The Cleft Palate–Craniofacial Journal 52(4) pp. 437–446 July 2015 Ó Copyright 2015 American Cleft Palate–Craniofacial Association

ORIGINAL ARTICLE The 100 Most-Cited Human Cleft Lip and Palate–Related Articles Published in Dentistry, Oral Surgery, and Medicine Journals Panagiotis Christou, D.D.S., Ph.D., Gregory S. Antonarakis, D.D.S., M.Sc., Ph.D. Objective: To identify the 100 most-cited articles pertaining to human cleft lip and palate research published in dentistry, oral surgery, and medicine journals and to identify their principal bibliometric characteristics. Design: Web-based bibliometric analysis. Main Outcome Measure: The Web of Science was searched to identify the 100 most-cited clinical articles related to cleft lip and/or palate. Information was extracted with regard to total number of citations, number of authors, affiliations, year, and journal of publication, Medical Subject Headings, type of study, specific area of study. Trends in citations were assessed. Results: The 100 most-cited articles identified received between 437 and 58 citations. The oldest was published in 1954 and the most recent in 2008. The number of authors ranged from 1 to 12, with an average of three authors per article. Most of the first authors were affiliated with institutions in the United States, with the most prolific institution being the University of Iowa. More than 70% of the studies appeared in The Cleft Palate–Craniofacial Journal. There was a significant negative correlation between average citations per year and time since publication (P , .001); whereas, a significant positive correlation was observed between average citations per year and number of total citations (P , .001). Conclusions: The 100 most-cited articles in human cleft lip and palate research published in dentistry, oral surgery, and medicine journals are listed and characterized. This can be used as a potential knowledge base for specialists in training or to produce relevant knowledge defining the direction of future research. KEY WORDS:

bibliometrics, citation analysis, cleft, impact factor, most-cited articles, publication, Web of Science

to inform the reader about pertinent results or to corroborate their findings and support their argument or hypothesis (Dawson, 1989; White and Wang, 1997; Nieri et al., 2007). The citation index of any scientific article is the number of citations it receives after its publication. There is speculation that the more often a publication is cited, the more impact it will have in the specialty community and the more it may subsequently influence clinical practice (Lefaivre et al., 2011; Feijoo et al., 2014). These publications, small in number but disproportionate in impact, may define the practice of the specialty and serve as a foundation for new methods, procedures, or concepts (Ponce and Lozano, 2010). No attempt to date has been made to identify the mostcited articles in cleft lip and palate research and evaluate them with bibliometric measures, although the necessary information is collected and available in online resources such as the Web of Science. Bibliometrics is a set of methods used to quantitatively analyze academic literature (De Bellis, 2009). The aim of the current study was to identify the 100 most-cited articles pertaining to human cleft lip and palate research published in dentistry, oral surgery, and medicine journals and to identify their principal bibliometric characteristics.

Cleft lip and palate–related research has seen tremendous development over the past 50 years. Scientific literature in this field can be found in many different journals and across many different specialties due to its multidisciplinary nature. The only dedicated, cleft-specific journal, The Cleft Palate–Craniofacial Journal (CPCJ), has grown in scope and thrived since its beginnings (Sharp, 2014). There is, nevertheless, a low publication rate within the field, with a relatively long lead time but with a higher publication rate in science- and surgery-based subjects (Moar and Butterworth, 2013). A measure of the academic importance of a published article in a specific discipline is the number of times it has been cited by other authors (Moed, 2009). Authors cite an existing publication for several reasons, including the desire

Dr. Christou, Private Practice, Centre Dentaire Lancy; and Dr. Antonarakis is Orthodontist, Department of Orthodontics, Dental School, University of Geneva, Geneva, Switzerland. Submitted March 2014; Revised May 2014; Accepted June 2014. Address correspondence to: Dr. Panagiotis Christou, Centre Dentaire Lancy, Chemin de la Caroline 18a, 1213 Petit Lancy, Geneva, Switzerland. E-mail [email protected]. DOI: 10.1597/14-085 437

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MATERIAL

AND

METHODS

Article Selection A database search was carried out using the Web of Science (WoS v5.12) in December 2013 to identify the 100 most-cited clinical articles related to cleft lip and/or palate published in peer-reviewed journals included in the WoS category ‘‘Dentistry, Oral Surgery and Medicine.’’ The only search term (keyword) used was ‘‘cleft’’ (using the truncation search ‘‘cleft$’’ in order to find both singular and plural), and the search was carried out using the WoS function pertaining to all databases as opposed to the core collection, with all years inserted in the timespan (1900 to 2013). No restrictions were set on language, date of publication, or article type. The database search and article selection were carried out by two independent reviewers and their output was compared. All results were cross-checked, and whenever there was a discrepancy between the two reviewers, a consensus decision was reached. Inclusion criteria were as follows: articles pertaining specifically to cleft lip and/or palate; any article type (including reviews, case reports, etc.); and articles on humans. Exclusion criteria were the following: animal studies; experimental laboratory (in vitro) or basic science studies that lacked a clinical component; non– cleft-specific studies (e.g., studies that included cleft subjects within their sample among other noncleft subjects; and studies describing techniques that can potentially be used with various patient groups including but not solely comprising cleft patients).

Articles were also classified as per specific area of study as follows: genetics; epidemiology; anatomy; classifications/indices; growth, tooth anomalies/formation, and occlusion; imaging; infant orthopedics; primary surgery; alveolar bone grafting; maxillofacial surgery; speech and otorhinolaryngology; comprehensive care; multicenter outcome studies; psychology and quality of life; syndromes and Pierre Robin sequence; cleft research reviews. Citation Index and Trends To overcome the likelihood that older articles have more citations purely because their citable period is longer (citation bias), the citation index was calculated for the first and last 10 articles identified based on citation ranking. This citation index was defined as the mean number of times articles were cited per year until 15 years after publication. For articles that had been published fewer than 15 years prior to the database search, the total number of citations since publication was divided by the number of years from publication (Loonen et al., 2008). Trends in citations were also evaluated. The impact factors of journals listed in the 2012 Journal Citation Reports Science Edition (http://thomsonreuters.com; accessed December 2013) were retrieved and adopted as quantitative tools for evaluating journals in which these articles were published. Nonparametric (Spearman) correlation was used to assess the association between years since publication (with reference to the year 2013), number of authors, the journal’s impact factor, and average citations per year.

Data Extraction and Elaboration RESULTS The following information was extracted from each of the 100 most-cited articles: total number of citations; ranking based on number of citations; number of authors; author affiliations; year of publication; journal of publication; Medical Subject Headings (MeSH terms) extracted from PubMed; type of study; and specific area of study. The affiliation of the first author or, when not available, the article’s reprint address, was used to define the geographic distribution of the studies as per the ‘‘absolute country counting’’ method (Egghe et al., 2000). The studies were classified according to type of study based on the pyramid of evidence in the following categories: systematic reviews and meta-analyses; randomized controlled trials; cohort studies; case-control studies; cross-sectional surveys; case reports/case series; and reports and reviews (Greenhalgh, 1997). The WoS does not provide a mechanism to restrict search by article type, and thus the two reviewers accomplished this by reading through the methodology of each of the selected articles.

The initial database search resulted in a total of 210,892 articles. The 200 most-cited articles published in dentistry, oral surgery, and medicine journals were selected, and the title, abstract, and if necessary, the full text were consulted. Based on the inclusion and exclusion criteria, the 100 mostcited articles were selected. The 100 most-cited articles identified (Table 1) were cited from 437 (most cited; Shprintzen et al., 1978) to 58 (100th most cited; Beirne et al., 1996) times. Among these articles, the oldest were published in 1954 (Graber, 1954; Harvold, 1954); whereas the most recent, in 2008 (Vieira, 2008). Eight of the articles were published during or after the year 2000. The average number of citations per year ranged from 1.1 (Harvold, 1954) to 15.6 (Vieira, 2008). Citation Index The citation index for the first 15 years after publication displayed a significant difference (P , .01) when comparing the 10 most-cited (mean, 4.4 citations per

Christou and Antonarakis, 100 MOST-CITED CLEFT LIP AND PALATE–RELATED ARTICLES

TABLE 1

439

100 Most-Cited Articles, in Order of Number of Citations per Article

Rank

Article Reference

Citations

1.

Shprintzen RJ, Goldberg RB, Lewin ML, Sidoti EJ, Berkman MD, Argamaso RV, Young D. A new syndrome involving cleft palate, cardiac anomalies, typical facies, and learning disabilities: velo-cardio-facial syndrome. Cleft Palate J. 1978;15:56–62. Tessier P. Anatomical classification facial, cranio-facial and latero-facial clefts. J Maxillofac Surg. 1976;4:69–92. Boyne PJ, Sands NR. Secondary bone grafting of residual alveolar and palatal clefts. J Oral Surg. 1972;30:87–92. Bergland O, Semb G, Abyholm FE. Elimination of the residual alveolar cleft by secondary bone grafting and subsequent orthodontic treatment. Cleft Palate J. 1986;23:175–205. Ross RB. Treatment variables affecting facial growth in complete unilateral cleft lip and palate. Cleft Palate J. 1987;24:5–77. Vanderas AP. Incidence of cleft lip, cleft palate, and cleft lip and palate among races: a review. Cleft Palate J. 1987;24:216–225. Shaw WC, Dahl E, Asher-McDade C, Brattstrom ¨ V, Mars M, McWilliam J, Mølsted K, Plint DA, Prahl-Andersen B, Roberts C, et al. A six-center international study of treatment outcome in patients with clefts of the lip and palate: Part 5. General discussion and conclusions. Cleft Palate Craniofac J. 1992;29:413–418. Golding-Kushner KJ, Argamaso RV, Cotton RT, Grames LM, Henningsson G, Jones DL, Karnell MP, Klaiman PG, Lewin ML, Marsh JL, et al. Standardization for the reporting of nasopharyngoscopy and multiview videofluoroscopy: a report from an International Working Group. Cleft Palate J. 1990;27:337–347. Mars M, Houston WJ. A preliminary study of facial growth and morphology in unoperated male unilateral cleft lip and palate subjects over 13 years of age. Cleft Palate J. 1990;27:7–10. Ranta R. A review of tooth formation in children with cleft lip/palate. Am J Orthod Dentofacial Orthop. 1986;90:11–18. Trost JE. Articulatory additions to the classical description of the speech of persons with cleft palate. Cleft Palate J. 1981;18:193–203. Wyszynski DF, Beaty TH, Maestri NE. Genetics of nonsyndromic oral clefts revisited. Cleft Palate Craniofac J. 1996;33:406– 417. Grayson BH, Santiago PE, Brecht LE, Cutting CB. Presurgical nasoalveolar molding in infants with cleft lip and palate. Cleft Palate Craniofac J. 1999;36:486–498. Boyne PJ, Sands NR. Combined orthodontic-surgical management of residual palato-alveolar cleft defects. Am J Orthod. 1976;70:20–37. Skoog T. The use of periosteal flaps in the repair of clefts of the primary palate. Cleft Palate J. 1965;2:332–339. Jensen BL, Kreiborg S, Dahl E, Fogh-Andersen P. Cleft lip and palate in Denmark, 1976–1981: epidemiology, variability, and early somatic development. Cleft Palate J. 1988;25:258–269. Mars M, Plint DA, Houston WJ, Bergland O, Semb G. The Goslon Yardstick: a new system of assessing dental arch relationships in children with unilateral clefts of the lip and palate. Cleft Palate J. 1987;24:314–322. Shprintzen RJ, Lewin ML, Croft CB, Daniller AI, Argamaso RV, Ship AG, Strauch B. A comprehensive study of pharyngeal flap surgery: tailor made flaps. Cleft Palate J. 1979;16:46–55. Shprintzen RJ. The implications of the diagnosis of Robin sequence. Cleft Palate Craniofac J. 1992;29:205–209. Hotz M, Gnoinski W. Comprehensive care of cleft lip and palate children at Zurich university: a preliminary report. Am J ¨ Orthod. 1976;70:481–504. Semb G. A study of facial growth in patients with unilateral cleft lip and palate treated by the Oslo CLP Team. Cleft Palate Craniofac J. 1991;28:1–21. Enemark H, Sindet-Pedersen S, Bundgaard M. Long-term results after secondary bone grafting of alveolar clefts. J Oral Maxillofac Surg. 1987;45:913–919. Wyszynski DF, Duffy DL, Beaty TH. Maternal cigarette smoking and oral clefts: a meta-analysis. Cleft Palate Craniofac J. 1997;34:206–210. Dalston RM, Warren DW, Dalston ET. Use of nasometry as a diagnostic tool for identifying patients with velopharyngeal impairment. Cleft Palate Craniofac J. 1991;28:184–188. Sindet-Pedersen S, Enemark H. Reconstruction of alveolar clefts with mandibular or iliac crest bone grafts: a comparative study. J Oral Maxillofac Surg. 1990;48:554–558. Stool SE, Randall P. Unexpected ear disease in infants with cleft palate. Cleft Palate J. 1967;4:99–103. Jones MC. Etiology of facial clefts: prospective evaluation of 428 patients. Cleft Palate J. 1988;25:16–20. Figueroa AA, Polley JW. Management of severe cleft maxillary deficiency with distraction osteogenesis: procedure and results. Am J Orthod Dentofacial Orthop. 1999;115:1–12. Sher AE. Mechanisms of airway obstruction in Robin sequence: implications for treatment. Cleft Palate Craniofac J. 1992;29:224–231. Skolnick ML. Videofluoroscopic examination of the velopharyngeal portal during phonation in lateral and base projections—a new technique for studying the mechanics of closure. Cleft Palate J. 1970;7:803–816. Kaplan EN. The occult submucous cleft palate. Cleft Palate J. 1975;12:356–368. Murray JC, Daack-Hirsch S, Buetow KH, Munger R, Espina L, Paglinawan N, Villanueva E, Rary J, Magee K, Magee W. Clinical and epidemiologic studies of cleft lip and palate in the Philippines. Cleft Palate Craniofac J. 1997;34:7–10. Schweckendiek W, Doz P. Primary veloplasty: long-term results without maxillary deformity. A twenty-five year report. Cleft Palate J. 1978;15:268–274. Shaw WC, Asher-McDade C, Brattstrom ¨ V, Dahl E, McWilliam J, Mølsted K, Plint DA, Prahl-Andersen B, Semb G, The RP. A six-center international study of treatment outcome in patients with clefts of the lip and palate: Part 1. Principles and study design. Cleft Palate Craniofac J. 1992;29:393–397. Lidral AC, Murray JC, Buetow KH, Basart AM, Schearer H, Shiang R, Naval A, Layda E, Magee K, Magee W. Studies of the candidate genes TGFB2, MSX1, TGFA, and TGFB3 in the etiology of cleft lip and palate in the Philippines. Cleft Palate Craniofac J. 1997;34:1–6. Hall HD, Posnick JC. Early results of secondary bone grafts in 106 alveolar clefts. J Oral Maxillofac Surg. 1983;41:289–294. Hogan VM. A clarification of the surgical goals in cleft palate speech and the introduction of the lateral port control (l.p.c.) pharyngeal flap. Cleft Palate J. 1973;10:331–345. Witzel MA, Salyer KE, Ross RB. Delayed hard palate closure: the philosophy revisited. Cleft Palate J. 1984;21:263–269. Cohen MM Jr. Syndromes with cleft lip and cleft palate. Cleft Palate J. 1978;15:306–328. Warren DW. Perci: a method for rating palatal efficiency. Cleft Palate J. 1979;16:279–285.

437

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.

302 273 256 224 187 150

154

148 140 133 121 120 116 117 115 115 114 113 112 112 107 106 100 100 98 96 94 94 94 93 93 93 91

90

89 88 87 86 86

440

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TABLE 1

Continued

Rank

Article Reference

Citations

41.

Turvey TA, Vig K, Moriarty J, Hoke J. Delayed bone grafting in the cleft maxilla and palate: a retrospective multidisciplinary analysis. Am J Orthod. 1984;86:244–256. Mars M, Asher-McDade C, Brattstrom ¨ V, Dahl E, McWilliam J, Mølsted K, Plint DA, Prahl-Andersen B, Semb G, Shaw WC, et al. A six-center international study of treatment outcome in patients with clefts of the lip and palate: Part 3. Dental arch relationships. Cleft Palate Craniofac J. 1992;29:405–408. Farkas LG, Hajnis K, Posnick JC. Anthropometric and anthroposcopic findings of the nasal and facial region in cleft patients before and after primary lip and palate repair. Cleft Palate Craniofac J. 1993;30:1–12. Warren DW. Compensatory speech behaviors in individuals with cleft palate: a regulation/control phenomenon? Cleft Palate J. 1986;23:251–260. Warren DW, Duany LF, Fischer ND. Nasal pathway resistance in normal and cleft lip and palate subjects. Cleft Palate J. 1969;6:134–140. Hardin MA, Van Demark DR, Morris HL, Payne MM. Correspondence between nasalance scores and listener judgments of hypernasality and hyponasality. Cleft Palate Craniofac J. 1992;29:346–351. Jordan RE, Kraus BS, Neptune CM. Dental abnormalities associated with cleft lip and/or palate. Cleft Palate J. 1966;3:22–55. Krogman WM, Mazaheri M, Harding RL, Ishiguro K, Bariana G, Meier J, Canter H, Ross P. A longitudinal study of the craniofacial growth pattern in children with clefts as compared to normal, birth to six years. Cleft Palate J. 1975;12:59–84. Ross RB. The clinical implications of facial growth in cleft lip and palate. Cleft Palate J. 1970;7:37–47. Vieira AR. Unraveling human cleft lip and palate research. J Dent Res. 2008;87:119–125. Asher-McDade C, Brattstrom ¨ V, Dahl E, McWilliam J, Mølsted K, Plint DA, Prahl-Andersen B, Semb G, Shaw WC, The RP. A six-center international study of treatment outcome in patients with clefts of the lip and palate: Part 4. Assessment of nasolabial appearance. Cleft Palate Craniofac J. 1992;29:409–412. Bishara SE, Krause CJ, Olin WH, Weston D, Ness JV, Felling C. Facial and dental relationships of individuals with unoperated clefts of the lip and/or palate. Cleft Palate J. 1976;13:238–252. Argamaso RV. Glossopexy for upper airway obstruction in Robin sequence. Cleft Palate Craniofac J. 1992;29:232–238. Derijcke A, Eerens A, Carels C. The incidence of oral clefts: a review. Br J Oral Maxillofac Surg. 1996;34:488–494. Mølsted K, Asher-McDade C, Brattstrom ¨ V, Dahl E, Mars M, McWilliam J, Plint DA, Prahl-Andersen B, Semb G, Shaw WC, et al. A six-center international study of treatment outcome in patients with clefts of the lip and palate: Part 2. Craniofacial form and soft tissue profile. Cleft Palate Craniofac J. 1992;29:398–404. Sindet-Pedersen S, Enemark H. Mandibular bone-grafts for reconstruction of alveolar clefts. J Oral Maxillofac Surg. 1988;46:533–537. Ayoub A, Garrahy A, Hood C, White J, Bock M, Siebert JP, Spencer R, Ray A. Validation of a vision-based, threedimensional facial imaging system. Cleft Palate Craniofac J. 2003;40:523–529. Croft CB, Shprintzen RJ, Daniller A, Lewin ML. Occult submucous cleft-palate and musculus uvulae. Cleft Palate J. 1978;15:150–154. Cosman B, Falk AS. Delayed hard palate repair and speech deficiencies: a cautionary report. Cleft Palate J. 1980;17:27–33. Endriga MC, Kapp-Simon KA. Psychological issues in craniofacial care: state of the art. Cleft Palate Craniofac J. 1999;36:3–11. Graber TM. The congenital cleft palate deformity. J Am Dent Assoc. 1954;48:375–395. Broder H, Strauss RP. Self-concept of early primary school age children with visible or invisible defects. Cleft Palate J. 1989;26:114–117. Cobourne MT. The complex genetics of cleft lip and palate. Eur J Orthod. 2004;26:7–16. Kriens OB. Fundamental anatomic findings for an intravelar veloplasty. Cleft Palate J. 1970;7:27–36. Stoll C, Alembik Y, Dott B, Roth MP. Associated malformations in cases with oral clefts. Cleft Palate Craniofac J. 2000;37:41– 47. Witsenburg B. The reconstruction of anterior residual bone defects in patients with cleft lip, alveolus and palate. A review. J Maxillofac Surg. 1985;13:197–208. Grayson BH, Cutting CB. Presurgical nasoalveolar orthopedic molding in primary correction of the nose, lip, and alveolus of infants born with unilateral and bilateral clefts. Cleft Palate Craniofac J. 2001;38:193–198. Kapp K. Self concept of the cleft lip and or palate child. Cleft Palate J. 1979;16:171–176. Richman LC. Behavior and achievement of cleft palate children. Cleft Palate J. 1976;13:4–10. Troxell JB, Fonseca RJ, Osbon DB. A retrospective study of alveolar cleft grafting. J Oral Maxillofac Surg. 1982;40:721–725. Kapp-Simon K. Self-concept of primary-school-age children with cleft lip, cleft palate, or both. Cleft Palate J. 1986;23:24–27. McComb HK, Coghlan BA. Primary repair of the unilateral cleft lip nose: completion of a longitudinal study. Cleft Palate Craniofac J. 1996;33:23–30. Enemark H, Krantz-Simonsen E, Schramm JE. Secondary bonegrafting in unilateral cleft lip palate patients: indications and treatment procedure. Int J Oral Surg. 1985;14:2–10. Harvold E. Cleft lip and palate: morphologic studies of the facial skeleton. Am J Orthod. 1954;40:493–506. Hayashi I, Sakuda M, Takimoto K, Miyazaki T. Craniofacial growth in complete unilateral cleft lip and palate: a roentgenocephalometric study. Cleft Palate J. 1976;13:215–237. ´ K. Maternal smoking and orofacial clefts. Cleft Palate Craniofac J. 1997;34:11–16. Kall ¨ en Williams AC, Bearn D, Mildinhall S, Murphy T, Sell D, Shaw WC, Murray JJ, Sandy JR. Cleft lip and palate care in the United Kingdom—the Clinical Standards Advisory Group (CSAG) Study. Part 2: dentofacial outcomes and patient satisfaction. Cleft Palate Craniofac J. 2001;38:24–29. Drommer R, Luhr HG. The stabilization of osteotomized maxillary segments with Luhr mini-plates in secondary cleft surgery. J Maxillofac Surg. 1981;9:166–169. Huddart AG, Bodenham RS. The evaluation of arch form and occlusion in unilateral cleft palate subjects. Cleft Palate J. 1972;9:194–209. Asher-McDade C, Roberts C, Shaw WC, Gallager C. Development of a method for rating nasolabial appearance in patients with clefts of the lip and palate. Cleft Palate Craniofac J. 1991;28:385–390. Oyama T, Nishimoto S, Tsugawa T, Shimizu F. Efficacy of platelet-rich plasma in alveolar bone grafting. J Oral Maxillofac Surg. 2004;62:555–558.

85

42.

43. 44. 45. 46. 47. 48. 49. 50. 51.

52. 53. 54. 55.

56. 57. 58. 59. 60. 61. 62. 63. 64. 65. 66. 67. 68. 69. 70. 71. 72. 73. 74. 75. 76. 77.

78. 79. 80. 81.

82

81 80 80 79 79 79 79 78 76

76 75 75 75

75 72 72 71 71 71 70 70 70 70 70 69 69 69 69 68 68 67 67 67 67 67

66 66 65 65

Christou and Antonarakis, 100 MOST-CITED CLEFT LIP AND PALATE–RELATED ARTICLES

TABLE 1

441

Continued

Rank

Article Reference

Citations

82.

Bishara SE. Cephalometric evaluation of facial growth in operated and non-operated individuals with isolated clefts of the palate. Cleft Palate J. 1973;10:239–246. Hunt O, Burden D, Hepper P, Johnston C. The psychosocial effects of cleft lip and palate: a systematic review. Eur J Orthod. 2005;27:274–285. Richman LC. Self-reported social, speech, and facial concerns and personality adjustment of adolescents with cleft lip and palate. Cleft Palate J. 1983;20:108–112. Smahel Z, Brejcha M. Differences in craniofacial morphology between complete and incomplete unilateral cleft lip and palate in adults. Cleft Palate J. 1983;20:113–127. Borstlap WA, Heidbuchel KL, Freihofer HP, Kuijpers-Jagtman AM. Early secondary bone grafting of alveolar cleft defects. A comparison between chin and rib grafts. J Craniomaxillofac Surg. 1990;18:201–205. Broder HL, Richman LC, Matheson PB. Learning disability, school achievement, and grade retention among children with cleft: a two-center study. Cleft Palate Craniofac J. 1998;35:127–131. Hotz MM, Gnoinski WM. Effects of early maxillary orthopaedics in coordination with delayed surgery for cleft lip and palate. J Maxillofac Surg. 1979;7:201–210. Spriestersbach DC, Dickson DR, Fraser FC, Horowitz SL, McWilliams BJ, Paradise JL, Randall P. Clinical research in cleft lip and cleft palate: the state of the art. Cleft Palate J. 1973;10:113–165. Hagberg C, Larson O, Milerad J. Incidence of cleft lip and palate and risks of additional malformations. Cleft Palate Craniofac J. 1998;35:40–45. Kortebein MJ, Nelson CL, Sadove AM. Retrospective analysis of 135 secondary alveolar cleft grafts using iliac or calvarial bone. J Oral Maxillofac Surg. 1991;49:493–498. Maull DJ, Grayson BH, Cutting CB, Brecht LL, Bookstein FL, Khorrambadi D, Webb JA, Hurwitz DJ. Long-term effects of nasoalveolar molding on three-dimensional nasal shape in unilateral clefts. Cleft Palate Craniofac J. 1999;36:391–397. Freihofer HP, Borstlap WA, Kuijpers-Jagtman AM, Voorsmit RA, van Damme PA, Heidbuchel KL, Borstlap-Engels VM. ¨ Timing and transplant materials for closure of alveolar clefts. A clinical comparison of 296 cases. J Craniomaxillofac Surg. 1993;21:143–148. Marsh JL, Grames LM, Holtman B. Intravelar veloplasty: a prospective study. Cleft Palate J. 1989;26:46–50. Mazaheri M, Harding RL, Cooper JA, Meier JA, Jones TS. Changes in arch form and dimensions of cleft patients. Am J Orthod. 1971;60:19–32. Bardach J, Eisbach KJ. The influence of primary unilateral cleft lip repair on facial growth. Cleft Palate J. 1977;14:88–97. Bishara SE, de Arrendondo RS, Vales HP, Jakobsen JR. Dentofacial relationships in persons with unoperated clefts: comparisons between three cleft types. Am J Orthod. 1985;87:481–507. Latham RA. Orthopedic advancement of the cleft maxillary segment: a preliminary report. Cleft Palate J. 1980;17:227–233. Richman LC, Eliason MJ, Lindgren SD. Reading disability in children with clefts. Cleft Palate J. 1988;25:21–25. Beirne JC, Barry HJ, Brady FA, Morris VB. Donor site morbidity of the anterior iliac crest following cancellous bone harvest. Int J Oral Maxillofac Surg. 1996;25:268–271.

64

83. 84. 85. 86. 87. 88. 89. 90. 91. 92. 93.

94. 95. 96. 97. 98. 99. 100.

year) with the 10 least-cited (mean, 2.2 citations per year) articles from the top 100 (Fig. 1). Number of Authors The number of authors ranged from 1 to 12, with an average of three authors per article. Two articles were authored by 12 authors and 30 articles, by a single author. The authors appearing most often within the

64 64 64 63 63 63 63 62 62 62 61

61 61 60 60 60 60 58

100 most-cited articles were Semb G (eight contributions) and Shaw WC (seven contributions) (Table 2). Contributors appearing most often as first authors in the top citations were Mars M (three times); Richman LC (three times); Shprintzen RJ (three times); Warren DW (three times). Geographic and Journal Distribution Most of the first authors were affiliated with institutions in the United States (n ¼ 57), followed by those in the U.K. (n ¼ 10) and Denmark (n ¼ 6) (Table TABLE 2 Authors Appearing Most Often Within the 100 MostCited Articles

FIGURE 1 Number of citations per year since publication for the 10 most-cited (black line) and least-cited (gray line) articles within the 100 most-cited articles for the first 15 years after publication.

Author

No. of Contributions

Semb G Shaw WC Asher-McDade C Plint DA Brattstrom ¨ V Dahl E Mars M McWilliam K Mølsted K Prahl-Andersen B

8 7 6 6 5 5 5 5 5 5

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TABLE 3 Geographic Distribution of Articles as per First Author Affiliation

TABLE 4

Institutional Affiliation of Articles as per First Author

Country

No. of Articles

Affiliation

No. of Articles

USA UK Denmark Canada Germany Norway Sweden The Netherlands Switzerland Australia Belgium Czechoslovakia Finland France Ireland Japan

57 10 6 5 4 3 3 3 2 1 1 1 1 1 1 1

University of Iowa, Iowa City, IA, USA Montefiore Medical Center, Bronx, NY, USA University of North Carolina, Chapel Hill, NC, USA Aarhus Cleft Palate Clinic, Aarhus, Denmark The Hospital for Sick Children, Toronto, Ontario, Canada New York University Medical Center, New York, NY, USA Hospital for Sick Children, London, UK Lancaster Cleft Palate Clinic, Lancaster, PA, USA University Hospital of Oslo, Oslo, Norway University of Illinois, Chicago, IL, USA University of Nijmegen, Nijmegen, The Netherlands University of Pittsburgh, Pittsburgh, PA, USA Children’s Hospital of Los Angeles, Los Angeles, CA, USA Johns Hopkins University, Baltimore, MD, USA Loma Linda University Medical Center, Loma Linda, CA, USA Northwestern University, Evanston, IL, USA Royal Dental College, Copenhagen, Denmark University of Manchester, Manchester, UK Zurich University, Zurich, Switzerland Academy of Sciences, Prague, Czechoslovakia Albany Medical College, Albany, NY, USA California State University, Sacramento, CA, USA Case Western Reserve University, Cleveland, OH, USA Children’s Hospital Philadelphia, Philadelphia, PA, USA GKT Dental Institute, London, UK Glasgow Dental Hospital and School, Glasgow, Scotland, UK Helsinki University, Helsinki, Finland Indiana University, Indianapolis, IN, USA Karolinska Institute, Solna, Sweden Kobe Children’s Hospital, Kobe, Japan Princess Margaret Hospital for Children, Perth, Australia Private practice, Marburg, Germany Queen’s University Belfast, Belfast, Northern Ireland, UK Stanford University, Stanford, CA, USA St James Hospital, Dublin, Ireland University Hospital, Hamburg, Germany University of Bristol, Bristol, UK University of California, Irvine, Irvine, CA, USA University of California, San Diego, CA, USA University of Gottingen, Gottingen, Germany ¨ ¨ University of Leuven, Leuven, Belgium University of Lund, Lund, Sweden University of Munster, Munster, Germany ¨ ¨ University of New Jersey, Newark, NJ, USA University of Strasbourg, Strasbourg, France University of Uppsala, Uppsala, Sweden University of Washington, Seattle, WA, USA University of Western Ontario, London, Ontario, Canada Upstate Medical Center, Syracuse, NY, USA Vanderbilt University Medical Center, Nashville, TN, USA Washington University Medical Center, St. Louis, MO, USA Wordsley Hospital, Worcestershire, UK

11 6 6 4 4 4 3 3 3 3 3 3 2 2 2 2 2 2 2 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1 1

3). The articles were derived from 52 departments/ institutions, and the most prolific were the University of Iowa (n ¼ 11) and the Center for Craniofacial Disorders, Montefiore Medical Center, New York (n ¼ 6) (Table 4). The studies appeared in 10 journals overall, led by the CPCJ (n ¼ 71) where the vast majority of studies were published (Table 5). MeSH Terms, Article Categories, and Types The most frequently used MeSH terms in the articles selected were ‘‘cleft palate’’ (n ¼ 139), ‘‘cleft lip’’ (n ¼ 111), ‘‘humans’’ (n ¼ 96) and ‘‘child’’ (n ¼ 80). Other common MeSH terms included ‘‘infant’’ (n ¼ 44), ‘‘male’’ (n ¼ 44), ‘‘female’’ (n ¼ 41), ‘‘adolescent’’ (n ¼ 36), ‘‘alveolar process’’ (n ¼ 24), ‘‘adult’’ (n ¼ 22), ‘‘cephalometric’’ (n ¼ 21), ‘‘bone transplantation’’ (n ¼ 20), ‘‘maxilla’’ (n ¼ 20), ‘‘maxillofacial development’’ (n ¼ 20), ‘‘nose’’ (n ¼ 19), ‘‘palate’’ (n ¼ 19), and ‘‘velopharyngeal insufficiency’’ (n ¼ 15). The MeSH terms were sometimes used more than once within a single article, with different subheadings, such as the use of the terms ‘‘cleft palate/surgery’’ and ‘‘cleft palate/ complications’’ within the same article. This explains why some MeSH terms appeared more than 100 times, although there were only 100 selected articles. The 100 most-cited articles could be classified most commonly in the following specific areas of study: alveolar bone grafting (n ¼ 16); growth, tooth anomalies/formation, and occlusion (n ¼ 13); speech and otorhinolaryngology (n ¼ 11); psychology and quality of life (n ¼ 9); epidemiology (n ¼ 9); anatomy (n ¼ 8); primary surgery (n ¼ 6); multicenter outcome studies (n ¼ 6); syndromes and Pierre Robin sequence (n ¼ 5); and infant orthopedics (n ¼ 4). The rest of the specific areas of study had three or fewer articles within each category. Most articles pertaining to anatomy as well as to growth, tooth anomalies/formation, and occlusion were published in the 1970s (38% and 62%, respectively).

Most articles pertaining to alveolar bone grafting as well as psychology and quality of life were published in the 1980s (50% and 44%, respectively), and most articles pertaining to infant orthopedics, epidemiology, multicenter outcome studies, and syndromes and Pierre Robin sequence were published in the 1990s (50%, 56%, 83%, and 60%, respectively). Articles pertaining to speech and otorhinolaryngology as well as primary surgery showed a more even distribution among the decades. Among the 100 most-cited articles, the types of studies varied with most common being cohort studies

Christou and Antonarakis, 100 MOST-CITED CLEFT LIP AND PALATE–RELATED ARTICLES

443

TABLE 5 Journals Where the 100 Most-Cited Articles Appear, With Only the Current Journal Titles Included (Impact Factor Stated Is as per the 2012 Journal Citation Reports Science Edition)

Journal Title The Cleft Palate–Craniofacial Journal American Journal of Orthodontics and Dentofacial Orthopedics Journal of Oral and Maxillofacial Surgery Journal of Cranio-Maxillo-Facial Surgery British Journal of Oral and Maxillofacial Surgery European Journal of Orthodontics International Journal of Oral and Maxillofacial Surgery Journal of Dental Research Journal of the American Dental Association

No. of Articles

Impact Factor (2012)

71

1.238

8 8 6 2 2

1.458 1.333 1.610 2.717 1.078

2 1 1

1.521 3.826 1.822

(n ¼ 38). The remaining studies were cross-sectional surveys (n ¼ 20), reviews (n ¼ 15), reports (n ¼ 13), casecontrol studies (n ¼ 6), case series (n ¼ 6), and systematic reviews (n ¼ 2). Correlations The Spearman correlation was used to assess the association between years since publication, numbers of authors, journal impact factor, and average citations per year. There was a significant negative correlation between average citations per year and time since publication (Spearman q ¼ .730; P , .001) (Fig. 2); whereas, significant positive correlation was observed between average citations per year and the number of total citations (Spearman q ¼ .512; P , .001) (Fig. 3). No significant correlation was found between the average citations per year and the number of authors (Spearman q ¼ 0.163; P ¼ .105) or average citations per

FIGURE 2 Scatterplot showing the number of citations per year in relation to the time since publication, with a line of best fit.

FIGURE 3 Scatterplot showing the number of citations per year in relation to the total number of citations, with a line of best fit.

year and the journal impact factor (Spearman q ¼.108; P ¼ .286). DISCUSSION In the present study we identified and analyzed the 100 most-cited clinical human articles in cleft lip and palate research, published in dentistry, oral surgery, and medicine journals, to date. By definition, a publication cited more than 400 times is considered a ‘‘citation classic’’ (Garfield, 1973). In our study, only the first of the studies could fulfill this criterion, highlighting the scarcity of research and the small size of the specific field. Therefore, we refer to the 100 most-cited articles as opposed to citation classics in the field. Similar to any other bibliometric study, certain general limitations are inherent in the present study design. The WoS as a search tool, which we rely on for its comprehensiveness and accuracy, may present certain biases. First, the results of searches conducted in the WoS via the resources of a particular institution may not match the results from another institution, given that each institution may subscribe to or have access to different resources within the WoS. Additionally, the WoS does not include citations in books (Dumont, 1989). Other, more general limitations not related to the WoS as a search tool but to citations in general include the possibility of some articles containing incomplete or erroneous citations and the observations that authors cite articles from journals in which they hope to publish and foreign language articles are often cited by articles written in the same language (Seglen, 1997). This may mean that important articles may be excluded from the Englishlanguage literature simply due to the language in which they were written. Regarding the limitations of the search, important and relevant articles may have been missed if

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published in journals not included in the WoS category ‘‘Dentistry, Oral Surgery and Medicine.’’ Articles using other terms than ‘‘cleft’’ for the same condition may have also been omitted. Citation analysis appears to be a valid and objective measure of the impact of scientific work on the rest of the scientific community (Garfield, 1973; Virgo, 1977). Critics of citation analysis, however, argue that citations are biased in favor of authors with previous major contributions in the field (May, 1967), authors who write review articles (Woodward and Hensman, 1976), and those whose articles are methodological or are in established fields with many researchers (Margolis, 1967). Our findings do not lend support to the aforementioned evidence. Most of the identified studies were cohort studies (n ¼ 38), with only 15 reviews. Furthermore, the highest reoccurrence of first authors in the identified articles was three, and no correlation between the number of authors and the citations received was observed. We could only find two studies that provided a high level of evidence, namely in the form of two systematic reviews. In the hierarchy of research study designs, systematic reviews, meta-analyses, and randomized controlled trials provide the highest quality of evidence, and the lowest grade is applied to case reports and expert opinions. However, one should keep in mind that all articles satisfied peer-review standards of the respective journal at the time of publication. Moreover, modern evaluation criteria can be applied with difficulty to studies published before evidence-based medicine came into existence. Furthermore, the nature of cleft lip and palate research presents certain challenges with regard to the ethics of carrying out randomized clinical trial designs that can yield high-quality evidence. The vast majority of studies (71%) pertaining to cleft lip and/or palate are published in the CPCJ, which is the most pertinent journal in the field, and is expected to attract most publications (Callaham et al., 2002). We could not find a relationship between study design (and other measures of quality), and the impact factor of the original publishing journal. This can be attributed to factors such as the scarcity of high-quality studies, the small number of pertinent journals, as well as the small difference in impact factors in the journals under the WoS category ‘‘Dentistry, Oral Surgery and Medicine.’’ The contributions to cleft research were dispersed across the different areas of cleft care including different aspects of treatment, psychology, epidemiology, anatomy, growth, and other areas. The largest amount of highly cited papers in our specialty appeared to concern alveolar bone grafting (n ¼ 16) and speech and otorhinolaryngology (n ¼ 11). The MeSH term analysis revealed that the most frequently appearing MeSH terms were ‘‘cleft palate’’ (n ¼ 139), ‘‘cleft lip’’ (n ¼ 111), ‘‘humans’’ (n ¼ 96), and ‘‘child’’ (n ¼ 80). These were often used in combination with subheadings pertinent to specific areas of study. The use of these general

terms is by no means surprising given the predefined inclusion criteria. These general MeSH terms were used by most articles, given the multidisciplinary nature of cleft lip and palate research where scientific fields overlap (Bornmann et al., 2008). The use of MeSH terms results in more precise and effective identification of articles for the purpose of meta-analyses (Jenuwine and Floyd, 2004) using relevant search strategies (Adams et al., 1994; Boynton et al., 1998; Nwosu et al., 1998; Watson et al., 1999). The output of the present study allows identification and evaluation of the characteristics of frequently cited articles in cleft lip and palate research. The citation of a scientific article in other disciplines usually follows a time course, not being cited until 1 to 2 years after publication and reaching a maximum after 7 to 10 years, after which citations decline (Marx et al., 2001; Baltussen and Kindler, 2004a). This time trend could be partially confirmed by our findings in cleft lip and palate research in that we observed an increasing trend in the first years after publication that—contrary to the other disciplines—continued after 10 years of publication. A reasonable explanation is the relatively narrow scope for high-quality evidence-based cleft lip and palate research in the past, with authors tending to refer to older articles for valuable information. Given the scarcity of innovative evidence in such a narrow field, under the current educational schemes old studies are often used as basis for the training of future specialists and therefore are accumulating value; whereas, it is not the same for recent publications (Hui et al., 2013). This trend is progressively changing following the recent collaborative multicenter efforts such as Dutchcleft, Scandcleft, Eurocleft, and Americleft. Six of the 100 most-cited articles identified in the present study pertained to such multicenter outcome studies. To overcome confounding search strings with multiple keywords we used the keyword cleft, using a truncation search ‘‘cleft$,’’ which is the most generic term possible for bibliometric research in the field. No other restrictions were applied, apart from the inclusion of articles only published in journals under the WoS category ‘‘Dentistry, Oral Surgery and Medicine’’ and pertaining to human clinical research, so that all relevant contributions could be assessed. Searches such as ‘‘cleft palate OR cleft lip’’ were not chosen to have the best likelihood of not missing any relevant articles. Despite that, some important studies published in the 1950s or 1960s may have been omitted, because the evidence they contributed has been assimilated into more recent articles, a process that is referred to as ‘‘obliteration by incorporation’’ (Garfield, 1987). It is interesting that we found only 29% dispersion of the research to non–cleft-specific journals, which is similar although lower than the findings of Mavropoulos and Kiliaridis (2003), who found 45% of orthodontic research published in various nonrelevant journals.

Christou and Antonarakis, 100 MOST-CITED CLEFT LIP AND PALATE–RELATED ARTICLES

Although no language criteria were set, all identified articles were written in the English language. This fact along with the overwhelming presence of research originating from the United States (57%) is comparable with findings on the origin of most-cited papers in other fields (Fenton et al., 2002; Paladugu et al., 2002; Baltussen and Kindler, 2004a, 2004b; Tsai et al., 2006; Hui et al., 2013). The influence of the United States on medical research has been attributed to the large active scientific and financial resources available to its scientific community compared with those of other countries (Fenton et al., 2002; Baltussen and Kindler, 2004b). Furthermore, the lack of publications from Africa and Asia (with the exception of Japan) is in accordance with previously published evidence (PerezIratxeta and Andrade, 2002). We should not neglect that a great amount of information published in journals of such countries are rarely cited in peer-reviewed articles because these journals do not fulfill criteria for inclusion in major bibliographic databases (Chinnock et al., 2005). CONCLUSIONS Our study identifies the 100 most-cited articles on cleft lip and palate published in ‘‘Dentistry, Oral Surgery and Medicine’’ journals. It can be a potential knowledge base for specialists wishing to train in the field of cleft lip and palate or produce relevant knowledge defining the direction of future research, because studies providing higher-quality evidence are required. It may also help in optimizing research allocation, perhaps by restricting research in specific fields and encouraging its productivity in others while reorienting research support. REFERENCES Adams CE, Power A, Frederick K, Lefebre C. An investigation of the adequacy of MEDLINE searches for randomized controlled trials (RCTs) of the effects of mental health care. Psychol Med. 1994;24:741–748. Baltussen A, Kindler CH. Citation classics in anesthetic journals. Anesth Analg. 2004a;98:443–451. Baltussen A, Kindler CH. Citation classics in critical care medicine. Intensive Care Med. 2004b;30:902–910. Beirne JC, Barry HJ, Brady FA, Morris VB. Donor site morbidity of the anterior iliac crest following cancellous bone harvest. Int J Oral Maxillofac Surg. 1996;25:268–271. Bornmann L, Mutz R., Neuhaus C, Daniel HD. Citation counts for research evaluation: standards of good practice for analyzing bibliometric data and presenting and interpreting results. Ethics Sci Environ Polit. 2008;8:93–102. Boynton J, Glanville J, McDaid D, Lefebre C. Identifying systematic reviews in MEDLINE: developing an objective approach to search strategy design. J Info Sci. 1998;24:137–157. Callaham M, Wears RL, Weber E. Journal prestige, publication bias, and other characteristics associated with citation of published studies in peer-reviewed journals. JAMA. 2002;287:2847–2850. Chinnock P, Siegfried N, Clarke M. Is evidence-based medicine relevant to the developing world? Systematic reviews have yet to achieve their potential as a resource for practitioners in developing countries. Evid Based Complement Alternat Med. 2005;2:321–324.

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