J Plast Surg Hand Surg, 2015; 49: 52—58 ©2015 Informa Healthcare ISSN: 2000-656X prin t/2000-6764 online DOI: 10.3109/2000656X.2014.951053

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ORIGINAL ARTICLE

A bibliometric analysis of the 50 most cited papers in cleft lip and palate Nicola A. Mahon & Cormac W. Joyce Department o f Plastic and Reconstructive Surgery, University Hospital Galway, Galway, Ireland

Abstract Citation analysis is an established bibliometric method which catalogues papers according to the number of times they have been referenced. It is believed that the total number of citations an article receives reflects its importance among its peers. Never before has a bibliometric analysis been performed in the area of Cleft Lip and Palate. Our citation analysis creates a comprehensive list of the 50 most influential papers in this field. Journals specializing in Cleft Palate, Craniofacial, Plastic Surgery, Maxillofacial Surgery, Aesthetics and Radiology were searched to establish which articles most enriched the specialty over the past 70 years. The results show an interesting collection of papers which reveal developing trends in surgical techniques. These landmark papers mould and influence management and decision-making today.

Key Words: Bibliometric analysis, citation analysis, most cited, top 50, cleft lip, cleft palate Introduction Cleft lip (cheiloschisis) and cleft palate (palatoschisis) occur in 1 in 500-700 births [1], This is due to the failure of fusion of the palate during the 6th week of embryonic life [2], It ultimately affects a child’s airway, feeding, appearance, dentition, and speech, thus requiring a multidisciplinary management approach. Over the years, the establishment of various cleft classification systems, as well as newly recognized associated syndromes and advances in surgical correction, have been documented in the literature by different specialties. Due to the diversity of specialties involved in this area, it is difficult to be aware of the most influential papers in cleft lip and palate. A published paper is considered to be influential within its field if it has been cited many times by subsequent authors. A citation is a numerical abbreviation embedded within the text of an article, which refers to and acknowledges another authors work. Citation analysis is an established bibliometric method which catalogues papers according to the number of times they have been referenced. In this way it is possible to ascertain how influential a paper is, by performing a citation analysis, which calculates the total number of times the paper has been cited. The purpose of a citation is to recognize another author for the important work they have published. This strengthens the author’s reputation in their clinical field and also augments the reputation of the journal that published the paper, by increasing the journal’s impact factor. The impact factor (IF) of a scientific journal is a measure of the number of citations it’s published articles have received [3]. It is a calculation based on the number of citations a journal has obtained in the current year to items published over the previous 2 years, divided by the substantive articles published over the previous 2 years [4,5], The higher the impact factor of a journal, the more prestigious it becomes within the scientific community which it serves [6], Some authors disagree that this is a valid method of evaluating a

journals worth [7,8]. However, scientific journals still aspire to increase their impact factor as a means of elevating their status, viewing it as a putative marker of journal quality. Several specialties have published articles on the most influential papers in their field, by way of a bibliometric analysis. These specialties include anaesthetics, general surgery, dermatology, otolaryngology, plastic surgery, radiology, and orthopaedics [9-18]. However, to our knowledge, there has never been a bibliometric analysis of the most cited papers in the field of cleft lip and palate. We have performed a citation analysis to evaluate the 50 most influential papers in cleft lip and palate over the past 70 years. Materials and methods The database of the Science Citation Index (SCI) of the Institute for Scientific information (ISI) was utilized in order to identify the most cited papers in cleft lip and palate [19]. This, is an online index of citations, available through the Web of Science database, which is part of the Web of Knowledge collection of databases. Twenty-four international scientific journals were included in our search, which was performed in January 2014 (Table I). The included journals consisted of cleft palate, craniofacial, plastics, maxillofacial, and radiology journals with high impact factors. Only eight of these 24 journals contributed to the top 50 most cited articles. The database was searched using filter terms of “cleft”, “cleft lip”, “cleft palate”, “cleft lip & palate”, and “prenatal cleft screening”. After identifying the 50 most cited papers, we performed a further analysis of each paper using criteria described by Paladugu et al. [10]. Each paper was individually analyzed to reveal it’s subject matter, authorship, article type, institution, year of publication, and level of evidence.

Correspondence: Dr Nicola Mahon, University Hospital Galway, Newcastle Road, Galway, Ireland. Tel: +00 353 1 6237922. Fax: +00 353 1 6268362. E-mail: [email protected] (Received 7 July 2014; accepted 24 July 2014)

Citation analysis o f papers in cleft lip and palate Results Table II shows the 50 most cited papers in cleft lip and palate, in descending rank order of citations received [2,20-68]. The total number of citations per articles ranged from 77-440. The most influential paper was cited 440 times and was published by Shprintzen et al. [20] in 1978. This seminal article was the first to describe velo-cardio facial syndrome. The second most cited paper by Bergland et al. [21] looked at eliminating the residual alveolar cleft defect by secondary bone grafting. The subject of secondary bone grafting was also discussed in the 4th most cited paper by Abyholm et al. [23] in 1981. It is the top cited cleft paper published in a plastic surgery journal and was published in the Scandinavian Journal o f Plastic and Reconstructive Sur­ gery. Furlow [24] published the 5th most cited paper in 1986, introducing a method of soft tissue repair by using a double opposing z-plasty. After almost 30 years this repair is still commonly used today [69]. The sixth most influential paper was the result of a six centre international study on treatment outcomes in patients with cleft lip and palate [25]. The strength of this paper was that it incorporated six different clinical institutions across Europe and the US. Each institution used different treatment protocols for managing patients with cleft defects, thus the long-term follow-up of various treatment modalities could be established within this one study. The 50th most cited paper was also part of the six centre international study. It was published by Asher-McDade et al. [68] in Man­ chester and was cited 77 times, it assessed the naso-labial appearance of patients with cleft lip and palate. The most recent paper was published in 2000 [65] discussing prenatal diagnosis of cleft lip/palate using ultrasound and was cited 80 times, whereas the oldest paper was published in 1946 by Tennison [30], describing the stencil method of repair, and has been cited 120 times to date. Plastic and Reconstructive Surgery (PRS) published 18 of the most cited papers (Table I), while the journals Cleft Palate and Cleft Palate Craniofacial both published 12 papers in the top 50 most cited list. The 1980s and 1990s contributed most papers to the top 50 list, having published 15 papers and 21 papers, respectively (Table III).

Table I. The journals and the number of papers each journal con­ tributed to the top 50 papers. n Plastics and Reconstructive Surgery Cleft Palate Journal Cleft Palate Craniofacial Journal Journal of Oral and Maxillofacial Surgery Scandinavian Journal of Plastics and Reconstructive Surgery British Journal of Plastic Surgery Prenatal Diagnosis Journal of Craniofacial Genetic and Developmental Biology

18 12 12 3 2 1 1 1

Other journals included in our search but which did not achieve top 50 status: Annals of Plastic Surgery, Clinics in Plastic Surgery, European Journal of Plastic Surgeiy, Facial Plastic Surgery, Aesthetic Plastic Surgery, Archives of Facial Plastic Surgery, Orthodontics Craniofacial Research, British Journal of Maxillo-facial Surgery, International Journal of Maxillo-facial Surgery, Aca­ demic Radiology, British Journal of Radiology, Clinical Radiology, Dentomaxillofacial Radiology, European Journal of Radiology, European Radiology, Investigative Radiology.

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Ten countries contributed to the top 50 papers. Most articles originated from the US, who were responsible for publishing 29 papers in the top 50 (Table IV). English-speaking countries produced 40 of the top 50 papers (US, UK, Canada, and Australia), whilst the remaining 10 articles originated from Table II. The 50 most cited papers in Cleft Lip and Palate. Rank 1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24 25 26 27 28 29 30 31 32 33 34 35 36 37 38 39 40 41 42 43 44 45 46 47 48 49 50

Reference Shprintzen et al. [20] Bergland et al. [21] Vanderas [22] Abyholm et al. [23] Furlow [24] Shaw et al. [25] Mars et al. [26] Polley and Figueroa [27] Trost [28] McComb [29] Tennison [30] Ortiz-Monasterio et al. [31] Wyszynski et al. [32] Cohen and Sulik [33] Mars et al. [34] Semb [35] Jensen et al. [36] Grayson et al. [37] Calnan [38] Shprintzen [39] Cohen et al. [40] Enemark et al. [41] Stark [2] Dorf and Curtin [42] Molina et al. [43] Sindetpedersen and Enemark [44] Millard and Latham [45] Salyer [46] Jones [47] Wyszynski et al. [48] Murray et al. [49] Kaplan [50] Hallock [51] Shaw et al. [52] Tajima and Maruyama [53] Hall and Posnick [54] Friede and Johanson [55] Cohen [56] Lidral et al. [57] Farkas et al. [58] Mars et al. [59] Cohen et al. [60] Orticoch [61] El Deeb et al. [62] Fara and Cvorak [63] Lemesurier [64] Clementi et al. [65] Krogman et al. [66] Mayer et al. [67] Asher-McDade et al. [68]

No of citations 440 245 180 170 154 147 145 137 127 121 120 120 118 115 114 113 112 111 105 105 104 104 103 102 101 100 98 98 98 97 91 91 91 90 90 87 86 85 84 82 82 82 82 81 81 80 80 79 78 77

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N. A. Mahon and C. W. Joyce

Table III. The decades where the top 50 papers originated from. Decade 1940s 1950s 1960s 1970s 1980s 1990s 2000s

Number of papers 2 2 2 7 15 21 1

Table IV. The countries of origin of the top 50 papers in cleft lip and palate. Nation USA UK Canada Denmark Norway Sweden Australia Japan Czech republic Italy

Number of papers 29 5 5 4 2 1 1 1 1 1

countries where English is not the first language (Denmark, Norway, Sweden, Japan, Czech Republic, and Italy). Table V displays the level of evidence of each paper, which was formulated by using the Oxford centre for Evidence based Medicine’s level of evidence table [70]. Levels of evidence are rankings of the most reliable evidence gained from different scientific methods to guide clinical decision-making. Almost half of the top 50 published papers were therapeutic papers. This highlights the fact that new innovations in surgical techniques have dominated the research literature in clefts over the last 70 years. Institutions such as the Department of Orthodontics in Sweden, Denmark, Holland, London, Manchester, and Oslo Table V. The type and level of evidence of the clinical studies in the top 50. Clinical study type Diagnostic Level 1 Level 2 Level 3 Level 4 Level 5 Prognostic Level 2 Level 4 Therapeutic Level 2 Level 3 Level 4 Level 5

Number of studies (n = 50) 15 3 1 2 3 6 11 10 1 24 10 3 1 10

Table VI. The institutions which contributed greater than one paper to the top 50 most cited papers in cleft lip and palate. Institution No of papers Department of Orthodontics, Vrije University, 4 Amsterdam, The Netherlands Department of Orthodontics, Hospital for Sick 4 Children, London, UK Department of Orthodontics, Royal Dental College, 4 Copenhagen Department of Orthodontics, Karolinska Institute, 4 Stockholm, Sweden Department of Orthodontics, University Dental 4 Hospital, Manchester, UK Children’s Hospital, Los Angeles, CA 4 University Aarhus, Denmark 3 Montefiore Hospital (craniofacial disorders), 2 Bronx, NY Deptodontol/Plastic Surgery, University of Oslo, Oslo, 2 Norway Department of Epidemiology, John Hopkins, MD 2 Dalhousie University, Halifax, Nova Scotia, Canada 2 Great Ormond Street Hospital, London, UK 2

contributed the highest number of articles to the 50 top cited papers (Table VI). All of the above were Dental institutions and were involved in the aforementioned multicentre study. The subject matter focused mainly on surgery (18 papers), epidemiology of cleft conditions (eight papers), and the diagnosis of associated syndromes (four papers). Facial growth patterns, effects on speech, and dental arch relationship were featured to a lesser extent, contributing three papers each to the top 50 papers [21,23,25,34,35,52,59,68], The author G. Semb from the cleft palate institute in Oslo was the most prolific author, having produced eight papers in the top 50 most cited list (Table VII). Discussion The management of cleft lip and palate has vastly improved over the past 70 years. This was once a poorly understood and lifethreatening condition. Nowadays, with the introduction of the multidisciplinary team approach to treatment, the care of these patients has been transformed [71]. Much of our improved knowledge and surgical skills are as a direct result of advances in scientific research in this field. However, as these patients are managed by multiple specialties, it is quite taxing to keep abreast of all new developments within each field. This study was undertaken to identify the 50 most cited papers published in the area of cleft lip and palate. As a result of this research, one can see how developments in this specialty have evolved over time. It becomes apparent which authors have made outstanding contributions in this area and led the way in terms of clinical research. It is now possible to appreciate the landmark papers that have contributed to the growth of the specialty. Although this study has included many of the prolific papers in cleft research, some important papers were not included in the top 50 fist. Choudhary et al. [72], in 2003, published an enlightening paper looking at the effect of the Veau-Wardill-Kilner type of cleft palate repair on long-term midfacial growth, but this paper did not achieve enough

Citation analysis o f papers in cleft lip and palate

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Table VII. The authors who contributed more than one article to the top 50 papers. Author Shprintzen, R. J. Bergland, O. Abyholm, F. E. Shaw, W. C. Mars, M. Cohen, M. M. Enemark H. Sindetpederson S. Semb, G. Wyzynski, D. F. Cohen, S. R. Asher McDade C. Plint, D. A. Houston, W. J. Buelow, K. H. Posnick, J. C. Ralph, P. S. Dahl, E. Magee, K. Magee, W. Brattstrom, V. McWilliams, J. Moisted, K. Prahl-Andersen, B. Beaty, T. H. Murray, J. C.

No. of papers 2 3 2 4 4 2 2 2 8 2 2 4 5 2 2 2 3 5 2 2 4 4 4 4 2 2

Position on authors list 1st author, 2-times 1st author, 2nd author, 4th author 1st author, 3rd author 1st author 2-times, 9th author, 10th author, 1st author 3-times, 5th author 1st author 2-times 1st author, 2nd author 1st author, 2nd author 1st author, 2nd author, 3rd author, 5th author, 8th author, 9th author 2-times, 11th author 1st author 2-times 1st author 2-times 1st author, 2nd author 2-times, 3rd author 2nd author, 6th author, 7th author 2-times, 8th author 2nd author, 3rd author 3rd author 2-times 2nd author, 3rd author 10th author, 11 author, 12th author 2nd author, 3rd author 2-times, 4th author 2-times 9th author 2-times 10th author 2-times 2nd author, 3rd author 2-times, 4th author 4th author, 5th author 2-times, 6th author 5th author, 6th author 2-times, 7th author 7th author, 8th author 2-times, 9th author 2nd author, 3rd author 1st author, 2nd author

citations to be included in the top 50 list. The 2003 highly influential paper by Sommerlad [73] on cleft repair was not included in the top 50 most cited papers also, possibly as a result of a phenomenon known as “obliteration by incorporation” [6,15], This is a theory whereby, as the subject matter of a published paper becomes “well known” and “common

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knowledge” within it’s particular field, it is no longer refer­ enced. This reflects some inherent limitations within this study design. To overcome this, it has been suggested that a more accurate evaluation of the most influential papers is to include the top 50 articles and the reference lists which they contain [15].

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Figure 1. Trend analysis of the numbers of published papers on cleft lip and palate in PubMed since 1910.

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N. A. Mahon and C. W. Joyce

It is commendable that almost 60% of the most cited articles originated from the US. This trend has been noted by many other citation analysis studies [9-12], According to the Institute of Scientific Information, the US holds the highest ranking in all 20 scientific disciplines [16,19], It is speculated that “national citing” and “auto citing” is responsible for this trend [74], US authors have gained a reputation for preferentially citing local papers and US reviewers often have a preference towards accepting local articles which have been submitted to their journals— “national bias” [74,75]. There is also an abundance of craniofacial specialist centres in the US in comparison to other countries, which may also account for their overwhelming representation in the top 50 list. It is pertinent to acknowledge the limitations of this study design. Obviously citations accumulate over time, therefore it is likely that older papers will have more citations, as they have had a longer period since publication to amass a greater number of citations [15,76,77]. Bohannon and Roberts [78] and Marx et al. [79] discussed the lifespan of published papers. Usually a scientific paper is first cited 1-2 years after it has been published and can take up to 10 years to gain it’s maximum quantity of citations. To overcome this problem, the “citation index” can alternatively be used [15]. This is calculated by dividing the number of citations an article has obtained by the number of years since the article was first published [80]. Citation analysis by virtue of its methodology can also be affected by different forms of bias. “Incomplete citing bias” can have a profound adverse effect on citation rates. It includes “self-citing”, “language bias” - citing only articles published in the English language, and “Omission bias” . “Omission bias” occurs when authors deliberately omit referencing another paper as both papers have conflicting results [7,81]. “Journal bias” occurs when high impact factor journals only publish articles that are guaranteed to be repeatedly referenced, such as sys­ tematic reviews, with the intention of increasing the journals impact factor [9,15]. The authors of this particular study acknowledge other potential limitations arising from this study design. First, the journals searched mainly included surgical journals; however, some papers on cleft may have been published in medical or paediatric journals. The web of knowledge is the most com­ monly used database for citation analysis but, had we used another database, our results may have been slightly different [15]. The reader should also be aware that this study reflects the top cited articles in January 2014, but this is a dynamic list as the included papers continue to be referenced at different rates which may alter their ranking over time. Despite these shortcomings, the value of performing a citation analysis is that it highlights the seminal papers that have historically influenced the specialty. By analyzing the top 50 list, future researchers gain a unique insight into the characteristics of a “classic paper” and can formulate future research ideas based on these characteristics to achieve mul­ tiple citations. From this study it is apparent that new surgical techniques are frequently published, so too are prospective cohort studies observing the long-term outcomes of cleft surgery. It is likely that papers written in the English language, originating from the US and involving multicentre studies will be published.

By performing a trend analysis it is possible to appreciate the changes in subject matter over time. The most cited papers of the 1950s and 1960s mainly dealt with the pathogenesis of cleft defects and the effects of untreated clefts in adulthood [2,31]. The 1970s articles introduced new syndromes and primary bone grafting [20,55,56]. This decade also began investigating facial growth patterns [65]. Further developments in bone grafting were published in the 1980s [21,41,54,62]. This era also introduced papers on experimental mice studies and the genetics of cleft [51]. Epidemiological studies discovering the variable incidences of cleft were prevalent in the 1980s, so too were the introduction of speech assessments [22,28,36,42]. By the 1990s animal experiments had developed into using canine subjects [66]. Fistula development was first explored at this stage [40]. Many papers covered the aesthetics of the nasal area [37,58,68]. Bone grafts were still evolving using the iliac crest bone [44]. It is at this point in time that external distraction methods are introduced and also further implications of the Robin Sequence [27,39,43,60], Finally in 2000 there is a shift to prenatal diagnosis of cleft defects [65], Similar to other citation studies, there is an upward trend in the number of papers being published in this specialty in recent years [81]. According to the Pubmed database there were 2162 and 3129 cleft publications in the 1990s and 2000s, respectively. This was not reflected in the top 50 most cited list, but this is likely due to their citable period being far shorter than their older counterparts (Figure 1). Conclusion Classic citation analysis has become a new valuable addition to the body of research within multiple specialties. It is a com­ prehensive guide to identify the most influential papers within its field. This study shows the historical evolution of cleft lip and palate research from 1945 to the present day. It emphasizes the highly regarded research articles that have influenced evidencebased clinical decision-making in this specialty. Acknowledgement The authors thank Mr J. McCann, Cleft Surgeon, Department of Plastic and Reconstructive Surgery, University Hospital Galway, Galway, Ireland. Declaration o f interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper. References [1] International Collaborative Research on Craniofacial Anoma­ lies. Available online at www.who.int/genomics/anomalies/en/. accessed January 2014. [2] Stark RB. The pathogenesis of harelip and cleft palate. Plast Reconstr Surg 1954;13:20-39. [3] Garfield E. The impact factor and its rightful use. Anaesthesist 1998;47:439-40. [4] Garfield E. Journal impact factor: a brief review. Can Med Assoc J 1999;161:979-80. [5] Saha S, Saint S, Christakis DA. Impact factor: a valid measure of journal quality? J Med Libr Assoc 2003;91:42-6. [6] Garfield E. 100 citation classics from the Journal of the Amer­ ican Medical Association. JAMA 1987;2:52-9. [7] Seglen PO. Why the impact factor of journals should not be used for evaluating research. BMJ 1997;314:(7079)498-502.

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A bibliometric analysis of the 50 most cited papers in cleft lip and palate.

Citation analysis is an established bibliometric method which catalogues papers according to the number of times they have been referenced. It is beli...
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