The Cleft Palate–Craniofacial Journal 52(3) pp. 277–286 May 2015 Ó Copyright 2015 American Cleft Palate–Craniofacial Association

ORIGINAL ARTICLE Dental Arch Relationship Outcomes in One- and Two-Stage Palatoplasty for Japanese Patients With Complete Unilateral Cleft Lip and Palate Tadashi Mikoya, D.D.S., Ph.D., Toyoko Shibukawa, D.D.S., Ph.D., Takafumi Susami, D.D.S., Ph.D., Yoshiaki Sato, D.D.S., Ph.D., Toshimoto Tengan, D.D.S., Ph.D., Hirotaka Katashima, D.D.S., Ph.D., Akihiko Oyama, M.D., Ph.D., Yusuke Matsuzawa, D.D.S., Ph.D., Yumi Ito, D.D.S., Emi Funayama, M.D., Ph.D. Objective: To compare dental arch relationship outcomes following one- and two-stage palatal repair. Design: Nonrandomized, clinical trial with concurrent control. Setting: Hokkaido University Hospital. Patients: Sixty-eight consecutively treated Japanese patients with complete unilateral cleft lip and palate. Interventions: Thirty-one of the 68 patients underwent two-stage palatoplasty with delayed hard palate closure, and 37 patients underwent one-stage pushback palatoplasty. Main Outcome Measures: Dental casts were taken at 4.9 to 6.3 (mean: 5.2) years of age in the two-stage group and at 4.0 to 6.3 (mean: 5.1) years of age in the one-stage group, and dental arch relationships were assessed using the 5-Year-Olds’ Index (5-Y) by four raters and the Huddart/ Bodenham Index (HB) by two raters. Results: Intrarater and interrater reliabilities evaluated using weighted kappa statistics were good or better for the 5-Y and HB ratings. The mean 5-Y score was 2.94 in the two-stage group and 3.13 in the one-stage group (P value was not significant). However, there was a significant difference in distributions between the groups (P , .05). The HB scores of molars were significantly greater in the two-stage group than in the one-stage group (P , .05). The rank correlation coefficients between the 5-Y and total HB score (q ¼0.840, P , .01) and between the 5-Y and the score of the incisors in the HB (q ¼ 0.814, P , .01) were significantly increased. Conclusions: These results suggest that the anteroposterior relationship was not significantly different between the groups, but the transversal relationship was better in the two-stage group than in the one-stage group. KEY WORDS:

5-Year-Olds’ Index, dental arch relationships, Huddart/Bodenham Index, Japanese infants, unilateral cleft lip and palate

Much attention has been focused on the adverse effects of surgical intervention in infants with cleft lip and palate (CLP) (Ross, 1987; Mars and Houston, 1990; Mars et al., 1992). It is thought that the timing and technique of palatal closure have a great influence on the acquisition of adequate palatal function for the development of normal speech without interfering significantly with maxillofacial growth. The main advantage of early palatal closure is to avoid the development of compensatory speech (Dorf and Curtin, 1982), but the disadvantage is impairment of midfacial growth (Rohrich et al., 1996). In a two-stage protocol, the soft and the hard palate are closed by two separate procedures at different times. Following this philosophy, the closure of the hard palate should be performed as late as possible. The main reason for preferring this technique is to facilitate favorable midfacial growth (Schmid et al., 1974; Schweckendiek, 1978; Hotz

Dr. Mikoya is Clinical Professor, Stomatognathic Function, Center for Advanced Oral Medicine, Hokkaido University Hospital. Dr. Shibukawa is Clinical Fellow, Dr. Matsuzawa is Assistant Professor, and Dr. Ito is Visiting Clinician, Department of Oral and Maxillofacial Surgery, Graduate School of Dentistry, Hokkaido University. Dr. Sato is Associate Professor, Department of Orthodontics, Graduate School of Dentistry, Hokkaido University. Dr. Oyama is Clinical Associate Professor, Dr. Funayama is Assistant Professor, Plastic and Reconstructive Surgery, Hokkaido University Hospital, Sapporo, Japan. Dr. Susami is Associate Professor, Department of Oral-Maxillofacial Surgery, Dentistry and Orthodontics, The University of Tokyo Hospital, Tokyo, Japan. Dr. Tengan is Lecturer and Dr. Katashima is Clinical Fellow, Department of Oral and Maxillofacial Functional Rehabilitation, Graduate School of Medicine, University of the Ryukyus, Okinawa, Japan. A part of this paper was presented at the 12th International Congress on Cleft Lip/Palate and Related Craniofacial Anomalies; Lake Buena Vista, Florida, May 5–10, 2013. This research was supported by JSPS KAKENHI Grant 23592911. Submitted November 2013; Accepted February 2014. Address correspondence to: Dr. Tadashi Mikoya, Clinic of Stomatognathic Function, Center for Advanced Oral Medicine, Hokkaido University Hospital, Kita 14, Nishi 5, Kita-Ku, Sapporo 060-8648, Japan. E-mail [email protected].

DOI: 10.1597/13-285 277

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and Gnoinski, 1979; Lilja et al., 2006), while a disadvantage is that the development of a speech problem is more likely (Cosman and Falk, 1980; Lohmander-Agerskov, 1998). A pioneering approach to this issue could be seen in the 1996 to 2000 Eurocleft project (Shaw et al., 2000). In the background while this intercenter research project was planned, a useful rating system for dental arch relationships was developed. This has become an important factor to assess the outcome of the primary repair, and an evaluation tool to make it possible to detect surgical problems early in the treatment process and to contribute to the improvement of the outcomes has been demanded. To estimate the overall degree of malocclusion, Huddart and Bodenham (1972) developed a numerical scoring system of the crossbite in deciduous dentition. The degrees of crossbite for individual teeth were calculated to give a total score. Based on the degree of crossbite of individual teeth, an overall score corresponding with the severity of the malocclusion was assigned to the patients. Unfortunately, because this scoring system was a method to evaluate interdental relationships and cannot evaluate the relationships of the apical bases, it has a relatively weak ability to discriminate cases with various prognoses (Fudalej et al., 2009). Indices such as the GOSLON Yardstick have been developed and adopted as a common standard in multicenter, comparative studies (Mars et al., 1987, 1992; Shaw et al., 1992a, 1992b; Williams et al., 2001). In the Eurocleft studies, the GOSLON Yardstick was found to be a more sensitive tool in discriminating differences in outcomes of dentofacial growth in the participating centers than in the cephalometric analysis of the same patients (Mars et al., 1992; Mølsted et al., 1992, 2005; Brattstrom ¨ et al., 2005). More recently, a further index also based on dental arch relationships has been developed specifically for the primary dentition (Atack et al., 1997a, 1997b). It is suggested that this index will provide an earlier assessment at an age at which the dental arches have not been affected by other treatments such as orthodontics or secondary bone grafting (Atack et al., 1998). The 5-Year-Olds’ Index is widely accepted as a simple and sensitive indicator of the early surgical outcome for the dental arch relationship and as a useful tool to compare treatment outcomes between different protocols and/or centers. There are two surgical teams for CLP at Hokkaido University Hospital, and each has a different treatment protocol. The acceptance of infants with CLP as outpatients is shared between the Department of Oral and Maxillofacial Surgery (OMS) and the Department of Plastic Surgery (PS), and the treatment systems for the patients are not yet unified due to the difference in the ways of thinking about palatal repair between the two departments. The OMS adopted a two-stage palatoplasty with delayed hard palate closure, and the PS used a one-stage pushback palatoplasty. Both types of patients received identical presurgical orthopedic treatment and lip repair, but palatal repair differed in timing and extent. The aim of

this study was to compare the dental arch relationship outcomes of the two surgical protocols using the 5-YearOlds’ Index that has been accepted as an international standard. Since the 5-Year-Olds’ Index has some ambiguous criteria, the Huddart/Bodenham Index, which could provide more detailed information regarding maxillary arch constriction, was also used. MATERIALS

AND

METHODS

Subjects The primary surgical management of patients with CLP in Hokkaido University Hospital has been performed using two different protocols by the OMS and PS teams. The difference depends on the way of thinking of each department. CLP patients who first visited the OMS outpatient clinic underwent two-stage palatoplasty, while those who first visited the PS outpatient clinic underwent one-stage palatoplasty. The OMS and PS treatment protocols are described in Table 1. The only difference between the treatment protocols was the palatoplasty technique itself. Of course, the rights that patient oneself chooses palatal repair method is guaranteed. These treatment protocols were continued from 1995 until 2007. For all patients of both protocols, the presurgical orthopedic treatment advocated by Hotz and Gnoinski (1979) was carried out at the OMS department, and lip repair using the modified Millard technique was performed at about 5 months of age at the PS department. Therefore, all patients of both treatment protocols underwent the same presurgical orthopedic and lip repair approaches by the same experienced hands, with the same techniques and timing. The two-stage palatoplasty group underwent soft palate closure at about 1.5 years of age. At the beginning, four patients in the two-stage group underwent soft palate repair using Widmaier-Perko’s method (Perko, 1979), and six patients in the same group underwent repair using Furlow’s method (Furlow, 1986). In conformity with the logic of two-stage palatal surgery with early velar closure and delayed hard palate repair, the first palate closure involved the soft palate to the posterior one third of the hard palate. In the latter 21 patients repaired using Furlow’s method, the soft palate and posterior half of the hard palate were closed simultaneously using an anterior-based vomerine mucoperiosteal flap (Kobus, 1984), in which the cleft margin incision anteriorly was extended onto the vomer to raise it, evading bone exposure. On the other hand, the one-stage palatoplasty group underwent soft and hard palate closure using the Wardill-Kilner pushback procedure at about 1.5 years of age, and a buccal mucosal flap based posteriorly to the maxillary tubercle was used for mucosal supplementation (Kaplan, 1975), with lengthening on the nasal side of soft palate. In both

Mikoya et al., COMPARISON OF DENTAL ARCH RELATIONSHIP OUTCOMES

TABLE 1

Treatment Protocols Two-Stage Group

Birth 3–5 months 18 months 5–7 years 9–11 years

One-Stage Group

Presurgical orthopedics by Hotz plate Lip repair by modified Millard Soft palate repair by Widmair-Perko or modified Furlow Hard and soft palate repair by Wardill-Kilner pushback Hard palate closure and alveolar bone grafting Alveolar bone grafting

protocols, the alveolar cleft was kept intact during primary lip repair and palate closure because alveolar bone grafting was to be done later. One hundred twenty-two patients with unilateral cleft lip and palate (UCLP) who were born from July 1995 to September 2006 visited both departments of Hokkaido University Hospital before any intervention. Of these, 81 consecutive patients were registered for this prospective study. The inclusion criteria were as follows: complete UCLP, excluding patients with Simonart’s band; Japanese ethnic background; full-term infant; and no associated congenital anomalies. Of the 81, 35 who first visited the OMS outpatient clinic were included in the two-stage group, and the remaining 46 who visited the PS outpatient clinic constituted the one-stage group. In 68 of the 81 patients who participated in this study, dental casts were duplicated during the treatment and dental arch relationships were evaluated. The remaining 13 patients were excluded from this study: five patients moved, two patients had too many decayed teeth, two patients failed to have their dental casts taken, and four patients had alveolar clefts closed using a vomerine flap during primary lip repair. A total of 68 patients in both groups underwent lip repair by three experienced plastic surgeons (H.I., K.K., and S.S.), followed by presurgical orthopedic treatment. The mean age at the time of cheiloplasty was 5.2 months (range 3.5 to 6.9 months) in the two-stage group and 5.3 months (range 3.3 to 7.9 months) in the one-stage group. Thirty-one patients in the two-stage group underwent soft palate closure by one experienced oral-maxillofacial surgeon (T.M.) at a mean age of 1 year 9 months (range 1 year 5 months to 2 years 1 month). On the other hand, 37 patients in the one-stage group underwent soft and hard palate closure simultaneously by three experienced plastic surgeons (H.I., K.K., and S.S.) at a mean age of 1 year 6 months (range, 1 year 5 months to 1 year 11 months). Thirtyone dental casts in the two-stage group were taken at a TABLE 2

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mean age of 5.2 years (range 4.9 to 6.3 years), and 37 casts in the one-stage group were taken at a mean age of 5.1 years (range 4.0 to 6.3 years). The characteristics are summarized in Table 2. No patient underwent orthodontic treatment, secondary alveolar bone grafting, or pharyngeal flap surgery prior to dental cast taking. All casts of both groups were duplicated with white stone and trimmed in the same manner. Assessment The casts were randomly numbered and assessed using the 5-Year-Olds’ Index (Atack et al., 1997a, 1997b) and the Huddart/Bodenham Index (Huddart and Bodenham, 1972). Scoring of the 5-Year-Olds’ Index was performed by four raters: two orthodontists experienced in cleft care for more than 20 years (T.S., Y.S.) and two oral and maxillofacial surgeons having more than seven years of clinical experience (T.S.’, H.K.). One orthodontist and one oral and maxillofacial surgeon were involved in cleft care at our hospital, and the remaining two raters were outsiders who participated from the other University Hospital. Four raters scored the casts from 1 to 5 individually with the reference casts provided by Bristol University twice with an interval of 1 day. All raters attended ‘‘The 5-YearOlds’ Index Calibration Course’’ by Dr. Atack and Prof. Sandy held by the Japanese Cleft Palate Association and had completed training in the use of the assessment (Sandy and Atack, 2012). Before the scoring sessions took place, each rater was calibrated using the test casts of UCLP patients other than the subjects. Each model was given a total of eight individual scores over two sessions. The Huddart/Bodenham Index was scored independently by two raters: one with more than 20 years of experience as an orthodontist (T.T.) who participated from the other hospital, and one oral and maxillofacial surgeon with more than seven years of

Sample Characteristics

Number of patients and sex distribution Presurgical orthopedics by Hotz plate Lip repair Palate repair Age at dental cast taking

Two-Stage Group

One-Stage Group

31 (14 male, 17 female) Yes Mean 5.2 mo (3.5–6.9 mo) Mean 21 mo (17–25 mo), soft palate closure Mean 5.2 y (4.9–6.3 y)

37 (15 male, 22 female) Yes Mean 5.3 mo (3.3–7.9 mo) Mean 18 mo (17–23 mo), soft and hard palate closure Mean 5.1 y (4.0–6.3 y)

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TABLE 3

Interpretation of Kappa Values (Altman, 1991)

Kappa Value

TABLE 5 Interrater Agreement (Weighted Kappa) for 5-YearOlds’ Index Scoring

Strength of Agreement Poor Fair Moderate Good Very good

,0.2 0.2–0.4 0.4–0.6 0.6–0.8 0.8–1.0

experience (T.S.’) who was engaged in cleft care at our hospital. Both raters carried out the scoring twice with an interval of 1 day. Each model was given a total of four individual scores over two sessions. The total crossbite score, by summing up all the scores for each pair of teeth, and the partial crossbite score, with the dental arch divided into five parts (central deciduous incisors, deciduous canines on the major and minor segments, and first and second deciduous molars on the major and minor segments), were calculated. None of the patients’ operators participated in the rating sessions for both indices. The results of the 5-Year-Olds’ Index in the one-stage and two-stage groups were visually compared with the results of other previous reports from domestic and overseas centers (DiBiase et al., 2002; Bongaarts et al., 2004; Clark et al., 2007; Hirano et al., 2007; Suzuki et al., 2007). Statistical Analysis Weighted kappa statistics were used to assess intrarater and interrater reliability scores for the four raters for the 5-Year-Olds’ Index and for the two raters for the Huddart/Bodenham Index. The interpretation of the kappa values was based on data according to Altman (1991) (Table 3). The unpaired t test was used to compare the mean values of the 5-Year-Olds’ Index scores and the Huddart/Bodenham Index scores between the two-stage group and the one-stage group. The chi-square test was performed to evaluate the difference in the score distribution of the 5-Year-Olds’ Index grades. P values , .05 were considered significant. The correlations of the Huddart/Bodenham Index scores with the 5-Year-Olds’ Index scores were tested using Spearman’s correlation coefficient.

Raters

Kappa Value

Standard Error of Kappa

95% Confidence Intervals

First rating T.S. 3 Y.S. T.S. 3 H.K. T.S. 3 T.S.’ Y.S. 3 H.K. Y.S. 3 T.S.’ H.K. 3 T.S.’

0.78 0.60 0.70 0.67 0.77 0.72

0.06 0.07 0.07 0.06 0.06 0.06

0.66–0.89 0.47–0.73 0.57–0.82 0.55–0.79 0.65–0.89 0.59–0.84

Second rating T.S. 3 Y.S. T.S. 3 H.K. T.S. 3 T.S.’ Y.S. 3 H.K. Y.S. 3 T.S.’ H.K. 3 T.S.’

0.83 0.75 0.62 0.82 0.69 0.63

0.05 0.05 0.07 0.05 0.06 0.07

0.74–0.93 0.64–0.86 0.49–0.75 0.73–0.91 0.58–0.81 0.49–0.77

Ethics This study was conducted in conformity with the Declaration of Helsinki and also approved by the Institutional Review Board and was performed in accordance with the ‘‘Ethical Guidance for Clinical Studies’’ of the Ministry of Health, Labour, and Welfare in Japan. RESULTS Weighted kappa statistics for double determinations of the 5-Year-Olds’ Index for four raters demonstrated values between 0.78 and 0.89 for intrarater agreement (Table 4) and between 0.60 and 0.83 for interrater agreement (Table 5), and weighted kappa statistics for double determinations of the Huddart/Bodenham Index for two raters were 0.78 and 0.82 for intrarater agreement (Table 6) and 0.62 and 0.77 for interrater agreement (Table 7). Tables 8 and 9 show the score distribution and mean scores of the 5-Year-Olds’ Index. In the two-stage group, 23.0% were scored 1 and 2, 56.0% were 3, and 21.0% were 4 and 5, with a mean score of 2.94. In the one-stage group, 23.7% were scored 1 and 2, 44.9% were 3, and 31.4% were 4 and 5, with a mean score of 3.13. The unpaired t test showed no significant difference in the mean scores between the groups. On the other hand, the chi-square test showed a significant difference in distributions between the groups (P , .05).

TABLE 4 Intrarater Agreement (Weighted Kappa) for 5-YearOlds’ Index Scoring

Raters

Kappa Value

Standard Error of Kappa

95% Confidence Intervals

T.S. Y.S. H.K. T.S.’

0.88 0.89 0.87 0.78

0.04 0.04 0.05 0.06

0.80–0.96 0.80–0.97 0.78–0.96 0.87–0.89

TABLE 6 Intrarater Agreement (Weighted Kappa) for Huddart/ Bodenham Index Scoring

Raters T.T. T.S.’

Kappa Value

Standard Error of Kappa

95% Confidence Intervals

0.79 0.82

0.07 0.06

0.65–0.91 0.70–0.94

Mikoya et al., COMPARISON OF DENTAL ARCH RELATIONSHIP OUTCOMES

TABLE 7 Interrater Agreement (Weighted Kappa) for Huddart/ Bodenham Index Scoring

Raters

Kappa Value

Standard Error of Kappa

95% Confidence Intervals

First rating T.T. 3 T.S.’

0.62

0.07

0.56–0.84

Second rating T.T. 3 T.S.’

0.78

0.07

0.65–0.90

Table 10 shows the mean Huddart/Bodenham Index scores. In the two-stage group, the total score, score of the incisors, scores of the canines on the major and minor segments, and the scores of the molars on the major and minor segments were 6.4, 3.7, 0.4, 1.4, 0.2, and 0.8, respectively. In the one-stage group, the total score, score of the incisors, scores of the canines on the major and minor segments, and the scores of the molars on the major and minor segments were 8.0, 3.2, 0.5, 1.7, 0.8, and 1.8, respectively. The unpaired t test showed significant differences in the total score (P , .05) and in the scores of molars on major and minor segments (P , .01) between the two groups. Table 11 demonstrates the correlations of the mean scores of the Huddart/Bodenham Index and the 5-YearOlds’ Index. The rank correlation coefficient was significantly higher between the 5-Year-Olds’ Index and the total Huddart/Bodenham score (q ¼ 0.840, P , .01), and between the 5-Year-Olds’ Index and the score of the incisors (q ¼ 0.814, P , .01). On the other hand, the correlations with the scores of the canines and the molars on the major and minor segments were significant but lower. Graphical comparisons of the results of this study with other previous reports are shown in Figure 1. The 5-YearOlds’ Index score distributions are summarized into three categories as follows: good (groups 1 and 2), fair (group 3), and poor (groups 4 and 5). The proportion of the sum of categories ‘‘good’’ and ‘‘fair’’ was higher in the two-stage group than in the one-stage group and other Japanese centers. On the other hand, the proportion of category ‘‘good’’ tended to be lower in Japanese centers including our hospital, but the category ‘‘poor’’ was higher than in European centers. TABLE 8 Numerical and Percentage Distributions of 5-YearOlds’ Index Scores Two-Stage Group Scores 1 2 3 4 5

(Excellent) (Good) (Fair) (Poor) (Very poor)

Frequency* 14 43 139 47 5

One-Stage Group

Percent 5.7 17.3 56.0 19.0 2.0



23.0

56.0 21.0

Frequency* 4 66 133 73 20

* Each model was given a total of eight individual scores.

Percent 1.4 22.3 44.9 24.7 6.8



23.7

44.9 31.4

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TABLE 9 Comparison of 5-Year-Olds’ Index Scores Between the Two-Stage and One-Stage Groups

Two-stage group One-stage group

Sample Size

Mean

SD

95% Confidence Intervals

31 37

2.94 3.13

0.775 0.823

2.66–3.23 2.86–3.41

DISCUSSION Study Design Under the present conditions at Hokkaido University Hospital, CLP infants are seen at the OMS outpatient clinic or the PS outpatient clinic, depending on the referring pediatrician’s choice. The surgical protocols for the patients of both are not yet unified due to the difference in the way of thinking about palatal repair between the two departments. That is why this study could be designed as a nonrandomized clinical trial with concurrent controls. Techniques and timing of lip repair and operators in charge of presurgical orthopedics and lip repair were the same for both groups, but not for palatal repair. To equalize the background factors of both groups as much as possible for this comparative study, subjects were limited to patients with complete UCLP, Japanese ethnic background, and full-term infants; patients with Simonart’s bands and associated congenital anomalies were excluded. Nollet et al. (2005a) reported in a meta-analysis of published GOSLON outcomes that studies that included patients with Simonart’s bands would have better outcomes, and they recommended that such patients be removed from the analysis. Therefore, it could be thought that the two samples were sufficiently similar at the outset. As factors that have an influence on maxillary development other than primary interventions, sex differences and family inheritance of facial pattern, especially Class III malocclusion, which is particularly common in Japanese, must be noted. Many reports have identified sex differences in the maxillary and mandibular arch dimensions of children without clefts in the primary dentition (Meredith and Hopp, 1956; Sillman, 1964; Moorees et al., 1969; Knott, 1972), with all arch dimensions being larger in boys. However, it was impossible to examine each outcome by sex in the present study because the power was limited due to the small sample size; fortunately, the sex ratio was almost the same in the two groups. From the basic point of view that both the 5-Year-Olds’ Index and the Huddart/ Bodenham Index are measures of relative relationships through which the whole or parts of the maxilla and the mandible are evaluated, it is thought that the influence of sex differences could be almost ignored. It has been reported that cleft patients who had a family history of Class III may tend to develop Class III malocclusion not

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TABLE 10

Mean Scores of the Huddart/Bodenham Index of Each Part Two-Stage Group

Total Incisors Canine on major segment Molars on major segment Canine on minor segment Molars on minor segment

Mean*

SD

6.43 3.69 0.34 0.23 1.41 0.77

3.336 1.707 0.561 0.575 0.688 1.062

One-Stage Group

95% Confidence Intervals 7.65 4.31 0.55 0.44 1.66 1.16

to to to to to to

5.20 3.06 0.13 0.02 1.16 0.38

Mean*

SD

8.01 3.17 0.52 0.89 1.66 1.78

4.285 2.242 0.742 1.305 0.557 1.314

95% Confidence Intervals 9.44 3.92 0.77 1.32 1.84 2.22

to to to to to to

6.59 2.42 0.27 0.45 0.45 1.47

Significance P P P P P P

, . . , . ,

.05 .05 .05 .01 .05 .01

* Mean of four individual scores given for each model.

only as an effect of primary surgery but also due to the genetic influence of excessive mandibular growth (Kajii et al., 2013). In the present study, a family history of Class III could not be considered because it was impossible to confirm. However, genetic influences may have little effect because facial growth is not fully expressed until after the pubertal growth spurt (Flinn et al., 2006; Mars et al., 2006). Of the 81 consecutive patients who were initially enrolled in this study, 13 patients who moved away, dropped out, or had too many decayed teeth were not available for the outcome study. Thus, 68 patients remained as evaluation subjects, for a follow-up rate of more than 84%. Shaw et al. (1992b) provided estimates of the sample size required to detect significant differences for the dental arch relationship outcome using the GOSLON score, and they demonstrated that, for a two-group comparison based on 5% probability and 80% power, detection of 0.5 and 0.75 GOSLON point differences required sample sizes of 42 and 19, respectively. Given that there were 31 subjects in the two-stage group and 37 in the one-stage group, the sample size requirements determined in the Eurocleft study appear to have been satisfied. Methods for Assessment Two methods were used to assess the dental arch relationship outcomes in this study. The 5-Year-Olds’ Index is widely accepted as a simple and sensitive indicator of the early surgical outcome for the dental arch relationship. It assesses the dental arch relationship in terms of anteroposterior, transverse, and vertical discrepancies using dental casts and classifies treatment outcomes in UCLP patients according to difficulty in correcting the malocclusion. The GOSLON scores have previously been shown to correlate well with cephalometric outcomes related to the positions of the maxilla and mandible (Mars and Plint, 1985). This characteristic is good for the 5-Year-Olds’ Index scoring. It is notable that the extent of maxillary growth impediment could be extrapolated from the state of the dental arch relationships. Moreover, confounding variables such as orthodontics and secondary surgeries are not performed yet, and genetic influences are also less of a factor because

facial growth is not fully expressed until after the pubertal growth spurt. Unless the patient has a severe skeletal discrepancy, the outcome measured in 5-year casts could assess the effects of primary surgery (Flinn et al., 2006; Mars et al., 2006). However, the use of the 5Year-Olds’ Index requires professional judgment regarding the potential for orthodontic management of the interarch discrepancy, and reference casts and a calibration course are absolutely necessary for the use of this tool. When the 5-Year-Olds’ Index scoring was performed in this study, each rater was trained in this scoring procedure by attending a 5-Year-Olds’ Index calibrating course (Sandy and Atack, 2012), with calibration just before actual scoring to reduce systematic bias. Both intrarater and interrater agreement evaluated by the weighted kappa statistics was ‘‘good,’’ so that the repeatability and reproducibility of the rating were ensured. On the other hand, although the 5-Year-Olds’ Index has become a very useful tool that must now be included in multicenter comparisons of treatment outcomes, the details of discrepancies are not clear, because it is a general overview measure. A rating scale such as the Huddart/Bodenham Index would be effective to make up for the 5-Year-Olds’ Index when a more detailed description of treatment outcome, such as absolute measurement of arch constriction, would be required (Bartzela et al., 2011). Mossey et al. (2003) reported that the Huddart/ Bodenham score had a high correlation with occlusal outcome on the reference model for the 5-Year-Olds’ Index and GOSLON Yardstick and was more sensitive to interarch discrepancies, but the correlation according to each segmental division was not reported. Morris et TABLE 11 Correlation Coefficients Between the 5-Year-Olds’ Index Score and the Huddart/Bodenham Index Score for Every Part Spearman Rank Correlation Coefficient (q) Total Incisors Canine on major segment Molars on major segment Canine on minor segment Molars on minor segment * P , .01.

0.84* 0.81* 0.49* 0.47* 0.41* 0.41*

Mikoya et al., COMPARISON OF DENTAL ARCH RELATIONSHIP OUTCOMES

FIGURE 1 Comparison of the 5-Year-Olds’ Index score distributions of the one-stage and two-stage groups in this study and previous reports.

al. (1994) reported that overjet explained 87% of the variance of the GOSLON score. Suzuki et al. (2007) reported that, in Japanese cases, the 5-Year-Olds’ Index would be mainly represented by the smallness of the maxillary dental arch length and anterior crossbite, and it had some relationship to the collapse of the maxillary segment, but it would not be as strong. Examining the correlation between the two scoring systems in the present study, the total score and the score of the incisors of the Huddart/Bodenham Index were found to have significantly high correlations with the 5-YearOlds’ Index. On the other hand, the scores of the canines and the molars on the major and minor segments were lower. Transverse collapse of the maxillary segments seems to be poorly reflected by the 5-Year-Olds’ Index scoring. Analysis of Outcomes In the present study, the two groups only varied in the method of palatal repair, with primary interventions such as presurgical orthopedics and lip repair being the same in both groups. Each treatment was carried out by highly experienced surgeons in the same department. Although Lehner et al. (2003) reported that the experience and skills of the surgeon in terms of tissue management may have more effect on craniofacial development than surgical technique or timing of hard palate closure, these differences can be excluded in this study. In the present study, despite the differences in the palatal repair technique and timing (i.e., one- or twostage palatal repair), both protocols resulted in similar 5-Year-Olds’ Index mean scores. However, the distribution of the scores differed significantly. The rate of cases classified as Excellent, Good, and Fair was significantly higher in the two-stage group than in the one-stage group. For the Huddart/Bodenham Index, the mean scores of molars on both segments were significantly greater in the two-stage group than in the onestage group, but the mean scores of the incisors and the

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canines in both groups were similar. When the comparison results of the mean and the distribution of the 5-Year-Olds’ Index scores are interpreted from the correlations of the 5-Year-Olds’ Index and Huddart/ Bodenham Index, the anteroposterior relationship was not significantly different between the groups, but the transversal relationship in the deciduous molar parts was better in the two-stage group than in the one-stage group. Thus, it can be thought that the two-stage group was able to avoid maxillary collapse. This result was just what was predicted. It is widely accepted that scar tissue that develops on denuded bone might increase the risk of subsequent maxillary growth impediment (Ross, 1970; Kim et al., 2002; Meng et al., 2007). It has been particularly pointed out that the greater insult to the hard palate caused by the Veau and Wardill-Kilner pushback procedure brings about more transverse collapse (Sommerlad, 2009). Scar tissue developing at the denuded bone sites after the V to Y pushback transpositions of the mucoperiosteal palatal flaps redirects contracting forces from a transverse direction to a sagittal direction (Kremenak et al., 1970; Ross, 1970; Wijdeveld et al., 1987, 1991). On the other hand, it has been accepted that delayed hard palate closure results in better growth (Schweckendiek, 1978; Bardach et al., 1984) because possible interference with maxillary growth is postponed to a later age when less growth remains (Friede and Enemark, 2001), and as areas of periosteal undermining and residual exposed palatal shelf shrink, the incisor relationships improve (Pigott et al., 2002). Contrary to expectations, the anteroposterior dental arch relationship did not show a significant difference between the groups. Although many reports have discussed the superiority and inferiority of dental arch relationship outcomes of different timings and methods of hard palate closure, the effect of the method of processing the pterygoid area for elongation of the soft palate and preparation of muscle sling on maxillary growth outcomes is not clear. According to Ross (1970), some dissection of tissues in the area around the pterygomaxillary suture causes a continuum of scar tissue that joins the maxilla, the palatine bone, and the pterygoid plates, which inhibits subsequent separation of these bones and, thereby, creates a condition that could be termed maxillary ankylosis. In most cases of the two-stage group adopting Furlow’s procedure, velar lengthening was accomplished by large entire velar Zplasties. In order to complete a tension-free closure, dissections into the space of Ernst (pterygoid-superior constrictor space) and displacing the periosteum from the inside aspect of the medial pterygoid plates were performed in all cases. On the other hand, in the one-stage group, the lengthening of the nasal side of velum was performed by inserting a pedicled buccal mucosal flap in the nasal side of the velum according to Kaplan (1975), which might

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decrease the surgical invasion to the pterygoid area. The two-stage procedure might cause more extensive invasion around the area of the pterygoid in comparison with the one-stage procedure, so that the possibility of a difference in the anteroposterior dental arch relationships is thought to disappear. The logic that the twostage procedure for palatal repair causes fewer maxillary developmental disorders than the one-stage procedure may not be simple. Rohrich et al. (1996) reported that the long-term benefits of late palatal repair on facial growth remain unproven, and this aspect of cleft care remains controversial. In the Eurocleft study (Mars et al., 1992; Mølsted et al., 1992; Shaw et al., 1992a, 1992b), three of the centers that showed lower levels of growth inhibition used a variety of techniques with different timings of primary repair. Thus, even if methods of primary repair were widely different, a relatively good result could be achieved if an expert team performed the repair. Furthermore, Nollet et al. (2005b) reported that the treatment outcome of Nijmegen patients with UCLP treated with a two-stage palatal closure, with delayed closure of the hard palate, was comparable with that of the Eurocleft centers with the best outcomes. However, the treatment protocol used in the centers did not have a major effect on the dental arch relationship; caseload or skill of the surgeon might be more important factors for the quality of the results. In the distributions of the 5-Year-Olds’ Index scores of European centers compared to Japanese centers including our hospital, the proportion of cases classified as excellent (group 1) and good (group 2) was very high, and the proportion of cases classified as poor (group 4) and very poor (group 5) was very low. The tendency of the score distribution among Japanese centers or among European centers seems not to have as large a difference as between the group of Japanese centers and the group of European centers. It is hard to assume that this discrepancy was caused by a difference in rating ability and surgery. Thus, it would be reasonable to think that the difference in dental arch relationship outcomes between the Japanese and the European centers rather derives from differences in ethnic backgrounds. A high prevalence of Class III malocclusion without clefts in northeastern Asian populations has been reported (Chan, 1974; Baik et al., 2000; Ishii et al., 2002). Enlow (1990) stated that white groups, with a tendency for a dolichocephalic head form, have a corresponding tendency toward class II malocclusion and a retrognathic profile, whereas Japanese groups, with a tendency for a brachycephalic head form, have a corresponding tendency toward class III malocclusion and a prognathic profile. Ishii et al. (2002) found a less favorable facial pattern in Japanese populations for correction of Class III malocclusion than in British whites. A naturally occurring higher incidence of

underlying skeletal Class III relationship may lead to a poorer 5-Year-Olds’ Index scoring that would not necessarily be related to primary surgery. Additionally, it may be thought that the threshold to get a skeletal Class III relationship due to maxillary growth impediment is lower in Japanese than in whites. Susami et al. (2006) emphasized that further studies are needed to clarify the above-mentioned issue more widely in Japan and other Asian countries. Careful consideration of the 5-Year-Olds’ Index will be needed to compare the outcomes among different ethnic groups. CONCLUSIONS The purpose of this study was to compare the dental arch relationship outcomes in two cohorts following one-stage or two-stage palatal repair. Matching of subjects excluding the palatal repair procedure was carried out to improve the equivalence of the groups compared. Dental arch relationships were judged and categorized using the 5-Year-Olds’ Index and the Huddart/Bodenham Index. Intrarater and interrater reliabilities were good or better for cast ratings using both indices. The mean scores of the 5Year-Olds’ Index between the groups were not significantly different. The distribution of the 5-Year-Olds’ Index scores and the Huddart/Bodenham scores on both deciduous molar parts showed better results in the two-stage group than in the one-stage group. It is suggested that the anteroposterior intermaxillary relationship was not significantly different between the groups, but the transversal relationship was better in the two-stage group than in the one-stage group. REFERENCES Altman DG. Practical Statistics for Medical Research. London: Chapman and Hall; 1991:404–408. Atack N, Hathorn I, Mars M, Sandy J. Study models of 5 year old children as predictors of surgical outcome in unilateral cleft lip and palate. Eur J Orthod. 1997a;19:165–170. Atack NE, Hathorn I, Dowell T, Sandy J, Semb G, Leach A. Early detection of differences in surgical outcome for cleft lip and palate. Br J Orthod. 1998;25:181–185. Atack NE, Hathorn IS, Semb G, Dowell T, Sandy JR. A new index for assessing surgical outcome in unilateral cleft lip and palate subjects aged five: reproducibility and validity. Cleft Palate Craniofac J. 1997b;34:242–246. Baik HS, Han HK, Kim DJ, Proffit WR. Cephalometric characteristics of Korean class III surgical patients and their relationship to plans for surgical treatment. Int J Adult Orthod Orthognath Surg. 2000;15:119–128. Bardach J, Morris HL, Olin WH. Late results of primary veloplasty: the Marburg project. Plast Reconstr Surg. 1984;73:207–215. Bartzela T, Leenarts C, Bronkhorst E, Borstlap W, Katsaros C, Kuijpers-Jagtman AM. Comparison of two scoring systems for evaluation of treatment outcome in patients with complete bilateral cleft lip and palate. Cleft Palate Craniofac J. 2011;48:455–461. Bongaarts CAM, Kuijpers-Jagtman AM, van’t Hof MA, PrahlAndersen B. The effect of infant orthopedics on the occlusion of the

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