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European Journal of Orthodontics, 2017, 641–645

doi:10.1093/ejo/cjx019
Advance Access publication 25 March 2017

Original article

Three-dimensional evaluation of the maxillary


arch and palate in unilateral cleft lip and palate
subjects using digital dental casts
Chiara Generali1, Jasmina Primozic2, Stephen Richmond3,
Maria Bizzarro1, Carlos Flores-Mir4, Maja Ovsenik2 and Letizia Perillo1
Multidisciplinary Department of Medical-Surgical and Dental Specialties, University of Campania “Luigi Vanvitelli”,
1

Naples, Italy, 2Department of Orthodontics and Jaw Orthopaedics, Medical Faculty, University of Ljubljana, Ljubljana,
Slovenia, 3Dental Health and Biological Sciences, Dental School, Cardiff University, Cardiff, UK and 4Department of
Dentistry, University of Alberta, Edmonton, Alberta, Canada

Correspondence to: Letizia Perillo, Multidisciplinary Department of Medical-Surgical and Dental Specialties, University of
Campania “Luigi Vanvitelli”, Via Luigi De Crecchio, 6, 80138 Naples, Italy. E-mail: letizia.perillo@unicampania.it

Summary
Objectives:  To assess arch width, palatal surface area, and volume in surgically treated unilateral
cleft lip and palate (UCLP) in mixed dentition children in comparison with non-cleft lip and palate
(NCLP) children using a 3D laser scanning.
Materials and Methods: 38 subjects (Caucasian origin), 5.63–11.9  years of age (mean,
9.33  ±  1.67  years), were included. 19 in each group (UCLP and NCLP). Digital dental casts were
obtained using a 3 Shape R700 laser scanner. Intercanine and intermolar widths (cusp and gingival
levels), palatal surface area and volume were measured. An independent sample Student’s t-test
and an ANOVA were undertaken with significance level set as P < 0.05.
Results:  Intercanine widths at the cusp (5.60 mm; P < 0.001) and at the gingival level (3.11 mm;
P = 0.014), palatal area (141.5 mm2; P = 0.009) and volume (890.7 mm3; P = 0.029) were significantly
lower in the UCLP compared to the control group.
Limitations:  A smaller part of the subjects was in late mixed dentition phase. To overcome this
limitation a matched control group was used. In seven subjects with UCLP, some teeth were missing,
which might have had an influence on the dental measurements. However, these subjects could
not be excluded because eliminating more severely affected subjects, would have introduced bias.
Conclusions:  Three-dimensional evaluation of the maxillary arch and palate highlighted significant
differences between UCLP and non-UCLP subjects in mixed dentition phase, suggesting that
orthopaedic maxillary expansion is advisable in UCLP.

Introduction and palate are frequently seen in CLP individuals (3, 4). The mor-
phology of the upper arch and palate has been widely investigated
Cleft lip and palate (CLP) is the most common craniofacial malfor-
in cleft patients mainly using conventional two-dimensional dental
mation that orthodontists will encounter (1) with an incidence of 1
cast analysis (5, 6). This method, although reliable, is very time-
out of 700 newborns (2). It arises due to the failure of fusion of the
consuming and limited to provide reliable volumetric data (7, 8).
maxillary processes and/or palatal shelves between the 4th and 12th
More recently, several studies used different three-dimensional (3D)
week of embryogenesis (2). Several dental, skeletal, aesthetic, and
imaging systems to accurately record the upper arch and palate in
functional discrepancies along with a constricted upper dental arch
cleft subjects (9–22). However, a high variability in subject selection

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642 European Journal of Orthodontics, 2017, Vol. 39, No. 6

and in the parameters examined, as well as some studies including and intermolar maxillary transverse widths at the cusp and gingival
only newborns (9, 10) without a control group (11–13), and assess- levels were measured (Figure  1). In addition, palatal surface area
ing only the palatal shape but not volume (14–17), are available in (Figure 2) and volume (Figure 3) were also considered. The palatal
the literature. A small number of studies (18–22) used 3D analysis surface area and the palatal volume were calculated with the gingival
to evaluate the upper arch and palate in cleft and non-cleft subjects and distal planes used as boundaries for the palate (7, 23). The gin-
during the period of expected orthodontic treatment. Comparisons gival plane was created by connecting the midpoints of the dentog-
of arch widths, palatal surface areas and volumes have been also ingival junction of all upper erupted deciduous and permanent teeth.
performed between untreated cleft children and controls in the The distal plane was created through two points at the distal of the
deciduous dentition phase (18). Other studies reported data regard- first upper permanent molars perpendicular to the gingival plane.
ing treated cleft patients in the permanent dentition, but they did
not consider palatal volume (19–22) and did not include a matched Statistical analysis
control group. However, to our knowledge, there is still insufficient The Statistical Package for Social Science 20.0 (SPSS Inc., Chicago,
evidence regarding arch width, palatal surface area and volume of Illinois, USA) was used for statistical analysis. Data were tested for
CLP patients during the mixed dentition phase, when a significant normal distribution according to Levene’s test. Analysis of variance
portion of the cleft orthodontic/orthopaedic treatment is likely to (ANOVA) test considered the effect of age, sex, group and their inter-
be started. actions on the parameters. An independent samples Student’s t-test
The aim of this study was therefore to evaluate and compare arch was used to calculate the statistical differences between the UCLP
width, palatal surface area, and volume of unilateral CLP (UCLP) and NCLP groups. The results were considered to be significant at
subjects and non-CLP subjects (NCLP) in the mixed dentition phase values P < 0.05.
using 3D laser scanning.

Method error
Material and methods The ICC and error of the measurements (in percentages) were
calculated and were for palatal surface area 1.0 and 2.1 per cent
Approval for this cross-sectional study was granted by the institu- (1.0–3.5%), while for palatal volume were 1.0 and 2.8 per cent
tional review board of the University of Campania “Luigi Vanvitelli”, (1.3–4.7%), respectively. Those differences were judged to be clini-
Naples, Italy and the Slovenian National Medical Ethics Committee. cally irrelevant.

Material and study design


A total of 38 Caucasian subjects aged 9.33 ± 1.67 years were included. Results
The UCLP group, selected from patients referred to the Division A post hoc analysis of the obtained power for each variable with
of Orthodontics at the University of Campania “Luigi Vanvitelli”, statistical significant differences showed a power of 99.9 per cent for
Naples, Italy, consisted of a sample of 19 subjects with a complete both intercanine measurements, of 66.6 per cent for the area differ-
UCLP (9 girls, 10 boys; aged 9.14 ± 1.4 years), all in the mixed denti- ence and of 82.9 per cent for the volumetric difference.
tion phase. All patients were treated at the Division of Maxillofacial The mean values and standard deviations of the intercanine and
Surgery at the University of Campania “Luigi Vanvitelli”, Naples, intermolar distances, palatal surface areas, and volumes for each
Italy, by the same surgeon, using the same protocol and method group and P-values for between group comparisons are reported in
as follows: lip surgery at 6  months according to the Delaire tech-
nique, soft palate surgery at 12 months and hard palate surgery at
18 months by pushback with two flaps. No infant orthopaedics was
carried out prior surgery, and no orthodontic/orthopaedic treatment
was performed after it. Furthermore, in none of the individuals an
alveolar bone graft was performed. Subjects with other cranio-facial
syndromes or previous or concomitant orthodontic treatment were
excluded.
The NCLP group consisted of 19 untreated matched non-cleft
subjects without malocclusion. These children were recruited from
a longitudinal growth study (7) carried out at the Department of
Pedodontics of the Dental Polyclinic of Kranj, Slovenia and they
were matched for sex, chronological age, and dentition phase. The
subjects had good general health, normal occlusion and no func-
tional impairment.

Three-dimensional evaluation of upper arch and


palate morphology
The study casts were scanned using a 3D scanner (3Shape
R700TM) with a reported manufacturing accuracy of 20 microns
(www.3shape.com). The 3D data were imported to a reverse model-
ling software package Rapidform™ 2006 (INUS Technology, Tokyo, Figure  1.  Assessment of the upper arch on digital models. Intercanine and
Japan). Each scan of a study cast was further manually pre-processed intermolar maxillary arch widths assessed at the cusp (red lines) and gingival
to remove unwanted data artefacts from the analysis. Intercanine (blue lines) level.

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C. Generali et al. 643

measurements have been previously reported as reliable indicators


of palatal and maxillary arch growth (22). The anatomical charac-
teristics of the upper arch and palate in UCLP subjects have been
previously evaluated by measuring only the intercanine and intermo-
lar distances (5, 6). Similarly to previous reports, the results of our
study confirmed that UCLP is associated with decreased intercanine
widths both at cusp and gingival levels. However, in the mixed den-
tition phase, no significant reduction of the intermolar widths was
measured. As expected, the differences between the UCLP and NCLP
groups were significant only for the intercanine widths because the
UCLP investigated in this study were complete unilateral clefts
extending to the anterior part of the palate beyond the incisive fora-
men. Thus, the less marked narrowing of intermolar widths in the
upper arch may be a reflection of a more significant hypoplasia with
compression of the anterior compared to the posterior portion of the
maxilla (21). In fact, Smahel et al. (20) reported reduced dentoalveo-
lar widths in isolated palatal clefts in the upper arch, but the reduc-
tion increased in the posterior direction compared with controls. In
the anterior part, the arch was therefore wider while in the posterior
part it was narrower than in UCLP, probably due to less compression
Figure 2.  Palatal surface area on digital models.
of the intact anterior part of the arch. On the other hand, a compari-
son between bilateral clefts and UCLP (22) evidenced an almost dou-
ble narrowing of the upper arch at the level of the canines in bilateral
clefts, although between molars, the difference was less evident. As
the assessment of maxillary morphology based on interdental meas-
urements does not fully consider the 3D maxillary morphological
characteristics and could be biased due to axial inclination of the
first molars and/or the alveolar bridge, palatal surface area and vol-
ume were used in the present study to better describe the maxillary
morphology. For this purpose, 3D laser technology has been used in
the present study as previously suggested (24). Both the palatal sur-
face area and volume were significantly smaller in UCLP than in the
NCLP group. Generally, the results of the present investigation are
in agreement with previous 3D studies (18–22). However, a direct
comparison with our findings was not possible due to the different
cleft types, size and age of samples, and 3D methods used. Okazaki
et al. (18) reported a greater reduction in arch widths, palatal surface
area and volume, but in the deciduous dentition phase and using
Moiré Fringes technique. Kilpelainen et  al. (19) and Smahel et  al.
(20–22) reported data in treated cleft subjects in the permanent den-
tition phase and did not include palatal volume in their investiga-
tion. Kilpelainen et al. (19) examined a very large sample, but with
a wide range of age, dentition and type of clefts using like Okazaki
Figure 3.  Palatal volume on digital models. The palate was delimited using a
et al. (18), the Moiré Fringes technique. Smahel et al. (21), although
gingival plane (blue) and a distal plane (light blue).
using a different 3D methodology, analyzed also UCLP, but the sub-
jects were in the permanent dentition phase. To our knowledge, the
Table 1. Intercanine widths were significantly smaller at the cusp level present study is the first attempt to evaluate the arch width, pala-
(5.60 mm; P < 0.001) and at the gingival level (3.11 mm; P = 0.014). tal surface area and volume in untreated UCLP subjects compared
On the other hand, no significant differences were observed between with untreated healthy children in mixed dentition phase using a 3D
the intermolar widths measured either at the cusp or gingival level (all analysis. Therefore we believe it adds a missing piece in the puzzle
p>0.05). Palatal surface area (141.5 mm2) and volume (890.7 mm3) in about maxillary skeletal and dentoalveolar changes in CLP individu-
the UCLP group were significantly smaller than in the NCLP group by als during the development of the dentition. It has to be considered
18 per cent (P=0.018) and 31 per cent (P=0.005), respectively. that the constriction in arch widths, palatal surface area and volume
in UCLP could be a result of a combination of both iatrogenic and
intrinsic factors (25) or other factors (9). In the literature, different
Discussion opinions regarding the importance of each factor (26) have been dis-
Orthopaedically or orthodontically untreated UCLP subjects in the cussed. It has been reported that a narrower upper arch is more often
mixed dentition show a significantly different upper arch morphol- seen in patients with UCLP that have undergone surgery treatment
ogy, with a higher degree of maxillary constriction. In this study, the (27) and that this narrowing is related to surgical closure (28). On
upper arch widths and palatal surface area and volume in UCLP the other hand, reduced upper widths were also found in untreated
were significantly smaller compared to NCLP subjects. These 3D adults with CLP (29). This would suggest that constriction of the

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644 European Journal of Orthodontics, 2017, Vol. 39, No. 6

Table 1.  Upper arch and palate parameters for UCLP and NCLP groups and respective group comparisons

UCLP group NCLP group

Parameter Mean SD Mean SD P Valuea

Intercanine width
  Cusp (mm) 27.4 3.4 33.0 2.3 0.000
  Gingival (mm) 22.4 4.2 25.5 1.8 0.014
Intermolar width
  Cusp (mm) 44.9 11.8 48.0 11.8 0.417
  Gingival (mm) 31.3 8.6 32.8 8.2 0.580
  Palatal surface area (mm2) 635.4 188.5 776.9 163.7 0.018
  Palatal volume (mm3) 1980.7 945.5 2871.4 898.7 0.005

a
Independent t-test was performed for comparison of the mean differences between the two groups. SD indicates standard deviation.

upper arch may be not only a result of early surgical procedures subjects. It seems that the presence of the cleft itself has a larger
(29), but also could be due to deficient palatal growth also due to the influence on the morphologic characteristics of the maxillary arch
pressure of surrounding tissues on maxillary segments, or due to the compared to sex (29).
unfilled space between palatal halves (22).
Moreover previous studies (30–32) reported also an osteogenic Limitations
defects in CLP patients showing an alteration of the Wnt/GSK3β/β- The majority of the subjects included herein were in the early mixed
catenin signalling which is involved in the osteogenic differentiation dentition phase while a smaller part was in late mixed dentition
of palatal shelves fusion. phase. To overcome this limitation a matched control group was
Another point worth consideration is the potential effect of scar- used. In seven subjects with UCLP, some teeth such as incisors and
ing on the palatal soft tissue after the cleft surgery. The strained tis- canines were missing on one or both sides, which might have had an
sue may not allow for proper palate transversal development (33, influence on the intercanine dental measurements. However, these
34). subjects could not be excluded because eliminating more severely
Since the present study investigated the upper arch and pala- affected subjects, with even smaller arch widths would have intro-
tal vault morphology in 9-year-old subjects in the mixed dentition duced bias.
phase, a clear influence of the impact of either iatrogenic or intrinsic
factors could not be determined. The decreased arch widths, palatal
surface area and volume are consistent with a lack of space for the Conclusions
tongue in UCLP patients, which could further influence maxillary Subjects with UCLP have significantly reduced intercanine max-
growth and development. In fact, although there is no data in the lit- illary arch widths, while intermolar widths are similar to those
erature on the postnatal size of the tongue in clefts, Kimes et al. (35) measured in matched subjects without malocclusion. Furthermore,
found a larger volume and length of the tongue as compared with a significantly smaller palatal surface area and volume is seen in
normal specimens prenatally in nine foetuses with cleft lip and pal- UCLP subjects in whom early orthodontic or orthopaedic treatment
ate. This suggests that smaller palatal volume is not associated with was not performed. Therefore, based on the results of the present
adaptive reduction of the tongue size (26), but the lack of space for study expansion of the anterior part of the maxillary arch could be
the tongue may contribute to several different problems including beneficial.
oral breathing, opening of the bite, posterior rotation of the mandi-
ble, speech and articulatory difficulties and speech is one of the main
concerns of cleft patients (18, 20). Conflict of Interest
Moreover, a narrower maxilla may lead to a lower tongue posi-
None to declare.
tion with an increase of the intermolar mandibular widths contribut-
ing to the development of a Class III malocclusion in CLP patients
(36). References
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