ORIGINAL ARTICLE

Facial Soft-Tissue Morphology of Adolescent Patients With Nonsyndromic Bilateral Cleft Lip and Palate Nadia Hasanzadeh, DDS, MS,* Mohammad Reza Majidi, MD,Þ Hamidreza Kianifar, MD,þ and Neda Eslami, DDS, MS§ Abstract: The purpose of this study was to cephalometrically evaluate the facial soft-tissue characteristics of adolescent patients with bilateral cleft lip and palate (BCLP) and to compare them with a noncleft control group. Lateral cephalometric radiographs obtained from 56 adolescents with nonsyndromic BCLP (29 boys and 27 girls) were analyzed and compared with 67 control subjects (29 boys and 38 girls) who were matched for sex, age, and ethnicity. All patients had been operated on before the age of 2 years for the surgical repair of cleft lip and palate. None had received any orthopedic or orthodontic treatment. Independent-samples t test revealed that patients with BCLP significantly differed from the control group by having a flatter facial profile, thinner and more retruded nasal base, flatter nasal tip (in males), and reduced upper-lip length. Furthermore, thicker lower-lip pit, shallower mentolabial sulcus, and increased inclination angles of the upper and lower lips relative to the horizontal plane were observed in female patients compared with the normal group. The findings of the current study suggested that adolescent patients with BCLP showed several facial soft-tissue deformities when compared with normal individuals with the same age, sex, and ethnic origin. This study provides objective measures that could lead to better treatment planning and prediction of the need for corrective surgeries in patients with BCLP. Key Words: Bilateral cleft lip and palate, facial soft tissue, cephalometry, morphology, nonsyndromic cleft (J Craniofac Surg 2014;25: 314Y317)

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lefts of the lip and palate are the fourth most common craniofacial birth defects.1 Patients with isolated or nonsyndromic cleft lip and/or palate has no other developmental or physical deformities. Orofacial clefts usually lead to various functional disturbances such as problems with feeding, hearing, speech, and dentofacial development. These defects may also cause psychosocial, emotional, and aesthetic problems.2,3 Treatment of patients affected with nonsyndromic cleft lip and/or palate needs a multidisciplinary team approach with a good interaction between different specialties: speech therapy, otolaryngology, psychology, audiology, orthodontics, maxillofacial surgery, and facial plastic surgery.4 The role of orthodontist in the management of these patients is substantial, particularly in the improvement of their facial aesthetics and dental occlusion.1 Currently, the key point in specific diagnosis and correction of dentofacial deformities is to incorporate the information from the clinical facial examination, plaster casts, panoramic radiographs, and cephalometric tracings.1,5 Facial analysis is used to identify positive and negative facial traits to optimize posttreatment facial alterations.6 A good relationship exists between soft-tissue morphology and underlying bony contours. When the skeletal pattern is so prominent that it can change the soft-tissue facial balance, tooth movement may not be sufficient, and orthognathic surgery would be indicated to achieve facial harmony.6,7 Soft-tissue evaluation on lateral cephalometric radiographs has been an effective tool for the orthodontist and maxillofacial surgeon to assess the facial growth and establish the most appropriate treatment options in patients with cleft lip and palate.8,9 Several previous studies have assessed facial morphology in patients with unilateral cleft lip and palate.9Y14 However, relatively few studies have addressed this issue for subjects with bilateral cleft lip and palate (BCLP). Thus, the information regarding soft-tissue cephalometric features of bilateral cleft patients is limited, based on the published literature. The aim of the present investigation was to cephalometrically evaluate the soft-tissue profile of Iranian patients with BCLP in comparison with normal population.

MATERIALS AND METHODS Study Subjects

From the *Dental Material Research Center, School of Dentistry; †Sinus and Surgical Endoscopic Research Center and ‡Ghaem Hospital, School of Medicine; and §Dental Research Center, School of Dentistry, Mashhad University of Medical Sciences, Mashhad, Iran. Received July 18, 2013. Accepted for publication September 17, 2013. Address correspondence and reprint requests to Neda Eslami, DDS, MS, Department of Orthodontics, School of Dentistry, Mashhad University of Medical Sciences, Vakilabad Blvd, Mashhad, Iran; E-mail: islamin@ mums.ac.ir The authors report no conflicts of interest. Copyright * 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000446

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A total of 123 adolescents between 13 and 19 years of age enrolled in the current study. Participants were divided into the cleft and control groups. The cleft group was composed of 56 Iranian patients (29 boys and 27 girls) with BCLP who had no other birth defect or syndrome. Subjects were recruited between 2007 and 2012 from the Cleft lip and Palate Clinic of Mashhad Dental School and Ghaem Medical Center in Mashhad, northeast of Iran. All patients had been operated on before the age of 2 years, for the surgical repair of cleft lip and palate. None of them had previously received orthodontic treatment or orthognathic surgery. The mean age of cleft patients was 15 years 6 months for boys and 16 years 1 month for girls. The control group consisted of 67 healthy age-matched adolescents (29 males and 38 girls) without cleft lip or palate or any other

The Journal of Craniofacial Surgery

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Bilateral Cleft Lip and Palate Patients

congenital anomalies, chosen from the Orthodontic Department of Mashhad University of Medical Sciences. They all had normal skeletal relationships, symmetrical faces, class I occlusion, and normal overjet or overbite with no history of orthodontic treatment, maxillofacial orthopedics, or craniofacial surgery. The mean age of control group was 17 years 2 months for boys and 17 years 1 month for girls.

Cephalometric Analysis Lateral cephalometric radiographs were obtained under standardized conditions with the head oriented along the Frankfort horizontal (FH) plane parallel to the floor. For all subjects, 18  24-cm films were used. The film-focus distance was 5 feet, and the distance from the midsagittal plane to the film was 13 cm. The subjects were asked to relax their lips and occlude their teeth in centric occlusion. Planmeca XC cephalometry x-ray machine (Helsinki, Finland) was used for all patients. The settings of the cephalostat machine and the magnification of cephalograms were the same for all cases throughout the investigation. All lateral radiographs were manually traced by the same orthodontist. The landmarks identified on the cephalograms and parameters measured for the analysis of soft-tissue profile, lips, and nasal morphology are shown in Figures 1 and 2. Moreover, definitions of these landmarks and variables are presented in Tables 1 and 2. For the evaluation of method error, 10 lateral cephalograms were randomly selected in each group and reassessed by the same researcher after 1 month. The Dahlberg formula was used for calculating method errors.15,16 The method error between registrations was less than 0.95 mm for linear parameters and less than 1 degree for angular measurements.

Statistical Analysis Mean and SD of all parameters measured in both groups were calculated using descriptive statistics. Independent-samples t test was used to compare the mean values of all variables between the cleft and control groups. Statistical significance was set as P G 0.05.

RESULTS The mean values, SDs, and Student t tests of the variables used for the analysis of soft-tissue morphology are shown in Tables 3 and 4. Relative to the control subjects, G-Prn-Pg¶ and G-Sn-Pg¶ were significantly larger in the BCLP group. Moreover, the projection

FIGURE 2. Cephalometric parameters used in the analysis of lip form and position and nasal morphology (see Tables 1 and 2 for definitions of landmarks and variables).

distance of points G and Sn in the FH plane was shorter in the cleft group, demonstrating a more retruded nasal base in these patients. The proportion of superior facial height/inferior facial height (G-Sn/ Sn-Me¶) was significantly larger in the male BCLP group, compared with the control subjects (P = 0.002). Other measurements such as GPg¶ and Pg-Pg¶ were not significantly different between the cleft and control groups (Tables 3 and 4). Mean values, SDs, and the results of Student t test for comparison of lip form and position, and nasal morphology are shown in Tables 5 and 6. TABLE 1. Cephalometric Landmarks, Reference Planes, and Definitions Abbreviation S N G N¶ Prn Cm Sn A¶ Ls Li Stms Stmi B¶ Pg¶ Me¶ Up Lp A B

FIGURE 1. Cephalometric landmarks and parameters determined in the analysis of soft-tissue profile (see Tables 1 and 2 for definitions of landmarks and variables).

Po Or FH EP

Definition Sella, center of the sella turcica Nasion, the most anterior part of the frontonasal suture in the median plane Soft-tissue glabella, the most anterior point on the soft-tissue glabella Soft-tissue nasion, the deepest point on the frontonasal curvature Pronasale, the most prominent point on the apex of the nose Columella, the most anterior point of columella Subnasale, the deepest point in the nasolabial curvature Soft-tissue subspinale, the point of greatest concavity in the midline of the upper lip Labrale superius, the most prominent point on the prolabium of the upper lip Labrale inferius, the most prominent point on the prolabium of the lower lip Stomion superius, the lowermost point of the upper lip Stomion inferius, the uppermost point of the lower lip Soft-tissue supramental, the point of the greatest concavity in the midline of the lower lip Soft-tissue pogonion, the most prominent point on the chin Soft-tissue menton, the most inferior point on the chin The most prominent point of the upper central incisor The most prominent point of the lower central incisor Subspinale, the point at the deepest midline concavity on the maxilla below the anterior nasal spine Supramentale, the point at the deepest midline concavity of mandibular symphysis Porion, the most superior point on the border of external auditory meatus Orbitale, the lowest point on the inferior margin of the orbit FH plane, the line connecting the porion and orbitale E plane, the line through points Prn and Pg¶

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TABLE 2. Angular and Linear Parameters Measured Variable

TABLE 4. Comparison of Soft-Tissue Morphology Measurements in Girls Between BCLP Patients (n = 27) and Control Subjects Without a Cleft (n = 38)

Definition

1. G-Prn-Pg¶ 2. G-Sn-Pg¶ 3. G-Sn 4. G-Pg¶ 5. G-Sn/Sn-Me¶ 6. Pg-Pg¶ 7. Cm-Sn-Ls 8. Sn-Stms/Stmi-Me¶ 9. Ls-EP 10. Li-EP 11. B¶-LiPg¶ 12. A-Sn 13. Up-Ls 14. Lp-Li 15. B-B¶ 16. Sn-Stms 17. Stmi-B¶ 18. A¶Ls-FH 19. LiB¶-FH 20. Prn-N¶-Sn 21. Prn-Sn

Angle of total facial convexity Angle of facial convexity The projection distance of points G, Sn in the FH plane The projection distance of points G, Pg¶ in the FH plane Superior facial height /inferior facial height Thickness of soft-tissue chin Nasolabial angle Upper-lip height/mentolabial height Point Ls to E plane, prominence of upper lip Point Li to E plane, prominence of lower lip Depth of mentolabial sulcus Thickness of nasal base Thickness of the upper labiate process Thickness of the lower labiate process Thickness of lower-lip pit Length of the upper lip Length of the lower lip The angle between A¶Ls line and FH plane The angle between LiB¶ and FH plane Nasal angle The projection distance of points Prn, Sn in the FH plane, prominence of nasal tip

In comparison with the control group, BCLP patients had significantly reduced upper-lip length (Sn-Stms) and thinner nasal base (A-Sn) in both male and female subjects. Female cleft patients had increased thickness of lower lip (LP-Li) and lower-lip pit (B-B¶) and shallower mentolabial sulcus (B¶-LiPg¶), relative to the matched control subjects. In addition, the variables A¶Ls-FH and LiB¶-FH were significantly increased in the female cleft group, representing the more retroclined position of both upper and lower lips compared with the control subjects. Reduced nasal projection (Prn-N¶-Sn) was observed in male BCLP group, indicating a flatter nose in these patients relative to the control subjects (Tables 5 and 6).

DISCUSSION In recent years, soft-tissue profile analysis has proven to be more effective in discriminating between different groups compared with conventional hard tissue analysis.10,17 Previous studies have used this method to assess soft-tissue morphology and provide some basic data for the management of patients with unilateral cleft lip and palate at different age ranges.9Y12 However, scientific data on this field are limited for patients affected with BCLP, possibly because of TABLE 3. Comparison of Soft-Tissue Morphology Measurements in Boys Between BCLP Patients (n = 29) and Control Subjects Without a Cleft (n = 29)

BCLP

BCLP/Control Subjects

Control Subjects

Variable*

Mean

SD

Mean

SD

t

P

G-Prn-Pg¶ G-Sn-Pg¶ G-Sn G-Pg¶ G-Sn/Sn-Me¶ Pg-Pg¶

145.31 171.62 3.81 8.50 1.03 12.00

6.60 9.10 3.63 5.77 0.10 2.85

134.77 165.00 6.48 7.44 0.95 11.48

18.29 4.19 3.98 4.98 0.08 2.49

2.917 3.556 j2.668 0.742 3.304 0.736

0.005† 0.001† 0.010‡ 0.461 0.002† 0.465

Distances are reported in millimeters and angles in degrees. *See Tables 1 and 2 for definitions of landmarks and variables. †P G 0.01. ‡P G 0.05.

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Control Subjects

BCLP

BCLP/Control Subjects

Variable*

Mean

SD

Mean

SD

t

P

G-Prn-Pg¶ G-Sn-Pg¶ G-Sn G-Pg¶ G-Sn/Sn-Me¶ Pg-Pg¶

147.81 175.81 3.66 7.59 0.97 11.00

7.38 7.93 3.19 4.64 0.13 3.16

137.90 164.81 7.38 6.76 0.96 11.07

5.09 5.62 4.27 4.63 0.09 2.03

6.405 6.548 j4.004 0.710 0.315 j0.114

0.000† 0.000† 0.000† 0.480 0.754 0.910

Distances are reported in millimeters and angles in degrees. *See Tables 1 and 2 for definitions of landmarks and variables. †P G 0.01.

the low incidence of this deformity.18,19 We attempted to investigate facial soft-tissue profile of adolescent patients with BCLP, before their preparation for the potential orthognathic surgery. Most of previous studies have compared soft-tissue profiles of BCLP patients between 2 and 3 cleft centers with different treatment protocols. However, they all have the shortcoming of lacking control subjects.20Y22 Therefore, their results could not be directly compared with the current investigation. The findings of the current study revealed a more retruded and thinner nasal base in cleft patients compared with the control subjects. Furthermore, the BCLP group had a significantly larger G-PrnPg¶ and smaller Prn-N¶-Sn angle relative to the control subjects, which indicates reduced nasal protrusion and flatter nose in the cleft group have occurred. Likewise, investigations on soft-tissue morphology of patients with unilateral cleft lip and palate have presented similar results.8Y10 This reduction in nasal growth may be related to a combination of factors including decreased forward growth of maxilla, surgical trauma, and postoperative scar tissue in palate of patients with cleft lip and palate disorder. In the current study, lips were more retroclined in female patients relative to the control group. Moreover, reduced upper-lip TABLE 5. Lip Form and Position and Nasal Morphology Comparison Between Male Patients With BCLP (n = 29) and Control Subjects (n = 29) Control Subjects

BCLP Variable* Cm-Sn-Ls Sn-Stms/Stmi-Me¶ Ls-EP Li-EP B¶-LiPg¶ A-Sn Up-Ls Lp-Li B-B¶ Sn-Stms Stmi-B¶ A¶Ls-FH LiB¶-FH Prn-N¶-Sn Prn-Sn

BCLP/Control Subjects

Mean

SD

Mean

SD

t

P

111.03 0.35 j2.31 0.48 4.79 12.46 12.43 14.70 12.51 17.15 17.74 86.00 51.72 19.25 15.96

18.57 0.10 6.88 3.26 1.89 4.92 2.74 2.41 2.54 3.31 4.14 20.00 11.30 2.78 3.39

106.75 0.47 j4.03 j1.39 5.24 16.36 12.60 14.31 12.58 21.63 18.00 78.55 48.17 21.41 15.70

7.43 0.16 2.80 3.23 1.32 2.57 2.88 1.58 2.73 3.09 3.57 10.49 9.92 2.13 2.66

1.151 j3.442 1.249 2.203 j1.044 j3.774 j0.233 0.740 j0.099 j5.325 j0.254 1.776 1.271 j3.310 0.323

0.257 0.001† 0.219 0.032‡ 0.301 0.000† 0.817 0.463 0.921 0.000† 0.800 0.083 0.209 0.002† 0.748

Distances are reported in millimeters and angles in degrees. *See Tables 1 and 2 for definitions of landmarks and variables. †P G 0.01. ‡P G 0.05.

* 2014 Mutaz B. Habal, MD

Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.

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TABLE 6. Lip Form and Position and Nasal Morphology Comparison Between Female Patients With BCLP (n = 27) and Control Subjects (n = 38) Control Subjects

BCLP Variable* Cm-Sn-Ls Sn-Stms/Stmi-Me¶ Ls-EP Li-EP B¶-LiPg¶ A-Sn Up-Ls Lp-Li B-B¶ Sn-Stms Stmi-B¶ A¶Ls-FH LiB¶-FH Prn-N¶-Sn Prn-Sn

BCLP/Control Subjects

Mean

SD

Mean

SD

t

P

102.77 0.35 j4.40 j0.81 4.09 11.03 10.53 13.88 12.50 16.50 17.09 82.92 55.48 20.55 14.81

21.64 0.06 5.71 2.86 1.81 3.41 2.37 1.70 2.57 3.68 4.12 13.03 9.01 3.38 3.58

104.68 0.45 j3.77 j1.47 4.97 14.14 10.92 12.92 11.15 19.60 16.73 76.36 48.34 20.93 14.03

10.06 0.06 2.14 2.26 1.50 2.09 1.93 1.71 1.28 1.91 2.78 8.79 9.00 1.91 1.53

j0.426 j5.841 j0.547 1.033 j2.134 j4.543 j0.718 2.247 2.496 j4.010 0.415 2.423 3.148 j0.524 1.055

0.673 0.000† 0.588 0.306 0.037‡ 0.000† 0.476 0.028‡ 0.017‡ 0.000† 0.679 0.018‡ 0.003† 0.603 0.299

Distances are reported in millimeters and angles in degrees. *See Tables 1 and 2 for definitions of landmarks and variables. †P G 0.01. ‡P G 0.05.

length was observed in both sexes with BCLP. This could be attributed to the deficiency in upper-lip tissue and the tension exerted by scar tissues formed following lip repair. Increased lower-lip thickness of female patients observed in the current study may be explained by the adaptation to the deficient form and function of upper lip. These findings are in agreement with the study of Liu et al9 on subjects with unilateral cleft lip and palate. We observed a larger ratio of G-Sn/Sn-Me¶ in male BCLP patients relative to the control subjects. However, in an investigation by van den Dungen et al19 on the craniofacial morphology of BCLP cases, the ratio ANS-Me/N-Me was larger in the BCLP group, indicating a relatively long lower facial height. In the current study, the facial height development of female BCLP patients did not significantly differ with that of the normal adolescent group. Therefore, BCLP patients did not seem to have compensatory mandibular vertical growth in our study. Moreover, the measurement of Pg-Pg¶ was statistically similar between the 2 groups, indicating a normal chin development in BCLP patients. Liu et al9 have shown comparable results with regard to patients with unilateral cleft in the mixed dentition stage. Based on the results of the current study, soft-tissue profile of adolescent patients with BCLP differed significantly from the noncleft control group. They showed retruded nasal base and concave facial profile, reduced upper-lip length, flatter nasal tip, reduced nasal protrusion, and increased lower-lip thickness. Although male and female patients were not directly compared in the current study, some sex differences in soft-tissue morphology were detected. This may not be related to cleft deformity but to the potential differences in facial shape and size or soft-tissue thickness between the 2 sexes in general population.23,24 For more detailed investigation on this issue, further studies with larger number of cleft patients are recommended. Patients with BCLP are a challenge for the interdisciplinary team involved in the treatment of this disorder. This study provides objective measures for the evaluation of facial deformities in BCLP patients that will lead to better treatment planning and prediction of the need for corrective surgeries. These cephalometric values can also be postoperatively used for the quantitative assessment of treatment results.

Bilateral Cleft Lip and Palate Patients

REFERENCES 1. Mercado AM, Vig KWL. The orthodontist’s role in a cleft palateYcraniofacial team. In: Graber LW, Vanarsdall RL, Vig KWL, eds. Orthodontics: Current Principles and Techniques. Philadelphia, PA: Mosby, 2012:965Y990 2. Mossey PA, Little J, Munger RG, et al. Cleft lip and palate. Lancet 2009;374:1773Y1785 3. Eslami N, Majidi MR, Aliakbarian M, et al. Oral health-related quality of life in children with cleft lip and palate. J Craniofac Surg 2013;24:e340Ye343 4. Schnitt DE, Agir H, David DJ. From birth to maturity: a group of patients who have completed their protocol management. Part I. Unilateral cleft lip and palate. Plast Reconstr Surg 2004;113:805Y817 5. Proffit WR, White RP, Sarver DM. Contemporary Treatment of Dentofacial Deformity. St Louis, MO: Mosby, 2003 6. Jacobson A, Vlachos C. Soft tissue evaluation. In : Jacobson A, Jacobson RL, eds. Radiographic Cephalometry from Basics to 3-D Imaging. Hanover Park, IL: Quintessence Publishing Co, 2006:205Y217 7. Nanda RS, Meng H, Kapila S, et al. Growth changes in the soft tissue facial profile. Angle Orthod 1990;60:177Y190 8. Naduwinmani JP, hallolli C, Naduwinmani SL. Cephalometric profile evaluation in patients with cleft lip and palate. Int J Contemp Dent 2011;2:63Y69 9. Liu RK, Wamalwa P, Lu DW, et al. Soft-tissue characteristics of operated unilateral complete cleft lip and palate patients in mixed dentition. J Craniofac Surg 2011;22:1275Y1279 10. Bearn DR, Sandy JR, Shaw WC. Cephalometric soft tissue profile in unilateral cleft lip and palate patients. Eur J Orthod 2002;24:277Y284 11. Toygar TU, Akc¸am MO, Arman A. A cephalometric evaluation of lower lip in patients with unilateral cleft lip and palate. Cleft Palate Craniofac J 2004;41:485Y489 12. Del Guercio F, Meazzini MC, Garattini G, et al. A cephalometric intercentre comparison of patients with unilateral cleft lip and palate at 5 and 10 years of age. Eur J Orthod 2010;32:24Y27 13. Johnston CD, Leonard AG, Burden DJ, et al. A comparison of craniofacial form in Northern Irish children with unilateral cleft lip and palate treated with different primary surgical techniques. Cleft Palate Craniofac J 2004;41:42Y46 14. Leonard AG, Kneafsey B, McKenna S, et al. A retrospective comparison of craniofacial form in Northern Irish children with unilateral cleft lip and palate. Cleft Palate Craniofac J 1998;35:402Y407 15. Pereira-Maxwell F. A-Z of Medical Statistic. 1st ed. New York: Oxford University Press, 1998:69Y70, 99Y101 16. Houston WJB. The analysis of errors in orthodontic measurements. Am J Orthod 1983;83:382Y390 17. Mølsted K, Asher-McDade C, Brattstro¨m V, et al. A six-center international study of treatment outcome in patients with clefts of the lip and palate: part 2. Craniofacial form and soft tissue profile. Cleft Palate Craniofac J 1992;29:398Y404 18. Sivertsen A, Wilcox A, Johnson GE, et al. Prevalence of major anatomic variations in oral clefts. Plast Reconstr Surg 2008;121:587Y595 19. van den Dungen GM, Ongkosuwito EM, Aartman IH, et al. Craniofacial morphology of Dutch patients with bilateral cleft lip and palate and noncleft controls at the age of 15 years. Cleft Palate Craniofac J 2008;45:661Y666 20. Bartzela TN, Katsaros C, Bronkhorst EM, et al. A two-centre study on facial morphology in patients with complete bilateral cleft lip and palate at nine years of age. Int J Oral Maxillofac Surg 2011;40:782Y789 21. Gaukroger MJ, Noar JH, Sanders R, et al. A cephalometric inter-centre comparison of growth in children with cleft lip and palate. J Orthod 2002;29:113Y117 22. Heidbu¨chel KL, Kuijpers-Jagtman AM, Freihofer HP. Facial growth in patients with bilateral cleft lip and palate: a cephalometric study. Cleft Palate Craniofac J 1994;31:210Y216 23. Wilkinson CM. In vivo facial tissue depth measurements for White British children. J Forensic Sci 2002;47:459Y465 24. Ferrario VF, Sforza C. Size and shape of soft-tissue facial profile: effects of age, gender, and skeletal class. Cleft Palate Craniofac J 1997;34: 498Y504

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Facial soft-tissue morphology of adolescent patients with nonsyndromic bilateral cleft lip and palate.

The purpose of this study was to cephalometrically evaluate the facial soft-tissue characteristics of adolescent patients with bilateral cleft lip and...
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