The Cleft Palate–Craniofacial Journal 52(1) pp. e8–e13 January 2015 Ó Copyright 2015 American Cleft Palate–Craniofacial Association
ORIGINAL ARTICLE The Need for Orthognathic Surgery in Nonsyndromic Patients With Repaired Isolated Cleft Palate Gregory S. Antonarakis, D.D.S., M.Sc., Ph.D., Guy Watts, M.B.B.S., F.R.A.C.S.(Plast.), John Daskalogiannakis, D.D.S., M.Sc., F.R.C.D.(C.) Objective: To determine the frequency of need for orthognathic surgery among nonsyndromic patients with isolated cleft palate repaired during infancy at The Hospital for Sick Children in Toronto, Canada. Design: Retrospective cohort study. Patients: Patients with nonsyndromic isolated cleft palate born between 1970 and 1997 with available records including a lateral cephalometric radiograph taken at 15 years of age. Methods: Patients who had undergone or were being prepared for orthognathic surgery were automatically counted as requiring surgery. For the remaining patients, lateral cephalometric radiographs were traced and analyzed. Arbitrarily set cephalometric criteria were used to identify the ‘‘objective’’ need for orthognathic surgery. Results: Of the 189 patients identified with nonsyndromic isolated cleft palate and for whom records were available, 25 (13.2%) were deemed to require orthognathic surgery. Of the surgical cohort, 92% required surgical correction for a Class III malocclusion. Similar percentages of males and females required orthognathic surgery. An apparently greater proportion of patients of Asian background (18.5%) than of white background (10.6%) required surgery, but this difference was not significant (P ¼ .205). Conclusions: The current results suggest that approximately one in eight patients at our institution with nonsyndromic isolated cleft palate requires orthognathic surgery. There is a tendency for this to be higher in patients of Asian descent and lower in patients of white descent. Variability in extent, severity, and phenotype of the cleft, which may be attributed largely to genetics, may play an important role in dictating the need for orthognathic surgery. KEY WORDS:
cephalometrics, isolated cleft palate, orthognathic surgery rate
Although the ideal timing for palatal repair continues to be debated, children with isolated cleft palate usually undergo a palatoplasty procedure at some point during their infancy in order to repair the palatal cleft and restore the velopharyngeal mechanism. Cleft-center protocols differ in the timing of palatal repair, opting for either early or late repair. Supporters of early palatal repair claim that this benefits speech development (Haapanen and Rantala, 1992); whereas, critics argue that early repair may result in a larger restriction of maxillary growth (Nystrom ¨ and Ranta, 1994). Staged palatoplasty protocols also exist and attempt to combine early closure of the soft palate with delayed closure of the hard palate. This concept was pioneered by Schweckendiek (Schweckendiek and Doz, 1978) and was later modified by Delaire (Markus et al., 1993). Some centers have reported advantages with staged protocols (Gundlach et al., 2013). The proportion of patients with isolated cleft palate deemed to require orthognathic surgery differs among studies. Posnick and Taylor (1994) speculated from extrapolation of data provided by Ross (1987b) that approximately 20% of white patients with isolated cleft palate repaired at infancy will develop maxillary hypoplasia
Isolated clefts of the palate occur in approximately 1 in 2000 live births regardless of ethnicity (Strong and Buckmiller, 2001), and nearly half are associated with a syndrome (van Aalst et al., 2008). The incidence of cleft palate, however, is higher in females than in males, and this has been explained by the fact that the process of anteriorto-posterior palatine fusion takes approximately 1 week longer in female fetuses, allowing for a longer window of time for exposure to potential teratogens (van Aalst et al., 2008).
Dr. Antonarakis is Craniofacial Orthodontic Fellow, Division of Orthodontics; and Dr. Watts is Craniofacial Fellow, Division of Plastic Surgery, The Hospital for Sick Children, Toronto, Ontario, Canada. Dr. Daskalogiannakis is Staff Orthodontist, Division of Orthodontics, The Hospital for Sick Children; Associate Professor, Department of Orthodontics, Faculty of Dentistry, University of Toronto; and Private practice, Toronto, Ontario, Canada. Submitted April 2013; Accepted October 2013. Address correspondence to: Dr. Gregory S. Antonarakis, Division of Orthodontics, The Hospital for Sick Children, 555 University Avenue, Toronto, Ontario M5G 1X8, Canada. E-mail gregory. [email protected]. DOI: 10.1597/13-080 8
Antonarakis et al., ISOLATED CLEFT PALATE AND ORTHOGNATHIC SURGERY
that will not be treatable by orthodontics alone. Results from studies presenting orthognathic surgical need of patients with cleft palate in different institutions vary from 0% (0/30 patients; Good et al., 2007) to 3.3% (4/122 patients; Oberoi et al., 2012) to 12.5% (4/32 patients; David et al., 2006) to a maximum of 40% (2/5 patients; DeLuke et al., 1997). Compiling all of these data together results in a combined reported need of orthognathic surgery in patients with isolated cleft palate of 5.3% (10/189 patients). A previous report on the need for orthognathic surgery among patients with a history of repaired complete unilateral and bilateral cleft lip and palate at The Hospital for Sick Children in Toronto, Canada, concluded that approximately 48% and 65% of patients with complete unilateral and bilateral cleft lip and palate, respectively, required orthognathic surgery (Daskalogiannakis and Mehta, 2009). The objective of the present study was to follow a similar methodology to determine the frequency of need for orthognathic surgery among nonsyndromic patients with a history of isolated cleft palate repaired at infancy at The Hospital for Sick Children by examining data collected over the last three decades. SUBJECTS
AND
METHODS
Patient Selection The present retrospective cohort study was approved by our institution’s Research Ethics Board. A review of The Hospital for Sick Children (Toronto, Canada) database of the Cleft Lip and Palate Program and the Orthodontic Clinic was undertaken, with reference to the initial diagnosis (type of cleft, accompanying anomalies) and the available records. Inclusion criteria were the following: patients with isolated cleft palate; availability of records (photographs at initial presentation and/or operative report describing the extent of the initial cleft condition); date of birth between 1970 and 1997; presence of a lateral cephalometric radiograph taken at a minimum age of 15 years. The age of 15 was considered to be an age where one could make a reasonable judgment as to the eventual need (or not) for orthognathic surgery based on the severity of skeletal malrelationships (Daskalogiannakis and Mehta, 2009). Exclusion criteria were as follows: patients whose cleft was part of a syndrome or patients with associated anomalies; patients with diagnosis of Pierre Robin sequence; patients who had their primary palate repair at other institutions; patients with clefts of the palate not requiring primary repair. At The Hospital for Sick Children the techniques used for the repair of clefts of the secondary palate during the period of the study varied among surgeons and among patients. Repair was carried out at or around the age of 12 months. A combination of von Langenbeck, two-flap
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palatoplasty, and Veau-Wardill-Kilner techniques, with the latter having varying degrees of push-back palatoplasty, were used. Push-back repairs fell out of favor in the later years of the study. The popularity of intravelar veloplasty increased after the 1980s, but the technique was not used by all surgeons. Need for Orthognathic Surgery Patients who had undergone orthognathic surgery or distraction osteogenesis, or who were in the process of presurgical orthodontic preparation at the time of record evaluation, were automatically classified as ‘‘needing surgery.’’ For the remaining patients, the most recent lateral cephalometric radiographs available (at a minimum age of 15 years) were manually traced and analyzed by one investigator (G.A.). The existing published reports on the need for orthognathic surgery were usually based on subjective assessment without an explanation of the criteria applied to make the determination (Rosenstein et al., 1991). As part of this investigation, an attempt was made to use set criteria in determining this ‘‘need,’’ so as to make the findings more ‘‘objective.’’ For the purposes of our study, a patient with an apparent Class III skeletal relationship was deemed to require orthognathic surgery based on the satisfaction of all three of the following criteria (selected arbitrarily): ANB (A point, nasion, B point) angle of 38 or lower; Harvold unit difference (CoGnCoANS ¼ condylion, gnathion - condylion, anterior nasal spine) of 34 mm or larger; and Wits appraisal result of 5 mm or lower (B-point ahead of A-point) (Linton, 1998; Daskalogiannakis and Mehta, 2009). Patients who fulfilled all three of these criteria were deemed to be objective orthognathic candidates, irrespective of whether or not they had actually undergone surgery. Patients with an apparent Class II skeletal relationship were deemed to require orthognathic surgery based on the satisfaction of all three of the following criteria (selected arbitrarily): ANBangle of 78 or higher; Harvold unit difference of 15 mm or smaller; and Wits appraisal result of 7 mm or larger (A-point ahead of Bpoint). Variables recorded for each patient were gender; ethnicity (white, Asian, African); decade of birth (1970s, 1980s, 1990s); surgeon performing the initial repair; objective need for orthognathic surgery; and type of jaw malrelationship for those with an objective need for orthognathic surgery (Class II or Class III skeletal relationships). Statistics Chi-square tests were used to test for statistically significant differences with regard to the need for
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orthognathic surgery in the different subgroups. These were gender, ethnicity, decade of birth, and surgeon performing the initial repair. Sensitivity and Specificity Because the cephalometric criteria that we applied were arbitrary, we tested the sensitivity and specificity of the method. In order to test for sensitivity, pretreatment cephalometric radiographs of all patients who had undergone surgery or were in the process of presurgical orthodontic preparation at the time of record evaluation were traced and analyzed to determine whether they would still be classified as needing surgery based on the arbitrary cephalometric parameters. In order to test for specificity, an evaluation was carried out to determine how many of the patients who had not undergone surgery were classified as needing surgery based on the arbitrary cephalometric parameters. Error of the Method An evaluation of the error of the method in cephalometric analysis was carried out by retracing and reanalyzing 15 randomly selected lateral cephalometric radiographs 3 weeks later. The paired t test was used to compare the duplicate recordings (Houston, 1983). RESULTS Need for Orthognathic Surgery The number of patients with isolated cleft palate born between 1970 and 1997 with charts available at our hospital totaled 664. Of these, 69 were born in the 1970s; 291 in the 1980s; and the remaining 304 in the 1990s. Only 189 patients satisfied the criteria of completeness of records and a lateral cephalometric radiograph at the age of 15 or older, and these ultimately formed the study sample. The distribution of the patient sample examined in the study is presented in Table 1. Twenty-five of these 189 patients (13.2%) had undergone orthognathic surgery already, were under presurgical orthodontic preparation, or were judged to have a definite need for orthognathic surgery based on the severity of their sagittal jaw discrepancy. Of the surgical cohort, 92% (23/25 patients) required surgical correction for Class III malocclusion; whereas, the remaining 8% (2/25 patients) required surgical correction for Class II malocclusion. There was no significant (P ¼ 1.000) gender predilection, with similar percentages of males (16/121 or 13.2%) and females (9/68 or 13.2%) requiring orthognathic surgery (Fig. 1). Differences in surgical need were identified, however, when comparing patients of white
TABLE 1
Nonsyndromic Isolated Cleft Palate Patient Sample
Variable
n
Gender Male Female Total
121 68 189
Ethnicity White Asian African Total
132 54 3 189
Birth decade 1970s 1980s 1990s Total
39 87 63 189
Surgeon A B C D E Other Total
40 14 31 30 57 17 189
background (14/132 or 10.6%) with those of Asian background (10/54 or 18.5%) (Fig. 2), but these difference were not significant (P ¼ .144). There were also differences depending on the birth decade (Fig. 3), but again, these were not statistically significant (P ¼ .248). Finally, some differences were identified among patients grouped by the surgeon performing the primary repair (Fig. 4), but these differences were once again not statistically significant (P ¼ .471). Sensitivity and Specificity Sensitivity was calculated to be 92%, meaning that 2 out of the 25 patients who had undergone surgery or were in the process of presurgical orthodontic preparation at the time of record evaluation did not satisfy all three of the arbitrary cephalometric parameters. Specificity was calculated to be 100% because none of the
FIGURE 1 Percentage of patients deemed to require orthognathic surgery, by gender.
Antonarakis et al., ISOLATED CLEFT PALATE AND ORTHOGNATHIC SURGERY
FIGURE 2 Percentage of patients deemed to require orthognathic surgery, by ethnicity (patients with an African background not included due to small numbers).
patients who had not undergone orthognathic surgery was objectively judged to require surgery based on the arbitrary cephalometric parameters. Error of the Method There were no statistically significant differences at the 5% level between the first and second cephalometric tracing and analysis for any of the cephalometric measurements. DISCUSSION Internal audits are an important quality assurance process in every cleft/craniofacial team so as to check that treatment protocols remain current and the results fare reasonably well against those of other centers. This includes the types and techniques of the procedures used, as well as their timing. The results of this study reveal that 13.2% of patients with nonsyndromic isolated cleft palate repaired during infancy present with an objective need for orthognathic surgery. This percentage certainly falls within the previously published range (0% to 40%) (DeLuke et al., 1997; David et al., 2006; Good et al., 2007; Oberoi et al., 2012).
FIGURE 3 Percentage of patients deemed to require orthognathic surgery, by birth decade.
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FIGURE 4 Percentage of patients deemed to require orthognathic surgery, by surgeon performing primary repair (surgeons performing fewer than 10 primary repairs among the sample were combined under ‘‘Other’’).
Although we reviewed 28 years of data, our final sample (n¼189) was somewhat limited due to the absence of lateral cephalometric radiographs taken at a minimum age of 15 for a large number of patients with isolated cleft palate (we were unable to find radiographs on 475 out of a total of 664 patients). The explanation for this may lie in the fact that many patients with isolated cleft palate presented with a mild malocclusion and chose to undergo their orthodontic treatment in the community, rather than in the hospital. Nevertheless, the present study comprises, to the best of our knowledge, the largest number of patients with isolated cleft palate (n ¼ 189) reported in the literature to date. Only approximately one in eight patients with nonsyndromic isolated cleft palate requires orthognathic surgery, and this implies that craniofacial growth patterns show a large variation between individual patients. Differences in craniofacial growth patterns in patients with clefts could be due to factors such as inherent developmental disturbances, growth disruption due to surgery (iatrogenic), functional distortions (due in part to altered muscular activity), and genetically inherited growth patterns (e.g., ethnic, familial) (Ross, 1987a). The relative contributions of the intrinsic developmental deficiency and the surgical iatrogenic effect on the ultimate craniofacial growth of a specific patient are hard to discern (Heliovaara ¨ and Rautio, 2011). Even so, it is well documented that patients with isolated cleft palate have different craniofacial characteristics compared with a noncleft population (Bishara, 1973). The craniofacial phenotype, which may be related to cranial base morphology (Singh et al., 2004), can be an important factor in determining the predisposition for surgical need in certain patients. The technique of palatal repair may potentially play a role in the amount of growth disturbance observed in isolated cleft palate patients. Different techniques are often used depending on the surgeon’s expertise and the extent and severity of the cleft, as well as other clinical judgment made on a case-by-case basis. Becker et al. (2001), however, found no obvious differences in facial skeletal morphology
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Cleft Palate–Craniofacial Journal, January 2015, Vol. 52 No. 1
of patients with isolated cleft palate (n ¼ 64) that could be attributed to surgical technique and emphasized the influence of gender, timing of cleft palate repair, and severity of cleft as other factors that could influence skeletal morphology. Timing of palatal repair may also play a role, but the methodological deficiencies in most existing studies and heterogeneity of samples and studies preclude definitive conclusions (Liao and Mars, 2006). Patients with isolated cleft palate may present with a variable phenotype and degree of severity of the cleft. However, patients with the most extensive clefts at birth have been reported by various authors to have the most marked deviations in craniofacial morphology, namely a smaller and more retrusive maxilla, both at an early age and ˇ in adulthood (Jonsson and Thilander, 1979; Smahel, 1984; Viteporn et al., 1991; Heliovaara and Ranta, 1993; Becker ¨ et al., 2001). The extent of the palatal cleft was not possible to ascertain in the present sample. The variable phenotype of patients with isolated cleft palate in our sample was associated with skeletal relationships ranging from Class II to Class III. Both maxillary and mandibular retrognathism have previously been reported ˇ ˇ (Smahel et al., 1992; Han et al., 1995; Smahel et al., 1999). Standard orthodontic treatment seems to suffice for a large number of patients; whereas, others will benefit from a combined orthodontic/orthognathic surgery approach. A published study of all nonsyndromic patients with a history of clefting who received orthognathic surgical care between 1997 and 2004 in an Australian institution reported that among these patients (n ¼ 103), three with isolated cleft palate received orthognathic surgery, namely a one-piece Le Fort I maxillary advancement (Chong et al., 2009). The percentage of patients with isolated cleft palate undergoing orthognathic surgery at our institution may also be a reflection of the ethnic background of the patients, despite the lack of statistical significance observed between different ethnic backgrounds. Chew (2006) investigated the spectrum of patients in a multiethnic Asian population who had undergone orthognathic surgery and concluded that the majority had a Class III skeletal pattern and that there is greater severity of dentofacial deformities in an Asian population. Ngan et al. (1997) compared Chinese with white surgical Class III patients and found that Chinese patients had a larger mandibular length, contributing to higher relative mandibular prognathism. Other institutions may deal with different populations who have different orthognathic surgical needs. Similarly to the findings of Daskalogiannakis and Mehta (2009), the results of the present study indicate higher, although not statistically significant, need for orthognathic surgery by the decade of birth. This may be a reflection of the growing popularity and acceptability of orthognathic surgery over time. Another possible explanation may lie in the fact that the proportion of the total patient population who were of Asian background increased by the birth decade. In the cohort born in the 1970s, approximately
10% was of Asian background. In the 1980s cohort this percentage increased to approximately 30% and then further increased to approximately 40% in the 1990s cohort. This is only, however, a speculation and is not supported by the statistical significance of the present results. In a multicultural and multiethnic country such as Canada, problems may arise when trying to subdivide a patient sample based on ethnic origin (white, Asian, or African). We attempted to do so using a rather crude distinction in order to evaluate possible differences in surgical need, realizing that there are inherent limitations and flaws in such a method. The findings, however, may provide an insight into general differences among patient subgroups. Another variable that may have influenced future need for orthognathic surgery conceptually by hindering subsequent growth of the midface would be the skill of the surgeon performing the initial repair. One must remember, however, that there are a large number of variables that may be implicated, such as the presenting phenotype and severity of the original cleft deformity, and this could not be taken into account in the present analysis. Furthermore, no statistically significant differences were found when comparing different surgeons in the present study. The decision to undertake orthognathic surgery is usually based on a subjective assessment without objective criteria and depends largely on experience and expectations; as such, it is likely to vary among centers (Rosenstein et al., 1991). Moreover, local financial factors such as cost and the nature of health care coverage in a particular institution can influence the decision to recommend orthognathic surgery (Daskalogiannakis and Mehta, 2009). The cost of orthognathic surgery may vary considerably among cleft centers (Heliovaara and Rautio, 2011). In Canada, the cost of the ¨ surgical procedure is covered entirely by the national health care plan, making this option more realistic for a large number of patients. In the present study we tried to overcome this by applying arbitrarily predefined criteria, with acceptable sensitivity and specificity, when evaluating the need for orthognathic surgery. CONCLUSIONS The goal of the current study was to report on the objective need for orthognathic surgery in patients with nonsyndromic isolated cleft palate treated at our institution. Approximately one in eight patients was found to require orthognathic surgery. There was a tendency, albeit not significant, for this to be higher in the subgroup of Asian descent and lower in the subgroup of white descent, perhaps due to differences in craniofacial phenotype. Variability in extent, severity, and phenotype of the cleft, which may be attributed largely to genetics, may play an important role in dictating the need for orthognathic surgery.
Antonarakis et al., ISOLATED CLEFT PALATE AND ORTHOGNATHIC SURGERY
REFERENCES Becker M, Svensson H, McWilliam J, Sarnas ¨ KV, Jacobsson S. Adult skeletal profile in isolated cleft palate: a comparison of the von Langenbeck and Wardill procedures for primary repair of the palate. Scand J Plast Reconstr Surg Hand Surg. 2001;35:387–397. 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. Chew MT. Spectrum and management of dentofacial deformities in a multiethnic Asian population. Angle Orthod. 2006;76:806–809. Chong DK, Portnof JE, Xu H, Salyer KE. Reviewing the orthognathic surgical care of the patients with cleft lip and palate: the single surgeon experience. J Craniofac Surg. 2009;20(suppl 2):1895–1904. Daskalogiannakis J, Mehta M. The need for orthognathic surgery in patients with repaired complete unilateral and complete bilateral cleft lip and palate. Cleft Palate Craniofac J. 2009;46:498–502. David DJ, Anderson PJ, Schnitt DE, Nugent MA, Sells R. From birth to maturity: a group of patients who have completed their protocol management. Part II. Isolated cleft palate. Plast Reconstr Surg. 2006;117:515–526. DeLuke DM, Marchand A, Robles EC, Fox P. Facial growth and the need for orthognathic surgery after cleft palate repair: literature review and report of 28 cases. J Oral Maxillofac Surg. 1997;55:694– 698. Good PM, Mulliken JB, Padwa BL. Frequency of Le Fort I osteotomy after repaired cleft lip and palate or cleft palate. Cleft Palate Craniofac J. 2007;44:396–401. Gundlach KK, Bardach J, Filippow D, Stahl-de Castrillon F, Lenz JH. Two-stage palatoplasty: is it still a valuable treatment protocol for patients with a cleft of lip, alveolus, and palate? J Craniomaxillofac Surg. 2013;41:62–70. Haapanen ML, Rantala SL. Correlation between the age at repair and speech outcome in patients with isolated cleft palate. Scand J Plast Reconstr Surg Hand Surg. 1992;26:71–78. Han BJ, Suzuki A, Tashiro H. Longitudinal study of craniofacial growth in subjects with cleft lip and palate: from cheiloplasty to 8 years of age. Cleft Palate Craniofac J. 1995;32:156–166. Heliovaara A, Ranta R. One-stage closure of isolated cleft palate with ¨ the Veau-Wardill-Kilner pushback procedure or the Cronin modification. III. Comparison of lateral craniofacial morphology. Acta Odontol Scand. 1993;51:313–321. Heliovaara A, Rautio J. A comparison of craniofacial cephalometric ¨ morphology and the later need for orthognathic surgery in 6-yearold cleft children. J Craniomaxillofac Surg. 2011;39:173–176. Houston WJ. The analysis of error in orthodontic measurements. Am J Orthod. 1983;83:382–390. Jonsson G, Thilander B. Occlusion, arch dimensions, and craniofacial morphology after palatal surgery in a group of children with clefts in the secondary palate. Am J Orthod. 1979;76:243–255.
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Liao YF, Mars M. Hard palate repair timing and facial growth in cleft lip and palate: a systematic review. Cleft Palate Craniofac J. 2006;43:563–570. Linton JL. Comparative study of diagnostic measures in borderline surgical cases of unilateral cleft lip and palate and noncleft class III malocclusions. Am J Orthod Dentofac Orthop. 1998;113:526–537. Markus AF, Smith WP, Delaire J. Primary closure of cleft palate: a functional approach. Br J Oral Maxillofac Surg. 1993;31:71–77. Ngan P, Hagg U, Yiu C, Merwin D, Wei SH. Cephalometric ¨ comparisons of Chinese and Caucasian surgical Class III patients. Int J Adult Orthodon Orthognath Surg. 1997;12:177–188. Nystrom M, Ranta R. Effect of timing and method of closure of ¨ isolated cleft palate on development of dental arches from 3 to 6 years of age. Eur J Orthod. 1994;16:377–383. Oberoi S, Hoffman WY, Chigurupati R, Vargervik K. Frequency of surgical correction for maxillary hypoplasia in cleft lip and palate. J Craniofac Surg. 2012;23:1665–1667. Posnick JC, Taylor M. Skeletal stability and relapse patterns after LeFort I osteotomy using miniplate fixation in patients with isolated cleft palate. Plast Reconstr Surg. 1994;94:51–58. Rosenstein S, Kernahan D, Dado D, Grasseschi M, Griffith BH. Orthognathic surgery in cleft patients treated by early bone grafting. Plast Reconstr Surg. 1991;87:835–892. Ross RB. Treatment variables affecting facial growth in complete unilateral cleft lip and palate. Part 1: treatment affecting growth. Cleft Palate J. 1987a;24:5–23. Ross RB. Treatment variables affecting facial growth in complete unilateral cleft lip and palate. Part 7: an overview of treatment and facial growth. Cleft Palate J. 1987b;24:71–77. Schweckendiek W, Doz P. Primary veloplasty: long-term results without maxillary deformity. A twenty-five year report. Cleft Palate J. 1978;15:268–274. Singh GD, Rivera-Robles J, de Jesus-Vinas J. Longitudinal craniofacial growth patterns in patients with orofacial clefts: geometric morphometrics. Cleft Palate Craniofac J. 2004;41:136–143. ˇ Smahel Z. Variations in craniofacial morphology with severity of isolated cleft palate. Cleft Palate J. 1984;21:140–158. ˇ Smahel Z, Hora´k I, Polivkova´ H, Skvalirova´ B. Configuration of facial profile in adults with isolated cleft palate. Acta Chir Plast. 1992;34:204–214. ˇ ´ Z. Multivariate comparison of Smahel Z, Hradisky´ D, Mullerova ¨ craniofacial morphology in different types of facial clefts. Acta Chir Plast. 1999;31:59–65. Strong EB, Buckmiller LM. Management of the cleft palate. Facial Plast Surg Clin North Am. 2001;9:15–25. van Aalst JA, Kolappa KK, Sadove M. Nonsyndromic cleft palate. Plast Reconstr Surg. 2008;121(suppl 1):1–14. Viteporn S, Enemark H, Melsen B. Postnatal craniofacial development following a pushback operation of patients with cleft palate. Cleft Palate Craniofac J. 1991;28:392–397.
ORIGINAL ARTICLE The Need for Orthognathic Surgery in Nonsyndromic Patients With Repaired Isolated Cleft Palate Gregory S. Antonarakis, D.D.S., M.Sc., Ph.D., Guy Watts, M.B.B.S., F.R.A.C.S.(Plast.), John Daskalogiannakis, D.D.S., M.Sc., F.R.C.D.(C.) Objective: To determine the frequency of need for orthognathic surgery among nonsyndromic patients with isolated cleft palate repaired during infancy at The Hospital for Sick Children in Toronto, Canada. Design: Retrospective cohort study. Patients: Patients with nonsyndromic isolated cleft palate born between 1970 and 1997 with available records including a lateral cephalometric radiograph taken at 15 years of age. Methods: Patients who had undergone or were being prepared for orthognathic surgery were automatically counted as requiring surgery. For the remaining patients, lateral cephalometric radiographs were traced and analyzed. Arbitrarily set cephalometric criteria were used to identify the ‘‘objective’’ need for orthognathic surgery. Results: Of the 189 patients identified with nonsyndromic isolated cleft palate and for whom records were available, 25 (13.2%) were deemed to require orthognathic surgery. Of the surgical cohort, 92% required surgical correction for a Class III malocclusion. Similar percentages of males and females required orthognathic surgery. An apparently greater proportion of patients of Asian background (18.5%) than of white background (10.6%) required surgery, but this difference was not significant (P ¼ .205). Conclusions: The current results suggest that approximately one in eight patients at our institution with nonsyndromic isolated cleft palate requires orthognathic surgery. There is a tendency for this to be higher in patients of Asian descent and lower in patients of white descent. Variability in extent, severity, and phenotype of the cleft, which may be attributed largely to genetics, may play an important role in dictating the need for orthognathic surgery. KEY WORDS:
cephalometrics, isolated cleft palate, orthognathic surgery rate
Although the ideal timing for palatal repair continues to be debated, children with isolated cleft palate usually undergo a palatoplasty procedure at some point during their infancy in order to repair the palatal cleft and restore the velopharyngeal mechanism. Cleft-center protocols differ in the timing of palatal repair, opting for either early or late repair. Supporters of early palatal repair claim that this benefits speech development (Haapanen and Rantala, 1992); whereas, critics argue that early repair may result in a larger restriction of maxillary growth (Nystrom ¨ and Ranta, 1994). Staged palatoplasty protocols also exist and attempt to combine early closure of the soft palate with delayed closure of the hard palate. This concept was pioneered by Schweckendiek (Schweckendiek and Doz, 1978) and was later modified by Delaire (Markus et al., 1993). Some centers have reported advantages with staged protocols (Gundlach et al., 2013). The proportion of patients with isolated cleft palate deemed to require orthognathic surgery differs among studies. Posnick and Taylor (1994) speculated from extrapolation of data provided by Ross (1987b) that approximately 20% of white patients with isolated cleft palate repaired at infancy will develop maxillary hypoplasia
Isolated clefts of the palate occur in approximately 1 in 2000 live births regardless of ethnicity (Strong and Buckmiller, 2001), and nearly half are associated with a syndrome (van Aalst et al., 2008). The incidence of cleft palate, however, is higher in females than in males, and this has been explained by the fact that the process of anteriorto-posterior palatine fusion takes approximately 1 week longer in female fetuses, allowing for a longer window of time for exposure to potential teratogens (van Aalst et al., 2008).
Dr. Antonarakis is Craniofacial Orthodontic Fellow, Division of Orthodontics; and Dr. Watts is Craniofacial Fellow, Division of Plastic Surgery, The Hospital for Sick Children, Toronto, Ontario, Canada. Dr. Daskalogiannakis is Staff Orthodontist, Division of Orthodontics, The Hospital for Sick Children; Associate Professor, Department of Orthodontics, Faculty of Dentistry, University of Toronto; and Private practice, Toronto, Ontario, Canada. Submitted April 2013; Accepted October 2013. Address correspondence to: Dr. Gregory S. Antonarakis, Division of Orthodontics, The Hospital for Sick Children, 555 University Avenue, Toronto, Ontario M5G 1X8, Canada. E-mail gregory. [email protected]. DOI: 10.1597/13-080 8
Antonarakis et al., ISOLATED CLEFT PALATE AND ORTHOGNATHIC SURGERY
that will not be treatable by orthodontics alone. Results from studies presenting orthognathic surgical need of patients with cleft palate in different institutions vary from 0% (0/30 patients; Good et al., 2007) to 3.3% (4/122 patients; Oberoi et al., 2012) to 12.5% (4/32 patients; David et al., 2006) to a maximum of 40% (2/5 patients; DeLuke et al., 1997). Compiling all of these data together results in a combined reported need of orthognathic surgery in patients with isolated cleft palate of 5.3% (10/189 patients). A previous report on the need for orthognathic surgery among patients with a history of repaired complete unilateral and bilateral cleft lip and palate at The Hospital for Sick Children in Toronto, Canada, concluded that approximately 48% and 65% of patients with complete unilateral and bilateral cleft lip and palate, respectively, required orthognathic surgery (Daskalogiannakis and Mehta, 2009). The objective of the present study was to follow a similar methodology to determine the frequency of need for orthognathic surgery among nonsyndromic patients with a history of isolated cleft palate repaired at infancy at The Hospital for Sick Children by examining data collected over the last three decades. SUBJECTS
AND
METHODS
Patient Selection The present retrospective cohort study was approved by our institution’s Research Ethics Board. A review of The Hospital for Sick Children (Toronto, Canada) database of the Cleft Lip and Palate Program and the Orthodontic Clinic was undertaken, with reference to the initial diagnosis (type of cleft, accompanying anomalies) and the available records. Inclusion criteria were the following: patients with isolated cleft palate; availability of records (photographs at initial presentation and/or operative report describing the extent of the initial cleft condition); date of birth between 1970 and 1997; presence of a lateral cephalometric radiograph taken at a minimum age of 15 years. The age of 15 was considered to be an age where one could make a reasonable judgment as to the eventual need (or not) for orthognathic surgery based on the severity of skeletal malrelationships (Daskalogiannakis and Mehta, 2009). Exclusion criteria were as follows: patients whose cleft was part of a syndrome or patients with associated anomalies; patients with diagnosis of Pierre Robin sequence; patients who had their primary palate repair at other institutions; patients with clefts of the palate not requiring primary repair. At The Hospital for Sick Children the techniques used for the repair of clefts of the secondary palate during the period of the study varied among surgeons and among patients. Repair was carried out at or around the age of 12 months. A combination of von Langenbeck, two-flap
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palatoplasty, and Veau-Wardill-Kilner techniques, with the latter having varying degrees of push-back palatoplasty, were used. Push-back repairs fell out of favor in the later years of the study. The popularity of intravelar veloplasty increased after the 1980s, but the technique was not used by all surgeons. Need for Orthognathic Surgery Patients who had undergone orthognathic surgery or distraction osteogenesis, or who were in the process of presurgical orthodontic preparation at the time of record evaluation, were automatically classified as ‘‘needing surgery.’’ For the remaining patients, the most recent lateral cephalometric radiographs available (at a minimum age of 15 years) were manually traced and analyzed by one investigator (G.A.). The existing published reports on the need for orthognathic surgery were usually based on subjective assessment without an explanation of the criteria applied to make the determination (Rosenstein et al., 1991). As part of this investigation, an attempt was made to use set criteria in determining this ‘‘need,’’ so as to make the findings more ‘‘objective.’’ For the purposes of our study, a patient with an apparent Class III skeletal relationship was deemed to require orthognathic surgery based on the satisfaction of all three of the following criteria (selected arbitrarily): ANB (A point, nasion, B point) angle of 38 or lower; Harvold unit difference (CoGnCoANS ¼ condylion, gnathion - condylion, anterior nasal spine) of 34 mm or larger; and Wits appraisal result of 5 mm or lower (B-point ahead of A-point) (Linton, 1998; Daskalogiannakis and Mehta, 2009). Patients who fulfilled all three of these criteria were deemed to be objective orthognathic candidates, irrespective of whether or not they had actually undergone surgery. Patients with an apparent Class II skeletal relationship were deemed to require orthognathic surgery based on the satisfaction of all three of the following criteria (selected arbitrarily): ANBangle of 78 or higher; Harvold unit difference of 15 mm or smaller; and Wits appraisal result of 7 mm or larger (A-point ahead of Bpoint). Variables recorded for each patient were gender; ethnicity (white, Asian, African); decade of birth (1970s, 1980s, 1990s); surgeon performing the initial repair; objective need for orthognathic surgery; and type of jaw malrelationship for those with an objective need for orthognathic surgery (Class II or Class III skeletal relationships). Statistics Chi-square tests were used to test for statistically significant differences with regard to the need for
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orthognathic surgery in the different subgroups. These were gender, ethnicity, decade of birth, and surgeon performing the initial repair. Sensitivity and Specificity Because the cephalometric criteria that we applied were arbitrary, we tested the sensitivity and specificity of the method. In order to test for sensitivity, pretreatment cephalometric radiographs of all patients who had undergone surgery or were in the process of presurgical orthodontic preparation at the time of record evaluation were traced and analyzed to determine whether they would still be classified as needing surgery based on the arbitrary cephalometric parameters. In order to test for specificity, an evaluation was carried out to determine how many of the patients who had not undergone surgery were classified as needing surgery based on the arbitrary cephalometric parameters. Error of the Method An evaluation of the error of the method in cephalometric analysis was carried out by retracing and reanalyzing 15 randomly selected lateral cephalometric radiographs 3 weeks later. The paired t test was used to compare the duplicate recordings (Houston, 1983). RESULTS Need for Orthognathic Surgery The number of patients with isolated cleft palate born between 1970 and 1997 with charts available at our hospital totaled 664. Of these, 69 were born in the 1970s; 291 in the 1980s; and the remaining 304 in the 1990s. Only 189 patients satisfied the criteria of completeness of records and a lateral cephalometric radiograph at the age of 15 or older, and these ultimately formed the study sample. The distribution of the patient sample examined in the study is presented in Table 1. Twenty-five of these 189 patients (13.2%) had undergone orthognathic surgery already, were under presurgical orthodontic preparation, or were judged to have a definite need for orthognathic surgery based on the severity of their sagittal jaw discrepancy. Of the surgical cohort, 92% (23/25 patients) required surgical correction for Class III malocclusion; whereas, the remaining 8% (2/25 patients) required surgical correction for Class II malocclusion. There was no significant (P ¼ 1.000) gender predilection, with similar percentages of males (16/121 or 13.2%) and females (9/68 or 13.2%) requiring orthognathic surgery (Fig. 1). Differences in surgical need were identified, however, when comparing patients of white
TABLE 1
Nonsyndromic Isolated Cleft Palate Patient Sample
Variable
n
Gender Male Female Total
121 68 189
Ethnicity White Asian African Total
132 54 3 189
Birth decade 1970s 1980s 1990s Total
39 87 63 189
Surgeon A B C D E Other Total
40 14 31 30 57 17 189
background (14/132 or 10.6%) with those of Asian background (10/54 or 18.5%) (Fig. 2), but these difference were not significant (P ¼ .144). There were also differences depending on the birth decade (Fig. 3), but again, these were not statistically significant (P ¼ .248). Finally, some differences were identified among patients grouped by the surgeon performing the primary repair (Fig. 4), but these differences were once again not statistically significant (P ¼ .471). Sensitivity and Specificity Sensitivity was calculated to be 92%, meaning that 2 out of the 25 patients who had undergone surgery or were in the process of presurgical orthodontic preparation at the time of record evaluation did not satisfy all three of the arbitrary cephalometric parameters. Specificity was calculated to be 100% because none of the
FIGURE 1 Percentage of patients deemed to require orthognathic surgery, by gender.
Antonarakis et al., ISOLATED CLEFT PALATE AND ORTHOGNATHIC SURGERY
FIGURE 2 Percentage of patients deemed to require orthognathic surgery, by ethnicity (patients with an African background not included due to small numbers).
patients who had not undergone orthognathic surgery was objectively judged to require surgery based on the arbitrary cephalometric parameters. Error of the Method There were no statistically significant differences at the 5% level between the first and second cephalometric tracing and analysis for any of the cephalometric measurements. DISCUSSION Internal audits are an important quality assurance process in every cleft/craniofacial team so as to check that treatment protocols remain current and the results fare reasonably well against those of other centers. This includes the types and techniques of the procedures used, as well as their timing. The results of this study reveal that 13.2% of patients with nonsyndromic isolated cleft palate repaired during infancy present with an objective need for orthognathic surgery. This percentage certainly falls within the previously published range (0% to 40%) (DeLuke et al., 1997; David et al., 2006; Good et al., 2007; Oberoi et al., 2012).
FIGURE 3 Percentage of patients deemed to require orthognathic surgery, by birth decade.
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FIGURE 4 Percentage of patients deemed to require orthognathic surgery, by surgeon performing primary repair (surgeons performing fewer than 10 primary repairs among the sample were combined under ‘‘Other’’).
Although we reviewed 28 years of data, our final sample (n¼189) was somewhat limited due to the absence of lateral cephalometric radiographs taken at a minimum age of 15 for a large number of patients with isolated cleft palate (we were unable to find radiographs on 475 out of a total of 664 patients). The explanation for this may lie in the fact that many patients with isolated cleft palate presented with a mild malocclusion and chose to undergo their orthodontic treatment in the community, rather than in the hospital. Nevertheless, the present study comprises, to the best of our knowledge, the largest number of patients with isolated cleft palate (n ¼ 189) reported in the literature to date. Only approximately one in eight patients with nonsyndromic isolated cleft palate requires orthognathic surgery, and this implies that craniofacial growth patterns show a large variation between individual patients. Differences in craniofacial growth patterns in patients with clefts could be due to factors such as inherent developmental disturbances, growth disruption due to surgery (iatrogenic), functional distortions (due in part to altered muscular activity), and genetically inherited growth patterns (e.g., ethnic, familial) (Ross, 1987a). The relative contributions of the intrinsic developmental deficiency and the surgical iatrogenic effect on the ultimate craniofacial growth of a specific patient are hard to discern (Heliovaara ¨ and Rautio, 2011). Even so, it is well documented that patients with isolated cleft palate have different craniofacial characteristics compared with a noncleft population (Bishara, 1973). The craniofacial phenotype, which may be related to cranial base morphology (Singh et al., 2004), can be an important factor in determining the predisposition for surgical need in certain patients. The technique of palatal repair may potentially play a role in the amount of growth disturbance observed in isolated cleft palate patients. Different techniques are often used depending on the surgeon’s expertise and the extent and severity of the cleft, as well as other clinical judgment made on a case-by-case basis. Becker et al. (2001), however, found no obvious differences in facial skeletal morphology
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Cleft Palate–Craniofacial Journal, January 2015, Vol. 52 No. 1
of patients with isolated cleft palate (n ¼ 64) that could be attributed to surgical technique and emphasized the influence of gender, timing of cleft palate repair, and severity of cleft as other factors that could influence skeletal morphology. Timing of palatal repair may also play a role, but the methodological deficiencies in most existing studies and heterogeneity of samples and studies preclude definitive conclusions (Liao and Mars, 2006). Patients with isolated cleft palate may present with a variable phenotype and degree of severity of the cleft. However, patients with the most extensive clefts at birth have been reported by various authors to have the most marked deviations in craniofacial morphology, namely a smaller and more retrusive maxilla, both at an early age and ˇ in adulthood (Jonsson and Thilander, 1979; Smahel, 1984; Viteporn et al., 1991; Heliovaara and Ranta, 1993; Becker ¨ et al., 2001). The extent of the palatal cleft was not possible to ascertain in the present sample. The variable phenotype of patients with isolated cleft palate in our sample was associated with skeletal relationships ranging from Class II to Class III. Both maxillary and mandibular retrognathism have previously been reported ˇ ˇ (Smahel et al., 1992; Han et al., 1995; Smahel et al., 1999). Standard orthodontic treatment seems to suffice for a large number of patients; whereas, others will benefit from a combined orthodontic/orthognathic surgery approach. A published study of all nonsyndromic patients with a history of clefting who received orthognathic surgical care between 1997 and 2004 in an Australian institution reported that among these patients (n ¼ 103), three with isolated cleft palate received orthognathic surgery, namely a one-piece Le Fort I maxillary advancement (Chong et al., 2009). The percentage of patients with isolated cleft palate undergoing orthognathic surgery at our institution may also be a reflection of the ethnic background of the patients, despite the lack of statistical significance observed between different ethnic backgrounds. Chew (2006) investigated the spectrum of patients in a multiethnic Asian population who had undergone orthognathic surgery and concluded that the majority had a Class III skeletal pattern and that there is greater severity of dentofacial deformities in an Asian population. Ngan et al. (1997) compared Chinese with white surgical Class III patients and found that Chinese patients had a larger mandibular length, contributing to higher relative mandibular prognathism. Other institutions may deal with different populations who have different orthognathic surgical needs. Similarly to the findings of Daskalogiannakis and Mehta (2009), the results of the present study indicate higher, although not statistically significant, need for orthognathic surgery by the decade of birth. This may be a reflection of the growing popularity and acceptability of orthognathic surgery over time. Another possible explanation may lie in the fact that the proportion of the total patient population who were of Asian background increased by the birth decade. In the cohort born in the 1970s, approximately
10% was of Asian background. In the 1980s cohort this percentage increased to approximately 30% and then further increased to approximately 40% in the 1990s cohort. This is only, however, a speculation and is not supported by the statistical significance of the present results. In a multicultural and multiethnic country such as Canada, problems may arise when trying to subdivide a patient sample based on ethnic origin (white, Asian, or African). We attempted to do so using a rather crude distinction in order to evaluate possible differences in surgical need, realizing that there are inherent limitations and flaws in such a method. The findings, however, may provide an insight into general differences among patient subgroups. Another variable that may have influenced future need for orthognathic surgery conceptually by hindering subsequent growth of the midface would be the skill of the surgeon performing the initial repair. One must remember, however, that there are a large number of variables that may be implicated, such as the presenting phenotype and severity of the original cleft deformity, and this could not be taken into account in the present analysis. Furthermore, no statistically significant differences were found when comparing different surgeons in the present study. The decision to undertake orthognathic surgery is usually based on a subjective assessment without objective criteria and depends largely on experience and expectations; as such, it is likely to vary among centers (Rosenstein et al., 1991). Moreover, local financial factors such as cost and the nature of health care coverage in a particular institution can influence the decision to recommend orthognathic surgery (Daskalogiannakis and Mehta, 2009). The cost of orthognathic surgery may vary considerably among cleft centers (Heliovaara and Rautio, 2011). In Canada, the cost of the ¨ surgical procedure is covered entirely by the national health care plan, making this option more realistic for a large number of patients. In the present study we tried to overcome this by applying arbitrarily predefined criteria, with acceptable sensitivity and specificity, when evaluating the need for orthognathic surgery. CONCLUSIONS The goal of the current study was to report on the objective need for orthognathic surgery in patients with nonsyndromic isolated cleft palate treated at our institution. Approximately one in eight patients was found to require orthognathic surgery. There was a tendency, albeit not significant, for this to be higher in the subgroup of Asian descent and lower in the subgroup of white descent, perhaps due to differences in craniofacial phenotype. Variability in extent, severity, and phenotype of the cleft, which may be attributed largely to genetics, may play an important role in dictating the need for orthognathic surgery.
Antonarakis et al., ISOLATED CLEFT PALATE AND ORTHOGNATHIC SURGERY
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