Soft-tissue profile growth in patients with repaired complete unilateral cleft lip and palate: A cephalometric comparison with normal controls at ages 7, 11, and 18 years.

ORIGINAL ARTICLE

Soft-tissue profile growth in patients with repaired complete unilateral cleft lip and palate: A cephalometric comparison with normal controls at ages 7, 11, and 18 years Iris Moreira,a Sunjay Suri,b∗ Bruce Ross,c Bryan Tompson,d David Fisher,e and Wendy Louf Toronto, Ontario, Canada

Introduction: In this retrospective longitudinal study, we aimed to study differences in the soft-tissue profiles in growing children with clefts in comparison with controls through the period of facial growth from 7 to 18 years. Methods: Lateral cephalometric measurements made at 7 years (T1), 11.1 years (T2), and 17.9 years (T3) of age of 70 white children (35 boys, 35 girls) with complete unilateral cleft lip and palate (UCLP) who received primary lip and palate repair surgeries at The Hospital for Sick Children, Toronto, were compared with those of a control group of similar ages, sexes, and racial backgrounds, and having skeletal Class I facial growth, selected from the Burlington Growth Study. None of the included subjects had received any surgeries other than the primary lip and palate repairs, and none had undergone nasal septum surgery or nasal molding during infancy. Between-group comparisons were made at each time point using generalized linear models adjusted for age and sex effects. Longitudinal comparisons across all time points were conducted using the mixed model approach, adjusting for these effects and their interactions with time. Results: Bimaxillary retrognathism, progressive maxillary retrognathism, and increasing lower anterior face height with downward and backward growth rotation of the mandible in the UCLP group were seen. Unlike the hard-tissue face height ratio, their soft-tissue face height ratio was not affected. The upper lips in the UCLP group were shorter by 1.81 mm at T2 (P \0.001) and by 1.16 mm at T3 (P 5 0.018), whereas their lower lips were 2.21 mm longer at T3 (P 5 0.003). A reduced upper lip to lower lip length ratio at T2 and T3 (P \0.001) resulted. Their upper lips were relatively retruded by 1.44 mm at T1, 1.66 mm at T2, and 1.86 mm at T3 (all, P \0.001), and their lower lips were relatively protruded by 1.07 mm at T1 (P 5 0.003), 1.40 mm at T2 (P \0.001), and 1.62 mm at T3 (P \0.001). Nose depths in the UCLP group were shallower by at least 1 mm from T1 to T3, and columellar length was shorter by almost 2 mm (all, P \0.001). Their columellae and nose tips rotated downward with growth, with the most significant rotations experienced from T2 to T3, and progressive reductions in their soft-tissue profile convexity were seen from T1 to T3 (P \0.001). Conclusions: Key attributes of the imbalance in the soft-tissue profile in children with repaired UCLP were identified in the lip and nose regions. Although many profile differences were visible as early as 7 years of age, they became more apparent by 11 years of age and increased in severity thereafter. The short upper lip combined with a long lower lip resulted in the characteristic lip length imbalance, whereas the progressively retruding upper lip and protruding lower lip led to developing a step relationship in the sagittal lip profile during the adolescent growth period. Their columellae and nose tips rotated downward during this time. (Am J Orthod Dentofacial Orthop 2014;145:341-58) a Research associate, Faculty of Dentistry, University of Toronto, Toronto, Ontario, Canada; former clinical orthodontic fellow, Hospital for Sick Children, Toronto, Ontario, Canada. b Associate professor, Discipline of Orthodontics, Faculty of Dentistry, University of Toronto, Toronto, Ontario, Canada; staff orthodontist, Hospital for Sick Children, Toronto, Ontario, Canada. c Professor, Discipline of Orthodontics, Faculty of Dentistry, University of Toronto, Toronto, Ontario, Canada; staff orthodontist, Hospital for Sick Children, Toronto, Ontario, Canada. d Associate professor and head, Discipline of Orthodontics, Faculty of Dentistry, University of Toronto, Toronto, Ontario, Canada; head, Division of Orthodontics, Hospital for Sick Children, Toronto, Ontario, Canada. e Medical director, Cleft Lip and Palate Program, Hospital for Sick Children, Toronto, Ontario, Canada; associate professor, Department of Surgery, University of Toronto, Toronto, Ontario, Canada. f Professor and head, Division of Biostatistics, Dalla Lana School of Public Health,

University of Toronto, Toronto, Ontario, Canada; Canada Research Chair in Statistical Methods for Health Care. Iris Moreira and Sunjay Suri are joint first authors and contributed equally to this work. All authors have completed and submitted the ICMJE Form for Disclosure of Potential Conflicts of Interest, and none were reported. Supported in part by the Ski for Kids campaign and a Biomedical Research Award from the American Association of Orthodontists Foundation. Presented in part at the 12th International Congress on Cleft Lip/Palate and Related Craniofacial Anomalies, May 5-10, 2013; Orlando, Fla. Address correspondence to: Sunjay Suri, 124 Edward St, Rm 519-B, Discipline of Orthodontics, Faculty of Dentistry, University of Toronto, Toronto, Canada M5G 1G6; e-mail, [email protected]. Submitted, January 2013; revised and accepted, November 2013. 0889-5406/$36.00 Copyright Ó 2014 by the American Association of Orthodontists. http://dx.doi.org/10.1016/j.ajodo.2013.11.018

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T

he effect of facial soft tissues on facial esthetics is well known and has been researched extensively.1-4 The nose, lips, and chin are the key regions of the face with the most significant impact on facial esthetics.5-8 In patients with repaired clefts, craniofacial growth is altered, and relative midfacial retrusion is a characteristic feature that becomes increasingly apparent with longitudinal growth.9,10 This retrusion is also seen in the upper lip that remains deficient in both height and depth, in contrast to the lower lip that is longer,11 protruded,12,13 and frequently everted.12,14,15 Sagittal and vertical nose-lip-chin imbalances are deterrents to facial esthetics in everyone.8,16 Rhinoplasty is the most commonly performed esthetic facial plastic surgery procedure, and botulin toxin and hyaluronic acid injections are the most common nonsurgical esthetic facial procedures performed by facial plastic surgeons in the United States.17 Not surprisingly, patients with clefts, who frequently have facial imbalances in the lip and nose regions, report these as having the most unfavorable impacts on their facial attractiveness, self-image, and self-esteem.7 The lip, nose, and columella also most frequently receive surgical revision in persons with clefts. One major goal of orthognathic surgery in these patients is to improve the soft-tissue profile esthetics by correcting the imbalances.18 Patients with facial malformations, including those with clefts, have been reported to suffer significant disadvantages in their social lives with higher prevalences of depression and anxiety.19-23 Although the importance of the soft-tissue profile in patients with clefts is well recognized, several limitations of previous studies have not allowed a complete understanding of this important aspect of their facial growth. The published literature comprises studies that either were cross-sectional or did not describe a sufficiently longitudinal comparison with control subjects of similar ages and sexes, or had fewer subjects or measurements, or fewer female subjects represented in the data, or were outcome studies that compared the effects of one surgical protocol with another and did not study the soft-tissue profile as their main focus. Table I includes a summary of literature reports that provided data on the soft-tissue profile in patients with unilateral cleft lip and palate (UCLP). We aimed to detail the differences in the soft-tissue profiles of patients with repaired complete UCLP from those of noncleft controls with comparable age, sex, and ethnicity at 7, 11, and 18 years, thus covering the entire period of active facial growth. We also aimed to analyze the differences longitudinally to determine whether the nature and magnitude of these differences changed over the growth period. Our

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objective was to study the soft-tissue profile differences and investigate the longitudinal differences in growth of the soft-tissue profile in general. We did not intend to evaluate the outcome of one surgical protocol or one surgeon with another. MATERIAL AND METHODS

After research ethics board approval, the lateral cephalograms of 70 white subjects (35 boys, 35 girls) with nonsyndromic complete UCLP who had received their lip and palate surgeries at the Hospital for Sick Children, Toronto, were retrospectively obtained from the archives of the hospital's craniofacial center. In this sample, 5 surgeons and their teams had performed the lip repairs using rectangular or lower triangular flaps or their modifications. Palate repair surgeries had been conducted by 5 surgeons and their teams using techniques that included push-back palatoplasty, von Langenbeck repair, or their modifications. No patients with Simonart's bands were included in the sample. To prevent effects of any other surgery than the primary lip and palate repairs on the soft-tissue profile, only patients who had not received any primary septoplasty, nasal revision, or alveolar bone graft surgery were selected. Furthermore, no subjects had received nasal molding in their presurgical maxillary orthopedic treatment during infancy. Lateral cephalograms available at 3 time points—T1 (7 years), T2 (11 years), and T3 (18 years)—were used (Table II). The T3 cephalograms had been taken before the orthognathic surgery if surgery had been provided for any subject. These steps ensured that our data were relatively homogeneous and free from any effects that secondary surgery or any ancillary procedure might have had on the skeletal or soft-tissue measurements. With the participation of experts from many disciplines involved in providing long-term craniofacial care of patients at the center, it is reasonable to state that our sample, which was under long-term follow-up and treatment of over 18 years at the center, included children who had been accurately diagnosed with nonsyndromic UCLP. The lateral cephalograms had been taken with the teeth in occlusion and the lips in repose. Cephalometric details, including soft-tissue profiles, were traced by 1 investigator (I.M.) and digitized by an experienced digitizer. For each subject with UCLP, radiographs from a white control subject with normal skeletal Class I growth and comparable age and sex were obtained from the Burlington Growth Center archives at the Faculty of Dentistry, University of Toronto, and were similarly traced and digitized. A customized cephalometric analysis was developed using the Dentofacial Planner software (Dentofacial Planner, Toronto, Ontario, Canada) to include

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Table I. Cephalometric studies reporting at least 5 soft-tissue profile parameters in repaired complete UCLP Author (year); study design Sadowsky et al11 (1973); mixed longitudinal

Ethnicity; region of sample White; Chicago

Pensler and Mulliken12 (1988); cross-sectional, mesh analysis

Mixed ethnicity; Boston

Enemark et al42 (1990); longitudinal

White; Copenhagen and Aarhus

Semb10 (1991); mixed longitudinal

White; Oslo

Friede et al43 (1991); cross-sectional

White; Goteborg, Aarhus, Oslo, Linkoping

Number of soft-tissue measurements reported UCLP sample Surgery Comparison group 15 n 5 75 (54 M, 21 F), Lip repair: Tennison, Noncleft data from no Simonart`s band LeMesurier, Millard Subtelny55 (1959); Palate repair: Wardill and Posen56 (1967): push-back, vonn 5 30; 15 M, 15 F, Langenbeck/vomer with normal skeletal flap profiles 7 n53 Not described Normal values for the mesh diagram analysis from Moorrees et al57 (1976) Noncleft data from 8 n 5 57 (42 M, 15 F), Lip repair: Tennison Michigan Growth ages 5, 8, 12, 16 Palate repair: pushStudy norms from and 21 y, no back palatoplasty Riolo et al58 (1974) and vomer flap Simonart`s band Other surgery: pharyngeal flap, alveolar bone grafting Bolton normal values 27 n 5 177 Lip repair: modified (white, 16 M, 16 F, (126 M, 51 F), LeMesurier, Millard excellence in static Hard palate: complete UCLP occlusion, without Simonart`s superiorly based esthetically favorable band single layer vomer faces)59 flap Posterior palate: von Langenbeck Other surgery: alveolar bone grafting; a few lip revisions, superiorly based pharyngeal flap (20%) Intercenter comparison Lip repair: not 11 15 consecutive described. patients at each Palate repair: center: Goteborg Linkoping used 1(10 M, 5 F), Oslo stage push-back. (11 M, 4 F), Aarhus Aarhus used 2 (12 M, 3 F), and stages—vomer flap Linkoping (15 M, 0 F). Age range, 7-10 and push-back. Oslo used 2 stages—vomer y, Simonart`s band flap and von included Langenbeck. Goteborg operated on velum first and delayed hard palate closure until mixed dentition



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Table I. Continued Author (year); study design Molsted et al38 (1992); cross-sectional

Ethnicity; region of sample White; Eurocleft study

Brattstrom et al44 (1992); cross-sectional

White; Stockholm, Oslo, and Toronto

Smahel et al45 (1993); mixed longitudinal and cross-sectional

White; Prague

Tindlund and Rygh46 (1993); cross-sectional

White; Bergen

Chaisrisookumporn et al32 (1995); cross-sectional

White; Texas


Number of soft-tissue measurements reported UCLP sample Surgery Comparison group Differences between 13 n 5 151; age, 8-9 y; Lip repair: Millard, centers; no n 5 18-41 for each Tennison normative group center. M:F varying Palate repair: between 1:0.8 and Bardach, Delaire, 1:0.3 Total number Wardill- Kilner, of surgeons 5 34 vomer flap, von Langenbeck Other surgery: alveolar bone grafting 27 n 5 85 (59 M, 26 F), Lip surgery: Millard and Intercenter comparison ages,16-18 y vomer flap, Skoog, Millard, Tennison and 4-flap technique, RandallTennison, le Mesurier Palate repair: von Langenbeck, Wardill- Kilner Other surgery: alveolar bone grafting 8 n 5 81 (ages, 8 y, 53; Lip repair: Tennison, 8-year- old UCLP were 10 y, 61; 12 y, 43; 14 Veau; compared with 5y, 38) and 24 adults Palate repair: pushyear-old normative back data from Smahel Other surgery: and Mullerova60 pharyngeal flap (1986); 10-14 year old children were compared with normal adult male volunteers reported in Smahel and Brejcha61 (1983) 36 Group 1 UCLP n 5 45; Lip repair: lip, primary Normative data taken group 2, various cleft and anterior palate. from Tindlund et al62 (1993); n 5 41 (27 types n 5 63. Mean Periosteoplasty M, 14 F). Normal age, 6 y (modified Skoog) growth, Palate repair: pushmaxillomandibular back. relationship (ANB No other surgery. angle, 0 -4 ), normal occlusion, matched for age and sex 8 10 bilateral, 20 Lip repair: Millard. Normative unilateral (15 M, 15 Palate repair: cephalometric data F); mean age, 20.8 y Wardill palatoplasty. taken from (range, 12-42 y) Other surgery: Scheideman et al63 velopharyngeal (1980) and Epker flaps, alveolar bone and Fish64 (1986) grafting


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Table I. Continued Ethnicity; region of sample

Number of soft-tissue measurements reported

Roberts-Harry et al (1996); crosssectional

White; Oslo and Bristol

22

Spyropoulos and Linder Aronson48 (1997); crosssectional

White; Greek and Swedish

8

Smahel et al49 (1998); crosssectional

White; Prague

9

Leonard et al50 (1998); Crosssectional

White; Irish

11

Author (year); study design 47

UCLP sample

Surgery

n 5 40 from Oslo (26 Oslo: lip repair: Millard M, 14 F; 9 with Palate repair: Simonart`s band) vomerine flap to and 32 from Bristol close anterior hard (23 M, 9 F; 7 with palate and von Simonart`s band) at Langenbeck for age 10 y posterior hard palate. Other surgery: alveolar bone grafting Bristol: lip repair: Millard with Muir or Veau type mucoperiosteal flaps and vomerine flap to repair the hard palate, nasal correction at the same time. Palate repair: 2 or 3 flap Veau type repair Other surgery: alveolar bone grafting n 5 23 (14 M, 9 F) Insufficient surgical from each center. details provided; Age range, 4-19 y some patients in the Swedish sample had received presurgical infant orthopedics and primary alveolar bone grafting n 5 84 M, older than Lip repair: Veau and 18 y Tennison-Randall. Palate repair: pushback, Schweckendiek. Other surgery: primary alveolar bone grafting, primary periosteoplasty n 5 25 (14 M, 11 F), Lip repair: Millard and mean age, 9.4 y, vomer flap, Skoog. Simonart`s band Palate repair: von included; compared Langenbeck, with Eurocleft study Wardill-Kilner. Other surgery: alveolar bone grafting at 9 y


Comparison group Comparative study between 2 centers (Frenchay Hospital, Bristol, United Kingdom, and the Rikshospitalet, Oslo, Norway), no control group

Intercenter comparison

Normal adult male volunteers reported in Smahel and Brejcha61 (1983)

Intercenter comparison


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Table I. Continued Author (year); study design 51

Ethnicity; region of sample


Smahel et al (1999); Crosssectional

White; Prague

5

Stoll et al52 (2002); Cross-sectional

White; Berlin

21

Bearn et al36 (2002); Cross-sectional

White; United Kingdon

Toygar et al15 (2004); Crosssectional

Turkish children

Dogan et al39 (2005); Crosssectional

White; Turkish children

22

Nollet et al53 (2008); longitudinal

White; Nijmegen

8


7 angles

10 lower lip measurements

UCLP sample

Surgery

Comparison group

No normative group n 5 30 M, mean age, Lip repair: Tennison (comparison 15.5 y; group 2 and primary between the 2 cleft without perioplasty. groups, with and repositioning of Palate repair: pushwithout nasal septum, n 5 back repositioning of 32 M, mean age, Other surgeries: nasal septum) 15.2 y pharyngeal flap, repositioning of nasal septum, corrective lip surgery n 5 58 (29 M, 29 F; n 5 84 (51 M, 33 F; Lip repair: Millard. mean age, 27 y), mean age, 20.9 y; Palate repair: Veauwhite subjects with range, 16-29 y) Rosenthal-Kriens. harmonious face in Other surgeries: terms of general velopharyngoplasty, perception of alveolar bone attractiveness; grafting complete dentition, including molar and a stable neutrocclusion in the canine and molar regions n 5 182 (ages, Surgical details not Published data from 12-14 y) provided, sample other studies taken from Clinical Standards Advisory Group Study (United Kingdom) n 5 24 (15 M, 9 F; Not described n 5 20 (M:F not mean age, 12.3 y) described; mean age, 12.3 y), harmonious profile and Class I neutral occlusion n 5 42 (20 M, 22 F; Lip repair: Millard. n 5 45 (21 M, 24 F; mean age, 13 y), Palate repair: von mean age, 15 y) almost all operations Langenbeck. done by same surgeon Descriptive data n 5 43 (27 M, 16 F), Lip repair: Millard; presented at 9, 12, ages, 9,12, and 18 y Soft palate repair: and 18 y; no modified von comparison group Langenbeck; Hard palate repair: delayed until 9-11 y. with alveolar bone grafting; Other surgeries: alveolar bone grafting, lip/nose correction (15-19 y)


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Table I. Continued Author (year); study design

Ethnicity; region of sample


Daskalogiannakis et al54 (2011); Cross-sectional

White; Americleft

7

This study; Longitudinal; comparison at ages 7, 11.1, and 17.9 y

White; Toronto

22

UCLP sample

Surgery

Comparison group

Intercenter comparison n 5 148 children, Lip repair: Millard, ages 8-9 y; Delaire, Tennison. n 5 18-40 for each Palate repair: center. Bardach, Delaire, M:F varying between Wardill Kilner, vomer 1:0.6 and 1:0.3 flap, von Langenbeck, Veau pushback, Furlow. n 5 70 (35 M, 35 F), Lip repair: rectangular n 5 70 (35 M, 35 F) comparable age and ages 7, 11.1, and or lower triangular sex from Burlington 17.9 y flaps or their Growth Center, modifications. Toronto Palate repair: pushback palatoplasty, von Langenbeck repair or their modifications

M, Male; F, female.

RESULTS

Table II. Age and sex details of the samples UCLP Time T1 (y) T2 (y) T3 (y)

Boys (n 5 35) 6.8 6 0.8 11.0 6 0.9 18.1 6 0.9

Girls (n 5 35) 7.3 6 0.7 11.3 6 1.1 17.5 6 1.6

Control Boys (n 5 35) 6.7 6 0.8 11.1 6 0.9 18.1 6 1.1

Girls (n 5 35) 7.1 6 0.8 11.3 6 1.0 17.7 6 1.5

relevant measurements from conventional analyses, with additional measurements specific for studying the softtissue profile, as demonstrated in Figure 1. No adjustment for radiographic magnification (9.66% in the radiographs from the Hospital for Sick Children and 9.84% in those from the Burlington Growth Center) was made. To estimate method error, a total of 45 cephalograms (15 radiographs from the UCLP and control groups at each of the 3 time points) were retraced and redigitized 8 weeks later. Intraclass correlation coefficients and Dahlberg's method24 of determining intraexaminer error (Table III) showed that the method errors were small, and the repeatability was excellent. Measurements between groups (UCLP and control) were compared at each time point using generalized linear models adjusted for age and sex effects. Longitudinal comparisons across all time points were conducted using the mixed model approach, adjusting for these effects and their interactions with time, to determine the effects of the group (UCLP and control) on growth differences.25 P \0.05 was considered significant.

Upper anterior face height was smaller in the UCLP group at T1 and T2 (Figs 2 and 3), but the differences decreased with adolescent growth so that they were not significant at T3 (Fig 4). However, lower anterior face height, which was not significantly different at T1, showed greater increments with growth and became distinctly longer in the UCLP group, especially during adolescence (Figs 3 and 4). The hard-tissue face height ratio remained 4% to 5% smaller in the UCLP group (Tables IV-VI). Although both jaws were relatively retrognathic in the UCLP group, the maxilla became particularly more retrognathic with growth, leading to a negative ANB angle at age 18 years (Fig 5, Table VI). This was despite the relatively larger mandibular plane angle (SN to GoGn) at all 3 times. The difference in the mandibular plane angle between the UCLP and control groups became progressively larger with growth. This angle was ultimately 5 larger in the UCLP group than in the control group at T3 (Fig 4, Table VI). The erupted height of the mandibular incisor to the mandibular border was not significantly different at any time point studied. The hard-tissue convexity (angle N-ANS-Me) was significantly larger in the UCLP group at T3 (Table VI). An overall growth effect was observed in the hardtissue measurements, with the differences between the groups generally increasing from T1 to T3. The longitudinal analysis of the hard-tissue measurements showed that overall, the growth changes from T1 to T3 were significantly smaller in the UCLP group for upper



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Fig 1. Definitions of specific landmarks and measurements. Landmarks: n0 , soft-tissue nasion; prn, pronasale; cm, midpoint of columella; sn0 , soft-tissue subnasale; A0 , soft-tissue A-point (superior labial sulcus); lab sup, labrale superius; sto sup, stomion superius; sto inf, stomion inferius; lab inf, labrale inferius; B0 , soft-tissue B-point (inferior labial sulcus); pg0 , soft-tissue pogonion; me0 , soft-tissue menton. Hard-tissue measurements: 1, anterior cranial base: s-n; 2, total cranial base: ba-n; 3, upper anterior face height: n-ANS; 4, lower anterior face height: ANS-me; 5, hard-tissue face height ratio: n-ANS/ ANS – me (ratio); 9, SN to GoGn: angle between s-n and go-gn; 10, hard-tissue convexity: angle between n-ANS-me; 11, maxillary incisor height 1 chin: distance from the mandibular incisor tip to go-gn. Soft-tissue measurements: 12, soft-tissue upper face height: vertical distance from n0 -sn0 ; 13, upper lip length: vertical distance from sn0 -sto sup; 14, soft-tissue lower face height: vertical distance from sn0 me0 ; 15, lower lip 1 soft-tissue chin height: vertical distance from sto inf-me0 ; 16, soft-tissue face height ratio: vertical distance from n0 -sn0 /vertical distance from sn0 -me0 (ratio); 17, upper lip:lower lip 1 chin: vertical distance from sn0 -sto sup/vertical distance from sto inf-me0 (ratio); 18, upper face height 1 upper lip: vertical distance from n0 -sto sup; 19, superior labial sulcus: distance from A0 to sn0 -lab sup; 20, inferior labial sulcus: distance of B0 to lab inf-pg0 ; 21, upper lip protrusion: distance from lab sup to sn0 pg0 ; 22, lower lip protrusion: distance from lab inf to sn0 -pg0 ; 23, lower lip projection: horizontal distance from lab inf to B measured parallel to the Frankfort horizontal plane; 24, ILS angle: angle between lab inf-B0 -pg0 ; 25, nose depth: distance from prn to n0 - sn0 ; 26, nose length: n0 -prn; 27, columella length: sn0 prn; 28, nasolabial angle: angle between lab sup-sn0 -cm; 29, columellar angle: angle between n0 -sn0 and sn0 -cm; 30, nose tip inclination: angle between sn0 -prm and Frankfort horizontal plane; 31, softtissue convexity without nose: angle between n0 -sn0 -pg0 ; 32, soft-tissue convexity with nose: angle between n0 -prn-pg0 ; 33, nasal tip convexity: angle between n0 -prn and sn0 -cm.

anterior face height, hard-tissue face-height ratio, and the SNA, SNB, and ANB angles, whereas the changes were greater for lower anterior face height and the mandibular plane angle. The soft-tissue upper face height was similar for the UCLP and the control groups at all 3 times (Figs 2-4, Tables IV-VI). The soft-tissue lower face height was significantly shorter in the UCLP group only at T1. When the upper and lower soft-tissue face heights were compared in the soft-tissue face height ratio, no significant differences were observed at any time. However, when the upper soft-tissue face height was


observed in combination with the length of the upper lip, it was significantly shorter in the UCLP group at T2. The upper lip length was smaller in the UCLP group at T2 and T3, and the magnitudes of difference were 8.3% at T2 and 5.3% at T3. The mean length of the lower lip and soft-tissue chin (measured together) was not significantly different in the UCLP group from the control group at T1 and T2 (Tables IV and V), but it was 2.21 mm longer at T3, and this difference was statistically significant (Table VI). The upper lip was relatively retruded throughout the growth period from T1 to T3, and the differences


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Table III. Intraexaminer repeatability analysis of measurements from 45 subjects Tracing 1 Measurement Hard tissues Anterior cranial base (mm) Total cranial base (mm) Upper anterior face height (mm) Lower anterior face height (mm) SNA ( ) SNB ( ) ANB ( ) SN to GoGn ( ) Hard-tissue convexity ( ) Maxillary incisor height 1 chin (mm) Soft tissues Upper anterior face height (mm) Upper lip length (mm) Lower soft-tissue face height (mm) Superior labial sulcus (mm) Inferior labial sulcus (mm) Upper lip protrusion (mm) Lower lip protrusion (mm) Lower lip projection (mm) Inferior labial sulcus angle ( ) Nose depth (mm) Nose length (mm) Columellar length (mm) Nasolabial angle ( ) Columellar angle ( ) Columellar inclination ( ) Soft-tissue convexity – nose ( ) Soft-tissue convexity 1 nose ( ) Nasal tip convexity ( )

Tracing 2

Mean

SD

Mean

SD

Intraexaminer measurement error

Intraexaminer ICC

P value

71.5 105.87 52.41 65.44 78.86 76.4 2.47 34.32 170.81 39.93

4.8 7.07 5.97 6.06 3.36 3.5 2.64 4.33 4.87 4.81

71.42 105.77 52.36 65.46 78.88 76.36 2.53 34.34 170.74 39.99

4.74 7.07 6.02 5.98 3.27 3.54 2.63 4.31 5.08 4.75

0.30 0.50 0.35 0.36 0.46 0.31 0.42 0.45 0.51 0.34

0.996 0.995 0.997 0.996 0.98 0.992 0.974 0.989 0.989 0.995

\0.001 \0.001 \0.001 \0.001 \0.001 \0.001 \0.001 \0.001 \0.001 \0.001

52.68 20.85 45.97 2.01 2.73 4.45 4.63 8.9 135.24 15.31 44.61 19.05 103.81 68.78 25 167.04 137.74 91.12

7.02 3.03 5.75 0.68 1.55 2.3 2.53 2.6 12.78 3.46 6.76 3.86 15.66 9.48 9.47 7.36 7.85 9.63

52.62 20.82 46.16 1.92 2.82 4.3 4.73 9.05 135.19 15.5 44.73 19.1 103.69 68.7 25.1 166.84 137.79 91.21

6.98 2.88 5.9 0.67 1.69 2.17 2.55 2.6 12.9 3.43 6.68 3.98 15.73 9.6 9.62 7.31 7.81 9.62

0.37 0.42 0.45 0.26 0.36 0.34 0.28 0.32 0.48 0.38 0.57 0.53 0.48 0.38 0.34 0.65 0.50 0.48

0.997 0.98 0.995 0.852 0.951 0.978 0.988 0.987 0.999 0.989 0.993 0.981 0.999 0.998 0.998 0.992 0.996 0.997

\0.001 \0.001 \0.001 \0.001 \0.001 \0.001 \0.001 \0.001 \0.001 \0.001 \0.001 \0.001 \0.001 \0.001 \0.001 \0.001 \0.001 \0.001

Fig 2. Superimposition of mean cephalometric tracings at T1 (on s-n registered at s).

became more apparent with growth (Tables IV-VI). The depth of the superior labial sulcus was not significantly different at any time. The lower lip, in contrast, was relatively more protruded in the UCLP group at all 3

times, and the protrusion increased with growth. The lower lip projection, which was measured as the horizontal distance from the soft-tissue B-point to labrale inferius, was not significantly different between the groups. The nose depth was smaller in the UCLP group at all 3 times by at least 1 mm, but nose length was not significantly different. Assessment of the soft-tissue profile convexity both with and without the nose showed larger angles in the UCLP group at all 3 times. These differences became larger with growth, indicating the progressive decrease in profile convexity during the 11-year growth period from ages 7 to 18 years (Tables IV-VII, Figs 2-6). Columellar length remained deficient in the UCLP group throughout the growth period, and the magnitude of the differences remained similar across the 3 time points. The columellar angle (measured between the soft-tissue nasion and softtissue subnasale plane and the plane of the columella) was larger in the UCLP group, whereas the nose tip inclination measured in relation to the Frankfort horizontal plane was markedly decreased, and the



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differences increased in magnitude from T1 to T3. The nasolabial angle became relatively smaller with growth, and the differences increased in magnitude from T1 to T3 (Tables IV-VII). DISCUSSION

One of the greatest challenges in treating patients with UCLP is to overcome the sequelae of the surgical scars and tissue deficiencies. Aberrant facial growth in patients with UCLP results from the long-term effects of the original defects in growth and patterning of the facial mesenchyme or epithelial fusion that led to clefting26 and the restrictive effects of the residual scar


tissue.27-29 These effects are readily visible in the appearance of the facial soft tissues. Imbalances in the nose, lips, and lower face have unfavorable impacts on the self-image and quality of life in people with clefts and facial deformities.7,8,29 Despite this, relatively fewer published studies have detailed soft-tissue facial growth compared with those that have detailed hardtissue facial growth in patients with UCLP. Our longitudinal study confirmed the well-recognized hard-tissue facial growth pattern in children with complete UCLP, including progressive maxillary retrognathism and decreasing profile convexity, particularly during adolescence. It also showed that the short upper anterior face height (hard tissue) in childhood in the UCLP group did not prevail during adolescence, but lower anterior face height became greater and clinically more apparent. This was compounded by the growth rotation of the mandible, which showed significantly less closure with growth in the UCLP group. These growth changes highlighted the vertical growth pattern seen in the UCLP group. Overlying these hard-tissue changes, the facial soft tissues exhibited many growth changes that were similar, and others that were remarkably different from those of the hard tissues. Whereas the hard-tissue face height ratio (upper anterior face height:lower anterior face height) was 4% to 5% smaller in the UCLP group, indicating a vertical skeletal growth pattern, the differences in the soft-tissue face height ratio were not statistically significant. However, when the lower face was viewed from the perspective of the upper lip length compared with the lengths of the lower lip and soft-tissue chin, a short upper lip and a long lower lip and chin were the major contributors to the soft-tissue imbalance in the lower face in the UCLP group. This imbalance was apparent by 11 years of age and persisted throughout adolescence to adulthood (Tables IV-VII). A short upper lip is a well-known feature of UCLP, and many factors can contribute to it. It has been described that the upper lip is short even before primary lip repair.30 However, lip repair and its resultant scar further impact the growth of the upper lip length, and this was indicated in our longitudinal data when the overall difference in growth from T1 through T3 was considered.27,28,31 Overall, the upper lip length increase from ages 7 to 18 years in the UCLP group was 59% of that in the control group. No subject had any secondary surgery or lip revision procedure that could affect lip length; thus, these results represent deficient upper lip growth because of a primary tissue deficiency and the long-term effects of primary lip repair surgery. We observed that the upper lip remained retruded in the UCLP sample throughout facial growth; this was similar to the findings of Semb.10 Chaisrisookumporn


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Table IV. Cephalometric comparison between the UCLP and control groups at T1 UCLP group (n 5 70) Measurement Hard tissues Anterior cranial base (mm) Total cranial base (mm) Upper anterior face height (mm) Lower anterior face height (mm) Hard-tissue face height ratio SNA ( ) SNB ( ) ANB ( ) SN to GoGn ( ) Hard-tissue convexity ( ) Mandibular incisor height 1 chin (mm) Soft tissues Upper soft-tissue face height (mm) Upper lip length (mm) Lower soft-tissue face height (mm) Lower lip 1 soft-tissue chin (mm) Soft-tissue face height ratio Upper lip:lower lip 1 soft-tissue chin (mm) Upper face height.1 upper lip length (mm) Superior labial sulcus (mm) Inferior labial sulcus (mm) Upper lip protrusion (mm) Lower lip protrusion (mm) Lower lip projection (mm) Inferior labial sulcus angle ( ) Nose depth (mm) Nose length (mm) Columellar length (mm) Nasolabial angle ( ) Columellar angle ( ) Nose tip inclination ( ) Soft-tissue convexity – nose ( ) Soft-tissue convexity 1 nose ( ) Nasal tip convexity ( )

Control group (n 5 70)

Estimated difference (UCLP – control)

Adjusted mean

95% CI

Adjusted mean

95% CI

Mean

SE

P value

66.82 97.51 44.46 61.21 0.73 78.93 74.57 4.36 36.65 166.29 35.64

(66.14, 67.51) (96.56, 98.45) (43.85, 45.06) (60.14, 62.29) (0.72, 0.75) (78.08, 79.78) (73.77, 75.38) (3.73, 4.98) (35.57, 37.72) (164.91, 167.68) (35.07, 36.2)

67.59 98.71 46.38 59.96 0.78 80.34 76.71 3.64 33.89 167.63 35.29

(66.9, 68.27) (97.77, 99.66) (45.77, 46.98) (58.88, 61.04) (0.76, 0.79) (79.49, 81.2) (75.91, 77.52) (3.01, 4.27) (32.81, 34.96) (166.25, 169.02) (34.73, 35.86)

0.77 1.21 1.92 1.25 0.04 1.41 2.14 0.71 2.76 1.34 0.34

0.49 0.68 0.43 0.77 0.01 0.61 0.58 0.45 0.77 0.99 0.40

0.121 0.077 \0.0001 0.108 0.000 0.022 0.000 0.115 0.001 0.178 0.396

(44.28, 45.85) (19.3, 20.6) (59.9, 62.04) (40.13, 41.91) (0.73, 0.76) (0.47, 0.51)

45.44 20.47 62.53 42.06 0.73 0.49

(44.65, 46.22) (19.82, 21.12) (61.46, 63.6) (41.17, 42.95) (0.71, 0.75) (0.47, 0.51)

0.38 0.52 1.56 1.04 0.01 0.00

0.56 0.47 0.77 0.64 0.01 0.01

0.505 0.263 0.043 0.104 0.280 0.796

65.01 (63.99, 66.03)

65.91

(64.89, 66.93)

0.90

0.73

0.220

(1.34, 1.6) (3.67, 4.19) (4.28, 5.07) (3.02, 4.01) (7.21, 8.18) (136.94, 142.89) (12.61, 13.31) (37.13, 38.7) (16.11, 16.97) (106.07, 112.1) (65.18, 69.24) (25.97, 30.21) (161.7, 164.74) (133.34, 135.63) (90.58, 94.72)

0.15 0.06 1.44 1.07 0.33 2.71 1.26 0.31 1.83 0.80 2.82 6.44 4.67 6.14 0.59

0.10 0.19 0.28 0.36 0.34 2.13 0.25 0.56 0.31 2.16 1.45 1.52 1.09 0.82 1.48

0.129 0.757 \0.0001 0.003 0.341 0.205 \0.0001 0.584 \0.0001 0.711 0.055 \0.0001 \0.0001 \0.0001 0.690

45.06 19.95 60.97 41.02 0.74 0.49

1.32 3.99 3.24 4.59 8.03 137.20 11.70 38.22 14.71 108.29 70.03 21.65 167.89 140.63 92.06

(1.19, 1.46) (3.72, 4.25) (2.84, 3.63) (4.09, 5.09) (7.54, 8.51) (134.23, 140.18) (11.35, 12.04) (37.44, 39.01) (14.28, 15.14) (105.27, 111.3) (68, 72.05) (19.53, 23.77) (166.37, 169.41) (139.48, 141.77) (89.99, 94.12)

1.47 3.93 4.67 3.52 7.70 139.92 12.96 37.92 16.54 109.09 67.21 28.09 163.22 134.49 92.65

Sig

z z * z z

*

z y z z z z z

Sig, Significance. *Significant; yhighly significant; zvery highly significant.

et al,32 however, demonstrated protrusion of the upper lip relative to a perpendicular dropped on the Frankfort horizontal plane from the soft-tissue subnasale in a sample of 20 subjects with UCLP. Their sample was cross-sectional and smaller, and it had a wide age range (12-42 years). In our study, the control group comprised normally growing skeletal Class I patients, who had not received fixed or functional orthodontic or orthopedic treatment, whereas all patients in the UCLP group had received orthodontic treatment, which could also have affected the upper and lower lip positions at T3. Furthermore, upper lip protrusion was also affected by the underlying relative maxillary retrognathism noted in the

UCLP group. Retrusion of the upper lip has been reported to improve after maxillary advancement in patients with UCLP.33 Although long-term data are presented in this report, less importance should be given to the measurements of upper lip protrusion at T1 (age 7 years), since many subjects in both groups were in a transitional stage of incisor development, and their maxillary permanent incisors had not erupted into their final positions. The lower lips in patients with clefts have been the subject of significant controversy. Some investigators have found shorter lower lips,15 whereas others have reported longer lower lips.11 In our study, the lower lip was



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Table V. Cephalometric comparison between the UCLP and control groups at T2 UCLP group (n 5 70) Measurement Hard tissues Anterior cranial base (mm) Total cranial base (mm) Upper anterior face height (mm) Lower anterior face height (mm) Hard-tissue face height ratio SNA ( ) SNB ( ) ANB ( ) SN to GoGn ( ) Hard-tissue convexity ( ) Maxillary incisor height 1 chin (mm) Soft tissues Upper soft-tissue face height (mm) Upper lip length (mm) Lower soft-tissue face height (mm) Lower lip 1 soft-tissue chin (mm) Soft-tissue face height ratio Upper lip:lower lip 1 soft-tissue chin (mm) Upper face height 1 upper lip length (mm) Superior labial sulcus (mm) Inferior labial sulcus (mm) Upper lip protrusion (mm) Lower lip protrusion (mm) Lower lip projection (mm) Inferior labial sulcus angle ( ) Nose depth (mm) Nose length (mm) Columellar length (mm) Nasolabial angle ( ) Columellar angle ( ) Nose tip inclination ( ) ST convexity – nose ( ) ST convexity 1 nose ( ) Nasal tip convexity ( )



Adjusted mean

95% CI

Adjusted mean

95% CI

Mean

SE

P value

Sig

70.26 104.11 50.38 65.23 0.78 76.19 74.48 1.72 36.60 171.18 39.19

(69.56, 70.97) (103.11, 105.11) (49.73, 51.03) (64.14, 66.33) (0.76, 0.79) (75.3, 77.08) (73.71, 75.24) (1.14, 2.29) (35.49, 37.71) (169.8, 172.55) (38.59, 39.78)

71.26 105.62 51.97 63.58 0.82 80.36 77.49 2.87 32.88 170.49 38.96

(70.56, 71.97) (104.62, 106.62) (51.31, 52.62) (62.48, 64.67) (0.8, 0.84) (79.47, 81.25) (76.72, 78.25) (2.3, 3.44) (31.77, 33.99) (169.12, 171.87) (38.36, 39.55)

1.00 1.51 1.58 1.66 0.04 4.16 3.01 1.15 3.72 0.68 0.23

0.51 0.71 0.47 0.78 0.01 0.64 0.55 0.41 0.79 0.98 0.43

0.049 0.036 0.001 0.036 0.000 \0.0001 \0.0001 0.006 \0.0001 0.488 0.589

* * z * z z z y z

51.02 21.84 67.14 45.30 0.76 0.49

(50.12, 51.92) (21.17, 22.51) (66, 68.28) (44.43, 46.17) (0.74, 0.78) (0.47, 0.5)

0.18 1.81 1.52 0.29 0.02 0.04

0.64 0.48 0.81 0.62 0.01 0.01

0.777 0.000 0.065 0.638 0.276 0.000

72.86

(71.82, 73.89)

1.99

0.74

0.008

(1.53, 1.84) (4.57, 5.13) (4.22, 5.11) (3.26, 4.28) (8.12, 9.08) (133.28, 138.54) (14.8, 15.65) (42.41, 44.31) (18.38, 19.38) (107.54, 113.14) (66.22, 69.94) (27.22, 31.22) (161.47, 164.27) (131.58, 133.94) (89.35, 93.12)

0.21 0.25 1.66 1.40 0.22 1.12 1.30 0.30 1.89 5.00 2.84 9.34 8.13 8.31 0.84

0.11 0.20 0.32 0.36 0.34 1.88 0.30 0.68 0.36 2.00 1.33 1.43 1.00 0.84 1.35

0.059 0.214 \0.0001 0.000 0.515 0.554 \0.0001 0.663 \0.0001 0.014 0.035 \0.0001 \0.0001 \0.0001 0.534

50.84 20.03 65.62 45.59 0.78 0.44

(49.94, 51.74) (19.36, 20.7) (64.48, 66.76) (44.73, 46.46) (0.76, 0.8) (0.43, 0.46)

70.86 (69.83, 71.9) 1.47 4.60 3.00 5.17 8.82 137.03 13.92 43.65 16.99 105.34 70.92 19.89 171.00 141.06 90.39

(1.32, 1.63) (4.32, 4.88) (2.55, 3.45) (4.67, 5.68) (8.35, 9.3) (134.4, 139.65) (13.49, 14.34) (42.7, 44.61) (16.49, 17.49) (102.54, 108.14) (69.06, 72.78) (17.88, 21.89) (169.6, 172.4) (139.88, 142.24) (88.51, 92.28)

1.68 4.85 4.66 3.77 8.60 135.91 15.22 43.36 18.88 110.34 68.08 29.22 162.87 132.76 91.23

z

z y

z z z z * * z z z


longer in the UCLP group by 2 mm at T3, with the most significant changes and differences occurring during adolescence. This could be the result of compensatory soft-tissue growth to achieve lip competence. Relative to the control group, the lower lips in the UCLP group became progressively longer and relatively more protruded than the upper lips with adolescent growth; this was similar to the findings of Pensler and Mulliken,12 Zhu et al,13 and Chaisrisookumporn et al.32 Observing the interrelationship of the upper and lower lips, it was clear that in the UCLP group, while the upper lip continued to become less prominent, the lower lip became more prominent with growth. The inferior labial


sulcus depth was not significantly different at T1 or T2, and although it was greater in the UCLP group at T3, the difference was only 0.5 mm. The lower lip projection was not significantly different at any time. The ILS angle (measured to assess, in association with lower lip projection, whether the lower lip was everted) had no significant differences at T1 and T2 but was 5 smaller than that of the controls at T3. These measurements indicated that gross lower lip eversion or redundancy was not common in this sample, although there was a tendency toward this effect during adolescent growth. The imbalance in the relative protrusions of the upper and lower lips created the characteristic step-like relationship in


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Table VI. Cephalometric comparison between the UCLP and control groups at T3 UCLP group (n 5 70) Measurement Hard tissues Anterior cranial base (mm) Total cranial base (mm) Upper anterior face height (mm) Lower anterior face height (mm) Hard-tissue face height ratio SNA ( ) SNB ( ) ANB ( ) SN to GoGn ( ) Hard-tissue convexity ( ) Mandibular incisor height 1 chin (mm) Soft tissues Upper soft-tissue face height (mm) Upper lip length (mm) Lower soft-tissue face height (mm) Lower lip 1 soft-tissue chin (mm) Soft-tissue face height ratio Upper lip:lower lip 1 soft-tissue chin (mm) Upper face height 1 upper lip length (mm) Superior labial sulcus (mm) Inferior labial sulcus (mm) Upper lip protrusion (mm) Lower lip protrusion (mm) Lower lip projection (mm) Inferior labial sulcus angle ( ) Nose depth (mm) Nose length (mm) Columellar length (mm) Nasolabial angle ( ) Columellar angle ( ) Nose tip inclination ( ) Soft-tissue convexity – nose ( ) Soft-tissue convexity 1 nose ( ) Nasal tip convexity ( )



Adjusted mean

95% CI

Adjusted mean

95% CI

Mean

SE

P value

Sig

74.37 111.08 56.16 72.60 0.78 74.52 75.38 -0.86 35.50 176.72 43.44

(73.52, 75.22) (110.01, 112.15) (55.39, 56.93) (71.42, 73.79) (0.76, 0.79) (73.58, 75.47) (74.55, 76.2) (-1.51, -0.21) (34.34, 36.66) (175.04, 178.39) (42.72, 44.15)

75.85 112.34 57.06 69.20 0.83 80.81 78.97 1.84 30.36 173.57 42.81

(75, 76.7) (111.27, 113.41) (56.29, 57.83) (68.02, 70.39) (0.81, 0.84) (79.87, 81.76) (78.15, 79.8) (1.19, 2.49) (29.19, 31.52) (171.9, 175.25) (42.09, 43.53)

1.48 1.26 0.91 3.40 0.05 6.29 3.60 2.69 5.15 3.15 0.63

0.61 0.76 0.55 0.85 0.01 0.67 0.59 0.46 0.83 1.20 0.51

0.016 0.103 0.102 0.000 \0.0001 \0.0001 \0.0001 \0.0001 \0.0001 0.010 0.222

*

(56.27, 58.31) (20.2, 21.54) (72.69, 75.27) (52.09, 54.14) (0.76, 0.8) (0.38, 0.41)

57.51 22.03 72.93 50.91 0.79 0.44

(56.49, 58.53) (21.35, 22.7) (71.64, 74.23) (49.89, 51.93) (0.77, 0.81) (0.42, 0.45)

0.22 1.16 1.05 2.21 0.01 0.04

0.73 0.48 0.92 0.73 0.01 0.01

0.762 0.018 0.258 0.003 0.436 0.000

78.16 (77.04, 79.28)

79.54

(78.42, 80.66)

1.38

0.80

0.087

0.04 0.53 1.86 1.62 0.52 5.22 1.12 0.41 1.89 9.65 5.03 12.41 11.26 10.26 3.43

0.13 0.24 0.34 0.42 0.40 2.03 0.37 0.73 0.45 2.12 1.41 1.51 1.12 0.96 1.38

0.750 0.027 \0.0001 0.000 0.198 0.011 0.003 0.570 \0.0001 \0.0001 0.001 \0.0001 \0.0001 \0.0001 0.014

57.29 20.87 73.98 53.11 0.78 0.39

2.03 5.92 2.17 5.03 9.30 130.12 17.49 50.22 20.45 100.36 74.93 16.07 175.42 140.94 84.61

(1.85, 2.21) (5.59, 6.25) (1.69, 2.65) (4.45, 5.61) (8.74, 9.86) (127.29, 132.96) (16.97, 18.01) (49.2, 51.24) (19.82, 21.08) (97.39, 103.33) (72.96, 76.9) (13.96, 18.18) (173.85, 176.98) (139.6, 142.28) (82.68, 86.54)

1.99 5.38 4.02 3.41 8.79 135.35 18.61 49.80 22.34 110.01 69.90 28.48 164.15 130.68 88.04

(1.8, 2.17) (5.05, 5.72) (3.55, 4.5) (2.83, 3.99) (8.23, 9.34) (132.51, 138.18) (18.09, 19.13) (48.78, 50.82) (21.71, 22.97) (107.04, 112.98) (67.93, 71.87) (26.37, 30.59) (162.59, 165.71) (129.35, 132.02) (86.11, 89.97)

z z z z z z y

* y z

* z z * y z z y z z z *


lip profiles, in contrast to the relative prominence of the upper lip in the control group (Figs 5 and 6). According to Pensler and Mulliken, the typical postural relationship of the lips in patients with UCLP can be detected immediately after lip repair in the first year of life and seen to progress in severity with age. More recently, Toygar et al,15 on the contrary, found that the lower lips of UCLP patients were significantly smaller, retruded, and curved, with a deep labiomental sulcus when compared with skeletal Class I subjects at approximately 12 years of age. When observing the longitudinal relationship of the lips, our data showed that the sagittal relationship between the upper and lower lips was affected in the

UCLP group even at T1 (7 years). At T1, the upper lip was still slightly prominent compared with the lower lip, although not as much as in the control group. At T2 (11.1 years), the relative prominence of the upper lip had disappeared; by T3 (17.9 years), a step relationship was visible, with the lower lip relatively more prominent than the upper lip (Fig 6). Another feature that persists as an esthetic challenge and has psychosocial effects in patients with clefts is the profile appearance of the nose. In any analysis, individual measurements provide a quantitative breakdown of a structure into various constituent dimensions, although the structure itself remains a composite of the different



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Fig 5. Longitudinal growth in the UCLP and control groups from T1 (black) to T2 (green) to T3 (red) by serial superimpositions of mean cephalometric tracings (on s-n registered at s).

measurements. Thus, greater emphasis should be placed on interpretations of regional measurements collectively rather than individually. This is especially relevant in the description of the nose. Our data showed that although both nasal depth and columella length were deficient in the UCLP sample, the length of the nose was not significantly different at any time. The nasal tip convexity measured in the UCLP group was smaller than in the control group, especially at T3, and this was most likely a long-term effect of the lip scar, columellar deficiency, and decreased columellar inclination. The nose depth in the UCLP group was 9.7% smaller at T1, 8.6% smaller at T2, and 6.0% smaller at T3. Considering all the nasal and columellar measurements (Tables IV-VII), attenuation of nasal cartilage growth was evident in the UCLP group.34,35 It appeared that the basilar nasal cartilages, attached to the retrognathic maxilla, and the lip scar might have led to a band-like effect throughout growth that also contributed to the downward rotation of the nasal tip and the columella. Facial convexity, when measured without the nose, is primarily influenced by maxillary retrusion, and the facial profile became flatter with growth in the UCLP group. The magnitude of differences increased from T1 to T3, and this was the result of the progressive maxillary retrognathism in relation to mandibular growth. The nasal tip convexity angle was smaller, affected by the markedly decreased nose tip inclination. When combined with the deficiencies in nasal depth and columellar length, it led to the characteristic nasal deformity viewed from the profile. This altered growth


pattern of the nose was already established by 11 years of age, but the downward rotations of the nasal tip and the columella became more severe during the adolescent years, when nasal septum growth is active.34 These nasal features, in association with a short and retruded upper lip and a reduced nasolabial angle, gave the UCLP group the characteristic midfacial profile appearance (Fig 6). Bearn et al36 also described that the low values of the nasolabial angle in patients with clefts led to the appearance of “hooked noses.” Decreased nose base angle and nasolabial angle have been found to be less esthetic, in both patients with repaired clefts and unaffected persons.37 Our comprehensive results also corroborate some findings reported in earlier studies.10,11,32,38,39 The clinical implications of these findings are clear. On average, it was seen that many soft-tissue profile features that were characteristically different in the UCLP group were established as early as 7 years, although they increased in severity during the adolescent period from 11 to 18 years. This is important to consider, since patients with clefts report higher incidences of teasing, and the greatest incidence is reported during this period.20-22 The psychological implications of the cleft deformity are greater in adolescence than in the preadolescent period.40 This is also the time when surgeons and orthodontists often confront the question of the timing of nasal revision surgery. Although the data have shown that profile differences in this region, such as nose tip inclination and nasolabial angle, which were established by 11 years of age, generally became more severe with adolescent facial growth, some


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Table VII. Cephalometric longitudinal comparisons across T1, T2, and T3 Difference in growth (UCLP – control) T1 vs T2 Measurement Hard tissues Anterior cranial base (mm) Total cranial base (mm) Upper anterior face height (mm) Lower anterior face height (mm) Hard-tissue face height ratio SNA ( ) SNB ( ) ANB ( ) SN to GoGn ( ) Hard-tissue convexity ( ) Mandibular incisor height 1 chin (mm) Soft tissues Upper soft-tissue face height (mm) Upper lip length (mm) Lower soft-tissue face height (mm) Lower lip 1 soft-tissue chin (mm) Soft-tissue face height ratio Upper lip:lower lip 1 soft-tissue chin (mm) Upper face height 1 upper lip length (mm) Superior labial sulcus (mm) Inferior labial sulcus (mm) Upper lip protrusion (mm) Lower lip protrusion (mm) Lower lip projection (mm) Inferior labial sulcus angle ( ) Nose depth (mm) Nose length (mm) Columellar length (mm) Nasolabial angle ( ) Columellar angle ( ) Nose tip inclination ( ) Soft-tissue convexity – nose ( ) Soft-tissue convexity 1 nose ( ) Nasal tip convexity ( )

EMD

SE

P value

0.23 0.36 0.23 0.38 0.00 2.68 0.80 1.86 0.87 1.99 0.17

0.21 0.274 0.51 0.482 0.36 0.523 0.58 0.512 0.01 0.939 0.34 \0.0001 0.31 0.012 0.35 \0.0001 0.46 0.062 0.78 0.012 0.30 0.579

0.04 1.25 0.13 1.40 0.00 0.05 1.23 0.05 0.30 0.20 0.35 0.09 3.70 0.06 0.17 0.07 4.29 0.07 2.83 3.47 2.16 0.22

0.63 0.949 0.43 0.004 0.60 0.825 0.59 0.019 0.01 0.851 0.01 0.001 0.67 0.070 0.11 0.640 0.21 0.168 0.28 0.465 0.34 0.312 0.33 0.780 2.20 0.095 0.24 0.812 0.64 0.796 0.36 0.838 2.05 0.038 1.52 0.966 1.55 0.071 0.78 \0.0001 0.61 0.001 1.44 0.879

T2 vs T3 Sig

z * z *

y * z

*

z z

EMD

SE

Overall

P value

0.39 0.45 0.79 1.77 0.00 2.11 0.54 1.57 1.32 2.50 0.46

0.39 0.309 0.66 0.499 0.48 0.102 0.56 0.002 0.01 0.615 0.38 \0.0001 0.37 0.143 0.38 \0.0001 0.49 0.008 0.86 0.004 0.30 0.126

0.16 0.57 2.58 2.01 0.03 0.00 0.73 0.25 0.80 0.21 0.22 0.29 6.43 0.23 0.29 0.06 4.58 2.12 2.97 3.12 1.93 2.51

0.67 0.41 0.66 0.59 0.01 0.01 0.69 0.12 0.23 0.31 0.34 0.33 1.98 0.35 0.65 0.42 1.85 1.35 1.42 0.82 0.67 1.33

0.814 0.167 0.000 0.001 0.034 0.977 0.294 0.043 0.001 0.502 0.525 0.376 0.002 0.507 0.658 0.885 0.015 0.117 0.039 0.000 0.004 0.060

Sig

y z z y y

z z *

* y

y

* * z y

EMD

SE

P value

Sig

1.06 1.27 1.43 2.10 0.04 3.98 2.92 1.06 3.86 0.87 0.41

0.54 0.67 0.43 0.73 0.01 0.60 0.53 0.38 0.75 0.92 0.43

0.051 0.059 0.001 0.005 \0.0001 \0.0001 \0.0001 0.005 \0.0001 0.346 0.342

y y z z z y z

0.21 1.19 0.71 0.48 0.01 0.03 1.40 0.11 0.10 1.68 1.34 0.34 2.15 1.23 0.41 1.88 5.07 3.61 9.44 8.02 8.25 1.64

0.56 0.41 0.75 0.59 0.01 0.01 0.66 0.09 0.16 0.26 0.31 0.30 1.58 0.26 0.57 0.30 1.72 1.08 1.18 0.94 0.78 1.11

0.715 0.004 0.346 0.416 0.547 0.006 0.035 0.209 0.523 \0.0001 \0.0001 0.256 0.175 \0.0001 0.475 \0.0001 0.004 0.001 \0.0001 \0.0001 \0.0001 0.144

y

y *

z z z z y y z z z

EMD, Estimated mean difference; Sig, significance. *Significant; yhighly significant; zvery highly significant.

measurements, such as nose depth and columellar length, on average, did not worsen significantly during this time, although they still remained significantly deficient at T3. The period of active nasal septum growth during adolescence did not lead to a clinically significant catch-up in these measurements to normal values.34 Keeping this in mind, decisions regarding the timing of nasal revision should be made based on the severity of the deformity and needs of each patient. The strengths and limits of these data should be discussed, as should several aspects of our study design. Access to the archives of one of the largest craniofacial centers in the world allowed us to collect a unique and large longitudinal sample in which both sexes were

equally represented. All subjects included in the UCLP sample had received surgical repairs at the hospital, according to well-defined protocols. We aimed to study the soft-tissue profile differences and investigate the longitudinal differences in growth of the soft-tissue profile in growing children with UCLP in general, and not to evaluate the outcome of one surgical protocol or the results of one surgeon with another. Using the generalized linear models adjusted for age and sex effects for comparing the UCLP group with the control group at each time point was preferred over the use of a t test because it allowed adjusting the analysis for age and sex and their interactions. Longitudinal comparisons across all time points were conducted with the mixed



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Fig 6. Longitudinal soft-tissue profile differences between the UCLP and control groups shown by superimposition of mean cephalometric tracings on n0 -pg0 (UCLP group, red; control group, black).

model approach, adjusting for these effects and their interactions with time, to determine the effects of the group (UCLP and control) on growth differences. In addition to adjusting for the effects of age and sex, the mixed model approach took into account both between- and within-individual variations over time. Since we did not aim to study the effect of surgical techniques, we did not include types of lip and palate surgery or surgeon as variables in the analysis. The longitudinal analysis allowed observing the changes in differences over time and accounting for individual subject variations. The results described here should therefore be considered a general representation of the typical long-term profile characteristics in children with complete UCLP after the types of reparative surgeries mentioned earlier that were conducted at the hospital. Inclusion of subjects who had received no prelip-repair nasal molding or any primary nasal septum surgery or any secondary surgeries ensured that our data would not reflect changes in profile and growth that could potentially occur after such primary ancillary procedures or surgeries, or any secondary lip or nasal revisions or alveolar bone grafting. Therefore, our results can be used for future studies exploring potential long-term effects of such ancillary procedures. Although the control group was selected to be comparable with the UCLP group, because of the relatively large number of subjects, the analysis of soft-tissue measurements in this normative group with Class I skeletal growth can be used as a


reference for studies evaluating treatment outcomes. Despite these strengths, the well-recognized limitations of 2-dimensional conventional cephalometrics should be considered when evaluating our results.41 Also, there are several methods of superimposing morphometric data related to growth and change, and our method of superimpositions of mean cephalometric tracings, on both the anterior cranial base and the soft-tissue profile (N0 -Pg0 ), should only be viewed as a visual representation of the mean data detailed in Tables IV through VI. The superimpositions did not affect the data or analyses shown in Tables IV through VI. Finally, the choice of a control group can be debated relentlessly in any comparative study. Although the normal population includes a range of growth patterns, and patients with clefts would similarly belong to such a group, upon which the effects of the cleft, the effects related to its primary cause, and its treatment would be superimposed, we chose to use a group with skeletal Class I growth of comparable age, sex, and ethnicity as the control group for homogeneity. Despite these clear strengths, the potential limitations of a normative control group should be kept in mind when interpreting the results. CONCLUSIONS

Key attributes of the imbalance in the soft-tissue profiles in children with repaired UCLP were identified in the


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lip and nose regions. When compared with their skeletal Class I control peers, many profile differences were visible as early as 7 years of age, but the most remarkable soft-tissue profile imbalance appeared by 11 years and increased in magnitude during adolescent growth. A short, retruded upper lip and, conversely, a longer and more protruded lower lip were the characteristic lip features in the UCLP group, and a step relationship developed between the upper and lower lips during adolescent facial growth. Nose depth and columellar length were smaller in the UCLP group at 7, 11, and 18 years of age, and the nose tip rotated downward with growth from 11 to 18 years. ACKNOWLEDGMENTS

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Preoperative Cleft Lip Measurements and Maxillary Growth in Patients With Unilateral Cleft Lip and Palate.

Cranial base morphology in cleft lip and palate: a cephalometric study from 7 to 18 years of age.

Presurgical cleft lip anthropometrics and dental arch relationships in patients with complete unilateral cleft lip and palate.

Influence of different palate repair protocols on facial growth in unilateral complete cleft lip and palate.

Nasal Airway Dimensions of Children With Repaired Unilateral Cleft Lip and Palate.

or palate and controls.

Long-term mid-facial growth of patients with a unilateral complete cleft of lip, alveolus and palate treated by two-stage palatoplasty: cephalometric analysis.

The Asher-McDade Aesthetic Index in Comparison With Two Scoring Systems in Nonsyndromic Complete Unilateral Cleft Lip and Palate Patients.

Improving the facial profile of a girl born with a right unilateral cleft lip and palate.

Nasoalveolar moulding for children with unilateral cleft lip and palate.

Occlusal Classification in Relation to Original Cleft Width in Patients With Unilateral Cleft Lip and Palate.

Speech outcome in complete unilateral cleft lip and palate - a comparison of three methods of the hard palate closure.

Effects of lip repair on maxillofacial morphology in patients with unilateral cleft lip with or without cleft palate.

Dental arch relationship outcomes in one- and two-stage palatoplasty for Japanese patients with complete unilateral cleft lip and palate.

Auditory complaints and audiologic assessment in children with surgically repaired cleft lip and palate.

A study of facial growth in patients with unilateral cleft lip and palate treated by the Oslo CLP Team.

Longitudinal study of growth of children with unilateral cleft lip and palate: 2 to 10 years of age.

Vertical maxillary growth after two different surgical protocols in unilateral cleft lip and palate patients.

Pre-surgical Alveolar Molding in A Newborn Patient with Complete Unilateral Cleft Lip and Palate-A Report.

Skeletal stability after Le Fort I maxillary advancement in patients with unilateral cleft lip and palate.

Development of permanent tooth length in patients with unilateral cleft lip and palate.

Alterations in tympanic membrane appearance and middle ear function in 11-year-old children with complete unilateral cleft lip and palate compared with healthy age-matched subjects.

A Comparative Study of Oral Microbiota in Infants with Complete Cleft Lip and Palate or Cleft Soft Palate.

The Americleft Project: A Proposed Expanded Nasolabial Appearance Yardstick for 5- to 7-Year-Old Patients With Complete Unilateral Cleft Lip and Palate (CUCLP).