The Cleft Palate–Craniofacial Journal 51(6) pp. 658–664 November 2014 Ó Copyright 2014 American Cleft Palate–Craniofacial Association
ORIGINAL ARTICLE Effects of Lip Repair on Maxillofacial Morphology in Patients With Unilateral Cleft Lip With or Without Cleft Palate Qinghua Shao, M.D., Zhengxi Chen, M.D., Yang Yang, M.D., Zhenqi Chen, D.D.S., Ph.D. Objective: To evaluate the effects of lip repair on maxillofacial development of patients with unilateral cleft lip with or without cleft palate. Design: Retrospective. Patients: A total of 75 patients were recruited, including 38 surgical patients with complete unilateral cleft lip and alveolus and 37 patients with complete unilateral cleft lip and palate who had lip but not palate repair. As controls, 38 patients with no cleft were selected. All subjects were divided according to two growth stages: before the pubertal peak (GS1) and after the pubertal peak (GS2). Interventions: Lateral cephalograms of all subjects were obtained. Main Outcome Measures: Cephalograms were analyzed and compared in the study and control groups. Results: The patients with unilateral cleft lip and palate in both GS1 and GS2 demonstrated an almost normal maxillary and mandibular growth with retroclined maxillary incisors. The patients with unilateral cleft lip and palate showed a shorter length of maxilla, a more clockwise-rotated mandible, and retroclined maxillary incisors. Conclusions: There was an almost normal maxillary and mandibular growth but retroclined maxillary incisors in patients with cleft lip with or without cleft palate who had received lip repair only, indicating that lip repair may not have a negative impact on the maxillofacial development and influences only the inclination of the maxillary incisors. The shorter anterior-posterior maxillary length and larger gonial angle in patients with unilateral cleft lip and palate compared with those in patients with unilateral cleft lip and alveolus suggest that these variations in maxillary and mandibular growth may be a consequence of the cleft itself. KEY WORDS: cephalogram, lip repair, maxillofacial morphology
Midfacial deformity and maxillary retrusion is a common finding in many patients with repaired unilateral cleft lip and palate (UCLP). It has been attributed to three possible causes: intrinsic developmental deficiencies, functional distortions affecting the position growth of both normal and abnormal parts, and iatrogenic factors introduced by treatments (Ross, 1987). Numerous reports on individuals with UCLP who did not have surgery (Mestre, 1960; Atherton, 1967; Capelozza et al., 1993;
Shetye and Evans, 2006) supported the claim that surgical repair of the cleft lip and palate was responsible for most of the abnormal maxillofacial growth. The effect of palatoplasty on sagittal maxillary growth has been generally acknowledged (Chen, 2012), but whether lip repair inhibits growth remains controversial. Some researchers considered that lip repair may have an influence on the maxillary incisors and the alveolar bone but not on the development of the maxilla (Mars and Houston, 1990; Liao and Mars, 2005). In contrast, Bardach proposed that lip repair had a negative effect on maxillary growth in experimental studies with animals (Bardach and Eisbach, 1977; Bardach et al., 1979; Bardach and Mooney, 1984). Bardach (1990) reported that increased lip pressure following lip repair resulted in some tension on the basal maxilla, which is believed to adversely affect the anteroposterior maxillary growth. However, the results from the animal studies are inconclusive and fail to relate to the human condition (Liao and Mars, 2005). There are few studies that have evaluated the long-term effects of lip repair on maxillofacial morphology in patients with UCLP (Mars and Houston, 1990; Capelozza Filho et al., 1996; Liao and Mars, 2005). The main problem is that
Dr. Shao is Resident, Department of Orthodontics, Affiliated Hospital of Stomatology, Nanjing Medical University, Jiangsu, China. Dr. Zhengxi Chen is Resident; Dr. Yang is Resident; and Dr. Zhenqi Chen is Professor, Department of Orthodontics, Ninth People’s Hospital, School of Stomatology, Shanghai Key Laboratory of Stomatology, Shanghai Jiao Tong University, Shanghai, China. Submitted January 2013; Revised August 2013; Accepted November 2013. Address correspondence to: Dr. Zhen-Qi Chen, Department of Orthodontics, Ninth People’s Hospital, School of Stomatology, Shanghai Key Laboratory of Stomatology, Shanghai Jiao Tong University, 639 Zhizaoju Road, Shanghai 200011, China. E-mail [email protected]. DOI: 10.1597/12-316 658
Shao et al., EFFECTS OF LIP REPAIR ON MAXILLOFACIAL MORPHOLOGY
FIGURE 1
Developmental stages of cervical vertebrae.
lip and palate repairs are routinely performed long before the growth of the facial complex ceases. The observed effect on maxillofacial morphology, therefore, is attributed to at least cleft lip and palate repairs together (Liao and Mars, 2005). Evaluating the maxillofacial morphology of patients with cleft lip and palate who have undergone lip repair only may provide an important tool for understanding the longterm, isolated effects of lip repair. Therefore, the purpose of this study was to describe the maxillofacial morphology of patients with unilateral cleft lip with or without palate who had received only lip surgery at different growth stages and to evaluate the effects of lip repair on their maxillofacial development. MATERIALS
AND
METHODS
Patient Groups A total of 113 patients with an eastern China ethnic background were included in this study. The study group was composed of 38 patients (23 males and 15 females) with complete unilateral cleft lip and alveolus but without cleft palate (UCLA) and 37 patients (32 males and five females) with complete unilateral cleft lip and palate (UCLP). Selection was determined according to the following criteria: (1) no congenital anomaly or any known syndrome; (2) no presurgical orthopedics; (3) one lip repair performed by the surgeons of the same team using the Millard surgical modality prior to the second year of age at Shanghai Jiao Tong University Cleft Center; (4) no palate repair; and (5) no orthodonTABLE 1
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tic treatment or other craniofacial surgery prior to cephalometric examination. The control group consisted of 38 patients (22 males and 16 females) with no cleft. None of them had ever undergone any orthodontic treatment or any craniofacial surgery. The principles outlined in the Declaration of Helsinki were followed. All of the subjects were grouped by stage of cervical vertebral maturation (CVMS), which was evaluated using the Baccetti method (Baccetti et al., 2002). This method depends on the morphology of three cervical vertebrae (C2, C3, and C4), which were subdivided into five consecutive stages of cervical maturation (Fig. 1). According to this method, CVMS I and CVMS II occur before the pubertal growth peak, and deceleration in skeletal growth occurs during the postpubertal intervals, from CVMS III through CVMS V. Thus, the two study groups and the control group were divided according to two growth stages: before the pubertal peak (GS1) and after the pubertal peak (GS2) (Table 1). Cephalometric Analysis Lateral cephalograms of all subjects were obtained with a Morita XH550 (J. Morita Mfg. Corp., Kyoto, Japan) according to standardized cephalometric guidelines, with natural head position and with teeth in centric occlusion. The images were traced with the Nemotec Dental Studio NX 2006 software (Nemotec S.L., Madrid, Spain) by one examiner. The cephalometric parameters were measured to indicate the skeletal
Distribution of Subjects According to Cleft Groups and Growth Stages* Before the Pubertal Peak (GS1)
UCLA group UCLP group Control group
After the Pubertal Peak (GS2)
Total
CVMS I
CVMS II
Total
CVMS III
CVMS IV
CVMS V
18 17 16
11 11 10
7 6 6
20 20 22
4 5 6
10 9 10
6 6 6
* GS1 ¼ before the pubertal peak; GS2 ¼ after the pubertal peak; CVMS ¼ cervical vertebral maturation stage; UCLA ¼ unilateral cleft lip and alveolus; UCLP ¼ unilateral cleft lip and palate.
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were not statistically significant with less than 0.98 and 1 mm, respectively. Statistical Analysis Statistical analysis was done using the Predictive Analytics Software Statistics Base 18.0 for Windows (2009; SPSS Inc., Chicago, IL). The mean and standard deviation were calculated for every parameter measured in each group. Multivariate analysis of variance was used to identify the differences in cephalometric parameters between the study and control groups. Furthermore, Duncan’s multiple comparison analysis was applied to indicate any significant differences in parameters between any two of the subgroups if the error variance of the dependent variable was equal across groups. If not, the Kruskal-Wallis test was applied. RESULTS
FIGURE 2 Landmarks and reference lines used on a lateral cephalogram. A ¼ A point; ANS ¼ anterior nasal spine; Ar ¼ articulare; B ¼ B point; Co ¼ condylion; FH ¼ Frankfort horizontal plane, a line through P and Or; Go ¼ gonion; LIA ¼ lower incisal apex; LIE ¼ lower incisal edge; Me ¼ menton; MP ¼ mandibular plane, a line from Me tangential to the postero-inferior border of the mandible; N ¼ nasion; Or ¼ orbitale; P ¼ porion; PMP ¼ posterior maxillary point, a construct created by dropping perpendicularly to the line from ANS passing through posterior hard palate from PTM; Pog ¼ pogonion; PP ¼ palatal plane, a line through ANS and PMP; PTM ¼ pterygomaxillary fissure; S ¼ estimated center of the hypophyseal fossa; SN ¼ anterior cranial base, a line through S and N; UIA ¼ upper incisal apex; UIE ¼ upper incisal edge.
changes in the maxillofacial regions. Figure 2 shows the landmarks and the reference lines that were used in the cephalometric analysis. The definitions of the parameters used are briefly described in Table 2. Considering normal variations in craniofacial size among patients, all of the linear parameters are expressed as a ratio compared to anterior cranial base length (S-N).
Cephalometric parameters were respectively examined to indicate the growth patterns of the maxillary midface and of the mandible in the groups in GS1 (Table 3) and GS2 (Table 4). Figures 3 and 4 depict the superimposition of cephalogram tracings of both cleft and control groups at each stage. In both GS1 and GS2, the UCLA and UCLP groups demonstrated a similar position of maxilla (SNA, SNANS, SN-PP) with the control. The maxillary length (ANSPMP/S-N) of the UCLP group was significantly shorter than that of the others. There were no significant differences in mandibular position (SNB, SN-Pog, SN-MP), shape (Ar-Go-Me), or size (Ar-Go/S-N, Go-Me/S-N, Co-Me/SN) among the three groups, except that the UCLP group exhibited a significantly larger gonial angle. Regarding the sagittal relationship between the maxilla and the mandible (ANB, convexity), no significant differences existed between any two of the groups, except that the UCLA group in GS2 showed a statistically increased ANB and convexity. The maxillary incisors (SN-U1) of both the UCLA and UCLP groups were statistically significantly more retroclined when compared with that of the control; although, there was no significant difference between the two experimental groups.
Error of Method DISCUSSION To determine the error in parameters, 10 radiographs were randomly selected and reassessed after at least a 1week interval by the same examiner. Method errors (MEs) were calculated using ffi the formula proposed by qX ffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffi Dahlberg ðME ¼ d2 =2nÞ (Houston, 1983), where P 2 d is the sum of the squared differences between the two mean values, and n is the number of double parameters. The MEs for angular and linear parameters
Repair of the unilateral cleft lip is one of the most dramatic surgical procedures that can improve the patients’ appearance to a great extent. However, controversy still remains as to whether lip repair inhibits maxillofacial growth (Bardach, 1990; Mars and Houston, 1990; Liao and Mars, 2005). Though several clinical arguments have been put forward to evaluate the effects of lip repair on maxillofacial morphology, they were all based on research
Shao et al., EFFECTS OF LIP REPAIR ON MAXILLOFACIAL MORPHOLOGY
TABLE 2
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Definitions of the Parameters
Parameters
Definitions
Cranial base length S-N, mm
Distance between S and N point
Maxillary skeletal SNA, 8 SN-ANS, 8 SN-PP, 8 ANS-PMP, mm ANS-PMP /S-N, %
Angle from S to N to A point Angle from S to N to ANS point Angle between the anterior cranial base (SN) and the palatal plane(ANS-PMP) Distance between the projections of point ANS and point PMP onto the frankfort horizontal plane (ANS-PMP / S-N) 3 100
Mandibular skeletal SNB, 8 SN-Pog, 8 SN-MP, 8 Ar-Go-Me, 8 Ar-Go, mm Ar-Go/S-N, % Go-Me, mm Go-Me/S-N, % Co-Me, mm Co-Me/S-N, %
Angle from S to N to B point Angle from S to N to Pog point Angle between the anterior cranial base (SN) and the mandibular plane Angle from Ar to Go to Me point Distance between Ar and Go point (Ar-Go / S-N) 3 100 Distance between Go and Me point (Go-Me / S-N) 3 100 Distance between Co and Me point (Co-Me / S-N) 3 100
Jaw relationship ANB, 8 Convexity, 8
Angle from A point to N to B point The intersection of line N-point A and point A-pogonion
Denture SN-U1, 8 L1-MP, 8
Angle between the anterior cranial base (SN) and the maxillary central incisor axis line Angle between the mandibular central incisor axis line and the mandibular plane
subjects with UCLP who had received lip repair only or both lip and palate surgery (Mars and Houston, 1990; Capelozza Filho et al., 1996; Liao and Mars, 2005). The observed effect must be attributed to several causes together, such as lip surgery, palate surgery, and even an intrinsic defect caused by the cleft of the palate. Therefore, in this study, patients with UCLA who had no cleft palate and had received lip repair only were selected to exclude TABLE 3
other influencing factors. Some authors reported that a comparison of various cleft subtypes showed different degrees of midfacial deficiency (Cronin and Hunter, 1980; Smahel and Brejcha, 1983; Chiu et al., 2011): mild in subjects with cleft lip and alveolus but severe in those with cleft lip and palate. Even so, the timing and technique of their lip repairs had no difference. Therefore, comparing maxillofacial morphology of patients with UCLA or
Cephalometric Parameters for 3 Groups in GS1 (Before the Pubertal Peak)
Parameters
UCLA Group Mean 6 SD
UCLP Group Mean 6 SD
Control Group Mean 6 SD
Maxillary skeletal SNA SN-ANS SN-PP ANS-PMP /S-N
81.6 83.9 8.9 71.6
6 6 6 6
3.3 3.2 3.6 3.1
80.1 83.4 10.4 67.4
6 6 6 6
3.0 3.1 5.1 4.4
80.4 84.3 8.2 71.3
6 6 6 6
Mandibular skeletal SNB SN-Pog SN-MP Ar-Go-Me Ar-Go/S-N Go-Me/S-N Co-Me/S-N
76.7 76.9 36.1 122.9 67.1 102.0 157.2
6 6 6 6 6 6 6
3.6 3.7 4.1 6.4 5.8 6.6 9.3
76.7 77.1 36.4 129.4 62.9 98.0 153.2
6 6 6 6 6 6 6
3.7 3.7 4.7 5.7 5.5 9.4 11.5
77.1 77.1 34.8 120.6 66.7 102.1 155.5
6 6 6 6 6 6 6
Jaw relationship ANB Convexity Denture SN-U1 L1-MP
4.8 6 2.5 9.8 6 5.5
3.4 6 3.3 6.5 6 6.9
93.4 6 8.1 92.3 6 6.9
86.9 6 11.6 89.0 6 5.9
P Value
Multiple Comparison†
4.4 4.9 2.1 4.9
.595 .726 .432 .024*
2,(3,1)
3.2 3.3 4.3 6.8 5.0 7.9 9.1
.786 .802 .538 .002* .094 .489 .658
3.3 6 1.8 7.2 6 3.8 104.8 6 5.5 93.4 6 4.6
(3,1),2
.090 .288 .000* .120
(2,1),3
† In the column of multiple comparison, 1, 2, and 3 mean UCLA (unilateral cleft lip and alveolus) group, UCLP (unilateral cleft lip and palate) group, and control group, respectively. * Significant differences (P .050) are presented in boldface.
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TABLE 4
Cephalometric Parameters for 3 Groups in GS2 (After the Pubertal Peak)
Parameters
UCLA Group Mean 6 SD
UCLP Group Mean 6 SD
Control Group Mean 6 SD
P Value
Multiple Comparison†
Maxillary skeletal SNA SN-ANS SN-PP ANS-PMP /S-N
83.2 87.1 10.0 75.1
6 6 6 6
3.7 3.7 4.5 4.3
79.4 82.8 8.7 68.8
6 6 6 6
5.0 5.5 3.9 6.6
82.4 86.6 8.2 73.4
6 6 6 6
2.5 2.0 3.2 4.1
.112 .056 .295 .006*
Mandibular skeletal SNB SN-Pog SN-MP Ar-Go-Me Ar-Go/S-N Go-Me/S-N Co-Me/S-N
78.7 79.2 33.8 119.3 74.8 106.5 166.9
6 6 6 6 6 6 6
3.7 3.7 4.7 6.4 9.1 8.1 10.0
77.9 78.6 34.5 123.0 74.0 106.7 167.3
6 6 6 6 6 6 6
4.0 4.3 8.5 8.6 8.0 6.5 7.8
79.8 80.5 32.5 116.2 79.6 110.6 171.3
6 6 6 6 6 6 6
2.9 2.8 3.8 6.4 6.6 5.2 9.3
.235 .237 .694 .016* .073 .061 .192
(3,1),(1,2)
2.6 6 1.2 4.1 6 2.6
.004* .005*
(2,3),1 (2,3),1
105.4 6 5.3 92.5 6 5.4
.001* .404
(1,2),3
Jaw relationship ANB Convexity Denture SN-U1 L1-MP
4.5 6 1.8 8.6 6 4.6 96.2 6 7.4 93.9 6 6.7
1.6 6 4.7 1.7 6 11.0 99.3 6 9.3 91.4 6 6.7
2,(3,1)
† In the column of multiple comparison, 1, 2, and 3 mean UCLA (unilateral cleft lip and alveolus) group, UCLP (unilateral cleft lip and palate) group, and control group, respectively. * Significant differences (P .050) are presented in boldface.
UCLP who have just undergone lip surgery with that of a person with no cleft can allow for evaluation of the role that lip repair plays in maxillofacial growth. The maxillofacial malformation of patients with cleft lip with or without cleft palate is closely related to their craniofacial skeletal growth. In previous studies, the midfacial morphology has been described according different chronological ages (Smahel et al., 1994; Han et al., 1995) or dental ages (Chen et al., 2012). But these indicators are not reliable predictors of an individual’s stage of skeletal development (Lewis, 1991; Flores-Mir et al., 2004). Cervical vertebral maturation is a method to evaluate the skeletal maturity on lateral cephalograms that has proven to be effective in assessing adolescent growth both in body height and mandibular size (Franchi et al., 2000; Baccetti et al., 2002). Consequently, considering the interaction of craniofacial morphology and skeletal growth, all the subjects have been grouped by CVMS using the Baccetti method (Baccetti et al., 2002). The results of this study showed equal length and protrusion of the maxilla in the UCLA groups and the controls, which supports the view that lip repair had no effect on the anteroposterior maxillary growth (Mars and Houston, 1990; Han et al., 1995; Liao and Mars, 2005). Seo (Seo et al., 2011), who investigated the growth pattern of patients with different types of cleft, found that surgical patients with UCLP and cleft palate had a more retruded maxilla than normally developed persons; whereas, those with UCLA demonstrated a nearly normal maxillary position. The authors then concluded that palatal scarring impaired maxillary growth more than lip scarring. In our study, cephalometric parameters of the UCLP group showed an almost normal maxillary sagittal position but
an obviously short anteroposterior maxilla length. It appears that there is a potential for normal maxillary growth in patients with UCLP who received lip surgery only, but the intrinsic defect caused by the cleft of the palate
FIGURE 3 Comparison of the cleft and control groups by superimposed cephalogram tracings (S-N at S) in GS1.
Shao et al., EFFECTS OF LIP REPAIR ON MAXILLOFACIAL MORPHOLOGY
FIGURE 4 Comparison of the cleft and control groups by superimposed cephalogram tracings (S-N at S) in GS2.
may have certain effects on sagittal maxillary growth. Contrary to our study, some researchers favor the view that major disturbance in maxillary growth was attributable to lip repair and studied patients with UCLP who had either only the lip operated upon or the lip as well as the palate repaired (Capelozza Filho et al., 1996; Kapucu et al., 1996; Li et al., 2006). But these studies ignored the intrinsic developmental deficiencies of these patients caused by the cleft palate and had certain limitations such as small sample size and overly broad age range. In the present study, no difference in mandibular position or morphology (SNB, SN-Pog, SN-MP, Ar-Go-Me, ArGo, Go-Me, Co-Me) was found among the UCLA and the control group. This is in accordance with previous studies (Bishara et al., 1985; Fudalej, 2007; Seo et al., 2011) and supports the claim that cleft mandibular growth is not related to lip repair (da Silva Junior et al., 1992; Capelozza Filho et al., 1996; Liao and Mars, 2005). We also found that the patients with UCLP had a normal mandibular position and length but a larger gonial angle. The results are similar to those of previous studies that observed a more clockwiserotated mandible in patients with UCLP who did not have surgical repair (Bishara et al., 1986; Mars and Houston, 1990; Capelozza Junior et al., 1993). These findings imply that shape and size of the mandible might be influenced by
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cleft type and support the hypothesis that variations in mandibular growth may be a consequence of the cleft itself (da Silva Filho et al., 1993). In the sagittal relationship between the maxilla and the mandible, the study groups were equal to the controls, except the UCLA group in GS2 had a larger ANB and convexity. This phenomenon may be cogenerated by the slightly protruding maxilla and receding mandible. Bishara et al. (1976), who conducted cephalometric measurements for patients with UCLA who did not have surgical repair, also found larger SNA, ANB, and convexity compared with persons without clefts. Therefore, it is reasonable to suggest that lip repair does not change the sagittal relationship between the maxilla and the mandible. But why the patients with UCLA showed this growth pattern cannot be well explained, and further studies are needed to clarify this notion. The surgical groups (UCLA and UCLP) showed retroclined maxillary incisors when compared with the controls. This is consistent with the cephalometric findings of Hagerty and Hill (1963) and Mars and Houston (1990). Liao and Mars (2005) thought that the tension of the repaired lip produced a bone-bending effect on the anterior maxillary alveolus, accompanied by controlled retroclination of maxillary incisors. There are some disagreements about the position of the mandibular incisors. Some authors reported that patients with lip repair demonstrated more retroclined mandibular incisors, which compensated for the retroclined maxillary incisors (Capelozza Filho et al., 1996; Lisson et al., 2004). In our study, no significant difference on these teeth was observed among the groups, which corroborates the findings of Mars and Houston (1990) and Liao and Mars (2005). CONCLUSION Based on elaborated assessment on cephalometric data, we found there was an almost normal maxillary and mandibular growth but retroclined maxillary incisors in patients with cleft lip with or without cleft palate who received lip repair only, indicating that lip repair may not have a negative impact on the maxillofacial development and influence only the inclination of maxillary incisors. The shorter anteroposterior maxillary length and larger gonial angle in patients with UCLP who have not had palate repair compared with that in patients with UCLA suggest that these variations in maxillary and mandibular growth may be a consequence of the cleft itself but not surgical repair. REFERENCES Atherton JD. Morphology of facial bones in skulls with unoperated unilateral cleft palate. Cleft Palate J. 1967;4:18–30. Baccetti T, Franchi L, McNamara JA Jr. An improved version of the cervical vertebral maturation (CVM) method for the assessment of mandibular growth. Angle Orthod. 2002;72:316–323.
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Bardach J. The influence of cleft lip repair on facial growth. Cleft Palate J. 1990;27:76–78. Bardach J, Eisbach KJ. The influence of primary unilateral cleft lip repair on facial growth. Cleft Palate J. 1977;14:88–97. Bardach J, Klausner EC, Eisbach KJ. The relationship between lip pressure and facial growth after cleft lip repair: an experimental study. Cleft Palate J. 1979;16:137–146. Bardach J, Mooney MP. The relationship between lip pressure following lip repair and craniofacial growth: an experimental study in beagles. Plast Reconstr Surg. 1984;73:544–555. Bishara SE, de Arrendondo RS, Vales HP, Jakobsen JR. Dentofacial relationships in persons with unoperated clefts: comparisons between three cleft types. Am J Orthod. 1985;87:481–507. Bishara SE, Jakobsen JR, Krause JC, Sosa-Martinez R. Cephalometric comparisons of individuals from India and Mexico with unoperated cleft lip and palate. Cleft Palate J. 1986;23:116–125. Bishara SE, Krause CJ, Olin WH, Weston D, Ness JV, Felling C. Facial and dental relationships of individuals with unoperated clefts of the lip and/or palate. Cleft Palate J. 1976;13:238–252. Capelozza Filho L, Normando AD, da Silva Filho OG. Isolated influences of lip and palate surgery on facial growth: comparison of operated and unoperated male adults with UCLP. Cleft Palate Craniofac J. 1996;33:51–56. Capelozza Junior L, Taniguchi SM, da Silva Junior ´ OG. Craniofacial morphology of adult unoperated complete unilateral cleft lip and palate patients. Cleft Palate Craniofac J. 1993;30:376–381. Chen ZQ, Wu J, Chen RJ. Sagittal maxillary growth pattern in unilateral cleft lip and palate patients with unrepaired cleft palate. J Craniofac Surg. 2012;23:491–493. 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. Cronin DG, Hunter WS. Craniofacial morphology in twins discordant for cleft lip and/or palate. Cleft Palate J. 1980;17:116–126. da Silva Filho OG, Normando AD, Capelozza Junior L. Mandibular growth in patients with cleft lip and/or cleft palate—the influence of cleft type. Am J Orthod Dentofacial Orthop. 1993;104:269–275. da Silva Junior OG, Normando AD, Capelozza Junior ´ L. Mandibular morphology and spatial position in patients with clefts: intrinsic or iatrogenic? Cleft Palate Craniofac J. 1992;29:369–375. Flores-Mir C, Nebbe B, Major PW. Use of skeletal maturation based on hand-wrist radiographic analysis as a predictor of facial growth: a systematic review. Angle Orthod. 2004;74:118–124. Franchi L, Baccetti T, McNamara JA Jr. Mandibular growth as related to cervical vertebral maturation and body height. Am J Orthod Dentofacial Orthop. 2000;118:335–340.
Fudalej P, Obłoj B, Miller-Drabikowska D, Samarcew-Krawczak A, Dudkiewicz Z. Cephalometric evaluation of craniofacial morphology in preadolescent children with a repaired unilateral cleft of the primary palate [abstract]. Med Wieku Rozwoj. 2007;11:247–253. Hagerty RF, Hill MJ. Facial growth and dentition in the unoperated cleft palate. J Dent Res. 1963;42:412–421. Han BJ, Suzuki A, Tashiro H. Longitudinal study of craniofacial growth in subjects with cleft lip and palate: from cheiloplasty to 8 years of age. Cleft Palate Craniofac J. 1995;32:156–166. Houston WJ. The analysis of errors in orthodontic measurements. Am J Orthod. 1983;83:382–390. Kapucu MR, Gursu KG, Enacar A, Aras S. The effect of cleft lip ¨ repair on maxillary morphology in patients with unilateral complete cleft lip and palate. Plast Reconstr Surg. 1996;97:1371–1375, discussion 1376–1378. Lewis AB. Comparisons between dental and skeletal ages. Angle Orthod. 1991;61:87–92. Li Y, Shi B, Song QG, Zuo H, Zheng Q. Effects of lip repair on maxillary growth and facial soft tissue development in patients with a complete unilateral cleft of lip, alveolus and palate. J Craniomaxillofac Surg. 2006;34:355–361. Liao YF, Mars M. Long-term effects of lip repair on dentofacial morphology in patients with unilateral cleft lip and palate. Cleft Palate Craniofac J. 2005;42:526–532. Lisson JA, Hanke I, Trankmann J. Vertical changes in patients with ¨ complete unilateral and bilateral cleft lip, alveolus and palate. J Orofac Orthop. 2004;65:246–258. Mars M, Houston WJ. A preliminary study of facial growth and morphology in unoperated male unilateral cleft lip and palate subjects over 13 years of age. Cleft Palate J. 1990;27:7–10. Mestre JC, Subtelny JD. Unoperated oral clefts at maturation. Angle Orthod. 1960;30:78–85. Ross RB. Treatment variables affecting facial growth in complete unilateral cleft lip and palate. Part 1: treatment affecting growth. Cleft Palate J. 1987;24:5–23. Seo YJ, Park JW, Kim YH, Baek SH. Initial growth pattern of children with cleft before alveolar bone graft stage according to cleft type. Angle Orthod. 2011;81:1103–1110. Shetye PR, Evans CA. Midfacial morphology in adult unoperated complete unilateral cleft lip and palate patients. Angle Orthod. 2006;76:810–816. Smahel Z, Brejcha M. Differences in craniofacial morphology between complete and incomplete unilateral cleft lip and palate in adults. Cleft Palate J. 1983;20:113–127. ´ Z, Skvarilova´ B, Stra´nska´ P. Development of Smahel Z, Mullerova ¨ overjet and dentoskeletal relations in unilateral cleft lip and palate before and during puberty. Cleft Palate Craniofac J. 1994;31:24–30.
ORIGINAL ARTICLE Effects of Lip Repair on Maxillofacial Morphology in Patients With Unilateral Cleft Lip With or Without Cleft Palate Qinghua Shao, M.D., Zhengxi Chen, M.D., Yang Yang, M.D., Zhenqi Chen, D.D.S., Ph.D. Objective: To evaluate the effects of lip repair on maxillofacial development of patients with unilateral cleft lip with or without cleft palate. Design: Retrospective. Patients: A total of 75 patients were recruited, including 38 surgical patients with complete unilateral cleft lip and alveolus and 37 patients with complete unilateral cleft lip and palate who had lip but not palate repair. As controls, 38 patients with no cleft were selected. All subjects were divided according to two growth stages: before the pubertal peak (GS1) and after the pubertal peak (GS2). Interventions: Lateral cephalograms of all subjects were obtained. Main Outcome Measures: Cephalograms were analyzed and compared in the study and control groups. Results: The patients with unilateral cleft lip and palate in both GS1 and GS2 demonstrated an almost normal maxillary and mandibular growth with retroclined maxillary incisors. The patients with unilateral cleft lip and palate showed a shorter length of maxilla, a more clockwise-rotated mandible, and retroclined maxillary incisors. Conclusions: There was an almost normal maxillary and mandibular growth but retroclined maxillary incisors in patients with cleft lip with or without cleft palate who had received lip repair only, indicating that lip repair may not have a negative impact on the maxillofacial development and influences only the inclination of the maxillary incisors. The shorter anterior-posterior maxillary length and larger gonial angle in patients with unilateral cleft lip and palate compared with those in patients with unilateral cleft lip and alveolus suggest that these variations in maxillary and mandibular growth may be a consequence of the cleft itself. KEY WORDS: cephalogram, lip repair, maxillofacial morphology
Midfacial deformity and maxillary retrusion is a common finding in many patients with repaired unilateral cleft lip and palate (UCLP). It has been attributed to three possible causes: intrinsic developmental deficiencies, functional distortions affecting the position growth of both normal and abnormal parts, and iatrogenic factors introduced by treatments (Ross, 1987). Numerous reports on individuals with UCLP who did not have surgery (Mestre, 1960; Atherton, 1967; Capelozza et al., 1993;
Shetye and Evans, 2006) supported the claim that surgical repair of the cleft lip and palate was responsible for most of the abnormal maxillofacial growth. The effect of palatoplasty on sagittal maxillary growth has been generally acknowledged (Chen, 2012), but whether lip repair inhibits growth remains controversial. Some researchers considered that lip repair may have an influence on the maxillary incisors and the alveolar bone but not on the development of the maxilla (Mars and Houston, 1990; Liao and Mars, 2005). In contrast, Bardach proposed that lip repair had a negative effect on maxillary growth in experimental studies with animals (Bardach and Eisbach, 1977; Bardach et al., 1979; Bardach and Mooney, 1984). Bardach (1990) reported that increased lip pressure following lip repair resulted in some tension on the basal maxilla, which is believed to adversely affect the anteroposterior maxillary growth. However, the results from the animal studies are inconclusive and fail to relate to the human condition (Liao and Mars, 2005). There are few studies that have evaluated the long-term effects of lip repair on maxillofacial morphology in patients with UCLP (Mars and Houston, 1990; Capelozza Filho et al., 1996; Liao and Mars, 2005). The main problem is that
Dr. Shao is Resident, Department of Orthodontics, Affiliated Hospital of Stomatology, Nanjing Medical University, Jiangsu, China. Dr. Zhengxi Chen is Resident; Dr. Yang is Resident; and Dr. Zhenqi Chen is Professor, Department of Orthodontics, Ninth People’s Hospital, School of Stomatology, Shanghai Key Laboratory of Stomatology, Shanghai Jiao Tong University, Shanghai, China. Submitted January 2013; Revised August 2013; Accepted November 2013. Address correspondence to: Dr. Zhen-Qi Chen, Department of Orthodontics, Ninth People’s Hospital, School of Stomatology, Shanghai Key Laboratory of Stomatology, Shanghai Jiao Tong University, 639 Zhizaoju Road, Shanghai 200011, China. E-mail [email protected]. DOI: 10.1597/12-316 658
Shao et al., EFFECTS OF LIP REPAIR ON MAXILLOFACIAL MORPHOLOGY
FIGURE 1
Developmental stages of cervical vertebrae.
lip and palate repairs are routinely performed long before the growth of the facial complex ceases. The observed effect on maxillofacial morphology, therefore, is attributed to at least cleft lip and palate repairs together (Liao and Mars, 2005). Evaluating the maxillofacial morphology of patients with cleft lip and palate who have undergone lip repair only may provide an important tool for understanding the longterm, isolated effects of lip repair. Therefore, the purpose of this study was to describe the maxillofacial morphology of patients with unilateral cleft lip with or without palate who had received only lip surgery at different growth stages and to evaluate the effects of lip repair on their maxillofacial development. MATERIALS
AND
METHODS
Patient Groups A total of 113 patients with an eastern China ethnic background were included in this study. The study group was composed of 38 patients (23 males and 15 females) with complete unilateral cleft lip and alveolus but without cleft palate (UCLA) and 37 patients (32 males and five females) with complete unilateral cleft lip and palate (UCLP). Selection was determined according to the following criteria: (1) no congenital anomaly or any known syndrome; (2) no presurgical orthopedics; (3) one lip repair performed by the surgeons of the same team using the Millard surgical modality prior to the second year of age at Shanghai Jiao Tong University Cleft Center; (4) no palate repair; and (5) no orthodonTABLE 1
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tic treatment or other craniofacial surgery prior to cephalometric examination. The control group consisted of 38 patients (22 males and 16 females) with no cleft. None of them had ever undergone any orthodontic treatment or any craniofacial surgery. The principles outlined in the Declaration of Helsinki were followed. All of the subjects were grouped by stage of cervical vertebral maturation (CVMS), which was evaluated using the Baccetti method (Baccetti et al., 2002). This method depends on the morphology of three cervical vertebrae (C2, C3, and C4), which were subdivided into five consecutive stages of cervical maturation (Fig. 1). According to this method, CVMS I and CVMS II occur before the pubertal growth peak, and deceleration in skeletal growth occurs during the postpubertal intervals, from CVMS III through CVMS V. Thus, the two study groups and the control group were divided according to two growth stages: before the pubertal peak (GS1) and after the pubertal peak (GS2) (Table 1). Cephalometric Analysis Lateral cephalograms of all subjects were obtained with a Morita XH550 (J. Morita Mfg. Corp., Kyoto, Japan) according to standardized cephalometric guidelines, with natural head position and with teeth in centric occlusion. The images were traced with the Nemotec Dental Studio NX 2006 software (Nemotec S.L., Madrid, Spain) by one examiner. The cephalometric parameters were measured to indicate the skeletal
Distribution of Subjects According to Cleft Groups and Growth Stages* Before the Pubertal Peak (GS1)
UCLA group UCLP group Control group
After the Pubertal Peak (GS2)
Total
CVMS I
CVMS II
Total
CVMS III
CVMS IV
CVMS V
18 17 16
11 11 10
7 6 6
20 20 22
4 5 6
10 9 10
6 6 6
* GS1 ¼ before the pubertal peak; GS2 ¼ after the pubertal peak; CVMS ¼ cervical vertebral maturation stage; UCLA ¼ unilateral cleft lip and alveolus; UCLP ¼ unilateral cleft lip and palate.
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were not statistically significant with less than 0.98 and 1 mm, respectively. Statistical Analysis Statistical analysis was done using the Predictive Analytics Software Statistics Base 18.0 for Windows (2009; SPSS Inc., Chicago, IL). The mean and standard deviation were calculated for every parameter measured in each group. Multivariate analysis of variance was used to identify the differences in cephalometric parameters between the study and control groups. Furthermore, Duncan’s multiple comparison analysis was applied to indicate any significant differences in parameters between any two of the subgroups if the error variance of the dependent variable was equal across groups. If not, the Kruskal-Wallis test was applied. RESULTS
FIGURE 2 Landmarks and reference lines used on a lateral cephalogram. A ¼ A point; ANS ¼ anterior nasal spine; Ar ¼ articulare; B ¼ B point; Co ¼ condylion; FH ¼ Frankfort horizontal plane, a line through P and Or; Go ¼ gonion; LIA ¼ lower incisal apex; LIE ¼ lower incisal edge; Me ¼ menton; MP ¼ mandibular plane, a line from Me tangential to the postero-inferior border of the mandible; N ¼ nasion; Or ¼ orbitale; P ¼ porion; PMP ¼ posterior maxillary point, a construct created by dropping perpendicularly to the line from ANS passing through posterior hard palate from PTM; Pog ¼ pogonion; PP ¼ palatal plane, a line through ANS and PMP; PTM ¼ pterygomaxillary fissure; S ¼ estimated center of the hypophyseal fossa; SN ¼ anterior cranial base, a line through S and N; UIA ¼ upper incisal apex; UIE ¼ upper incisal edge.
changes in the maxillofacial regions. Figure 2 shows the landmarks and the reference lines that were used in the cephalometric analysis. The definitions of the parameters used are briefly described in Table 2. Considering normal variations in craniofacial size among patients, all of the linear parameters are expressed as a ratio compared to anterior cranial base length (S-N).
Cephalometric parameters were respectively examined to indicate the growth patterns of the maxillary midface and of the mandible in the groups in GS1 (Table 3) and GS2 (Table 4). Figures 3 and 4 depict the superimposition of cephalogram tracings of both cleft and control groups at each stage. In both GS1 and GS2, the UCLA and UCLP groups demonstrated a similar position of maxilla (SNA, SNANS, SN-PP) with the control. The maxillary length (ANSPMP/S-N) of the UCLP group was significantly shorter than that of the others. There were no significant differences in mandibular position (SNB, SN-Pog, SN-MP), shape (Ar-Go-Me), or size (Ar-Go/S-N, Go-Me/S-N, Co-Me/SN) among the three groups, except that the UCLP group exhibited a significantly larger gonial angle. Regarding the sagittal relationship between the maxilla and the mandible (ANB, convexity), no significant differences existed between any two of the groups, except that the UCLA group in GS2 showed a statistically increased ANB and convexity. The maxillary incisors (SN-U1) of both the UCLA and UCLP groups were statistically significantly more retroclined when compared with that of the control; although, there was no significant difference between the two experimental groups.
Error of Method DISCUSSION To determine the error in parameters, 10 radiographs were randomly selected and reassessed after at least a 1week interval by the same examiner. Method errors (MEs) were calculated using ffi the formula proposed by qX ffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffi Dahlberg ðME ¼ d2 =2nÞ (Houston, 1983), where P 2 d is the sum of the squared differences between the two mean values, and n is the number of double parameters. The MEs for angular and linear parameters
Repair of the unilateral cleft lip is one of the most dramatic surgical procedures that can improve the patients’ appearance to a great extent. However, controversy still remains as to whether lip repair inhibits maxillofacial growth (Bardach, 1990; Mars and Houston, 1990; Liao and Mars, 2005). Though several clinical arguments have been put forward to evaluate the effects of lip repair on maxillofacial morphology, they were all based on research
Shao et al., EFFECTS OF LIP REPAIR ON MAXILLOFACIAL MORPHOLOGY
TABLE 2
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Definitions of the Parameters
Parameters
Definitions
Cranial base length S-N, mm
Distance between S and N point
Maxillary skeletal SNA, 8 SN-ANS, 8 SN-PP, 8 ANS-PMP, mm ANS-PMP /S-N, %
Angle from S to N to A point Angle from S to N to ANS point Angle between the anterior cranial base (SN) and the palatal plane(ANS-PMP) Distance between the projections of point ANS and point PMP onto the frankfort horizontal plane (ANS-PMP / S-N) 3 100
Mandibular skeletal SNB, 8 SN-Pog, 8 SN-MP, 8 Ar-Go-Me, 8 Ar-Go, mm Ar-Go/S-N, % Go-Me, mm Go-Me/S-N, % Co-Me, mm Co-Me/S-N, %
Angle from S to N to B point Angle from S to N to Pog point Angle between the anterior cranial base (SN) and the mandibular plane Angle from Ar to Go to Me point Distance between Ar and Go point (Ar-Go / S-N) 3 100 Distance between Go and Me point (Go-Me / S-N) 3 100 Distance between Co and Me point (Co-Me / S-N) 3 100
Jaw relationship ANB, 8 Convexity, 8
Angle from A point to N to B point The intersection of line N-point A and point A-pogonion
Denture SN-U1, 8 L1-MP, 8
Angle between the anterior cranial base (SN) and the maxillary central incisor axis line Angle between the mandibular central incisor axis line and the mandibular plane
subjects with UCLP who had received lip repair only or both lip and palate surgery (Mars and Houston, 1990; Capelozza Filho et al., 1996; Liao and Mars, 2005). The observed effect must be attributed to several causes together, such as lip surgery, palate surgery, and even an intrinsic defect caused by the cleft of the palate. Therefore, in this study, patients with UCLA who had no cleft palate and had received lip repair only were selected to exclude TABLE 3
other influencing factors. Some authors reported that a comparison of various cleft subtypes showed different degrees of midfacial deficiency (Cronin and Hunter, 1980; Smahel and Brejcha, 1983; Chiu et al., 2011): mild in subjects with cleft lip and alveolus but severe in those with cleft lip and palate. Even so, the timing and technique of their lip repairs had no difference. Therefore, comparing maxillofacial morphology of patients with UCLA or
Cephalometric Parameters for 3 Groups in GS1 (Before the Pubertal Peak)
Parameters
UCLA Group Mean 6 SD
UCLP Group Mean 6 SD
Control Group Mean 6 SD
Maxillary skeletal SNA SN-ANS SN-PP ANS-PMP /S-N
81.6 83.9 8.9 71.6
6 6 6 6
3.3 3.2 3.6 3.1
80.1 83.4 10.4 67.4
6 6 6 6
3.0 3.1 5.1 4.4
80.4 84.3 8.2 71.3
6 6 6 6
Mandibular skeletal SNB SN-Pog SN-MP Ar-Go-Me Ar-Go/S-N Go-Me/S-N Co-Me/S-N
76.7 76.9 36.1 122.9 67.1 102.0 157.2
6 6 6 6 6 6 6
3.6 3.7 4.1 6.4 5.8 6.6 9.3
76.7 77.1 36.4 129.4 62.9 98.0 153.2
6 6 6 6 6 6 6
3.7 3.7 4.7 5.7 5.5 9.4 11.5
77.1 77.1 34.8 120.6 66.7 102.1 155.5
6 6 6 6 6 6 6
Jaw relationship ANB Convexity Denture SN-U1 L1-MP
4.8 6 2.5 9.8 6 5.5
3.4 6 3.3 6.5 6 6.9
93.4 6 8.1 92.3 6 6.9
86.9 6 11.6 89.0 6 5.9
P Value
Multiple Comparison†
4.4 4.9 2.1 4.9
.595 .726 .432 .024*
2,(3,1)
3.2 3.3 4.3 6.8 5.0 7.9 9.1
.786 .802 .538 .002* .094 .489 .658
3.3 6 1.8 7.2 6 3.8 104.8 6 5.5 93.4 6 4.6
(3,1),2
.090 .288 .000* .120
(2,1),3
† In the column of multiple comparison, 1, 2, and 3 mean UCLA (unilateral cleft lip and alveolus) group, UCLP (unilateral cleft lip and palate) group, and control group, respectively. * Significant differences (P .050) are presented in boldface.
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TABLE 4
Cephalometric Parameters for 3 Groups in GS2 (After the Pubertal Peak)
Parameters
UCLA Group Mean 6 SD
UCLP Group Mean 6 SD
Control Group Mean 6 SD
P Value
Multiple Comparison†
Maxillary skeletal SNA SN-ANS SN-PP ANS-PMP /S-N
83.2 87.1 10.0 75.1
6 6 6 6
3.7 3.7 4.5 4.3
79.4 82.8 8.7 68.8
6 6 6 6
5.0 5.5 3.9 6.6
82.4 86.6 8.2 73.4
6 6 6 6
2.5 2.0 3.2 4.1
.112 .056 .295 .006*
Mandibular skeletal SNB SN-Pog SN-MP Ar-Go-Me Ar-Go/S-N Go-Me/S-N Co-Me/S-N
78.7 79.2 33.8 119.3 74.8 106.5 166.9
6 6 6 6 6 6 6
3.7 3.7 4.7 6.4 9.1 8.1 10.0
77.9 78.6 34.5 123.0 74.0 106.7 167.3
6 6 6 6 6 6 6
4.0 4.3 8.5 8.6 8.0 6.5 7.8
79.8 80.5 32.5 116.2 79.6 110.6 171.3
6 6 6 6 6 6 6
2.9 2.8 3.8 6.4 6.6 5.2 9.3
.235 .237 .694 .016* .073 .061 .192
(3,1),(1,2)
2.6 6 1.2 4.1 6 2.6
.004* .005*
(2,3),1 (2,3),1
105.4 6 5.3 92.5 6 5.4
.001* .404
(1,2),3
Jaw relationship ANB Convexity Denture SN-U1 L1-MP
4.5 6 1.8 8.6 6 4.6 96.2 6 7.4 93.9 6 6.7
1.6 6 4.7 1.7 6 11.0 99.3 6 9.3 91.4 6 6.7
2,(3,1)
† In the column of multiple comparison, 1, 2, and 3 mean UCLA (unilateral cleft lip and alveolus) group, UCLP (unilateral cleft lip and palate) group, and control group, respectively. * Significant differences (P .050) are presented in boldface.
UCLP who have just undergone lip surgery with that of a person with no cleft can allow for evaluation of the role that lip repair plays in maxillofacial growth. The maxillofacial malformation of patients with cleft lip with or without cleft palate is closely related to their craniofacial skeletal growth. In previous studies, the midfacial morphology has been described according different chronological ages (Smahel et al., 1994; Han et al., 1995) or dental ages (Chen et al., 2012). But these indicators are not reliable predictors of an individual’s stage of skeletal development (Lewis, 1991; Flores-Mir et al., 2004). Cervical vertebral maturation is a method to evaluate the skeletal maturity on lateral cephalograms that has proven to be effective in assessing adolescent growth both in body height and mandibular size (Franchi et al., 2000; Baccetti et al., 2002). Consequently, considering the interaction of craniofacial morphology and skeletal growth, all the subjects have been grouped by CVMS using the Baccetti method (Baccetti et al., 2002). The results of this study showed equal length and protrusion of the maxilla in the UCLA groups and the controls, which supports the view that lip repair had no effect on the anteroposterior maxillary growth (Mars and Houston, 1990; Han et al., 1995; Liao and Mars, 2005). Seo (Seo et al., 2011), who investigated the growth pattern of patients with different types of cleft, found that surgical patients with UCLP and cleft palate had a more retruded maxilla than normally developed persons; whereas, those with UCLA demonstrated a nearly normal maxillary position. The authors then concluded that palatal scarring impaired maxillary growth more than lip scarring. In our study, cephalometric parameters of the UCLP group showed an almost normal maxillary sagittal position but
an obviously short anteroposterior maxilla length. It appears that there is a potential for normal maxillary growth in patients with UCLP who received lip surgery only, but the intrinsic defect caused by the cleft of the palate
FIGURE 3 Comparison of the cleft and control groups by superimposed cephalogram tracings (S-N at S) in GS1.
Shao et al., EFFECTS OF LIP REPAIR ON MAXILLOFACIAL MORPHOLOGY
FIGURE 4 Comparison of the cleft and control groups by superimposed cephalogram tracings (S-N at S) in GS2.
may have certain effects on sagittal maxillary growth. Contrary to our study, some researchers favor the view that major disturbance in maxillary growth was attributable to lip repair and studied patients with UCLP who had either only the lip operated upon or the lip as well as the palate repaired (Capelozza Filho et al., 1996; Kapucu et al., 1996; Li et al., 2006). But these studies ignored the intrinsic developmental deficiencies of these patients caused by the cleft palate and had certain limitations such as small sample size and overly broad age range. In the present study, no difference in mandibular position or morphology (SNB, SN-Pog, SN-MP, Ar-Go-Me, ArGo, Go-Me, Co-Me) was found among the UCLA and the control group. This is in accordance with previous studies (Bishara et al., 1985; Fudalej, 2007; Seo et al., 2011) and supports the claim that cleft mandibular growth is not related to lip repair (da Silva Junior et al., 1992; Capelozza Filho et al., 1996; Liao and Mars, 2005). We also found that the patients with UCLP had a normal mandibular position and length but a larger gonial angle. The results are similar to those of previous studies that observed a more clockwiserotated mandible in patients with UCLP who did not have surgical repair (Bishara et al., 1986; Mars and Houston, 1990; Capelozza Junior et al., 1993). These findings imply that shape and size of the mandible might be influenced by
663
cleft type and support the hypothesis that variations in mandibular growth may be a consequence of the cleft itself (da Silva Filho et al., 1993). In the sagittal relationship between the maxilla and the mandible, the study groups were equal to the controls, except the UCLA group in GS2 had a larger ANB and convexity. This phenomenon may be cogenerated by the slightly protruding maxilla and receding mandible. Bishara et al. (1976), who conducted cephalometric measurements for patients with UCLA who did not have surgical repair, also found larger SNA, ANB, and convexity compared with persons without clefts. Therefore, it is reasonable to suggest that lip repair does not change the sagittal relationship between the maxilla and the mandible. But why the patients with UCLA showed this growth pattern cannot be well explained, and further studies are needed to clarify this notion. The surgical groups (UCLA and UCLP) showed retroclined maxillary incisors when compared with the controls. This is consistent with the cephalometric findings of Hagerty and Hill (1963) and Mars and Houston (1990). Liao and Mars (2005) thought that the tension of the repaired lip produced a bone-bending effect on the anterior maxillary alveolus, accompanied by controlled retroclination of maxillary incisors. There are some disagreements about the position of the mandibular incisors. Some authors reported that patients with lip repair demonstrated more retroclined mandibular incisors, which compensated for the retroclined maxillary incisors (Capelozza Filho et al., 1996; Lisson et al., 2004). In our study, no significant difference on these teeth was observed among the groups, which corroborates the findings of Mars and Houston (1990) and Liao and Mars (2005). CONCLUSION Based on elaborated assessment on cephalometric data, we found there was an almost normal maxillary and mandibular growth but retroclined maxillary incisors in patients with cleft lip with or without cleft palate who received lip repair only, indicating that lip repair may not have a negative impact on the maxillofacial development and influence only the inclination of maxillary incisors. The shorter anteroposterior maxillary length and larger gonial angle in patients with UCLP who have not had palate repair compared with that in patients with UCLA suggest that these variations in maxillary and mandibular growth may be a consequence of the cleft itself but not surgical repair. REFERENCES Atherton JD. Morphology of facial bones in skulls with unoperated unilateral cleft palate. Cleft Palate J. 1967;4:18–30. Baccetti T, Franchi L, McNamara JA Jr. An improved version of the cervical vertebral maturation (CVM) method for the assessment of mandibular growth. Angle Orthod. 2002;72:316–323.
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