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Midfacial morphology in children with unilateral cleft lip and palate treated by different surgical protocols
Int. J. Oral Maxillofac. Surg. 2002; 31: 13–22 doi:10.1054/ijom.2001.0177, available online at http://www.idealibrary.com on
Leading clinical paper: Congenital deformities
Midfacial morphology in children with unilateral cleft lip and palate treated by different surgical protocols
G. Swennen1, J. L. Berten2, H. Schliephake3, C. Treutlein2, R. Dempf1, C. Malevez4, A. De Mey5 1
Department of Oral and Maxillofacial Surgery; Department of Orthodontics, Medical University, Hannover, Carl-Neuberg-Strasse 1, 30625 Hannover, Germany; 3Department of Oral and Maxillofacial Surgery; Georg-August-University, Go¨ttingen, Robert-Koch-Strasse 40, 37075 Go¨ttingen, Germany; 4Department of Oral and Maxillofacial Surgery; 5Plastic and Reconstructive Surgery, University of Brussels (ULB), Children’s Hospital, Avenue J.J. Crocq 15, 1020 Brussels, Belgium 2
G. Swennen, J. L. Berten, H. Schliephake, C. Treutlein, R. Dempf, C. Malevez, A. De Mey: Midfacial morphology in children with unilateral cleft lip and palate treated by different surgical protocols. Int. J. Oral Maxillofac. Surg. 2002; 31: 13–22. 2002 International Association of Oral and Maxillofacial Surgeons Abstract. The purpose of this study was to compare cranio-facial, particularly midfacial morphology, in two groups of children with complete unilateral cleft lip and palate (UCLP) treated at two different cleft centres (Hannover, Germany and Brussels, Belgium) following different surgical treatment protocols. A total of 62 children (40 males; 22 females) with non-syndromic UCLP were included in this study at approximately the age of 10. The Hannover group comprised 36 children, who had repair of the lip at a mean age of 5.831.16 months, followed by repair of the hard and soft palate at a mean age of 29.084.68 and 32.254.29 months, respectively. The Brussels group consisted of 26 children who underwent surgical treatment according to the Malek protocol: the soft palate was closed at a mean age of 3.040.20 months, followed by simultaneous repair of the lip and hard palate at a mean age of 6.150.68 months. Midfacial morphology was evaluated by means of cephalometric analysis according to Ross. The children in the Hannover UCLP group did not differ significantly from those in the Brussels group in the anteroposterior dimension of the midface. However, the maxillary plane was significantly more open in the Brussels group due to less posterior vertical maxillary development.
Introduction Many different surgical treatment protocols and adjunctive orthopaedic/ orthodontic procedures have been proposed to treat children with unilateral cleft lip and palate (UCLP)11,21,27. S & S24 reported in 1998 that the register of the Eurocleft Biomed II Project, although incomplete at the time of publication, consisted of 178 cleft 0901-5027/02/010013+10 $35.00/0
teams with 171 different treatment protocols for treating patients with UCLP alone. All those treatment protocols vary from each other through variations and modifications regarding surgical technique, timing, sequence and the application of presurgical orthopaedic and/or adjunctive orthodontic procedures. Evaluation of treatment outcomes achieved by different cleft centres is essential in cleft lip–palate research.
Key words: cleft lip palate; cranio-facial morphology; cephalometry. Accepted for publication 1 October 2001 Published online 7 January 2002
Although the literature provides many reports on cranio-facial morphology in patients with cleft disorders, only few intercentre comparative studies have been carried out in an attempt to elucidate the effects of different treatments on cranio-facial morphology7,8,16,17,20,29. The six-centre study of treatment outcome conducted by the European Cleft Lip and Palate Research Group1,15,16,25,26 and the multicentre
2002 International Association of Oral and Maxillofacial Surgeons
14
Swennen et al. Hannover Lip Hard palate Soft palate
40.00
32.62
32.04
30.00
29.54
Months
28.83
20.00
10.00 5.96
0.00
5.62
Males
Females
Fig. 1. Data on primary surgery for the children in the Hannover UCLP group.
study performed by R19 have added a major contribution to the literature on cleft lip–palate research. However, comparison of cephalometric data with the published results from these multicentre studies is scientifically questionable due to potential interobserver error19. Hence, the purpose of this study was to compare cranio-facial, particularly midfacial morphology in children with UCLP, who were treated at two different cleft centres following different surgical protocols, to a non-cleft control group.
Material and methods The Cleft databases from the Department of OMF Surgery, Medical University, Hannover, Germany, and the Department of OMF, Plastic and Reconstructive Surgery, Children’s Hospital, Brussels University (ULB), Belgium, were consulted for children who met the following inclusion criteria: 1. Complete unilateral cleft lip, alveolus and palate. Patients with Simonart’s bands were included 2. Consecutive cases 3. No associated syndrome or mental retardation 4. Caucasian ethnicity 5. Presurgical infant orthopaedics with passive removable appliances 6. All children must have received their primary and secondary surgery at the concerned cleft centre
7. Lateral cephalometric radiographs available of each patient at the age of 10 years must be. Hannover sample
The Hannover database revealed that 115 children with complete UCLP were treated between 1 February 1981 and 1 March 1991. A total of 36 (31.3%) children (23 males, 13 females) who met the above mentioned criteria were included in this study; nine had right and 27 left complete UCLP; six had Simonart’s bands. The surgical treatment protocol of the Hannover group consisted of lip closure according to the Tennison–Randall technique at a mean age of 5.831.16 months (males 5.960.97, females 5.621.45). The hard palate was repaired by a vomer flap according to Pichler at a mean age of 29.084.68 months (males 28.834.84, females 29.544.54). Soft palate repair was carried out following a modified Widmaier technique at a mean age of 32.254.29 months (males 32.044.25, females 32.624.52) (Fig. 1). Different OMF surgeons were involved but followed all the same well-defined surgical technique meticulously. No primary nose surgery was performed in this group of cleft children. Brussels sample
A total of 90 children with complete UCLP, treated between November 1981 and November 1990, were found in the
Brussels cleft database; 36 (40.0%) fulfilled the inclusion criteria. A total of 26 children (17 males, nine females) who were operated on following the Malek protocol were included in this study; 13 had right and 13 left complete UCLP, and four had Simonart’s bands. There were 10 children with smaller clefts who underwent closure of their cleft following the all-in-one procedure during the same period of time, who were excluded. The soft palate was closed according to the Malek protocol at a mean age of 3.040.20 months (males 3.060.24, females 3.000.00); the lip was repaired following Tennisson–Randall at a mean age of 6.150.68 months (males 6.24 0.56, females 6.000.87) and the hard palate was closed using a vomer flap according to the Malek protocol simultaneously with the lip closure (Fig. 2). Almost all primary surgical procedures (96.2%) were performed by two plastic surgeons; one surgeon (ADM) carried out 28 (53.9%) of the primary operations. Children operated on from 1985 (n=15, 57.7%) had primary nose correction according to the McComb technique simultaneously with lip closure. Data on orthopaedic/orthodontic treatment and secondary surgical procedures for both groups are shown in Tables 1 and 2, respectively. Cephalometric analysis
Cranio-facial, particularly midfacial morphology was evaluated by means of conventional lateral cephalometric radiographs carried out on the patients at approximately 10 years of age; Hannover (age range 9.0–11.1 years, mean age 9.970.53), and Brussels (age range 8.8–11.2 years, mean age 10.400.70). The cleft samples were compared to a sample of 40 untreated children (20 males and 20 females, ranging in age from 9.2–11.0 years, mean age 10.290.42). These patients had normal skeletal relationships (ANB angle within 1SD of the mean), randomly selected from the files of the Department of Orthodontics, Medical University Hannover and matched according to age and gender with the cleft samples. Two investigators made acetate tracings and digitized all original X-rays independently. Discrepancies between the tracings of the two investigators were carefully revaluated and agreed upon after joint discussion. Landmarks and cephalometric analysis according to R19 were used. All linear variables were adjusted to the cranial
15
Midfacial morphology in UCLP children Brussels Soft palate Lip/hard palate
7.00
6.24
6.00
N
Months
6.00
S
5.00
NB
Co PTM Ba
PMP
4.00
UIapex
ANS A
LI
3.00
Go 3.06
3.00
UI B
Pog Gn Men
2.00
Males
Females
Fig. 2. Data on primary surgery for the children in the Brussels UCLP group.
base length (Ba-N) in order to calibrate different enlargement factors generated at both cleft centres. The cephalometric reference points used are shown in Fig. 3. Statistical analysis
Calculation of the method error associated with the cephalometric procedure was based on the variances of duplicate measurements. Analysis of variance (ANOVA with post-hoc Tukey’s test) was used to compare the mean cephalometric values of the children in the noncleft, Hannover cleft and Brussels cleft groups matched according to gender. Further comparisons (unpaired t-test) were carried out to compare male with female children within the non-cleft, Hannover cleft and Brussels cleft groups. Differences were defined as statistically significant at the 0.05 level. All computations were performed using the SPSS statistical package (SPSS 10.0 for Windows). Results Method error
In order to determine the reliability of tracing and digitizing, measurements were made twice of all subjects independently. A total of 7140 measurements (3060 angular, 3246 linear and 816 relations) were performed. Analysis of duplicate measurements revealed method errors of less than 1.50 and 1.20 mm. This is in agreement with
method errors of other studies17. Bigger method errors were found for the angular variables N-UI-Uiapex (1.58), Ba-NANS (1.51) and ANS-N-Pog (1.55). The difficulty in locating the ANS point on the lateral cephalometric radiograph explains the bigger method errors for these two variables. Significant differences
Results of the cephalometric analysis of male and female children are summarized in Tables 3 and 4, respectively. It is recognized that the variable sample sizes might have influenced the statistical power of the pair-wise post-hoc Tukey’s test between the cleft centres. Because only one variable (N-ANS/N-Men in the Brussels cleft group) reached a significant level (P<0.001), male and female children were pooled for the non-cleft, the Hannover cleft and the Brussels cleft group and reanalyzed using the same statistical procedure (Table 5). Facial profile The nasal bone angle (Ba-N-NB) was decreased in both cleft groups compared to the non-cleft group (Hannover P<0.001; Brussels P=0.002) (Table 5). Maxillary protrusion at ANS related to the cranial base (Ba-N) showed no significant differences for both cleft groups compared to the non-cleft group (Table 5) (Fig. 4). However, maxillary protrusion of the A point was significantly
Fig. 3. Lateral cephalometric skeletal landmarks: ANS=anterior nasal spine; A=point of greatest concavity of the alveolar process of the maxilla; Ba=basion, anterior lip of the foramen magnum; B=point of greatest concavity of the mandibular alveolar process; Co=condylion, posterior superior point on the outline of the condyle; Gn=gnathion, point on the symphysis between pogonion and menton farthest from the condyle; Go=gonion, constructed on the outline of the mandible by bisecting the ramus plane and body plan; LI=lower incisor; Men=menton, most inferior point on the symphysis; N=nasion, junction of the nasal and frontal bone; NB=lip of nasal bone; PMP=posterior maxillary point, a construct created by dropping a perpendicular to the maxillary plane from PTM; Pog=Pogonion, most prominent point on the chin; PTM=pterygomaxillary fissure, the inferior point in the fissure; R=registration point, point of crossing of the greater wing of the sphenoid and planium sphenoidale; S=Sella, estimated centre of the hypophyseal fossa; UI=upper incisor; UI-apex=upper incisor apex; MdPl= mandibular plane, line from Men through Go; MxPl=maxillary plane, line from ANS passing through the posterior hard palate.
less in both cleft groups in relation to the S-N plane (Hannover P=0.004; Brussels P<0.001) and the Ba-N plane (Hannover P=0.035; Brussels P=0.041) compared to the non-cleft group (Table 5). Mandibular protrusion at B point (Ba-N-B, SNB) and Pogonion (Ba-NPog) related to the cranial base were significantly less (P<0.001) in both cleft groups compared to the non-cleft group (Fig. 5, Table 5). Moreover, the Brussels cleft group was significantly more retruded at SNB (P=0.047) compared
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Swennen et al.
Table 1. Orthopaedic and orthodontic treatment Presurgical orthopaedics Deciduous dentition Mixed dentition
Hannover
Brussels
Passive appliance Removable, active appliance* Removable, active appliance*
Passive appliance Removable, active appliance* Removable, active appliance*
to the Hannover cleft group. Anteroposterior position of the incisors (BaN-UI, Ba-N-LI) was significantly less (P<0.001) in both cleft samples compared to the non-cleft group (Fig. 6, Table 5).
*Removable active appliances are used to correct malocclusion if necessary.
Sagittal relations Table 2. Incidence of secondary surgery in both cleft samples Hannover
Brussels
Males
Females
Males
Females
Sample size
23
13
17
9
Procedure Lip revision Rhinoplasty VPP Fistula closure Bone grafting*
— — 2 3 —
— — — 2 1
5 3 1 9 5
3 1 2 6 3
VPP: velopharyngoplasty; *Secondary alveolar bone grafting.
Evaluation of the maxillomandibular relationship showed no significant differences at the skeletal (ANS-N-Pog) and dento-alveolar (ANB) level. At the dental (overjet) level, a significant difference (P=0.033) was found in the Hannover cleft group compared to the non-cleft group (Table 5). Facial depths Maxillary length (PMP-A), anteroposterior development of the maxillary
Table 3. Mean values and standard deviations of angular, indexed linear measurements and relations in male UCLP and non-cleft children. Results of one-way ANOVA (p) and post-hoc Tukey test
Sample size
Unit
Mean age N-Ba Profile Ba-N-NB Ba-N-ANS Ba-N-A SNA Ba-N-UI Ba-N-LI Ba-N-B SNB Ba-N-Pog Sagittal relations ANB ANS-N-Pog Overjet Facial depths Ba-ANS Ba-A Ba-UI Ba-LI Ba-PMP PMP-A Co-Gn Vertical relations N-ANS N-ANS/N-Men ANS-UI/N-Men ANS-Men/Ba-Pog Men-Li/N-Men R-PMP MdPl/SN MxPl/SN Others N-UI-Ui apex N-S-Ba
Year mm
P
Hannover
Brussels
Non-cleft
23
17
20
Tukey
Mean
SD
Mean
SD
Mean
SD
9.85 94.07
0.52 2.85
10.37 101.08
0.67 6.97
10.25 94.77
0.49 3.38
H-B
H-C
B-C
deg deg deg deg deg deg deg deg deg
0.000 0.205 0.131 0.026 0.000 0.000 0.001 0.000 0.000
82.87 65.04 59.76 79.15 59.13 56.59 55.65 75.13 56.61
4.01 4.43 4.02 4.31 3.19 3.33 2.70 3.49 2.63
84.30 65.32 60.35 78.80 59.47 57.29 54.85 73.12 55.44
4.63 3.31 2.53 3.24 3.03 3.10 2.55 3.38 2.38
89.60 66.90 61.68 81.80 64.53 61.38 58.05 77.93 58.98
4.44 2.39 2.13 3.25 2.98 2.89 2.40 2.95 2.68
ns ns ns ns ns ns ns ns ns
‡ ns ns ns ‡ ‡ † * *
† ns ns * ‡ † † ‡ ‡
deg deg mm
0.071 0.661 0.071
4.04 8.37 2.79
3.57 4.43 2.80
5.65 9.32 4.05
1.90 2.93 2.82
3.78 8.43 4.38
1.53 2.82 0.71
ns ns ns
ns ns ns
ns ns ns
mm mm mm mm mm mm mm
0.068 0.057 0.000 0.000 0.031 0.569 0.311
90.83 86.39 88.20 85.08 39.70 47.28 100.58
3.47 3.47 4.62 3.73 2.18 2.92 4.38
90.98 86.93 89.28 86.02 39.95 47.85 100.29
2.38 2.16 3.59 3.77 2.24 2.22 4.66
92.73 88.38 95.02 90.34 41.41 46.94 102.43
2.30 2.06 3.46 3.34 2.15 2.44 5.07
ns ns ns ns ns ns ns
ns ns ‡ ‡ * ns ns
ns ns ‡ † ns ns ns
mm % % % % mm deg deg
0.070 0.000 0.116 0.000 0.023 0.000 0.000 0.000
46.17 42.84 25.40 66.73 36.00 41.35 35.70 9.54
2.01 1.87 1.35 4.72 1.34 3.98 4.50 3.26
47.14 42.61 25.71 69.23 36.18 38.55 38.35 11.47
2.57 1.45 1.61 4.15 1.34 3.69 4.19 3.37
47.75 45.01 24.66 61.25 34.96 43.90 31.03 6.38
2.13 2.29 1.75 5.13 1.65 2.55 5.47 2.22
ns ns ns ns ns * ns ns
ns † ns † ns ns † †
ns † ns ‡ * ‡ ‡ ‡
deg deg
0.000 0.007
4.91 130.26
3.13 4.86
4.94 132.29
1.64 4.61
9.23 126.98
3.01 5.44
ns ns
‡ ns
‡ †
*P<0.05; †P<0.01; ‡P<0.001; ns: not significant; deg: degrees.
17
Midfacial morphology in UCLP children
Table 4. Mean values and standard deviations of angular, indexed linear measurements and relations in female UCLP and non-cleft children. Results of one-way ANOVA (p) and post-hoc Tukey test
Sample size
Unit
Mean Age N-Ba Profile Ba-N-NB Ba-N-ANS Ba-N-A SNA Ba-N-UI Ba-N-LI Ba-N-B SNB Ba-N-Pog Sagittal relations ANB ANS-N-Pog Overjet Facial depths Ba-ANS Ba-A Ba-UI Ba-LI Ba-PMP PMP-A Co-Gn Vertical relations N-ANS N-ANS/N-Men ANS-UI/N-Men ANS-Men/Ba-Pog Men-Li/N-Men R-PMP MdPl/SN MxPl/SN Others N-UI-Ui apex N-S-Ba
Year mm
P
Hannover
Brussels
Non-cleft
13
9
20
Tukey
Mean
SD
Mean
SD
Mean
SD
10.17 92.04
0.52 4.57
10.46 95.67
0.81 6.72
10.33 92.32
0.35 3.31
H-B
H-C
B-C
deg deg deg deg deg deg deg deg deg
0.450 0.665 0.074 0.000 0.000 0.001 0.011 0.000 0.017
87.08 65.15 60.50 78.04 60.00 57.46 56.35 74.00 57.50
5.03 4.26 3.65 4.03 3.16 3.54 2.71 3.95 2.64
87.28 65.00 59.06 75.89 58.17 56.17 55.22 72.22 57.00
3.30 4.88 1.93 1.98 1.62 4.10 2.82 2.58 2.75
88.80 66.20 61.98 80.83 64.50 61.60 58.80 77.55 60.10
3.90 3.29 3.26 1.66 3.52 3.45 3.37 2.01 3.38
ns ns ns ns ns ns ns ns ns
ns ns ns * † † ns † ns
ns ns ns ‡ ‡ † * ‡ *
deg deg mm
0.635 0.495 0.230
4.08 7.50 3.39
2.82 4.06 1.75
3.67 6.33 2.83
2.78 3.58 4.27
3.30 6.25 4.34
1.55 1.87 1.17
ns ns ns
ns ns ns
ns ns ns
mm mm mm mm mm mm mm
0.212 0.040 0.000 0.004 0.481 0.855 0.005
87.65 87.08 87.37 85.83 39.54 46.03 99.70
11.29 2.96 8.15 3.94 2.22 6.26 5.62
89.75 85.63 87.58 79.88 39.30 46.50 96.43
2.04 1.77 2.01 15.26 2.50 2.45 2.26
91.89 88.58 95.78 90.87 40.94 46.85 103.63
3.00 3.13 4.78 4.38 5.20 2.59 6.07
ns ns ns ns ns ns ns
ns ns † * ns ns ns
ns * † † ns ns †
mm % % % % mm deg deg
0.010 0.047 0.778 0.112 0.738 0.007 0.159 0.000
46.41 43.62 24.99 64.47 34.74 40.94 36.27 9.73
2.44 2.10 1.87 5.09 1.30 5.05 4.12 3.56
48.99 48.27 25.36 61.77 35.74 40.76 37.06 12.61
1.10 8.33 5.34 7.82 5.91 2.39 6.07 2.09
48.69 45.16 24.58 60.21 35.14 44.56 33.78 7.48
2.39 2.00 1.57 4.62 1.53 2.78 4.44 2.26
* * ns ns ns ns ns *
* ns ns ns ns * ns ns
ns ns ns ns ns * ns ‡
deg deg
0.000 0.004
6.35 132.46
2.90 4.86
5.00 135.22
2.44 1.79
9.38 129.53
2.30 4.35
ns ns
† ns
‡ †
*P<0.05; †P<0.01; ‡P<0.001; ns: not significant; deg: degrees.
bone (Ba-ANS) and pharyngeal depth (Ba-PMP), indicating the anterior position of the maxilla, showed no significant differences between both cleft groups and the non-cleft group (Table 5). However, antero-posterior development of the dento-alveolar process (Ba-A) was significantly less in both cleft groups compared to the control group (Hannover P=0.012; Brussels P=0.013) (Table 5). Measurements indicating the forward protrusion of the upper (Ba-UI) and lower (Ba-LI) incisors were significantly less (P<0.001) in both cleft groups compared to the matched controls (Table 5). These findings are consistent with the above mentioned angular variables (Ba-N-UI, Ba-N-LI) and can be explained by the more upright inclination of upper (N-UIUiapex significant at P<0.001 for both Hannover and Brussel cleft groups)
(Table 5) and lower incisors. Both cleft groups showed a significantly smaller mandibular length (Co-Gn) compared to the non-cleft group (Hannover P=0.048; Brussels P=0.005) (Table 5). Vertical relations Posterior maxillary height (R-PMP) was significantly decreased in both cleft groups compared to the non-cleft group, but more so in the Brussels cleft group (P<0.001) (Table 5). There was a significant decrease in anterior maxillary height (N-ANS) in the Hannover cleft group compared to the non-cleft (P=0.001) and Brussels cleft (P=0.026) groups (Table 5). The mandibular (MdPl/SN) and maxillary (MxPl/SN) planes were more open in the Hannover cleft group compared to the control group (P=0.005 and P<0.001, respect-
ively), but even more open in the Brussels group (P<0.001 and P<0.001, respectively) (Table 5) (Fig. 7). Moreover, the inclination of the maxillary plane reached a significant value (P=0.008) in the Hannover and Brussels comparison data (Table 5). The more open mandibular plane in the Brussels cleft children explains the above mentioned more retruded mandible (SNB) in the Brussels group (Table 5). The anterior maxillary height related to the total face height (N-ANS/N-Men) was the only variable that showed a difference of gender and was therefore not pooled. The contribution of the anterior maxillary vertical height (ANS-UI/NMen) and anterior mandibular vertical height (Me-Li/N-Men) to the total facial height showed in both cleft groups no significant differences compared to the non-cleft group (Table 5). The
18
Swennen et al.
Table 5. Mean values and standard deviations of angular, indexed linear measurements and relations in pooled UCLP and non-cleft children. Results of one-way ANOVA (p) and post-hoc Tukey test
Sample size
Unit
Mean Age N-Ba Profile Ba-N-NB Ba-N-ANS Ba-N-A SNA Ba-N-UI Ba-N-LI Ba-N-B SNB Ba-N-Pog Sagittal relations ANB ANS-N-Pog Overjet Facial depths Ba-ANS Ba-A Ba-UI Ba-LI Ba-PMP PMP-A Co-Gn Vertical relations N-ANS ANS-UI/N-Men ANS-Men/Ba-Pog Men-Li/N-Men R-PMP MdPl/SN MxPl/SN Others N-UI-Ui apex N-S-Ba
Year mm
P
Hannover
Brussels
Non-cleft
36
26
40
Tukey
Mean
SD
Mean
SD
Mean
SD
9.97 93.34
0.53 3.64
10.40 99.21
0.70 7.24
10.29 93.54
0.42 3.53
H-B
H-C
B-C
deg deg deg deg deg deg deg deg deg
0.000 0.170 0.016 0.000 0.000 0.000 0.000 0.000 0.000
84.39 65.08 60.03 78.75 59.44 56.90 55.90 74.72 56.93
4.79 4.31 3.85 4.18 3.16 3.39 2.69 3.65 2.64
85.33 65.21 59.90 77.79 59.02 56.90 54.98 72.81 55.98
4.40 3.83 2.38 3.16 2.67 3.44 2.60 3.10 2.57
89.20 66.55 61.83 81.31 64.51 61.49 58.43 77.74 59.54
4.15 2.86 2.72 2.60 3.22 3.14 2.91 2.50 3.06
ns ns ns ns ns ns ns * ns
‡ ns * † ‡ ‡ ‡ ‡ ‡
† ns * ‡ ‡ ‡ ‡ ‡ ‡
deg deg mm
0.080 0.497 0.043
4.06 8.06 3.01
3.28 4.26 2.47
4.96 8.29 3.63
2.39 3.42 3.36
3.54 7.34 4.36
1.54 2.61 0.95
ns ns ns
ns ns *
ns ns ns
mm mm mm mm mm mm mm
0.057 0.004 0.000 0.000 0.054 0.780 0.004
89.68 86.63 87.90 85.35 39.64 46.83 100.27
7.33 3.27 6.03 3.77 2.16 4.38 4.80
90.56 86.48 88.69 83.90 39.72 47.38 98.95
2.30 2.09 3.20 9.62 2.31 2.34 4.36
92.31 88.48 95.40 90.61 41.17 46.90 103.03
2.67 2.62 4.13 3.85 3.94 2.48 5.55
ns ns ns ns ns ns ns
ns * ‡ ‡ ns ns *
ns * ‡ ‡ ns ns †
mm % % % mm deg deg
0.001 0.178 0.000 0.212 0.000 0.000 0.000
46.26 25.26 65.92 35.55 41.20 35.90 9.61
2.14 1.54 4.91 1.45 4.33 4.31 3.32
47.78 25.59 66.65 36.03 39.31 37.90 11.87
2.33 3.29 6.61 3.52 3.42 4.84 3.00
48.22 24.62 60.73 35.05 44.23 32.40 6.93
2.28 1.64 4.85 1.57 2.65 5.11 2.28
* ns ns ns ns ns †
† ns ‡ ns † † ‡
ns ns ‡ ns ‡ ‡ ‡
deg deg
0.000 0.000
5.43 131.06
3.08 4.91
4.96 133.31
1.90 4.08
9.30 128.25
2.64 5.03
ns ns
‡ *
‡ ‡
*P<0.05; †P<0.01; ‡P<0.001; ns: not significant; deg: degrees.
relationship of the anterior vertical mandibular height to Ba-Pog was in both cleft groups significantly increased (P<0.001) (Table 5). The cranial base angle (N-S-Ba) was significantly increased in both cleft groups but more so in the Brussels cleft group compared to the non-cleft group (Hannover P=0.032; Brussels P<0.001) (Table 5). Secondary surgery
Data on secondary surgery performed during the investigation period are listed in Table 2. In the Hannover cleft group no lip revision or rhinoplasty was performed. A total of eight (30.8%) children in the Brussels cleft group underwent lip correction at a mean age of 4.382.13 years, while four (15.4%) children had a rhinoplasty at 4.832.71 years (no septoplasty). A velopharyngoplasty was
carried out in two (5.6%) children in the Hannover cleft group and in three (11.5%) children in the Brussels cleft group at a mean age of 7.752.19 and 5.070.32 years, respectively. In the Hannover cleft group, a total of five (13.9%) children underwent closure of oronasal fistulas at a mean age of 3.040.63 years: three were closed with a vomer flap, one using a von Langenbeck palatoplasty and one with a veloplasty according to Widmeier. In one child, fistula closure with a vomer flap was not successful and a von Langenbeck palatoplasty was performed. A total of eight (22.2%) children had fistulas (four oronasal, four vestibulonasal) that were not closed during the investigation period because they caused no functional problems. In the Brussels group, however, all fistulas were closed; a total of 17 fistula closures (eight oronasal,
nine vestibulonasal) were performed in 15 (57.7%) children at a mean age of 3.311.57 years. Closure of vestibulonasal fistulas was carried out in four children with a hinge flap, two with a small rotational flap, one with a vomer flap, one with a conventional primary suture and one during alveolar bone grafting. Oronasal fistulas were closed in four cases through a vomer flap, in two with a hinge flap, in one with a small rotation flap and in one with a conventional suture. In one child, closure of an oronasal fistula with a primary suture was not successful and a small rotation flap was performed. During the period of investigation, only one (2.8%) child underwent secondary alveolar bone grafting in the Hannover cleft group at the age of 5.5 years, while in the Brussels group eight (30.8%) children had bone grafting at a mean age of 8.41.05 years.
Midfacial morphology in UCLP children Maxillary protrusion SNA Ba-N-ANS Ba-N-A
90.00
80.00
81.31
Degrees
78.75
77.79
70.00 66.55 65.21
65.08 61.83
60.00
50.00
60.03
Non-cleft
Hannover
59.90
Brussels
Fig. 4. Comparison of maxillary protrusion in pooled UCLP and non-cleft patients.
Mandibular protusion SNB Ba-N-Pog Ba-N-B
80.00 77.74 74.72 72.81
Degrees
70.00
60.00 59.54 58.42 56.93 55.90
55.98 54.98
50.00
Non-cleft
Hannover
Brussels
Fig. 5. Comparison of mandibular protrusion in pooled UCLP and non-cleft patients.
Discussion Surgical technique, timing and sequence of cleft closure and the application of presurgical orthopaedics and/or adjunctive orthodontics in the treatment of children with UCLP in reference to midfacial growth impairment, have been major concerns of cleft centres throughout the last decades. Different treatment protocols may have their specific influence on cranio-facial morphology. In
this study we compared cranio-facial morphology of children with UCLP treated according to different surgical protocols, particularly regarding timing and sequence but having identical orthopaedic/orthodontic regimens. In Hannover, lip closure is followed by a two-stage palatoplasty5,9,10,22,23,28 (Fig. 1). The children in the Brussels cleft group in this study underwent cleft closure following a new approach
19
advocated by Rene´ Malek: early veloplasty followed by simultaneous lip and hard palate repair4,12,13,20 (Fig. 2). Cephalometric analysis revealed that the children with UCLP in the Hannover cleft group did not differ significantly from the children with UCLP in the Brussels group in the anteroposterior dimension of the midface. The angle of maxillary protrusion (Ba-N-ANS) reflecting the forward translation of the maxilla and the length of its basal bone giving a good indication of anteroposterior growth was excellent in both cleft groups. The effective length of the maxilla (PMP-A), which includes both basal maxillary length and the position of the alveolus on it, was likewise excellent in both cleft groups. However, as far as the vertical dimension is concerned, a significant difference (MxPl/SN at P=0.008) was found in the Hannover– Brussels comparison data. Although the maxillary plane was more open in both cleft groups compared to the non-cleft group, it was significantly more open in the Brussels group compared to the Hannover group. This can be explained because the posterior maxillary development (R-PMP) was significantly less in the Brussels group (P<0.001), while the anterior maxillary development (N-ANS) was significantly less in the Hannover group (P=0.001) (Table 5). The posterior vertical development of the maxilla (R-PMP) was decreased in both cleft groups; this is consistent with the literature2,19. However, the posterior maxillary vertical height was significantly less in the children in the Brussels cleft group (Table 5). In this group, soft palate closure (3.040.20 months) was performed much earlier compared to the children in the Hannover cleft group (32.254.29 months). The soft palate repair according to Malek consists of a veloplasty using a transverse incision at the level of the posterior edges of the palatal shelves with the dissection performed posterior to the neurovascular bundle. To allow easier closure of the cleft at the level of the posterior margin of the palatal shelves where the tension is the greatest, a small mucoperiosteal vomer flap is raised, rotated 180 and sutured to the nasal mucosa of the velum12,13. The inhibiting effect of soft palate repair on posterior maxillary vertical height has been described in the literature19. E & H6 showed that in the first 3 years the direction of maxillary growth is primarily vertical. We suggest that the Malek soft palate repair is probably related with more
20
Swennen et al. Dental relations Ba-N-UI Ba-N-LI UI-UIapex
70.00 64.51
60.00
61.49 59.44
59.02
56.90
56.90
Degrees
50.00
40.00
30.00
20.00
10.00 9.30 5.43
0.00
Non-cleft
Hannover
4.96
Brussels
Fig. 6. Comparison of dental relations in pooled UCLP and non-cleft patients.
Inclination of maxilla and mandible 40.00
MxPI/SN MdPI/SN 37.90
35.00
35.90 32.40
30.00
Degrees
25.00 20.00 15.00 11.87
10.00 9.61
5.00 0.00
6.93
Non-cleft
Hannover
Brussels
Fig. 7. Comparison of maxillar and mandibular plane inclination in pooled UCLP and non-cleft patients.
cicatrised soft tissues, which might provide resistance to posterior vertical growth. In combination with the earlier timing of soft palate repair, this might explain the less vertical posterior maxillary development in the Brussels group. The mandible was more open in both cleft groups and more in the Brussels group (P<0.001) (Table 5). Presurgical orthopaedic treatment with a passive appliance has been reported to influence
the inclination of the mandible17,19,24. Mø et al.17 hypothesized that the tongue position is altered by the appliance and therefore the mandible is inclined in a more backward direction. Although both cleft groups in this study underwent presurgical orthopaedic treatment with a passive appliance, we do not believe that these appliances are responsible for the more open mandibular angle. We suggest that the increased
inclination of the mandibular plane in both cleft groups is a consequence of mouth breathing and maxillary morphology. Therefore, the increased inclination of the maxillary plane in the Brussels cleft group explains its more open mandibular plane. It has been reported that vomerine mucosal flaps result in a reduction of maxillary vertical height, a more retruded maxilla and an overclosure of the mandible due to the scar tissue adhesions that unite the vomer to the palatal shelves at the site of the repaired cleft3,14,18. Although correction of the hard palate defect using vomer flaps was performed in the children in the Hannover and Brussels cleft groups, both cleft groups showed excellent anteroposterior maxillary morphology and an even more open mandible. However, as already mentioned, both cleft groups showed a decreased vertical development. The veloplasty according to Malek, which includes a small vomer flap, might be an additional cause for the decreased posterior development in the Brussels group. The results of this study support the hypothesis of Ross that variations in the timing of hard and soft palate repair within the first decade do not influence midfacial growth in the antero-posterior dimension19. Contrary to R, this study shows that variation in the timing of soft palate repair might influence posterior vertical growth. However, it is not entirely clear whether the decreased posterior maxillary development in the Brussels group is caused by the earlier timing of soft palate closure or by the surgical technique. This point requires further analysis. There may be many reasons why the UCLP children of both cleft centres in this study have good midfacial morphology. Many cleft routines include procedures that have been assumed to be associated with impaired midfacial growth, such as primary bone grafting, periosteal grafts and active presurgical orthopaedics19,26,29. The treatment protocols of both cleft centres in this study did not include these procedures. As far as palatal repair is concerned, the surgical procedures of both cleft centres are characterized by the absence of denudation of the thick palatal mucoperiosteum, which is associated with growth-impairing palatal scar tissue14,18. Furthermore, we agree with S et al.25,26 that the good results obtained in a cleft centre most likely depend more on its well-organized and centralized structure that ensures uniformity in its
Midfacial morphology in UCLP children treatment protocol, rather than on individual components of its treatment protocol. A higher number of secondary revisionary procedures were found in the Brussels cleft group (Table 2). Interpretation of these data, however, should be done with caution. During the investigation period, no secondary lip or nose corrections were performed in the children in the Hannover cleft group because at that time those procedures were delayed until they had reached the age of 13 years. As far as fistulas are concerned, the Brussels cleft group showed a higher incidence of fistula closures (57.7%). It is known that the Malek protocol is associated more frequently with palatal fistulas4,12, although it provides excellent craniofacial growth20. The fact that all fistulas were closed in the Brussels group also suggests that patient and surgeon requirements may be different in both cleft centres. Furthermore, the combination of secondary surgical procedures is essential in the Brussels treatment philosophy of cleft children. Hence, closure of fistulas was often combined with small lip or nose corrections. Because our primary goal in this study was to compare the surgical treatment protocols of two cleft centres, we attempted to provide detailed surgical data on the primary and secondary procedures of both centres. However, the potential influence of these secondary procedures on cranio-facial morphology was not analyzed in this study. Studies comparing the influence of different surgical protocols on craniofacial morphology in cleft patients are often likely to be unreliable because of methodological biases. S & S24 defined potential biases as follows: detection, exclusion, analysis, reporting, susceptibility and proficiency bias. In this study, we did attempt to overcome those biases in that: 1. The cleft databases of both cleft centres were consulted and all patients that fulfilled the abovementioned criteria were included (detection and exclusion bias) 2. All cleft patients were compared to a non-cleft control group, matched according to age and gender. Linear measurements were adjusted to the cranial base length (Ba-N) and reliability of the cephalometric analysis was proved through calculations of the method error (analysis bias)
3. The complete data (cephalometric measurements, age, primary and secondary surgery) of both cleft samples were reported (reporting bias) 4. Susceptibility bias could only be partially resolved. Although both cleft samples were equivalent in type (complete non-syndromic UCLP) and race (Caucasian), the difference in severity of the initial cleft could not be assessed because infant dental casts were not available for all children 5. Although both cleft centres meticulously followed a well-defined surgical protocol, different surgeons were involved in both centres, so that proficiency bias was only partially resolved. Proficiency bias, however, cannot be solved even in welldesigned prospective randomized intercentre studies, as the surgical skill of surgeons will inevitably be different. Furthermore, the possibility of sampling bias exists if two or more treatment protocols are being used concurrently in one centre26. Contrary to the Hannover cleft group, there is clearly a sample bias in the Brussels group, as only the Malek cases were included and the all-in-one cases were excluded in this study. Despite our effort to perform a welldesigned comparative study, this study still has several shortcomings. Firstly, cranio-facial morphology was evaluated and compared cross-sectionally at 10 years of age, before the beginning of their pubertal growth spurt. As the cephalometric analysis we used does not take into account cranial predisposition, errors of interpretation are also possible18. Secondly, because of the retrospective design and the strict inclusion criteria, the size of the cleft groups is still too small in this study to discern statistically minor surgical effects using multiple comparison tests. Finally, evaluation of cranio-facial growth through longitudinal architectural cranio-facial cephalometry, anthropometry and dental casts analyses combined with clinical findings, video analysis, speech and hearing evaluation would be the ideal setting to compare the aesthetic and functional outcome between different cleft centres. Acknowledgment. The authors thank Professor Hartmut Hecker, Department of Biometry, Medical University Hannover, Germany, for his assistance
21
in the statistical analysis of the cephalometric data. References 1. A-M C, B¨ V, D E, MW J, Mø K, P DA, P-A B, S G, S WC. The RPS. A six-center international study of treatment outcome in patients with clefts of the lip and palate: Part 4. Assessment of nasiolabial appearance. Cleft Palate Craniofac J 1992: 29: 409– 412. 2. B PH, S JN, G E, S KE. Growth status of children treated for unilateral cleft lip and palate. Plast Reconstr Surg 1991: 88: 413–420. 3. D J, P D. Avoidance of the use of vomerine mucosa in primary surgical management of velopalatine clefts. Oral Surg Oral Med Oral Pathol 1985: 60: 589–597. 4. D M A, L B, M C, M AL, L M. Treatment of cleft lip and palate: long-term results. Ann Chir Plast Esthet 1992: 37: 174–178. 5. D R, T T, K FJ, H JE. Alveolar bone grafting in patients with complete clefts—a comparative study between secondary and tertiary bone grafting. Cleft Palate Craniofac J (Accepted for publication). 6. E DH, H MG. Essentials of facial growth. Philadelphia: W.B. Saunders 1996: 79–98. 7. F H, E H, S G, P G, A F, B S, L J, O L. Craniofacial and occlusal characteristics in unilateral cleft lip and palate patients from four Scandinavian centres. Scand J Plast Reconstr Hand Surg 1991: 25: 269–276. 8. F H, P D, M¨ M, M I, L J, B B. Comparisons of facial growth in patients with unilateral cleft lip and palate treated by different regimens for two-stage palatal repair. Scand J Plast Reconstr Hand Surg 1999: 33: 73–81. 9. H JE. Surgical rehabilitation of patients with cleft lip, jaw and palate. Quintessenz 1986: 37: 195–203. 10. H JE, S R. LippenKiefer-Gaumenspalten. In: Hausamen JE, Machtens E, Reuther J, ed.: Mund-, Kiefer- und Gesichtschirurgie. Berlin: Springer 1995: 299–357. 11. LR D. The state of the art in cleft palate surgery. Cleft Palate Craniofac J 2000: 37: 225–228. 12. M R, P J. New concept of the chronology and surgical technic in the treatment of cleft lip and palate. Results in 220 cases. Ann Chir Plast Esthet 1983: 28: 237–247. 13. M R, M H, M MR, T C. Multidisciplinary management of cleft lip and palate in Paris, France. In: Bardach J, Morris HL, eds:
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Swennen et al. Multidisciplinary Approach of Cleft Lip and Palate Patients. Philadelphia: W.B. Saunders 1990: 1–10. M AF, P DS. Effect of primary surgery for cleft lip and palate on mid-facial growth. Br J Oral Maxillofac Surg 1997: 35: 6–10. M M, A-MD C, B¨ V, D E, MW J, Mø K, P DA, PA B, S G, S WC. The RPS. A six-center international study of treatment outcome in patients with clefts of the lip and palate: Part 3. Dental arch relationships. Cleft Palate Craniofac J 1992: 29: 405–408. Mø K, A-MD C, B¨ V, D E, M M, MW J, P DA, PA B, S G, S WC. The RPS. 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: 398–404. Mø K, D E, S LT, A-MD C, B¨ V, MC A, P-A B, S G, S B. The RPS. A multicenter comparison of treatment regimens for unilateral cleft lip and palate using a multiple regression model. Scand J Plast Reconstr Hand Surg 1993: 27: 277–284. P DS, D J. Clinical observations of cleft lip and palate. Oral Surg Oral Med Oral Pathol 1993: 75: 141– 151.
19. R RB. Treatment variables affecting facial growth in complete unilateral cleft lip and palate. Cleft Palate Craniofac J 1987: 24: 5–77. 20. R RB. Growth of the facial skeleton following the Malek repair for unilateral cleft lip and palate. Cleft Palate Craniofac J 1995: 32: 194–198. 21. S SA. Unilateral cleft lip repair: State of the art. Cleft Palate Craniofac J 2000: 37: 335–341. 22. S H, N FW, R RH, B P. Bone repair of lipmaxillary-palatal cleft patients in adulthood. Fortschr Kiefer Gesichtschir 1993: 38: 92–94. 23. S¨ R, L JA, S¨ B, E A, P M, T¨ J, H JE. A retrospective study of hearing, speech and language function in children with clefts following palatoplasty and veloplasty procedures at 18–24 months. Int J Pediatr Otorhinolaryngol 1999: 50: 205–217. 24. S G, S WC. Facial growth after different methods of surgical intervention in patients with cleft lip and palate. Acta Odontol Scand 1998: 56: 352–355. 25. S WC, A-MD C, B¨ V, D E, MW J, Mø K, P DA, PA B, S G. The RPS. A sixcenter international study of treatment outcome in patients with clefts of the lip and palate: Part 1. Principles and study design. Cleft Palate Craniofac J 1992: 29: 393–397.
26. S WC, D E, A-MD C, B¨ V, M M, MW J, Mø K, P DA, PA B, R C, S G. The RPS. A six-center international study of treatment outcome in patients with clefts of the lip and palate: Part 5. General discussion and conclusion. Cleft Palate Craniofac J 1992: 29: 413–418. 27. S B, P BL, B P, K L. State of the art in oral and maxillofacial surgery: Treatment of maxillary hypoplasia and anterior palatal and alveolar clefts. Cleft Palate Craniofac J 1999: 36: 283–291. 28. T¨ J. Presurgical orthodontic treatment in cheilognatho-palatoschisis. Fortschr Kieferorthop 1986: 47: 370–379. 29. T CA, R EL J, R SW, M C, J LE J. Comparison of facial form in primary alveolar bone-grafted an nongrafted unilateral cleft lip and palate patients: Intercenter retrospective study. Cleft Palate Craniofac J 1996: 33: 91–95. Address: Gwen Swennen, MD, DDS Department of Oral and Maxillofacial Surgery Medizinische Hochschule Hannover Carl-Neuberg-Strasse 1 30625 Hannover Germany Tel: +49 511 532 47 48 Fax: +49 511 532 47 40 E-mail: [email protected]
Leading clinical paper: Congenital deformities
Midfacial morphology in children with unilateral cleft lip and palate treated by different surgical protocols
G. Swennen1, J. L. Berten2, H. Schliephake3, C. Treutlein2, R. Dempf1, C. Malevez4, A. De Mey5 1
Department of Oral and Maxillofacial Surgery; Department of Orthodontics, Medical University, Hannover, Carl-Neuberg-Strasse 1, 30625 Hannover, Germany; 3Department of Oral and Maxillofacial Surgery; Georg-August-University, Go¨ttingen, Robert-Koch-Strasse 40, 37075 Go¨ttingen, Germany; 4Department of Oral and Maxillofacial Surgery; 5Plastic and Reconstructive Surgery, University of Brussels (ULB), Children’s Hospital, Avenue J.J. Crocq 15, 1020 Brussels, Belgium 2
G. Swennen, J. L. Berten, H. Schliephake, C. Treutlein, R. Dempf, C. Malevez, A. De Mey: Midfacial morphology in children with unilateral cleft lip and palate treated by different surgical protocols. Int. J. Oral Maxillofac. Surg. 2002; 31: 13–22. 2002 International Association of Oral and Maxillofacial Surgeons Abstract. The purpose of this study was to compare cranio-facial, particularly midfacial morphology, in two groups of children with complete unilateral cleft lip and palate (UCLP) treated at two different cleft centres (Hannover, Germany and Brussels, Belgium) following different surgical treatment protocols. A total of 62 children (40 males; 22 females) with non-syndromic UCLP were included in this study at approximately the age of 10. The Hannover group comprised 36 children, who had repair of the lip at a mean age of 5.831.16 months, followed by repair of the hard and soft palate at a mean age of 29.084.68 and 32.254.29 months, respectively. The Brussels group consisted of 26 children who underwent surgical treatment according to the Malek protocol: the soft palate was closed at a mean age of 3.040.20 months, followed by simultaneous repair of the lip and hard palate at a mean age of 6.150.68 months. Midfacial morphology was evaluated by means of cephalometric analysis according to Ross. The children in the Hannover UCLP group did not differ significantly from those in the Brussels group in the anteroposterior dimension of the midface. However, the maxillary plane was significantly more open in the Brussels group due to less posterior vertical maxillary development.
Introduction Many different surgical treatment protocols and adjunctive orthopaedic/ orthodontic procedures have been proposed to treat children with unilateral cleft lip and palate (UCLP)11,21,27. S & S24 reported in 1998 that the register of the Eurocleft Biomed II Project, although incomplete at the time of publication, consisted of 178 cleft 0901-5027/02/010013+10 $35.00/0
teams with 171 different treatment protocols for treating patients with UCLP alone. All those treatment protocols vary from each other through variations and modifications regarding surgical technique, timing, sequence and the application of presurgical orthopaedic and/or adjunctive orthodontic procedures. Evaluation of treatment outcomes achieved by different cleft centres is essential in cleft lip–palate research.
Key words: cleft lip palate; cranio-facial morphology; cephalometry. Accepted for publication 1 October 2001 Published online 7 January 2002
Although the literature provides many reports on cranio-facial morphology in patients with cleft disorders, only few intercentre comparative studies have been carried out in an attempt to elucidate the effects of different treatments on cranio-facial morphology7,8,16,17,20,29. The six-centre study of treatment outcome conducted by the European Cleft Lip and Palate Research Group1,15,16,25,26 and the multicentre
2002 International Association of Oral and Maxillofacial Surgeons
14
Swennen et al. Hannover Lip Hard palate Soft palate
40.00
32.62
32.04
30.00
29.54
Months
28.83
20.00
10.00 5.96
0.00
5.62
Males
Females
Fig. 1. Data on primary surgery for the children in the Hannover UCLP group.
study performed by R19 have added a major contribution to the literature on cleft lip–palate research. However, comparison of cephalometric data with the published results from these multicentre studies is scientifically questionable due to potential interobserver error19. Hence, the purpose of this study was to compare cranio-facial, particularly midfacial morphology in children with UCLP, who were treated at two different cleft centres following different surgical protocols, to a non-cleft control group.
Material and methods The Cleft databases from the Department of OMF Surgery, Medical University, Hannover, Germany, and the Department of OMF, Plastic and Reconstructive Surgery, Children’s Hospital, Brussels University (ULB), Belgium, were consulted for children who met the following inclusion criteria: 1. Complete unilateral cleft lip, alveolus and palate. Patients with Simonart’s bands were included 2. Consecutive cases 3. No associated syndrome or mental retardation 4. Caucasian ethnicity 5. Presurgical infant orthopaedics with passive removable appliances 6. All children must have received their primary and secondary surgery at the concerned cleft centre
7. Lateral cephalometric radiographs available of each patient at the age of 10 years must be. Hannover sample
The Hannover database revealed that 115 children with complete UCLP were treated between 1 February 1981 and 1 March 1991. A total of 36 (31.3%) children (23 males, 13 females) who met the above mentioned criteria were included in this study; nine had right and 27 left complete UCLP; six had Simonart’s bands. The surgical treatment protocol of the Hannover group consisted of lip closure according to the Tennison–Randall technique at a mean age of 5.831.16 months (males 5.960.97, females 5.621.45). The hard palate was repaired by a vomer flap according to Pichler at a mean age of 29.084.68 months (males 28.834.84, females 29.544.54). Soft palate repair was carried out following a modified Widmaier technique at a mean age of 32.254.29 months (males 32.044.25, females 32.624.52) (Fig. 1). Different OMF surgeons were involved but followed all the same well-defined surgical technique meticulously. No primary nose surgery was performed in this group of cleft children. Brussels sample
A total of 90 children with complete UCLP, treated between November 1981 and November 1990, were found in the
Brussels cleft database; 36 (40.0%) fulfilled the inclusion criteria. A total of 26 children (17 males, nine females) who were operated on following the Malek protocol were included in this study; 13 had right and 13 left complete UCLP, and four had Simonart’s bands. There were 10 children with smaller clefts who underwent closure of their cleft following the all-in-one procedure during the same period of time, who were excluded. The soft palate was closed according to the Malek protocol at a mean age of 3.040.20 months (males 3.060.24, females 3.000.00); the lip was repaired following Tennisson–Randall at a mean age of 6.150.68 months (males 6.24 0.56, females 6.000.87) and the hard palate was closed using a vomer flap according to the Malek protocol simultaneously with the lip closure (Fig. 2). Almost all primary surgical procedures (96.2%) were performed by two plastic surgeons; one surgeon (ADM) carried out 28 (53.9%) of the primary operations. Children operated on from 1985 (n=15, 57.7%) had primary nose correction according to the McComb technique simultaneously with lip closure. Data on orthopaedic/orthodontic treatment and secondary surgical procedures for both groups are shown in Tables 1 and 2, respectively. Cephalometric analysis
Cranio-facial, particularly midfacial morphology was evaluated by means of conventional lateral cephalometric radiographs carried out on the patients at approximately 10 years of age; Hannover (age range 9.0–11.1 years, mean age 9.970.53), and Brussels (age range 8.8–11.2 years, mean age 10.400.70). The cleft samples were compared to a sample of 40 untreated children (20 males and 20 females, ranging in age from 9.2–11.0 years, mean age 10.290.42). These patients had normal skeletal relationships (ANB angle within 1SD of the mean), randomly selected from the files of the Department of Orthodontics, Medical University Hannover and matched according to age and gender with the cleft samples. Two investigators made acetate tracings and digitized all original X-rays independently. Discrepancies between the tracings of the two investigators were carefully revaluated and agreed upon after joint discussion. Landmarks and cephalometric analysis according to R19 were used. All linear variables were adjusted to the cranial
15
Midfacial morphology in UCLP children Brussels Soft palate Lip/hard palate
7.00
6.24
6.00
N
Months
6.00
S
5.00
NB
Co PTM Ba
PMP
4.00
UIapex
ANS A
LI
3.00
Go 3.06
3.00
UI B
Pog Gn Men
2.00
Males
Females
Fig. 2. Data on primary surgery for the children in the Brussels UCLP group.
base length (Ba-N) in order to calibrate different enlargement factors generated at both cleft centres. The cephalometric reference points used are shown in Fig. 3. Statistical analysis
Calculation of the method error associated with the cephalometric procedure was based on the variances of duplicate measurements. Analysis of variance (ANOVA with post-hoc Tukey’s test) was used to compare the mean cephalometric values of the children in the noncleft, Hannover cleft and Brussels cleft groups matched according to gender. Further comparisons (unpaired t-test) were carried out to compare male with female children within the non-cleft, Hannover cleft and Brussels cleft groups. Differences were defined as statistically significant at the 0.05 level. All computations were performed using the SPSS statistical package (SPSS 10.0 for Windows). Results Method error
In order to determine the reliability of tracing and digitizing, measurements were made twice of all subjects independently. A total of 7140 measurements (3060 angular, 3246 linear and 816 relations) were performed. Analysis of duplicate measurements revealed method errors of less than 1.50 and 1.20 mm. This is in agreement with
method errors of other studies17. Bigger method errors were found for the angular variables N-UI-Uiapex (1.58), Ba-NANS (1.51) and ANS-N-Pog (1.55). The difficulty in locating the ANS point on the lateral cephalometric radiograph explains the bigger method errors for these two variables. Significant differences
Results of the cephalometric analysis of male and female children are summarized in Tables 3 and 4, respectively. It is recognized that the variable sample sizes might have influenced the statistical power of the pair-wise post-hoc Tukey’s test between the cleft centres. Because only one variable (N-ANS/N-Men in the Brussels cleft group) reached a significant level (P<0.001), male and female children were pooled for the non-cleft, the Hannover cleft and the Brussels cleft group and reanalyzed using the same statistical procedure (Table 5). Facial profile The nasal bone angle (Ba-N-NB) was decreased in both cleft groups compared to the non-cleft group (Hannover P<0.001; Brussels P=0.002) (Table 5). Maxillary protrusion at ANS related to the cranial base (Ba-N) showed no significant differences for both cleft groups compared to the non-cleft group (Table 5) (Fig. 4). However, maxillary protrusion of the A point was significantly
Fig. 3. Lateral cephalometric skeletal landmarks: ANS=anterior nasal spine; A=point of greatest concavity of the alveolar process of the maxilla; Ba=basion, anterior lip of the foramen magnum; B=point of greatest concavity of the mandibular alveolar process; Co=condylion, posterior superior point on the outline of the condyle; Gn=gnathion, point on the symphysis between pogonion and menton farthest from the condyle; Go=gonion, constructed on the outline of the mandible by bisecting the ramus plane and body plan; LI=lower incisor; Men=menton, most inferior point on the symphysis; N=nasion, junction of the nasal and frontal bone; NB=lip of nasal bone; PMP=posterior maxillary point, a construct created by dropping a perpendicular to the maxillary plane from PTM; Pog=Pogonion, most prominent point on the chin; PTM=pterygomaxillary fissure, the inferior point in the fissure; R=registration point, point of crossing of the greater wing of the sphenoid and planium sphenoidale; S=Sella, estimated centre of the hypophyseal fossa; UI=upper incisor; UI-apex=upper incisor apex; MdPl= mandibular plane, line from Men through Go; MxPl=maxillary plane, line from ANS passing through the posterior hard palate.
less in both cleft groups in relation to the S-N plane (Hannover P=0.004; Brussels P<0.001) and the Ba-N plane (Hannover P=0.035; Brussels P=0.041) compared to the non-cleft group (Table 5). Mandibular protrusion at B point (Ba-N-B, SNB) and Pogonion (Ba-NPog) related to the cranial base were significantly less (P<0.001) in both cleft groups compared to the non-cleft group (Fig. 5, Table 5). Moreover, the Brussels cleft group was significantly more retruded at SNB (P=0.047) compared
16
Swennen et al.
Table 1. Orthopaedic and orthodontic treatment Presurgical orthopaedics Deciduous dentition Mixed dentition
Hannover
Brussels
Passive appliance Removable, active appliance* Removable, active appliance*
Passive appliance Removable, active appliance* Removable, active appliance*
to the Hannover cleft group. Anteroposterior position of the incisors (BaN-UI, Ba-N-LI) was significantly less (P<0.001) in both cleft samples compared to the non-cleft group (Fig. 6, Table 5).
*Removable active appliances are used to correct malocclusion if necessary.
Sagittal relations Table 2. Incidence of secondary surgery in both cleft samples Hannover
Brussels
Males
Females
Males
Females
Sample size
23
13
17
9
Procedure Lip revision Rhinoplasty VPP Fistula closure Bone grafting*
— — 2 3 —
— — — 2 1
5 3 1 9 5
3 1 2 6 3
VPP: velopharyngoplasty; *Secondary alveolar bone grafting.
Evaluation of the maxillomandibular relationship showed no significant differences at the skeletal (ANS-N-Pog) and dento-alveolar (ANB) level. At the dental (overjet) level, a significant difference (P=0.033) was found in the Hannover cleft group compared to the non-cleft group (Table 5). Facial depths Maxillary length (PMP-A), anteroposterior development of the maxillary
Table 3. Mean values and standard deviations of angular, indexed linear measurements and relations in male UCLP and non-cleft children. Results of one-way ANOVA (p) and post-hoc Tukey test
Sample size
Unit
Mean age N-Ba Profile Ba-N-NB Ba-N-ANS Ba-N-A SNA Ba-N-UI Ba-N-LI Ba-N-B SNB Ba-N-Pog Sagittal relations ANB ANS-N-Pog Overjet Facial depths Ba-ANS Ba-A Ba-UI Ba-LI Ba-PMP PMP-A Co-Gn Vertical relations N-ANS N-ANS/N-Men ANS-UI/N-Men ANS-Men/Ba-Pog Men-Li/N-Men R-PMP MdPl/SN MxPl/SN Others N-UI-Ui apex N-S-Ba
Year mm
P
Hannover
Brussels
Non-cleft
23
17
20
Tukey
Mean
SD
Mean
SD
Mean
SD
9.85 94.07
0.52 2.85
10.37 101.08
0.67 6.97
10.25 94.77
0.49 3.38
H-B
H-C
B-C
deg deg deg deg deg deg deg deg deg
0.000 0.205 0.131 0.026 0.000 0.000 0.001 0.000 0.000
82.87 65.04 59.76 79.15 59.13 56.59 55.65 75.13 56.61
4.01 4.43 4.02 4.31 3.19 3.33 2.70 3.49 2.63
84.30 65.32 60.35 78.80 59.47 57.29 54.85 73.12 55.44
4.63 3.31 2.53 3.24 3.03 3.10 2.55 3.38 2.38
89.60 66.90 61.68 81.80 64.53 61.38 58.05 77.93 58.98
4.44 2.39 2.13 3.25 2.98 2.89 2.40 2.95 2.68
ns ns ns ns ns ns ns ns ns
‡ ns ns ns ‡ ‡ † * *
† ns ns * ‡ † † ‡ ‡
deg deg mm
0.071 0.661 0.071
4.04 8.37 2.79
3.57 4.43 2.80
5.65 9.32 4.05
1.90 2.93 2.82
3.78 8.43 4.38
1.53 2.82 0.71
ns ns ns
ns ns ns
ns ns ns
mm mm mm mm mm mm mm
0.068 0.057 0.000 0.000 0.031 0.569 0.311
90.83 86.39 88.20 85.08 39.70 47.28 100.58
3.47 3.47 4.62 3.73 2.18 2.92 4.38
90.98 86.93 89.28 86.02 39.95 47.85 100.29
2.38 2.16 3.59 3.77 2.24 2.22 4.66
92.73 88.38 95.02 90.34 41.41 46.94 102.43
2.30 2.06 3.46 3.34 2.15 2.44 5.07
ns ns ns ns ns ns ns
ns ns ‡ ‡ * ns ns
ns ns ‡ † ns ns ns
mm % % % % mm deg deg
0.070 0.000 0.116 0.000 0.023 0.000 0.000 0.000
46.17 42.84 25.40 66.73 36.00 41.35 35.70 9.54
2.01 1.87 1.35 4.72 1.34 3.98 4.50 3.26
47.14 42.61 25.71 69.23 36.18 38.55 38.35 11.47
2.57 1.45 1.61 4.15 1.34 3.69 4.19 3.37
47.75 45.01 24.66 61.25 34.96 43.90 31.03 6.38
2.13 2.29 1.75 5.13 1.65 2.55 5.47 2.22
ns ns ns ns ns * ns ns
ns † ns † ns ns † †
ns † ns ‡ * ‡ ‡ ‡
deg deg
0.000 0.007
4.91 130.26
3.13 4.86
4.94 132.29
1.64 4.61
9.23 126.98
3.01 5.44
ns ns
‡ ns
‡ †
*P<0.05; †P<0.01; ‡P<0.001; ns: not significant; deg: degrees.
17
Midfacial morphology in UCLP children
Table 4. Mean values and standard deviations of angular, indexed linear measurements and relations in female UCLP and non-cleft children. Results of one-way ANOVA (p) and post-hoc Tukey test
Sample size
Unit
Mean Age N-Ba Profile Ba-N-NB Ba-N-ANS Ba-N-A SNA Ba-N-UI Ba-N-LI Ba-N-B SNB Ba-N-Pog Sagittal relations ANB ANS-N-Pog Overjet Facial depths Ba-ANS Ba-A Ba-UI Ba-LI Ba-PMP PMP-A Co-Gn Vertical relations N-ANS N-ANS/N-Men ANS-UI/N-Men ANS-Men/Ba-Pog Men-Li/N-Men R-PMP MdPl/SN MxPl/SN Others N-UI-Ui apex N-S-Ba
Year mm
P
Hannover
Brussels
Non-cleft
13
9
20
Tukey
Mean
SD
Mean
SD
Mean
SD
10.17 92.04
0.52 4.57
10.46 95.67
0.81 6.72
10.33 92.32
0.35 3.31
H-B
H-C
B-C
deg deg deg deg deg deg deg deg deg
0.450 0.665 0.074 0.000 0.000 0.001 0.011 0.000 0.017
87.08 65.15 60.50 78.04 60.00 57.46 56.35 74.00 57.50
5.03 4.26 3.65 4.03 3.16 3.54 2.71 3.95 2.64
87.28 65.00 59.06 75.89 58.17 56.17 55.22 72.22 57.00
3.30 4.88 1.93 1.98 1.62 4.10 2.82 2.58 2.75
88.80 66.20 61.98 80.83 64.50 61.60 58.80 77.55 60.10
3.90 3.29 3.26 1.66 3.52 3.45 3.37 2.01 3.38
ns ns ns ns ns ns ns ns ns
ns ns ns * † † ns † ns
ns ns ns ‡ ‡ † * ‡ *
deg deg mm
0.635 0.495 0.230
4.08 7.50 3.39
2.82 4.06 1.75
3.67 6.33 2.83
2.78 3.58 4.27
3.30 6.25 4.34
1.55 1.87 1.17
ns ns ns
ns ns ns
ns ns ns
mm mm mm mm mm mm mm
0.212 0.040 0.000 0.004 0.481 0.855 0.005
87.65 87.08 87.37 85.83 39.54 46.03 99.70
11.29 2.96 8.15 3.94 2.22 6.26 5.62
89.75 85.63 87.58 79.88 39.30 46.50 96.43
2.04 1.77 2.01 15.26 2.50 2.45 2.26
91.89 88.58 95.78 90.87 40.94 46.85 103.63
3.00 3.13 4.78 4.38 5.20 2.59 6.07
ns ns ns ns ns ns ns
ns ns † * ns ns ns
ns * † † ns ns †
mm % % % % mm deg deg
0.010 0.047 0.778 0.112 0.738 0.007 0.159 0.000
46.41 43.62 24.99 64.47 34.74 40.94 36.27 9.73
2.44 2.10 1.87 5.09 1.30 5.05 4.12 3.56
48.99 48.27 25.36 61.77 35.74 40.76 37.06 12.61
1.10 8.33 5.34 7.82 5.91 2.39 6.07 2.09
48.69 45.16 24.58 60.21 35.14 44.56 33.78 7.48
2.39 2.00 1.57 4.62 1.53 2.78 4.44 2.26
* * ns ns ns ns ns *
* ns ns ns ns * ns ns
ns ns ns ns ns * ns ‡
deg deg
0.000 0.004
6.35 132.46
2.90 4.86
5.00 135.22
2.44 1.79
9.38 129.53
2.30 4.35
ns ns
† ns
‡ †
*P<0.05; †P<0.01; ‡P<0.001; ns: not significant; deg: degrees.
bone (Ba-ANS) and pharyngeal depth (Ba-PMP), indicating the anterior position of the maxilla, showed no significant differences between both cleft groups and the non-cleft group (Table 5). However, antero-posterior development of the dento-alveolar process (Ba-A) was significantly less in both cleft groups compared to the control group (Hannover P=0.012; Brussels P=0.013) (Table 5). Measurements indicating the forward protrusion of the upper (Ba-UI) and lower (Ba-LI) incisors were significantly less (P<0.001) in both cleft groups compared to the matched controls (Table 5). These findings are consistent with the above mentioned angular variables (Ba-N-UI, Ba-N-LI) and can be explained by the more upright inclination of upper (N-UIUiapex significant at P<0.001 for both Hannover and Brussel cleft groups)
(Table 5) and lower incisors. Both cleft groups showed a significantly smaller mandibular length (Co-Gn) compared to the non-cleft group (Hannover P=0.048; Brussels P=0.005) (Table 5). Vertical relations Posterior maxillary height (R-PMP) was significantly decreased in both cleft groups compared to the non-cleft group, but more so in the Brussels cleft group (P<0.001) (Table 5). There was a significant decrease in anterior maxillary height (N-ANS) in the Hannover cleft group compared to the non-cleft (P=0.001) and Brussels cleft (P=0.026) groups (Table 5). The mandibular (MdPl/SN) and maxillary (MxPl/SN) planes were more open in the Hannover cleft group compared to the control group (P=0.005 and P<0.001, respect-
ively), but even more open in the Brussels group (P<0.001 and P<0.001, respectively) (Table 5) (Fig. 7). Moreover, the inclination of the maxillary plane reached a significant value (P=0.008) in the Hannover and Brussels comparison data (Table 5). The more open mandibular plane in the Brussels cleft children explains the above mentioned more retruded mandible (SNB) in the Brussels group (Table 5). The anterior maxillary height related to the total face height (N-ANS/N-Men) was the only variable that showed a difference of gender and was therefore not pooled. The contribution of the anterior maxillary vertical height (ANS-UI/NMen) and anterior mandibular vertical height (Me-Li/N-Men) to the total facial height showed in both cleft groups no significant differences compared to the non-cleft group (Table 5). The
18
Swennen et al.
Table 5. Mean values and standard deviations of angular, indexed linear measurements and relations in pooled UCLP and non-cleft children. Results of one-way ANOVA (p) and post-hoc Tukey test
Sample size
Unit
Mean Age N-Ba Profile Ba-N-NB Ba-N-ANS Ba-N-A SNA Ba-N-UI Ba-N-LI Ba-N-B SNB Ba-N-Pog Sagittal relations ANB ANS-N-Pog Overjet Facial depths Ba-ANS Ba-A Ba-UI Ba-LI Ba-PMP PMP-A Co-Gn Vertical relations N-ANS ANS-UI/N-Men ANS-Men/Ba-Pog Men-Li/N-Men R-PMP MdPl/SN MxPl/SN Others N-UI-Ui apex N-S-Ba
Year mm
P
Hannover
Brussels
Non-cleft
36
26
40
Tukey
Mean
SD
Mean
SD
Mean
SD
9.97 93.34
0.53 3.64
10.40 99.21
0.70 7.24
10.29 93.54
0.42 3.53
H-B
H-C
B-C
deg deg deg deg deg deg deg deg deg
0.000 0.170 0.016 0.000 0.000 0.000 0.000 0.000 0.000
84.39 65.08 60.03 78.75 59.44 56.90 55.90 74.72 56.93
4.79 4.31 3.85 4.18 3.16 3.39 2.69 3.65 2.64
85.33 65.21 59.90 77.79 59.02 56.90 54.98 72.81 55.98
4.40 3.83 2.38 3.16 2.67 3.44 2.60 3.10 2.57
89.20 66.55 61.83 81.31 64.51 61.49 58.43 77.74 59.54
4.15 2.86 2.72 2.60 3.22 3.14 2.91 2.50 3.06
ns ns ns ns ns ns ns * ns
‡ ns * † ‡ ‡ ‡ ‡ ‡
† ns * ‡ ‡ ‡ ‡ ‡ ‡
deg deg mm
0.080 0.497 0.043
4.06 8.06 3.01
3.28 4.26 2.47
4.96 8.29 3.63
2.39 3.42 3.36
3.54 7.34 4.36
1.54 2.61 0.95
ns ns ns
ns ns *
ns ns ns
mm mm mm mm mm mm mm
0.057 0.004 0.000 0.000 0.054 0.780 0.004
89.68 86.63 87.90 85.35 39.64 46.83 100.27
7.33 3.27 6.03 3.77 2.16 4.38 4.80
90.56 86.48 88.69 83.90 39.72 47.38 98.95
2.30 2.09 3.20 9.62 2.31 2.34 4.36
92.31 88.48 95.40 90.61 41.17 46.90 103.03
2.67 2.62 4.13 3.85 3.94 2.48 5.55
ns ns ns ns ns ns ns
ns * ‡ ‡ ns ns *
ns * ‡ ‡ ns ns †
mm % % % mm deg deg
0.001 0.178 0.000 0.212 0.000 0.000 0.000
46.26 25.26 65.92 35.55 41.20 35.90 9.61
2.14 1.54 4.91 1.45 4.33 4.31 3.32
47.78 25.59 66.65 36.03 39.31 37.90 11.87
2.33 3.29 6.61 3.52 3.42 4.84 3.00
48.22 24.62 60.73 35.05 44.23 32.40 6.93
2.28 1.64 4.85 1.57 2.65 5.11 2.28
* ns ns ns ns ns †
† ns ‡ ns † † ‡
ns ns ‡ ns ‡ ‡ ‡
deg deg
0.000 0.000
5.43 131.06
3.08 4.91
4.96 133.31
1.90 4.08
9.30 128.25
2.64 5.03
ns ns
‡ *
‡ ‡
*P<0.05; †P<0.01; ‡P<0.001; ns: not significant; deg: degrees.
relationship of the anterior vertical mandibular height to Ba-Pog was in both cleft groups significantly increased (P<0.001) (Table 5). The cranial base angle (N-S-Ba) was significantly increased in both cleft groups but more so in the Brussels cleft group compared to the non-cleft group (Hannover P=0.032; Brussels P<0.001) (Table 5). Secondary surgery
Data on secondary surgery performed during the investigation period are listed in Table 2. In the Hannover cleft group no lip revision or rhinoplasty was performed. A total of eight (30.8%) children in the Brussels cleft group underwent lip correction at a mean age of 4.382.13 years, while four (15.4%) children had a rhinoplasty at 4.832.71 years (no septoplasty). A velopharyngoplasty was
carried out in two (5.6%) children in the Hannover cleft group and in three (11.5%) children in the Brussels cleft group at a mean age of 7.752.19 and 5.070.32 years, respectively. In the Hannover cleft group, a total of five (13.9%) children underwent closure of oronasal fistulas at a mean age of 3.040.63 years: three were closed with a vomer flap, one using a von Langenbeck palatoplasty and one with a veloplasty according to Widmeier. In one child, fistula closure with a vomer flap was not successful and a von Langenbeck palatoplasty was performed. A total of eight (22.2%) children had fistulas (four oronasal, four vestibulonasal) that were not closed during the investigation period because they caused no functional problems. In the Brussels group, however, all fistulas were closed; a total of 17 fistula closures (eight oronasal,
nine vestibulonasal) were performed in 15 (57.7%) children at a mean age of 3.311.57 years. Closure of vestibulonasal fistulas was carried out in four children with a hinge flap, two with a small rotational flap, one with a vomer flap, one with a conventional primary suture and one during alveolar bone grafting. Oronasal fistulas were closed in four cases through a vomer flap, in two with a hinge flap, in one with a small rotation flap and in one with a conventional suture. In one child, closure of an oronasal fistula with a primary suture was not successful and a small rotation flap was performed. During the period of investigation, only one (2.8%) child underwent secondary alveolar bone grafting in the Hannover cleft group at the age of 5.5 years, while in the Brussels group eight (30.8%) children had bone grafting at a mean age of 8.41.05 years.
Midfacial morphology in UCLP children Maxillary protrusion SNA Ba-N-ANS Ba-N-A
90.00
80.00
81.31
Degrees
78.75
77.79
70.00 66.55 65.21
65.08 61.83
60.00
50.00
60.03
Non-cleft
Hannover
59.90
Brussels
Fig. 4. Comparison of maxillary protrusion in pooled UCLP and non-cleft patients.
Mandibular protusion SNB Ba-N-Pog Ba-N-B
80.00 77.74 74.72 72.81
Degrees
70.00
60.00 59.54 58.42 56.93 55.90
55.98 54.98
50.00
Non-cleft
Hannover
Brussels
Fig. 5. Comparison of mandibular protrusion in pooled UCLP and non-cleft patients.
Discussion Surgical technique, timing and sequence of cleft closure and the application of presurgical orthopaedics and/or adjunctive orthodontics in the treatment of children with UCLP in reference to midfacial growth impairment, have been major concerns of cleft centres throughout the last decades. Different treatment protocols may have their specific influence on cranio-facial morphology. In
this study we compared cranio-facial morphology of children with UCLP treated according to different surgical protocols, particularly regarding timing and sequence but having identical orthopaedic/orthodontic regimens. In Hannover, lip closure is followed by a two-stage palatoplasty5,9,10,22,23,28 (Fig. 1). The children in the Brussels cleft group in this study underwent cleft closure following a new approach
19
advocated by Rene´ Malek: early veloplasty followed by simultaneous lip and hard palate repair4,12,13,20 (Fig. 2). Cephalometric analysis revealed that the children with UCLP in the Hannover cleft group did not differ significantly from the children with UCLP in the Brussels group in the anteroposterior dimension of the midface. The angle of maxillary protrusion (Ba-N-ANS) reflecting the forward translation of the maxilla and the length of its basal bone giving a good indication of anteroposterior growth was excellent in both cleft groups. The effective length of the maxilla (PMP-A), which includes both basal maxillary length and the position of the alveolus on it, was likewise excellent in both cleft groups. However, as far as the vertical dimension is concerned, a significant difference (MxPl/SN at P=0.008) was found in the Hannover– Brussels comparison data. Although the maxillary plane was more open in both cleft groups compared to the non-cleft group, it was significantly more open in the Brussels group compared to the Hannover group. This can be explained because the posterior maxillary development (R-PMP) was significantly less in the Brussels group (P<0.001), while the anterior maxillary development (N-ANS) was significantly less in the Hannover group (P=0.001) (Table 5). The posterior vertical development of the maxilla (R-PMP) was decreased in both cleft groups; this is consistent with the literature2,19. However, the posterior maxillary vertical height was significantly less in the children in the Brussels cleft group (Table 5). In this group, soft palate closure (3.040.20 months) was performed much earlier compared to the children in the Hannover cleft group (32.254.29 months). The soft palate repair according to Malek consists of a veloplasty using a transverse incision at the level of the posterior edges of the palatal shelves with the dissection performed posterior to the neurovascular bundle. To allow easier closure of the cleft at the level of the posterior margin of the palatal shelves where the tension is the greatest, a small mucoperiosteal vomer flap is raised, rotated 180 and sutured to the nasal mucosa of the velum12,13. The inhibiting effect of soft palate repair on posterior maxillary vertical height has been described in the literature19. E & H6 showed that in the first 3 years the direction of maxillary growth is primarily vertical. We suggest that the Malek soft palate repair is probably related with more
20
Swennen et al. Dental relations Ba-N-UI Ba-N-LI UI-UIapex
70.00 64.51
60.00
61.49 59.44
59.02
56.90
56.90
Degrees
50.00
40.00
30.00
20.00
10.00 9.30 5.43
0.00
Non-cleft
Hannover
4.96
Brussels
Fig. 6. Comparison of dental relations in pooled UCLP and non-cleft patients.
Inclination of maxilla and mandible 40.00
MxPI/SN MdPI/SN 37.90
35.00
35.90 32.40
30.00
Degrees
25.00 20.00 15.00 11.87
10.00 9.61
5.00 0.00
6.93
Non-cleft
Hannover
Brussels
Fig. 7. Comparison of maxillar and mandibular plane inclination in pooled UCLP and non-cleft patients.
cicatrised soft tissues, which might provide resistance to posterior vertical growth. In combination with the earlier timing of soft palate repair, this might explain the less vertical posterior maxillary development in the Brussels group. The mandible was more open in both cleft groups and more in the Brussels group (P<0.001) (Table 5). Presurgical orthopaedic treatment with a passive appliance has been reported to influence
the inclination of the mandible17,19,24. Mø et al.17 hypothesized that the tongue position is altered by the appliance and therefore the mandible is inclined in a more backward direction. Although both cleft groups in this study underwent presurgical orthopaedic treatment with a passive appliance, we do not believe that these appliances are responsible for the more open mandibular angle. We suggest that the increased
inclination of the mandibular plane in both cleft groups is a consequence of mouth breathing and maxillary morphology. Therefore, the increased inclination of the maxillary plane in the Brussels cleft group explains its more open mandibular plane. It has been reported that vomerine mucosal flaps result in a reduction of maxillary vertical height, a more retruded maxilla and an overclosure of the mandible due to the scar tissue adhesions that unite the vomer to the palatal shelves at the site of the repaired cleft3,14,18. Although correction of the hard palate defect using vomer flaps was performed in the children in the Hannover and Brussels cleft groups, both cleft groups showed excellent anteroposterior maxillary morphology and an even more open mandible. However, as already mentioned, both cleft groups showed a decreased vertical development. The veloplasty according to Malek, which includes a small vomer flap, might be an additional cause for the decreased posterior development in the Brussels group. The results of this study support the hypothesis of Ross that variations in the timing of hard and soft palate repair within the first decade do not influence midfacial growth in the antero-posterior dimension19. Contrary to R, this study shows that variation in the timing of soft palate repair might influence posterior vertical growth. However, it is not entirely clear whether the decreased posterior maxillary development in the Brussels group is caused by the earlier timing of soft palate closure or by the surgical technique. This point requires further analysis. There may be many reasons why the UCLP children of both cleft centres in this study have good midfacial morphology. Many cleft routines include procedures that have been assumed to be associated with impaired midfacial growth, such as primary bone grafting, periosteal grafts and active presurgical orthopaedics19,26,29. The treatment protocols of both cleft centres in this study did not include these procedures. As far as palatal repair is concerned, the surgical procedures of both cleft centres are characterized by the absence of denudation of the thick palatal mucoperiosteum, which is associated with growth-impairing palatal scar tissue14,18. Furthermore, we agree with S et al.25,26 that the good results obtained in a cleft centre most likely depend more on its well-organized and centralized structure that ensures uniformity in its
Midfacial morphology in UCLP children treatment protocol, rather than on individual components of its treatment protocol. A higher number of secondary revisionary procedures were found in the Brussels cleft group (Table 2). Interpretation of these data, however, should be done with caution. During the investigation period, no secondary lip or nose corrections were performed in the children in the Hannover cleft group because at that time those procedures were delayed until they had reached the age of 13 years. As far as fistulas are concerned, the Brussels cleft group showed a higher incidence of fistula closures (57.7%). It is known that the Malek protocol is associated more frequently with palatal fistulas4,12, although it provides excellent craniofacial growth20. The fact that all fistulas were closed in the Brussels group also suggests that patient and surgeon requirements may be different in both cleft centres. Furthermore, the combination of secondary surgical procedures is essential in the Brussels treatment philosophy of cleft children. Hence, closure of fistulas was often combined with small lip or nose corrections. Because our primary goal in this study was to compare the surgical treatment protocols of two cleft centres, we attempted to provide detailed surgical data on the primary and secondary procedures of both centres. However, the potential influence of these secondary procedures on cranio-facial morphology was not analyzed in this study. Studies comparing the influence of different surgical protocols on craniofacial morphology in cleft patients are often likely to be unreliable because of methodological biases. S & S24 defined potential biases as follows: detection, exclusion, analysis, reporting, susceptibility and proficiency bias. In this study, we did attempt to overcome those biases in that: 1. The cleft databases of both cleft centres were consulted and all patients that fulfilled the abovementioned criteria were included (detection and exclusion bias) 2. All cleft patients were compared to a non-cleft control group, matched according to age and gender. Linear measurements were adjusted to the cranial base length (Ba-N) and reliability of the cephalometric analysis was proved through calculations of the method error (analysis bias)
3. The complete data (cephalometric measurements, age, primary and secondary surgery) of both cleft samples were reported (reporting bias) 4. Susceptibility bias could only be partially resolved. Although both cleft samples were equivalent in type (complete non-syndromic UCLP) and race (Caucasian), the difference in severity of the initial cleft could not be assessed because infant dental casts were not available for all children 5. Although both cleft centres meticulously followed a well-defined surgical protocol, different surgeons were involved in both centres, so that proficiency bias was only partially resolved. Proficiency bias, however, cannot be solved even in welldesigned prospective randomized intercentre studies, as the surgical skill of surgeons will inevitably be different. Furthermore, the possibility of sampling bias exists if two or more treatment protocols are being used concurrently in one centre26. Contrary to the Hannover cleft group, there is clearly a sample bias in the Brussels group, as only the Malek cases were included and the all-in-one cases were excluded in this study. Despite our effort to perform a welldesigned comparative study, this study still has several shortcomings. Firstly, cranio-facial morphology was evaluated and compared cross-sectionally at 10 years of age, before the beginning of their pubertal growth spurt. As the cephalometric analysis we used does not take into account cranial predisposition, errors of interpretation are also possible18. Secondly, because of the retrospective design and the strict inclusion criteria, the size of the cleft groups is still too small in this study to discern statistically minor surgical effects using multiple comparison tests. Finally, evaluation of cranio-facial growth through longitudinal architectural cranio-facial cephalometry, anthropometry and dental casts analyses combined with clinical findings, video analysis, speech and hearing evaluation would be the ideal setting to compare the aesthetic and functional outcome between different cleft centres. Acknowledgment. The authors thank Professor Hartmut Hecker, Department of Biometry, Medical University Hannover, Germany, for his assistance
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