve. It also suggested that an increasing number of researchers are becoming interested in the topic of HSCR.

Publications are distributed globally, while productivity in some areas is modest. Figure 2A depicts the geographic distribution of publications on HSCR research. The USA has the highest NPs [574], followed by China [453], Japan [152] and England [134]. Among the top ten countries/regions, the USA rated first in NP, indicating that it is a very prolific country in the field of HSCR. In HSCR research, five institutions and three scholars from the USA ranked in the top ten affiliations and authors, indicating that the USA has exceptional institutions and specialist scholars. This may explain why the USA has had so much influence on the field over the last decade. Furthermore, the USA has a higher H-index and NC compared to China. This was due to American researchers revealing the spatiotemporal regulation network of ENCC’s proliferation, differentiation and migration, addressing the essential role of NCCs in the development of ENS (2,4,16). In addition, the USA has been more intensively researched than the rest of the world. It is suggested that Chinese scholars and affiliates in this field should improve the quality of their work. Similarly, there is a contradictory in the quantity and quality of publications in Japan.

Among the top ten journals publishing on the HSCR, only two had an IF >3. This result indicates that publishing research on HSCR in high-quality journals is a challenge. There are several reasons for this issue. Firstly, HSCR is one of the neurocristopathies, a large heterogeneous group of rare diseases caused by abnormal development of neural crest cells. Most research on the pathogenesis of HSCR has been implemented into neurocristopathies and has not solely focused on HSCR. Secondly, while the treatment of HSCR has been matured with advances in pediatric surgery, there is still a lack of large-scale, high-quality clinical trials to support it, and studies related to postoperative outcomes and quality of life are still in a relative void at this time. In addition, as these journals are professional periodicals with high recognition and influence in the field of pediatric surgery, scholars can more easily promote their ideals or views in the scientific field, allowing them to discuss and exchange their ideas with their peers in order to improve their academic level and scientific competence. Finally, these journals have relatively shorter review cycles, making it more likely that academic papers will be submitted. According to this trend, the journals listed in Table 2 may remain the primary source for future findings in this field. This also encourages scholars with an interest in the subject to thoroughly review these publications.

With regard to affiliation, almost all of the top ten institutions are from the top five countries with the most publications, half of which are in the USA, indicating that the country has good academic capacity in this field. Weibing Tang, Prem Puri, and Allan M. Goldstein are the three most published scholars in HSCR research. Therefore, to keep up with the latest developments in the field, we should pay more attention to their work and give it higher priority. The University System of Ohio (81 publications, cited 988 times) is the most prolific institution, followed closely by Harvard University (76 publications, cited 1,850 times), with most of the papers produced by Allan M. Goldstein. His highly cited paper summarizes our current understanding of the factors involved in early development of the ENS and suggests areas for further investigation (17). And his latest study indicates that hindgut ENS development is perturbed by overexpression or inhibition of bone morphogenetic protein-4 (BMP4) in ceca, and that glial cell line-derived neurotrophic factor (GDNF) plays a critical regulatory role, suggesting that both signaling pathways are necessary for normal migration of enteric neural crest derived cells and formation of enteric ganglia in developing hindgut ENS (18). This study illustrates the regulation mechanism of BMP4 between GDNF in the colorectal development of ENS, shedding light on the pathogenesis of HSCR.

Among the top ten most cited articles, 60% (6/10) focused on the pathogenesis of HSCR, indicating that this is a popular research topic. Table 5 shows that Puisieux A, Wilkins AS, and Workman MJ are among the three scholars whose articles have been cited more than 380 times. The article of Puisieux et al. had the highest NC, ranking first. His research showed that EMT is dependent on contextual microenvironmental signals and is regulated by a network of EMT-inducing transcription factors (EMT-TFs). The aberrant reactivation or repression of EMT-TFs not only promotes cancer cell plasticity, contributing to tumorigenesis and metastasis, but is also implicated in a variety of human developmental disorders (11). Additionally, Wilkins et al. proposed an interesting hypothesis on the nature and origin of “domestication syndrome” (general combinations of traits in domesticated mammals). The hypothesis suggests that the different traits of “domestication syndromes” share a common developmental linkage through NCCs (19). Their hypothesis, which explains the domestication syndrome through an analytical summary of successive experiments, has been widely accepted by their peers and is therefore frequently cited. Notably, Workman et al. received more GCS for their research, which successfully recapitulated normal intestinal ENS development by combining hPSC-derived neural crest cells and developing human intestinal organoids (HIOs). The ENS-containing HIOs, cultivated in vivo, exhibited neuroglial structures resembling a myenteric and submucosal plexus. Furthermore, the cells featured functional interstitial cells of Cajal (ICCs) and demonstrated electromechanical coupling that regulated waves of propagating contraction (20).

Citation burst analysis, provided by CiteSpace, is a methodological tool used to identify significant fluctuations in references and keywords within a defined time span. In citation burst analysis, citation strength is a metric that indicates the gravitational attraction of a particular reference or keyword. It indicates its widespread adoption and discussion in the scientific discourse. Additionally, the duration of a citation burst signifies the temporal extent of its prominence and its ongoing relevance within the research community. This analytical method provides valuable insights into emerging research topics within a particular field. The keywords with the latest burst until 2023 in this study were “pediatric surgery”, “care”, “health”, and “case report”. These keywords also appeared in the time-overlapping analysis, as shown in Figure 6B. As for references, there were three cited publications since 2021 and the burst has persisted until 2023. Reference hotspots were consistent with keywords hotspots. One of these publications is the guideline for the diagnosis and management of rectosigmoid HSCR proposed by the European Reference Network for rare Inherited and Congenital (digestive and gastrointestinal) Anomalies (ERNICA), which includes 52 European health care providers (21). Another publication is a multicenter study of long-term bowel function in 200 postoperative patients with rectosigmoid HSCR. The result indicated that there are a large number of HSCR patients suffering from bowel problems, emphasizing the necessity for long-term follow-up (22). The last study successfully induced enteric neurogenesis using GDNF in three different mouse models of HSCR, demonstrating the therapeutic potential of GDNF for HSCR (23). Furthermore, it was observed that Wang et al. investigated the correlation between genetic variants in the IKBKAP gene and the susceptibility to Hirschsprung’s disease in southern Chinese children. This investigation identified the rs2275630 locus as a specific susceptibility marker for total colonic aganglionosis (TCA) patients for the first time (24). Another study systematically screens for differentially expressed circular RNAs (circRNAs) in Hirschsprung’s disease and constructs a circRNA-miRNA-mRNA regulatory network, revealing their potential roles in the pathogenesis of the disorder (25). A recent study has yielded promising results, as human ENS progenitor cells derived from hPSCs were successfully generated and transplanted into the tissues of patients with Hirschsprung’s spondylosis, demonstrating an enhanced functional response. This approach represents a novel strategy for the treatment of Hirschsprung’s disease, with the potential to restore intestinal motor function through the use of stem cell technology (26).

On the basis of the bibliometric analysis and the visualization, the following directions of research are of interest for future studies: (I) excavating the deep mechanism of ENCC proliferation, differentiation, and migration associated with HSCR; (II) exploring effective prevention and treatment of postoperative complications; (III) improving the long-term quality of life for postoperative patients. However, it must be acknowledged that there are several limitations to this review. Firstly, as a bibliometric analysis, the data collection and processing heavily rely on software. Although this analysis cannot completely replace manual retrieval, it facilitates a comprehensive analysis of large data sets. Secondly, this study only collected articles and reviews written in English from the WoSCC database, which means that some valuable studies may have been missed. Given WoSCC’s high coverage rate of the majority of studies, it is unlikely that any oversights would significantly impact general trends. Additionally, it is important to note that citation impact may be delayed, potentially leading to underestimation of the impact of recently published high-quality studies. These studies should be closely monitored and updated in future research. Despite all these, this study will academically help researchers to understand the developing trend and hotspots and frontiers in the field of HSCR.

Conclusions

This analysis of bibliometrics shows that the research of HSCR has a minor fluctuation in the number of articles published over the last decade. However, the overall trend is toward more and more articles being published. The research of HSCR has a good research prospect.

Future research areas of high interest include: (I) the mechanism of ENCC proliferation, differentiation and migration associated to HSCR; (II) prevention and treatment of postoperative complications; (III) the long-term quality of life for postoperative patients. By focusing on these areas of investigation, it may be possible to address current clinical challenges, such as the reduction of postoperative complications and the improvement of patient outcomes, thus responding to urgent clinical needs.

Our study offers valuable insights into the current state of HSCR research from a macro perspective, facilitating the identification of research trends, influential studies, and potential gaps in knowledge. By providing a systematic analysis of the existing literature, this study can inform clinicians and researchers in prioritizing research areas with the greatest potential for clinical impact. We hope that this study will serve as a reference point for future research efforts, ultimately contributing to improved diagnostic and therapeutic approaches for HSCR.

Acknowledgments

We gratefully acknowledge the contributions of all reviewers and peers who provided suggestions for revisions to this article.

Footnote

Peer Review File: Available at https://pm.amegroups.com/article/view/10.21037/pm-24-23/prf

Funding: None.

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://pm.amegroups.com/article/view/10.21037/pm-24-23/coif). The authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

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