The Cleft Palate–Craniofacial Journal 52(3) pp. 269–276 May 2015 Ó Copyright 2015 American Cleft Palate–Craniofacial Association
ORIGINAL ARTICLE Presurgical Cleft Lip Anthropometrics and Dental Arch Relationships in Patients With Complete Unilateral Cleft Lip and Palate Gregory S. Antonarakis, D.D.S., M.Sc., Ph.D., Alex Adibfar, B.Sc., Bryan D. Tompson, D.D.S., D. Paedo, D. Ortho., John Daskalogiannakis, D.D.S., M.Sc., F.R.C.D.(C.), David M. Fisher, M.D., M.F.R.C.S.(C.), F.A.C.S. Objective: To investigate associations between anthropometric lip measurements and dental arch relationships in patients with complete unilateral cleft lip and palate (CUCLP). Design: Retrospective cross-sectional study. Patients: Children with CUCLP. Methods: Anthropometric lip measurements, made immediately prior to lip repair, were available for each patient. The dental arch relationships were evaluated on dental study casts (8.6 6 0.9 years) taken prior to any orthodontic treatment and prior to alveolar bone graft, using the modified Huddart and Bodenham (MHB) scoring system. The presence of associations between anthropometric lip measurements and dental arch relationships was determined using linear regression analysis. Results: In the 63 patients included in the study, the cleft lateral lip element was deficient in height in 87% and in transverse width in 86% of patients. Patients with more deficient cleft-side lateral lip height were more likely to present with more negative MHB scores (r ¼ .443; P , .001). Conversely, patients with more deficient cleft-side lateral lip transverse width more often presented with more positive MHB scores (r ¼ .281; P ¼ .025). Conclusions: In patients with CUCLP, there is a wide variability in the degree of deficiency of the cleft-side lateral lip element, both in the vertical and in the transverse dimension. The extent of this deficiency may, in part, predict the resulting dental arch relationships. KEY WORDS:
cleft lip anthropometry, dental arch relationship, scoring system, unilateral cleft lip and palate
Maxillary hypoplasia, and the consequent midface retrusion, are a common occurrence in patients with complete unilateral cleft lip and palate (CUCLP). Approximately 48% of patients with CUCLP in our institution
were found to have midface retrusion that was severe enough to require orthognathic surgery (Daskalogiannakis and Mehta, 2009). Two factors have been proposed to play a role in the etiology of this growth deficiency (Ross, 1987; Chiu et al., 2011): (1) the intrinsic factor, such as a developmental deficiency leading to the formation of a cleft and the growth potential of the midfacial skeleton, and (2) the iatrogenic factor, introduced via treatments, mainly from surgical repair. Surgeon experience and the influence of surgical technique have often been implicated in the severity of maxillary hypoplasia and the variation seen among patients (Shaw et al., 1992). However, even when considering patients treated by the same surgeon and with the same protocol (thus eliminating variation due to iatrogenic factors), varying results are observed with respect to maxillary hypoplasia (Meazzini et al., 2011). Despite similar iatrogenic influences, maxillofacial growth deficiency is encountered in certain patients but not in others (Nakamura et al., 2005). This inconsistency suggests that individual intrinsic factors may play an important role in the maxillary growth potential. Wide phenotypic variation is exhibited in CUCLP with regard to severity of the birth deformity. Moreover, throughout the postnatal growth period, one can observe
Dr. Antonarakis is Craniofacial Orthodontic Fellow, Division of Orthodontics, The Hospital for Sick Children, Toronto, Ontario, Canada, and Department of Orthodontics, Faculty of Dentistry, University of Geneva, Switzerland. Mr. Adibfar is summer student, Division of Plastic Surgery, The Hospital for Sick Children, Toronto, Ontario, Canada. Dr. Tompson is Head, Division of Orthodontics, The Hospital for Sick Children, Toronto, Ontario, Canada, and Associate Professor and Head, Department of Orthodontics, Faculty of Dentistry, University of Toronto, Ontario, Canada. Dr. Daskalogiannakis is Staff Orthodontist, Division of Orthodontics, The Hospital for Sick Children, Toronto, Ontario, Canada, and Associate Professor, Department of Orthodontics, Faculty of Dentistry, University of Toronto, Ontario, Canada and Private practice, Toronto, Ontario, Canada. Dr. Fisher is Medical Director, Cleft Lip and Palate Program, Division of Plastic Surgery, The Hospital for Sick Children, Toronto, Ontario, Canada, and Associate Professor, Department of Surgery, University of Toronto, Toronto, Ontario, Canada. Submitted October 2013; Revised December 2013, February 2014; Accepted February 2014. 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: 10.1597/13-272 269
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a wide variation in maxillary skeletal growth and resulting dental occlusion. The severity of the CUCLP at birth may reflect differing contributions of tissue deficiency and/or tissue displacement. The critical factors responsible for the variable maxillary hypoplasia and associated malocclusion are not known. If the initial deformity and maxillary skeletal growth are linked, it is plausible to assume that the midface growth problem associated with tissue deficiency will play a role throughout the postnatal growth period (Peltomaki ¨ et al., 2001). Evidently, one must also keep in mind that maxillary hypoplasia may be related to horizontal, vertical, and/or transverse growth. To investigate a possible association between the severity of the cleft and the midface growth potential of a patient with CUCLP, a methodology for assessment of cleft severity is necessary. Cleft severity is often determined on the basis of cleft or palate size (Chiu et al., 2011), but this can be influenced by factors such as infant orthopedics, aberrant function (such as the modeling effect of the tongue on the maxillary arch during the prenatal and neonatal period; Delestan et al., 2013), sleeping position (Huang et al., 1994), or differences in the general size of the infant. Liao et al. (2010) postulated that the cleft size primarily reflects tissue displacement, mainly through tongue force, rather than tissue deficiency. The extent of cleft-side lateral lip element deficiency has not attracted much attention in either the clinical or the research setting. Anthropometry is an option for quantifying this type of deficiency. What is required is a relative measurement of the ratio of the cleft lip to the noncleft-side lip. This can provide a more reliable estimation of cleft severity in patients with CUCLP as compared with absolute measurements. Patients with unilateral clefts have been shown to exhibit large variability in lip anthropometric measurements (Boorer et al., 2011). We hypothesize that the physical dimensions of the lip on the cleft side have a predictive role in determining dental arch relationships. The aim of the current study was thus to investigate the presence of any association between the size or extent of the unilateral cleft lip deficiency and dental arch relationships in patients with CUCLP. To the best of the authors’ knowledge, no previous attempt at exploring such an association has been made to date. MATERIALS
AND
METHODS
The current study design was retrospective and crosssectional in nature and approved by the Research Ethics Board of the Hospital for Sick Children. A chart review was carried out to identify children born with CUCLP between the years 2000 and 2005. Inclusion criteria comprised the following: primary unilateral cleft lip repair carried out by the senior author (D.M.F.) and dental study casts available at the age of 6 years or older with patients in their mixed dentition, before any orthodontic treatment or alveolar bone grafting. The following were our exclusion criteria:
children whose cleft was part of a craniofacial syndrome; children who had secondary surgical treatment prior to the evaluation of the dental study casts; children who had teeth extracted prematurely due to caries, orthodontics, or trauma; dental study casts of insufficient diagnostic quality due to damage (such as broken teeth); and dental study casts available with patients in their deciduous dentition due to the increase in the incidence of crossbite in the transition from the deciduous to the early mixed dentition (Bergland and Sidhu, 1974). All of the children had been treated according to the following treatment protocol: presurgical infant orthopedics using the nasoalveolar molding technique described by Grayson et al. (1999) for a period of approximately 12 to 24 weeks (until primary repair), primary unilateral cleft lip repair at approximately 3 to 6 months of age using the technique of Fisher (2005), and primary repair of the cleft palate at approximately 12 months of age using the hybrid palatoplasty technique (Gillet and Clarke, 1996). Determination of Cleft Phenotype Determination of CUCLP phenotype was based on clinical diagnosis. All diagnoses were made by the senior author (D.M.F.) at or soon after birth and confirmed at the time of primary unilateral cleft lip repair. Laterality of the cleft (left- or right-sided) was also recorded. Lip Anthropometric Measurements The senior author (D.M.F.) performed all of the anthropometric lip measurements, with calipers, immediately prior to primary cleft lip repair with the patient under general anesthesia. Vertical height and transverse width measurements of both the cleft and noncleft sides of the lip were recorded to the nearest 0.5 mm. The anthropometric points used for measurements are shown in Figure 1. The medial lip height was measured from the midline of the labiocolumellar groove (subnasale) to the peaks of Cupid’s bow. The lateral lip height of the noncleft side was measured from the lowest point of the alar base (subalare) to the peak of Cupid’s bow. The lateral lip height of the cleft side was measured from the lowest point of the alar base (subalare) to the proposed peak of Cupid’s bow. The proposed peak of Cupid’s bow was defined, according to Noordhoff, as the point along the vermilion-cutaneous junction where the cutaneous roll and red line (vermilion-mucosal junction) begin to converge medially (Noordhoff, 1997). The lateral lip transverse width of the noncleft side was measured from the oral commissure to the peak of Cupid’s bow. The lateral lip transverse width of the cleft side was measured from the oral commissure to the proposed peak of Cupid’s bow at Noordhoff’s point.
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of the five-point score for each of the 10 individual teeth (ranging from 3 to 1). Two examiners (G.S.A. and A.A.), previously calibrated in the use of the MHB system, scored the study casts independently on two separate occasions, under similar conditions, with a 1-week interval between the two scoring sessions. The averages of the two scores for each examiner were recorded, and the average score between the two examiners was used for the analysis. Analysis
FIGURE 1 The medial lip heights (A) and (B) were measured from the midline of the labiocolumellar groove (subnasale) to the peaks of Cupid’s bow. The noncleft-side lateral lip height (C) was measured from the lowest point of the alar base (subalare) to the peak of Cupid’s bow. The cleft-side lateral lip height (D) was measured from the lowest point of the alar base (subalare) to the proposed peak of Cupid’s bow. The noncleftside lateral lip transverse width (E) was measured from the oral commissure to the peak of Cupid’s bow. The cleft-side lateral lip transverse width (F) was measured from the oral commissure to the proposed peak of Cupid’s bow. (Reproduced with permission from Boorer et al., 2011.)
Dental Arch Relationships Dental study casts of patients, all of good quality and accurately trimmed, were used to measure dental arch relationships. The modified Huddart/Bodenham (MHB) scoring system (Mossey et al., 2003; Gray and Mossey, 2005; Dobbyn et al., 2012), a modified version of the original Huddart/Bodenham scoring system (Huddart and Bodenham, 1972), was used to score each set of models. According to this method, maxillary tooth positions or arch attributes are scored relative to the mandibular arch in maximum intercuspation. Each maxillary tooth, from the first permanent molar forward excluding the lateral incisor, or midpoint of the arch (if the tooth is missing) is scored according to its relationship with the corresponding tooth in the mandible. A cumulative score (the sum of the scores for each individual tooth) is derived from 10 categorical assessments in the mixed dentition, giving the total arch constriction MHB score. The more negative the score, the more severe the arch constriction. The cumulative MHB score uses a 40-point ordinal quasi-continuous scale ranging from 30 to þ10, corresponding to the sum
All data were analyzed using the Statistical Package for Social Sciences, Version 21.0 for Windows (SPSS, Inc., Chicago, IL). Statistical significance was set at the P , .05 level. Descriptive statistics (mean, standard deviation, range) were calculated for the anthropometric lip measurements, and independent-sample t tests were used to investigate the presence of statistically significant differences between lip measurements and gender or laterality. Descriptive statistics were also calculated for dental arch relationships using the MHB scoring system (mean, standard deviation, median, mode, range). The presence of associations between anthropometric lip measurements and MHB scores was subsequently examined. Possible confounding determinants of facial growth, including gender, laterality, age at primary unilateral cleft lip repair, age at primary cleft palate repair, age at which dental study casts were taken, and surgeon’s experience (date at which primary repair was carried out), were investigated to identify the possible presence of any statistically significant association between these factors and the MHB scores. Similar to the methods used by Doucet et al. (2013), all variables were divided into continuous and categorical variables. Categorical variables were further subdivided into dichotomous and polytomous variables. Pearson chi-square tests were used to evaluate two dichotomous categorical variables or a dichotomous with a polytomous categorical variable. Student’s t tests were used to study a dichotomous categorical variable with a continuous variable. Regression analysis was used to examine polytomous categorical variables with continuous variables. In the latter scenario, the continuous variable was used as the independent variable (predictor), while the polytomous categorical variable (assumed to be a quasi-continuous variable) was used as the dependent variable (outcome). Analysis of Error and Reliability Intraexaminer reliability was assessed using Cohen’s weighted Kappa statistic, by rescoring all of the dental study casts, under similar conditions, after a 1-week
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interval. Interrater reliability was similarly calculated using Cohen’s weighted Kappa statistic. The concordance was considered to be very good with a kappa 0.8, good with a kappa between 0.6 and 0.8, average with a kappa between 0.4 and 0.6, and poor with a kappa ,0.4 (Landis and Koch, 1977). Systematic error was assessed for each examiner by using one-sample t tests (Houston, 1983) comparing the duplicate MHB scorings. Random error was minimized by averaging the results of the duplicate measurements (Houston, 1983) for each examiner. RESULTS Sample A total of 194 children with CUCLP, born between 2000 and 2005, who had their primary lip repair carried out by the senior author (D.M.F.), were identified. Sixty-three of these children fit the inclusion and exclusion criteria. The sample comprised 46 boys (31 left- and 15 right-sided clefts) and 17 girls (12 left- and 5 right-sided clefts). The overall left:right ratio was 43:20. The mean age at which the dental study casts were taken was 8.6 (60.9) years. Lip Anthropometric Data Mean height and transverse width measurements, as well as differences between the cleft and noncleft sides, for the total study sample are shown in Table 1. The same table also presents the cleft-side anthropometric measurements as a percentage of the same measurements on the noncleft side. No gender or cleft-side differences were found for the lip measurements. The entire patient sample had a cleft-side medial lip height deficiency. Severe cleft-side medial lip height deficiency (defined as .1 standard deviation from the mean) was seen in two patients. A total of 55 of 63 (87%) patients had a cleft-side lateral lip height deficiency, while 54 of 63 (86%) patients had a cleftside lateral lip transverse width deficiency. Severe cleftside lateral lip height deficiency was seen in 11 patients. Severe cleft-side lateral lip transverse width deficiency was observed in six patients. A combined cleft-side lateral lip element deficiency (both height and transverse width) was found in 46 of 63 (73%) patients, but no patient was found to have a combined severe cleft-side lateral lip element deficiency.
TABLE 1
Measurement
Mean (SD)
Range
Medial lip height Noncleft side (mm) Cleft side (mm) Difference (mm) Cleft side/noncleft side (%)
9.3 6.9 2.4 74
(1.5) (1.2) (0.7) (6)
6.5 to 13 4.5 to 10.5 1 to 4.5 63 to 88
Lateral lip height Noncleft side (mm) Cleft side (mm) Difference (mm) Cleft side/noncleft side (%)
11.0 8.3 2.7 77
(1.6) (1.7) (2.0) (16)
7 5 4 46
to to to to
15.5 17 6.5 131
Lateral lip transverse width Noncleft side (mm) Cleft side (mm) Difference (mm) Cleft side/noncleft side (%)
17.5 15.3 2.2 88
(2.6) (1.9) (1.8) (9)
13 11 1 58
to to to to
26 22 10 107
* Negative values in range indicate a cleft-side value greater than a noncleft-side value.
for detection of systematic error, there was no statistically significant difference, at the 5% level, between the first and second measurements. The MHB scores are presented in Table 2. When calculating the MHB score, the midpoint of the arch was used instead of the tooth, in cases in which a tooth was missing, and in two patients where one of the maxillary central incisors was not yet erupted. No significant differences were found when comparing boys to girls or left-sided to right-sided clefts. No significant associations were found between MHB scores and any other of the recorded confounding determinants of facial growth. Associations Correlations were observed between lateral lip height measurements and MHB scores (Table 3). Specifically, the percentage of lateral lip height of the cleft to the noncleft side correlated to the MHB score (r ¼ .443; P , .001) in that patients with a smaller percentage (smaller relative cleft-side lateral lip height) were more likely to have more negative MHB scores (Fig. 2). Conversely, when considering the percentage of lateral lip transverse width of cleft side to noncleft side, patients with a smaller percentage were more likely to have more positive MHB scores (r ¼ .281; P ¼ .025). No TABLE 2
MHB Scores MHB Score
Dental Arch Relationships The weighted Kappa score was calculated to be 0.885 (95% confidence interval [CI] ¼ 0.785, 0.985) for intrarater reliability and 0.862 (95% CI ¼ 0.771, 0.971) for interrater reliability. Using repeated measurements
Summary of Anthropometric Data*
Mean SD Median Mode Minimum Maximum
8.2 5.8 7 7 23 2
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TABLE 3
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Associations Between Anthopometric Lip Measurements and MHB Scores
Anthropometric Lip Measurement
Correlation Coefficient (r)
P Value
Medial lip height cleft/noncleft side (%) Lateral lip height cleft/noncleft side (%) Lateral lip transverse width cleft/noncleft side (%)
.170 .443 .281
.183 ,.001 .025
correlations were observed with medial lip height measurements. DISCUSSION The results of the present study suggest that in patients with CUCLP, the physical dimensions of the cleft lip may have a predictive role in determining dental arch relationships, using the MHB score. Patients with a more deficient cleft-side lateral lip height and less deficient cleft-side lateral lip transverse width are more likely to present more negative MHB scores. One must keep in mind, however, that associations here are relatively weak. An attempt was made in our study to control for other iatrogenic factors that could affect maxillary hypoplasia and dental arch relationships by considering patients treated by the same surgeon and with the same protocol. No significant associations were found between MHB scores and any of the measured confounding determinants of facial growth. Several previous studies have attempted to correlate the severity or extent of the cleft with later maxillary growth or dental arch relationships, but all have judged cleft severity according to the size of the palatal cleft rather than that of the labial cleft. Many of these studies support the assertion that more severe clefts are associated with more maxillary retrusion. Suzuki et al. (1993), using posteroanterior and lateral cephalograms
FIGURE 2 Scatter plot showing the correlation between the lateral lip height cleft-/noncleft-side ratio and the MHB score (P , .001). A line of best fit and the correlation coefficient (r) are also shown.
Standardized Coefficient MHB Score 17.301 15.803 18.648
95% Confidence Interval 42.977, 8.375 7.614, 12.993 34.930, 2.365
and dental casts, demonstrated that palatal cleft width significantly correlated with wider upper facial width and posterior dental arch width and vertical hypoplasia of the upper face in patients with CUCLP. Using cephalometrics, Peltomaki et al. (2001) showed that ¨ patients with CUCLP who had larger clefts and smaller arch circumference or arch length exhibited less favorable maxillary projection at 5 years of age. Honda et al. (2002) also found that patients with CUCLP who have more severe palatal tissue deficiency exhibited poorer maxillofacial growth. Liao et al. (2010) cephalometrically determined that patients with a large cleft at the time of palate repair have a shorter and more retrusive maxilla by 9 years of age than those with a small cleft. Also using cephalometric radiographic data, Chiu et al. (2011) demonstrated that a large cleft at birth increases the tendency for maxillary retrusion. On the other hand, using the GOSLON yardstick on dental study models, Hsieh et al. (2012) found that patients with CUCLP who have wide initial clefts are more likely to develop poor dental arch relationships by age 5. Some studies, however, do not support the idea that more severe clefts are associated with greater maxillary retrusion. Johnson et al. (2000) found that initial cleft severity in patients with CUCLP did not correlate with outcome in terms of dental arch relationship using the 5year-old index. Meazzini et al. (2008) looked for a cause for differences of maxillary growth impairment in patients with CUCLP by comparing worst to best growers and found no significant differences in initial cleft width. The same team also examined a consecutive sample of patients with CUCLP and found no association between maxillary growth and initial cleft severity (Meazzini et al., 2011). Reiser et al. (2010) found that large cleft sizes at the level of the canine points in infancy are associated with fewer anterior and posterior crossbites at 5 years in patients with CUCLP. In a systematic review, Chiu and Liao (2012) concluded that cleft severity is negatively correlated with later maxillary growth. This assertion was, however, based on only four studies, three of which were from the same group. Moreover, the quality of the included studies was judged to be medium to low. Seckel et al. (1995) stated that there are difficulties in measuring initial cleft size and that reproducible landmark positioning on the infant maxilla can be a reality only with optimal cast quality and an experienced investigator. This assertion emphasizes the complexity in measuring cleft severity based on the palatal cleft. However, studies
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evaluating maxillary hypoplasia or dental arch relationships in relation to the labial cleft size in patients with CUCLP are limited. Nakamura et al. (2005), in a study looking at three-dimensional analysis of facial deformities in patients with CUCLP, found that patients with greater upper lip tissue volume at the time of lip repair showed better maxillary growth by the age of 15 years. The extent of the cleft lip may affect the difficulty of surgical repair, thereby indirectly influencing the outcome via greater generation of scar tissue or amount of surgical tissue movement. The amount of lip deficiency may also correlate with the amount of palatal or alveolar tissue deficiency and hence a more difficult palatal surgical repair, again indirectly affecting outcome. Wiggman et al. (2013) concluded that maxillary development in patients with CUCLP is mainly dependent on the treatment performed rather than the severity of the cleft. Undoubtedly, all cases of CUCLP are not inherently identical and do not share the same prognosis. This probably has an influence on the growth potential, regardless of treatment protocol. Midfacial growth depends on numerous factors, such as the initial anatomic/clinical form (Peltomaki ¨ et al., 2001; Reiser et al., 2010; Chiu et al., 2011), the treatment protocol and surgical techniques used (Wiggman et al., 2013), and the biological variation of the craniofacial complex and its growth (Peltomaki et al., ¨ 2001), explaining interindividual variation. A more detailed analysis of the different cleft suptypes seems necessary to distinguish between the role of therapeutic action and the growth potential of the child. It is thus important to establish a very precise initial evaluation of the malformation to avoid comparing different entities and making errors of appreciation (Delestan et al., 2013). Clinical criteria used to establish differences within a population with CUCLP can be based on the extent of the nasolabial deformity, the width of the cleft, and the cleft palate severity (Doucet et al., 2013). The heterogeneity of CUCLP and its characterization by cleft severity can have important clinical implications. Treatment outcome, and favorable or unfavorable maxillary growth, can be anticipated according to initial cleft severity; consequently, different severities of CUCLP can be directed to different treatment protocols (Peltomaki ¨ et al., 2001). Perhaps every child needs individual treatment based on individual presenting morphology, rather than conforming to a predetermined surgical treatment protocol (Reiser et al., 2010). For example, in the case of a child with a more severe cleft, prone sleep position, later palate closure, or a staged palatal closure might be proposed (Hsieh et al., 2012). Moreover, growth prognosis may be identified early, and those with the worst prognosis may require special attention in order to receive appropriate treatment (Chiu et al., 2011; Doucet et al., 2013). In a recent systematic review evaluating indices used to assess malocclusion in patients with cleft lip and palate, the authors conclude that current evidence suggests that the
FIGURE 3 Bar chart showing the average lateral lip height cleft-/ noncleft-side ratio for each of the translated GOSLON Yardstick scores. Whiskers represent one standard deviation from the mean.
MHB scoring system equals or outperforms the other indices, according to World Health Organization criteria, and hence should be considered as the standard to measure outcomes in patients with clefts (Altalibi et al., 2013). The advantages of the MHB scoring system are its objectivity, relative simplicity, and avoidance of the need for a calibration course. The MHB uses a 40-point ordinal quasi-continuous scale, which allows differentiation of outcome between cases that would be identified as the same with the 5-year-old or GOSLON 5-point indices. Potential disadvantages of the MHB system are that it does not take into account the underlying anteroposterior skeletal discrepancy or incisor inclination and that it is based on crossbites, some of which may be orthodontically treatable. However, a high correlation has been found between the sagittal relationship and the anterior tooth relationship (Staudt and Kiliaridis, 2009). Despite the performance and ease of use of the MHB scoring system, a large number of studies and authors use the GOSLON yardstick score because of its traditional preference in cleft research. On these grounds, based on the method defined by Dobbyn et al. (2012), we translated the MHB score into a GOSLON yardstick score. This resulted in the translation of our MHB scores into GOSLON scores of 1 (n ¼ 12), 2 (n ¼ 20), 3 (n ¼ 12), 4 (n ¼ 16), and 5 (n ¼ 3). Correlations were observed between lip anthropometric measurements and translated GOSLON yardstick scores. Patients with a smaller relative cleft-side lateral lip height were more likely to have higher translated GOSLON yardstick scores (r¼.503; P , .001; Fig. 3), while those with
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a smaller relative cleft-side transverse width were more likely to have lower translated GOSLON yardstick scores (r ¼ .278; P ¼ .028). The present study highlights the variation seen in cleftside lateral lip element deficiency, both in the vertical and transverse dimensions, and the potential correlations that this deficiency may have with dental arch relationships. Within the spectrum of the deformity for patients with CUCLP, the degree of cleft-side lateral lip element hypoplasia seems predictive of the resulting dental arch relationships and perhaps even predictive of global lateral segment hypoplasia. One could speculate that cleft-side lateral lip element deficiency may ultimately correlate with maxillary growth. Further research in this direction is required to confirm such possible correlations. Our findings may also have implications in the design of research protocols for cleft lip and palate growth studies. A revision of the traditional pooling of patients (Peltomaki et al., ¨ 2001) may be beneficial, such that children with CUCLP could be sorted by severity before correlating specific treatment variables with varying outcomes. CONCLUSIONS In patients with CUCLP, there is a wide variability in the degree of deficiency of the cleft-side lateral lip element, both in the vertical and in the transverse dimension. We have demonstrated that the extent of this deficiency may predict the resulting dental arch relationships despite associations being rather weak. REFERENCES Altalibi M, Saltaji H, Edwards R, Major PW, Flores-Mir C. Indices to assess malocclusion in patients with cleft lip and palate. Eur J Orthod. 2013;35:772–782. Bergland O, Sidhu SS. Occlusal changes from the deciduous to the early mixed dentition in unilateral complete clefts. Cleft Palate J. 1974;11:317–326. Boorer CJ, Cho DC, Vijayasekaran VS, Fisher DM. Presurgical unilateral cleft lip anthropometrics: implications for the choice of repair technique. Plast Reconstr Surg. 2011;127:774–780. Chiu YT, Liao YF. Is cleft severity related to maxillary growth in patients with unilateral cleft lip and palate? Cleft Palate Craniofac J. 2012;49:535–540. Chiu YT, Liao YF, Chen PK. Initial cleft severity and maxillary growth in patients with complete unilateral cleft lip and palate. Am J Orthod Dentofacial Orthop. 2011;140:189–195. 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. Delestan C, Montoya P, Doucet JC, Bigorre M, Baumler C, Herlin C, ¨ Daures JP, Captier G. New neonatal classification of unilateral cleft lip and palate—part 1: to predict primary lateral incisor agenesis and inherent tissue hypoplasia. Cleft Palate Craniofac J. 2014;51:392–399. Dobbyn LM, Weir JT, Macfarlane TV, Mossey PA. Calibration of the modified Huddart and Bodenham scoring system against the GOSLON/5-year-olds’ index for unilateral cleft lip and palate. Eur J Orthod. 2012;34:762–767.
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Doucet JC, Delestan C, Montoya P, Matei L, Bigorre M, Herlin C, Baumler C, Daures JP, Captier G. New neonatal classification of ¨ unilateral cleft lip and palate—part 2: to predict permanent lateral incisor agenesis and maxillary growth. Cleft Palate Craniofac J. 2014;51:533–539. Fisher DM. Unilateral cleft lip repair: an anatomical subunit approximation technique. Plast Reconstr Surg. 2005;116:61–71. Gillet DA, Clarke HM. The hybrid palatoplasty: a preliminary report. Canadian J Plast Surg. 1996;4:157–160. Gray D, Mossey PA. Evaluation of a modified Huddart/Bodenham scoring system for assessment of maxillary arch constriction in unilateral cleft lip and palate subjects. Eur J Orthod. 2005;27:507– 511. 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. Honda Y, Suzuki A, Nakamura N, Ohishi M. Relationship between primary palatal form and maxillofacial growth in Japanese children with unilateral cleft lip and palate: infancy to adolescence. Cleft Palate Craniofac J. 2002;39:527–534. Houston WJ. The analysis of errors in orthodontic measurements. Am J Orthod. 1983;83:382–390. Hsieh YJ, Liao YF, Shetty A. Predictors of poor dental arch relationship in young children with unilateral cleft lip and palate. Clin Oral Investig. 2012;16:1261–1266. Huang CS, Cheung HC, Chen YR, Noordhoff MS. Maxillary dental arch affected by different sleep positions in unilateral complete cleft lip and palate infants. Cleft Palate Craniofac J. 1994;31:179–184. Huddart AG, Bodenham RS. The evaluation of arch form and occlusion in unilateral cleft palate subjects. Cleft Palate J. 1972;9:194–209. Johnson N, Williams A, Singer S, Southall P, Sandy J. Initial cleft size does not correlate with outcome in unilateral cleft lip and palate. Eur J Orthod. 2000;22:93–100. Landis JR, Koch GG. An application of hierarchical kappa-type statistics in the assessment of majority agreement among multiple observers. Biometrics. 1977;33:363–374. Liao YF, Prasad NK, Chiu YT, Yun C, Chen PK. Cleft size at the time of palate repair in complete unilateral cleft lip and palate as an indicator of maxillary growth. Int J Oral Maxillofac Surg. 2010;39:956–961. Meazzini MC, Donati V, Garattini G, Brusati R. Maxillary growth impairment in cleft lip and palate patients: a simplified approach in the search for a cause. J Craniofac Surg. 2008;19:1302–1307. Meazzini MC, Tortora C, Morabito A, Garattini G, Brusati R. Factors that affect variability in impairment of maxillary growth in patients with cleft lip and palate treated using the same surgical protocol. J Plast Surg Hand Surg. 2011;45:188–193. Mossey PA, Clark JD, Gray D. Preliminary investigation of a modified Huddart/Bodenham scoring system for assessment of maxillary arch constriction in unilateral cleft lip and palate subjects. Eur J Orthod. 2003;25:251–257. Nakamura N, Suzuki A, Takahashi H, Honda Y, Sasaguri M, Ohishi M. A longitudinal study on influence of primary facial deformities on maxillofacial growth in patients with cleft lip and palate. Cleft Palate Craniofac J. 2005;42:633–640. Noordhoff MS. The Surgical Technique for the Unilateral Cleft LipNasal Deformity. Taipei, Taiwan: Noordhoff Craniofacial Foundation; 1997. Peltomaki T, Vendittelli BL, Grayson BH, Cutting CB, Brecht LE. ¨ Associations between severity of clefting and maxillary growth in patients with unilateral cleft lip and palate treated with infant orthopedics. Cleft Palate Craniofac J. 2001;38:582–586. Reiser E, Skoog V, Gerdin B, Andlin-Sobocki A. Association between cleft size and crossbite in children with cleft palate and unilateral cleft lip and palate. Cleft Palate Craniofac J. 2010;47:175–181.
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Ross RB. Treatment variables affecting facial growth in complete unilateral cleft lip and palate. Cleft Palate J. 1987;24:5–77. Seckel NG, van der Tweel I, Elema GA, Specken TF. Landmark positioning on maxilla of cleft lip and palate infant—a reality? Cleft Palate Craniofac J. 1995;32:434–441. 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. Staudt CB, Kiliaridis S. Divergence in prevalence of mesiocclusion
caused by different diagnostic criteria. Am J Orthod Dentofacial Orthop. 2009;135:323–327. Suzuki A, Mukai Y, Ohishi M, Miyanoshita Y, Tashiro H. Relationship between cleft severity and dentocraniofacial morphology in Japanese subjects with isolated cleft palate and complete unilateral cleft lip and palate. Cleft Palate Craniofac J. 1993;30:175–181. Wiggman K, Larson M, Larson O, Semb G, Brattstrom V. The ¨ influence of the initial width of the cleft in patients with unilateral cleft lip and palate related to final treatment outcome in the maxilla at 17 years of age. Eur J Orthod. 2013;35:335–340.
ORIGINAL ARTICLE Presurgical Cleft Lip Anthropometrics and Dental Arch Relationships in Patients With Complete Unilateral Cleft Lip and Palate Gregory S. Antonarakis, D.D.S., M.Sc., Ph.D., Alex Adibfar, B.Sc., Bryan D. Tompson, D.D.S., D. Paedo, D. Ortho., John Daskalogiannakis, D.D.S., M.Sc., F.R.C.D.(C.), David M. Fisher, M.D., M.F.R.C.S.(C.), F.A.C.S. Objective: To investigate associations between anthropometric lip measurements and dental arch relationships in patients with complete unilateral cleft lip and palate (CUCLP). Design: Retrospective cross-sectional study. Patients: Children with CUCLP. Methods: Anthropometric lip measurements, made immediately prior to lip repair, were available for each patient. The dental arch relationships were evaluated on dental study casts (8.6 6 0.9 years) taken prior to any orthodontic treatment and prior to alveolar bone graft, using the modified Huddart and Bodenham (MHB) scoring system. The presence of associations between anthropometric lip measurements and dental arch relationships was determined using linear regression analysis. Results: In the 63 patients included in the study, the cleft lateral lip element was deficient in height in 87% and in transverse width in 86% of patients. Patients with more deficient cleft-side lateral lip height were more likely to present with more negative MHB scores (r ¼ .443; P , .001). Conversely, patients with more deficient cleft-side lateral lip transverse width more often presented with more positive MHB scores (r ¼ .281; P ¼ .025). Conclusions: In patients with CUCLP, there is a wide variability in the degree of deficiency of the cleft-side lateral lip element, both in the vertical and in the transverse dimension. The extent of this deficiency may, in part, predict the resulting dental arch relationships. KEY WORDS:
cleft lip anthropometry, dental arch relationship, scoring system, unilateral cleft lip and palate
Maxillary hypoplasia, and the consequent midface retrusion, are a common occurrence in patients with complete unilateral cleft lip and palate (CUCLP). Approximately 48% of patients with CUCLP in our institution
were found to have midface retrusion that was severe enough to require orthognathic surgery (Daskalogiannakis and Mehta, 2009). Two factors have been proposed to play a role in the etiology of this growth deficiency (Ross, 1987; Chiu et al., 2011): (1) the intrinsic factor, such as a developmental deficiency leading to the formation of a cleft and the growth potential of the midfacial skeleton, and (2) the iatrogenic factor, introduced via treatments, mainly from surgical repair. Surgeon experience and the influence of surgical technique have often been implicated in the severity of maxillary hypoplasia and the variation seen among patients (Shaw et al., 1992). However, even when considering patients treated by the same surgeon and with the same protocol (thus eliminating variation due to iatrogenic factors), varying results are observed with respect to maxillary hypoplasia (Meazzini et al., 2011). Despite similar iatrogenic influences, maxillofacial growth deficiency is encountered in certain patients but not in others (Nakamura et al., 2005). This inconsistency suggests that individual intrinsic factors may play an important role in the maxillary growth potential. Wide phenotypic variation is exhibited in CUCLP with regard to severity of the birth deformity. Moreover, throughout the postnatal growth period, one can observe
Dr. Antonarakis is Craniofacial Orthodontic Fellow, Division of Orthodontics, The Hospital for Sick Children, Toronto, Ontario, Canada, and Department of Orthodontics, Faculty of Dentistry, University of Geneva, Switzerland. Mr. Adibfar is summer student, Division of Plastic Surgery, The Hospital for Sick Children, Toronto, Ontario, Canada. Dr. Tompson is Head, Division of Orthodontics, The Hospital for Sick Children, Toronto, Ontario, Canada, and Associate Professor and Head, Department of Orthodontics, Faculty of Dentistry, University of Toronto, Ontario, Canada. Dr. Daskalogiannakis is Staff Orthodontist, Division of Orthodontics, The Hospital for Sick Children, Toronto, Ontario, Canada, and Associate Professor, Department of Orthodontics, Faculty of Dentistry, University of Toronto, Ontario, Canada and Private practice, Toronto, Ontario, Canada. Dr. Fisher is Medical Director, Cleft Lip and Palate Program, Division of Plastic Surgery, The Hospital for Sick Children, Toronto, Ontario, Canada, and Associate Professor, Department of Surgery, University of Toronto, Toronto, Ontario, Canada. Submitted October 2013; Revised December 2013, February 2014; Accepted February 2014. 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: 10.1597/13-272 269
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a wide variation in maxillary skeletal growth and resulting dental occlusion. The severity of the CUCLP at birth may reflect differing contributions of tissue deficiency and/or tissue displacement. The critical factors responsible for the variable maxillary hypoplasia and associated malocclusion are not known. If the initial deformity and maxillary skeletal growth are linked, it is plausible to assume that the midface growth problem associated with tissue deficiency will play a role throughout the postnatal growth period (Peltomaki ¨ et al., 2001). Evidently, one must also keep in mind that maxillary hypoplasia may be related to horizontal, vertical, and/or transverse growth. To investigate a possible association between the severity of the cleft and the midface growth potential of a patient with CUCLP, a methodology for assessment of cleft severity is necessary. Cleft severity is often determined on the basis of cleft or palate size (Chiu et al., 2011), but this can be influenced by factors such as infant orthopedics, aberrant function (such as the modeling effect of the tongue on the maxillary arch during the prenatal and neonatal period; Delestan et al., 2013), sleeping position (Huang et al., 1994), or differences in the general size of the infant. Liao et al. (2010) postulated that the cleft size primarily reflects tissue displacement, mainly through tongue force, rather than tissue deficiency. The extent of cleft-side lateral lip element deficiency has not attracted much attention in either the clinical or the research setting. Anthropometry is an option for quantifying this type of deficiency. What is required is a relative measurement of the ratio of the cleft lip to the noncleft-side lip. This can provide a more reliable estimation of cleft severity in patients with CUCLP as compared with absolute measurements. Patients with unilateral clefts have been shown to exhibit large variability in lip anthropometric measurements (Boorer et al., 2011). We hypothesize that the physical dimensions of the lip on the cleft side have a predictive role in determining dental arch relationships. The aim of the current study was thus to investigate the presence of any association between the size or extent of the unilateral cleft lip deficiency and dental arch relationships in patients with CUCLP. To the best of the authors’ knowledge, no previous attempt at exploring such an association has been made to date. MATERIALS
AND
METHODS
The current study design was retrospective and crosssectional in nature and approved by the Research Ethics Board of the Hospital for Sick Children. A chart review was carried out to identify children born with CUCLP between the years 2000 and 2005. Inclusion criteria comprised the following: primary unilateral cleft lip repair carried out by the senior author (D.M.F.) and dental study casts available at the age of 6 years or older with patients in their mixed dentition, before any orthodontic treatment or alveolar bone grafting. The following were our exclusion criteria:
children whose cleft was part of a craniofacial syndrome; children who had secondary surgical treatment prior to the evaluation of the dental study casts; children who had teeth extracted prematurely due to caries, orthodontics, or trauma; dental study casts of insufficient diagnostic quality due to damage (such as broken teeth); and dental study casts available with patients in their deciduous dentition due to the increase in the incidence of crossbite in the transition from the deciduous to the early mixed dentition (Bergland and Sidhu, 1974). All of the children had been treated according to the following treatment protocol: presurgical infant orthopedics using the nasoalveolar molding technique described by Grayson et al. (1999) for a period of approximately 12 to 24 weeks (until primary repair), primary unilateral cleft lip repair at approximately 3 to 6 months of age using the technique of Fisher (2005), and primary repair of the cleft palate at approximately 12 months of age using the hybrid palatoplasty technique (Gillet and Clarke, 1996). Determination of Cleft Phenotype Determination of CUCLP phenotype was based on clinical diagnosis. All diagnoses were made by the senior author (D.M.F.) at or soon after birth and confirmed at the time of primary unilateral cleft lip repair. Laterality of the cleft (left- or right-sided) was also recorded. Lip Anthropometric Measurements The senior author (D.M.F.) performed all of the anthropometric lip measurements, with calipers, immediately prior to primary cleft lip repair with the patient under general anesthesia. Vertical height and transverse width measurements of both the cleft and noncleft sides of the lip were recorded to the nearest 0.5 mm. The anthropometric points used for measurements are shown in Figure 1. The medial lip height was measured from the midline of the labiocolumellar groove (subnasale) to the peaks of Cupid’s bow. The lateral lip height of the noncleft side was measured from the lowest point of the alar base (subalare) to the peak of Cupid’s bow. The lateral lip height of the cleft side was measured from the lowest point of the alar base (subalare) to the proposed peak of Cupid’s bow. The proposed peak of Cupid’s bow was defined, according to Noordhoff, as the point along the vermilion-cutaneous junction where the cutaneous roll and red line (vermilion-mucosal junction) begin to converge medially (Noordhoff, 1997). The lateral lip transverse width of the noncleft side was measured from the oral commissure to the peak of Cupid’s bow. The lateral lip transverse width of the cleft side was measured from the oral commissure to the proposed peak of Cupid’s bow at Noordhoff’s point.
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of the five-point score for each of the 10 individual teeth (ranging from 3 to 1). Two examiners (G.S.A. and A.A.), previously calibrated in the use of the MHB system, scored the study casts independently on two separate occasions, under similar conditions, with a 1-week interval between the two scoring sessions. The averages of the two scores for each examiner were recorded, and the average score between the two examiners was used for the analysis. Analysis
FIGURE 1 The medial lip heights (A) and (B) were measured from the midline of the labiocolumellar groove (subnasale) to the peaks of Cupid’s bow. The noncleft-side lateral lip height (C) was measured from the lowest point of the alar base (subalare) to the peak of Cupid’s bow. The cleft-side lateral lip height (D) was measured from the lowest point of the alar base (subalare) to the proposed peak of Cupid’s bow. The noncleftside lateral lip transverse width (E) was measured from the oral commissure to the peak of Cupid’s bow. The cleft-side lateral lip transverse width (F) was measured from the oral commissure to the proposed peak of Cupid’s bow. (Reproduced with permission from Boorer et al., 2011.)
Dental Arch Relationships Dental study casts of patients, all of good quality and accurately trimmed, were used to measure dental arch relationships. The modified Huddart/Bodenham (MHB) scoring system (Mossey et al., 2003; Gray and Mossey, 2005; Dobbyn et al., 2012), a modified version of the original Huddart/Bodenham scoring system (Huddart and Bodenham, 1972), was used to score each set of models. According to this method, maxillary tooth positions or arch attributes are scored relative to the mandibular arch in maximum intercuspation. Each maxillary tooth, from the first permanent molar forward excluding the lateral incisor, or midpoint of the arch (if the tooth is missing) is scored according to its relationship with the corresponding tooth in the mandible. A cumulative score (the sum of the scores for each individual tooth) is derived from 10 categorical assessments in the mixed dentition, giving the total arch constriction MHB score. The more negative the score, the more severe the arch constriction. The cumulative MHB score uses a 40-point ordinal quasi-continuous scale ranging from 30 to þ10, corresponding to the sum
All data were analyzed using the Statistical Package for Social Sciences, Version 21.0 for Windows (SPSS, Inc., Chicago, IL). Statistical significance was set at the P , .05 level. Descriptive statistics (mean, standard deviation, range) were calculated for the anthropometric lip measurements, and independent-sample t tests were used to investigate the presence of statistically significant differences between lip measurements and gender or laterality. Descriptive statistics were also calculated for dental arch relationships using the MHB scoring system (mean, standard deviation, median, mode, range). The presence of associations between anthropometric lip measurements and MHB scores was subsequently examined. Possible confounding determinants of facial growth, including gender, laterality, age at primary unilateral cleft lip repair, age at primary cleft palate repair, age at which dental study casts were taken, and surgeon’s experience (date at which primary repair was carried out), were investigated to identify the possible presence of any statistically significant association between these factors and the MHB scores. Similar to the methods used by Doucet et al. (2013), all variables were divided into continuous and categorical variables. Categorical variables were further subdivided into dichotomous and polytomous variables. Pearson chi-square tests were used to evaluate two dichotomous categorical variables or a dichotomous with a polytomous categorical variable. Student’s t tests were used to study a dichotomous categorical variable with a continuous variable. Regression analysis was used to examine polytomous categorical variables with continuous variables. In the latter scenario, the continuous variable was used as the independent variable (predictor), while the polytomous categorical variable (assumed to be a quasi-continuous variable) was used as the dependent variable (outcome). Analysis of Error and Reliability Intraexaminer reliability was assessed using Cohen’s weighted Kappa statistic, by rescoring all of the dental study casts, under similar conditions, after a 1-week
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interval. Interrater reliability was similarly calculated using Cohen’s weighted Kappa statistic. The concordance was considered to be very good with a kappa 0.8, good with a kappa between 0.6 and 0.8, average with a kappa between 0.4 and 0.6, and poor with a kappa ,0.4 (Landis and Koch, 1977). Systematic error was assessed for each examiner by using one-sample t tests (Houston, 1983) comparing the duplicate MHB scorings. Random error was minimized by averaging the results of the duplicate measurements (Houston, 1983) for each examiner. RESULTS Sample A total of 194 children with CUCLP, born between 2000 and 2005, who had their primary lip repair carried out by the senior author (D.M.F.), were identified. Sixty-three of these children fit the inclusion and exclusion criteria. The sample comprised 46 boys (31 left- and 15 right-sided clefts) and 17 girls (12 left- and 5 right-sided clefts). The overall left:right ratio was 43:20. The mean age at which the dental study casts were taken was 8.6 (60.9) years. Lip Anthropometric Data Mean height and transverse width measurements, as well as differences between the cleft and noncleft sides, for the total study sample are shown in Table 1. The same table also presents the cleft-side anthropometric measurements as a percentage of the same measurements on the noncleft side. No gender or cleft-side differences were found for the lip measurements. The entire patient sample had a cleft-side medial lip height deficiency. Severe cleft-side medial lip height deficiency (defined as .1 standard deviation from the mean) was seen in two patients. A total of 55 of 63 (87%) patients had a cleft-side lateral lip height deficiency, while 54 of 63 (86%) patients had a cleftside lateral lip transverse width deficiency. Severe cleftside lateral lip height deficiency was seen in 11 patients. Severe cleft-side lateral lip transverse width deficiency was observed in six patients. A combined cleft-side lateral lip element deficiency (both height and transverse width) was found in 46 of 63 (73%) patients, but no patient was found to have a combined severe cleft-side lateral lip element deficiency.
TABLE 1
Measurement
Mean (SD)
Range
Medial lip height Noncleft side (mm) Cleft side (mm) Difference (mm) Cleft side/noncleft side (%)
9.3 6.9 2.4 74
(1.5) (1.2) (0.7) (6)
6.5 to 13 4.5 to 10.5 1 to 4.5 63 to 88
Lateral lip height Noncleft side (mm) Cleft side (mm) Difference (mm) Cleft side/noncleft side (%)
11.0 8.3 2.7 77
(1.6) (1.7) (2.0) (16)
7 5 4 46
to to to to
15.5 17 6.5 131
Lateral lip transverse width Noncleft side (mm) Cleft side (mm) Difference (mm) Cleft side/noncleft side (%)
17.5 15.3 2.2 88
(2.6) (1.9) (1.8) (9)
13 11 1 58
to to to to
26 22 10 107
* Negative values in range indicate a cleft-side value greater than a noncleft-side value.
for detection of systematic error, there was no statistically significant difference, at the 5% level, between the first and second measurements. The MHB scores are presented in Table 2. When calculating the MHB score, the midpoint of the arch was used instead of the tooth, in cases in which a tooth was missing, and in two patients where one of the maxillary central incisors was not yet erupted. No significant differences were found when comparing boys to girls or left-sided to right-sided clefts. No significant associations were found between MHB scores and any other of the recorded confounding determinants of facial growth. Associations Correlations were observed between lateral lip height measurements and MHB scores (Table 3). Specifically, the percentage of lateral lip height of the cleft to the noncleft side correlated to the MHB score (r ¼ .443; P , .001) in that patients with a smaller percentage (smaller relative cleft-side lateral lip height) were more likely to have more negative MHB scores (Fig. 2). Conversely, when considering the percentage of lateral lip transverse width of cleft side to noncleft side, patients with a smaller percentage were more likely to have more positive MHB scores (r ¼ .281; P ¼ .025). No TABLE 2
MHB Scores MHB Score
Dental Arch Relationships The weighted Kappa score was calculated to be 0.885 (95% confidence interval [CI] ¼ 0.785, 0.985) for intrarater reliability and 0.862 (95% CI ¼ 0.771, 0.971) for interrater reliability. Using repeated measurements
Summary of Anthropometric Data*
Mean SD Median Mode Minimum Maximum
8.2 5.8 7 7 23 2
Antonarakis et al., CLEFT LIP ANTHROPOMETRICS AND DENTAL ARCH RELATIONSHIPS
TABLE 3
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Associations Between Anthopometric Lip Measurements and MHB Scores
Anthropometric Lip Measurement
Correlation Coefficient (r)
P Value
Medial lip height cleft/noncleft side (%) Lateral lip height cleft/noncleft side (%) Lateral lip transverse width cleft/noncleft side (%)
.170 .443 .281
.183 ,.001 .025
correlations were observed with medial lip height measurements. DISCUSSION The results of the present study suggest that in patients with CUCLP, the physical dimensions of the cleft lip may have a predictive role in determining dental arch relationships, using the MHB score. Patients with a more deficient cleft-side lateral lip height and less deficient cleft-side lateral lip transverse width are more likely to present more negative MHB scores. One must keep in mind, however, that associations here are relatively weak. An attempt was made in our study to control for other iatrogenic factors that could affect maxillary hypoplasia and dental arch relationships by considering patients treated by the same surgeon and with the same protocol. No significant associations were found between MHB scores and any of the measured confounding determinants of facial growth. Several previous studies have attempted to correlate the severity or extent of the cleft with later maxillary growth or dental arch relationships, but all have judged cleft severity according to the size of the palatal cleft rather than that of the labial cleft. Many of these studies support the assertion that more severe clefts are associated with more maxillary retrusion. Suzuki et al. (1993), using posteroanterior and lateral cephalograms
FIGURE 2 Scatter plot showing the correlation between the lateral lip height cleft-/noncleft-side ratio and the MHB score (P , .001). A line of best fit and the correlation coefficient (r) are also shown.
Standardized Coefficient MHB Score 17.301 15.803 18.648
95% Confidence Interval 42.977, 8.375 7.614, 12.993 34.930, 2.365
and dental casts, demonstrated that palatal cleft width significantly correlated with wider upper facial width and posterior dental arch width and vertical hypoplasia of the upper face in patients with CUCLP. Using cephalometrics, Peltomaki et al. (2001) showed that ¨ patients with CUCLP who had larger clefts and smaller arch circumference or arch length exhibited less favorable maxillary projection at 5 years of age. Honda et al. (2002) also found that patients with CUCLP who have more severe palatal tissue deficiency exhibited poorer maxillofacial growth. Liao et al. (2010) cephalometrically determined that patients with a large cleft at the time of palate repair have a shorter and more retrusive maxilla by 9 years of age than those with a small cleft. Also using cephalometric radiographic data, Chiu et al. (2011) demonstrated that a large cleft at birth increases the tendency for maxillary retrusion. On the other hand, using the GOSLON yardstick on dental study models, Hsieh et al. (2012) found that patients with CUCLP who have wide initial clefts are more likely to develop poor dental arch relationships by age 5. Some studies, however, do not support the idea that more severe clefts are associated with greater maxillary retrusion. Johnson et al. (2000) found that initial cleft severity in patients with CUCLP did not correlate with outcome in terms of dental arch relationship using the 5year-old index. Meazzini et al. (2008) looked for a cause for differences of maxillary growth impairment in patients with CUCLP by comparing worst to best growers and found no significant differences in initial cleft width. The same team also examined a consecutive sample of patients with CUCLP and found no association between maxillary growth and initial cleft severity (Meazzini et al., 2011). Reiser et al. (2010) found that large cleft sizes at the level of the canine points in infancy are associated with fewer anterior and posterior crossbites at 5 years in patients with CUCLP. In a systematic review, Chiu and Liao (2012) concluded that cleft severity is negatively correlated with later maxillary growth. This assertion was, however, based on only four studies, three of which were from the same group. Moreover, the quality of the included studies was judged to be medium to low. Seckel et al. (1995) stated that there are difficulties in measuring initial cleft size and that reproducible landmark positioning on the infant maxilla can be a reality only with optimal cast quality and an experienced investigator. This assertion emphasizes the complexity in measuring cleft severity based on the palatal cleft. However, studies
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evaluating maxillary hypoplasia or dental arch relationships in relation to the labial cleft size in patients with CUCLP are limited. Nakamura et al. (2005), in a study looking at three-dimensional analysis of facial deformities in patients with CUCLP, found that patients with greater upper lip tissue volume at the time of lip repair showed better maxillary growth by the age of 15 years. The extent of the cleft lip may affect the difficulty of surgical repair, thereby indirectly influencing the outcome via greater generation of scar tissue or amount of surgical tissue movement. The amount of lip deficiency may also correlate with the amount of palatal or alveolar tissue deficiency and hence a more difficult palatal surgical repair, again indirectly affecting outcome. Wiggman et al. (2013) concluded that maxillary development in patients with CUCLP is mainly dependent on the treatment performed rather than the severity of the cleft. Undoubtedly, all cases of CUCLP are not inherently identical and do not share the same prognosis. This probably has an influence on the growth potential, regardless of treatment protocol. Midfacial growth depends on numerous factors, such as the initial anatomic/clinical form (Peltomaki ¨ et al., 2001; Reiser et al., 2010; Chiu et al., 2011), the treatment protocol and surgical techniques used (Wiggman et al., 2013), and the biological variation of the craniofacial complex and its growth (Peltomaki et al., ¨ 2001), explaining interindividual variation. A more detailed analysis of the different cleft suptypes seems necessary to distinguish between the role of therapeutic action and the growth potential of the child. It is thus important to establish a very precise initial evaluation of the malformation to avoid comparing different entities and making errors of appreciation (Delestan et al., 2013). Clinical criteria used to establish differences within a population with CUCLP can be based on the extent of the nasolabial deformity, the width of the cleft, and the cleft palate severity (Doucet et al., 2013). The heterogeneity of CUCLP and its characterization by cleft severity can have important clinical implications. Treatment outcome, and favorable or unfavorable maxillary growth, can be anticipated according to initial cleft severity; consequently, different severities of CUCLP can be directed to different treatment protocols (Peltomaki ¨ et al., 2001). Perhaps every child needs individual treatment based on individual presenting morphology, rather than conforming to a predetermined surgical treatment protocol (Reiser et al., 2010). For example, in the case of a child with a more severe cleft, prone sleep position, later palate closure, or a staged palatal closure might be proposed (Hsieh et al., 2012). Moreover, growth prognosis may be identified early, and those with the worst prognosis may require special attention in order to receive appropriate treatment (Chiu et al., 2011; Doucet et al., 2013). In a recent systematic review evaluating indices used to assess malocclusion in patients with cleft lip and palate, the authors conclude that current evidence suggests that the
FIGURE 3 Bar chart showing the average lateral lip height cleft-/ noncleft-side ratio for each of the translated GOSLON Yardstick scores. Whiskers represent one standard deviation from the mean.
MHB scoring system equals or outperforms the other indices, according to World Health Organization criteria, and hence should be considered as the standard to measure outcomes in patients with clefts (Altalibi et al., 2013). The advantages of the MHB scoring system are its objectivity, relative simplicity, and avoidance of the need for a calibration course. The MHB uses a 40-point ordinal quasi-continuous scale, which allows differentiation of outcome between cases that would be identified as the same with the 5-year-old or GOSLON 5-point indices. Potential disadvantages of the MHB system are that it does not take into account the underlying anteroposterior skeletal discrepancy or incisor inclination and that it is based on crossbites, some of which may be orthodontically treatable. However, a high correlation has been found between the sagittal relationship and the anterior tooth relationship (Staudt and Kiliaridis, 2009). Despite the performance and ease of use of the MHB scoring system, a large number of studies and authors use the GOSLON yardstick score because of its traditional preference in cleft research. On these grounds, based on the method defined by Dobbyn et al. (2012), we translated the MHB score into a GOSLON yardstick score. This resulted in the translation of our MHB scores into GOSLON scores of 1 (n ¼ 12), 2 (n ¼ 20), 3 (n ¼ 12), 4 (n ¼ 16), and 5 (n ¼ 3). Correlations were observed between lip anthropometric measurements and translated GOSLON yardstick scores. Patients with a smaller relative cleft-side lateral lip height were more likely to have higher translated GOSLON yardstick scores (r¼.503; P , .001; Fig. 3), while those with
Antonarakis et al., CLEFT LIP ANTHROPOMETRICS AND DENTAL ARCH RELATIONSHIPS
a smaller relative cleft-side transverse width were more likely to have lower translated GOSLON yardstick scores (r ¼ .278; P ¼ .028). The present study highlights the variation seen in cleftside lateral lip element deficiency, both in the vertical and transverse dimensions, and the potential correlations that this deficiency may have with dental arch relationships. Within the spectrum of the deformity for patients with CUCLP, the degree of cleft-side lateral lip element hypoplasia seems predictive of the resulting dental arch relationships and perhaps even predictive of global lateral segment hypoplasia. One could speculate that cleft-side lateral lip element deficiency may ultimately correlate with maxillary growth. Further research in this direction is required to confirm such possible correlations. Our findings may also have implications in the design of research protocols for cleft lip and palate growth studies. A revision of the traditional pooling of patients (Peltomaki et al., ¨ 2001) may be beneficial, such that children with CUCLP could be sorted by severity before correlating specific treatment variables with varying outcomes. CONCLUSIONS In patients with CUCLP, there is a wide variability in the degree of deficiency of the cleft-side lateral lip element, both in the vertical and in the transverse dimension. We have demonstrated that the extent of this deficiency may predict the resulting dental arch relationships despite associations being rather weak. REFERENCES Altalibi M, Saltaji H, Edwards R, Major PW, Flores-Mir C. Indices to assess malocclusion in patients with cleft lip and palate. Eur J Orthod. 2013;35:772–782. Bergland O, Sidhu SS. Occlusal changes from the deciduous to the early mixed dentition in unilateral complete clefts. Cleft Palate J. 1974;11:317–326. Boorer CJ, Cho DC, Vijayasekaran VS, Fisher DM. Presurgical unilateral cleft lip anthropometrics: implications for the choice of repair technique. Plast Reconstr Surg. 2011;127:774–780. Chiu YT, Liao YF. Is cleft severity related to maxillary growth in patients with unilateral cleft lip and palate? Cleft Palate Craniofac J. 2012;49:535–540. Chiu YT, Liao YF, Chen PK. Initial cleft severity and maxillary growth in patients with complete unilateral cleft lip and palate. Am J Orthod Dentofacial Orthop. 2011;140:189–195. 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. Delestan C, Montoya P, Doucet JC, Bigorre M, Baumler C, Herlin C, ¨ Daures JP, Captier G. New neonatal classification of unilateral cleft lip and palate—part 1: to predict primary lateral incisor agenesis and inherent tissue hypoplasia. Cleft Palate Craniofac J. 2014;51:392–399. Dobbyn LM, Weir JT, Macfarlane TV, Mossey PA. Calibration of the modified Huddart and Bodenham scoring system against the GOSLON/5-year-olds’ index for unilateral cleft lip and palate. Eur J Orthod. 2012;34:762–767.
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Doucet JC, Delestan C, Montoya P, Matei L, Bigorre M, Herlin C, Baumler C, Daures JP, Captier G. New neonatal classification of ¨ unilateral cleft lip and palate—part 2: to predict permanent lateral incisor agenesis and maxillary growth. Cleft Palate Craniofac J. 2014;51:533–539. Fisher DM. Unilateral cleft lip repair: an anatomical subunit approximation technique. Plast Reconstr Surg. 2005;116:61–71. Gillet DA, Clarke HM. The hybrid palatoplasty: a preliminary report. Canadian J Plast Surg. 1996;4:157–160. Gray D, Mossey PA. Evaluation of a modified Huddart/Bodenham scoring system for assessment of maxillary arch constriction in unilateral cleft lip and palate subjects. Eur J Orthod. 2005;27:507– 511. 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. Honda Y, Suzuki A, Nakamura N, Ohishi M. Relationship between primary palatal form and maxillofacial growth in Japanese children with unilateral cleft lip and palate: infancy to adolescence. Cleft Palate Craniofac J. 2002;39:527–534. Houston WJ. The analysis of errors in orthodontic measurements. Am J Orthod. 1983;83:382–390. Hsieh YJ, Liao YF, Shetty A. Predictors of poor dental arch relationship in young children with unilateral cleft lip and palate. Clin Oral Investig. 2012;16:1261–1266. Huang CS, Cheung HC, Chen YR, Noordhoff MS. Maxillary dental arch affected by different sleep positions in unilateral complete cleft lip and palate infants. Cleft Palate Craniofac J. 1994;31:179–184. Huddart AG, Bodenham RS. The evaluation of arch form and occlusion in unilateral cleft palate subjects. Cleft Palate J. 1972;9:194–209. Johnson N, Williams A, Singer S, Southall P, Sandy J. Initial cleft size does not correlate with outcome in unilateral cleft lip and palate. Eur J Orthod. 2000;22:93–100. Landis JR, Koch GG. An application of hierarchical kappa-type statistics in the assessment of majority agreement among multiple observers. Biometrics. 1977;33:363–374. Liao YF, Prasad NK, Chiu YT, Yun C, Chen PK. Cleft size at the time of palate repair in complete unilateral cleft lip and palate as an indicator of maxillary growth. Int J Oral Maxillofac Surg. 2010;39:956–961. Meazzini MC, Donati V, Garattini G, Brusati R. Maxillary growth impairment in cleft lip and palate patients: a simplified approach in the search for a cause. J Craniofac Surg. 2008;19:1302–1307. Meazzini MC, Tortora C, Morabito A, Garattini G, Brusati R. Factors that affect variability in impairment of maxillary growth in patients with cleft lip and palate treated using the same surgical protocol. J Plast Surg Hand Surg. 2011;45:188–193. Mossey PA, Clark JD, Gray D. Preliminary investigation of a modified Huddart/Bodenham scoring system for assessment of maxillary arch constriction in unilateral cleft lip and palate subjects. Eur J Orthod. 2003;25:251–257. Nakamura N, Suzuki A, Takahashi H, Honda Y, Sasaguri M, Ohishi M. A longitudinal study on influence of primary facial deformities on maxillofacial growth in patients with cleft lip and palate. Cleft Palate Craniofac J. 2005;42:633–640. Noordhoff MS. The Surgical Technique for the Unilateral Cleft LipNasal Deformity. Taipei, Taiwan: Noordhoff Craniofacial Foundation; 1997. Peltomaki T, Vendittelli BL, Grayson BH, Cutting CB, Brecht LE. ¨ Associations between severity of clefting and maxillary growth in patients with unilateral cleft lip and palate treated with infant orthopedics. Cleft Palate Craniofac J. 2001;38:582–586. Reiser E, Skoog V, Gerdin B, Andlin-Sobocki A. Association between cleft size and crossbite in children with cleft palate and unilateral cleft lip and palate. Cleft Palate Craniofac J. 2010;47:175–181.
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Cleft Palate–Craniofacial Journal, May 2015, Vol. 52 No. 3
Ross RB. Treatment variables affecting facial growth in complete unilateral cleft lip and palate. Cleft Palate J. 1987;24:5–77. Seckel NG, van der Tweel I, Elema GA, Specken TF. Landmark positioning on maxilla of cleft lip and palate infant—a reality? Cleft Palate Craniofac J. 1995;32:434–441. 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. Staudt CB, Kiliaridis S. Divergence in prevalence of mesiocclusion
caused by different diagnostic criteria. Am J Orthod Dentofacial Orthop. 2009;135:323–327. Suzuki A, Mukai Y, Ohishi M, Miyanoshita Y, Tashiro H. Relationship between cleft severity and dentocraniofacial morphology in Japanese subjects with isolated cleft palate and complete unilateral cleft lip and palate. Cleft Palate Craniofac J. 1993;30:175–181. Wiggman K, Larson M, Larson O, Semb G, Brattstrom V. The ¨ influence of the initial width of the cleft in patients with unilateral cleft lip and palate related to final treatment outcome in the maxilla at 17 years of age. Eur J Orthod. 2013;35:335–340.
