967016 JOO Journal of OrthodonticsJohal et al.

Scientific Section

Journal of Orthodontics

The influence of mild versus severe 1­–9

https://doi.org/10.1177/1465312520967016
DOI: 10.1177/1465312520967016
© The Author(s) 2020
hypodontia on facial soft tissues? Article reuse guidelines:
sagepub.com/journals-permissions
A three-dimensional optical laser journals.sagepub.com/home/joo

scanning-based cohort study

Ama Johal , Eiman Hasan, Li Fong Zou, Ferranti Wong,

Shakeel Shahdad and Ryad Al-Klash

Abstract
Objective: To determine whether there are differences in the facial soft tissue morphology between participants with
mild (up to two) or severe (six or more) hypodontia.
Design and Setting: A prospective hospital-based cohort study.
Participants and Methods: Ninety-two participants, aged 11–16 years, with confirmed hypodontia were recruited.
Participants were sub-grouped based on the severity (mild, two or less and severe, six or more) and distribution of the
missing teeth and age. They underwent a three-dimensional (3D) optical surface scan of the facial soft tissues. Facial
surface scans were compared quantitatively, applying landmark measurements and surface-based analysis.
Results: In total, 92 participants, with an equal distribution between the mild (n=46) and severe (n=46) categories, were
recruited. Patients with severe hypodontia displayed a reduced alar base, lower facial height, nasolabial angle (P = 0.02)
and transgonial width (P < 0.001) compared to those with milder hypodontia. Furthermore, significant differences were
observed between mild-male and severe-female groups regarding alar base, lower anterior face height and transgonial
width and between mild-male and mild-female groups regarding nasolabial angle and transgonial width.
Conclusion: Significant reductions were seen in the 3D soft tissue morphology of participants with severe hypodontia,
in terms of the nasolabial angle, lower facial height, alar base and transgonial widths, emphasising the importance of using
facial scanning as a relatively simple non-invasive method of assessment.

Keywords
restorative–orthodontic interface, interdisciplinary treatment, three-dimensional imaging, imaging and cephalometry,
hypodontia, aetiology of malocclusion and growth

Date received: 26 June 2020; revised: 14 September 2020; accepted: 28 September 2020

Introduction further classified by its severity and/or aetiological associa-

Hypodontia is defined as the developmental absence of at
least one permanent tooth, excluding third molars, with a
reported prevalence in the range of 6.4%−8.5% in the perma-
nent dentition and importantly is regarded as the most com-
mon congenital dental anomaly (Endo et al., 2006; Khalaf
et al., 2014). A range of additional terms have been applied to
describe the absence of teeth and these are based on their
numbers. Oligodontia is defined as the absence of six or
more teeth, excluding third molars, whereas anodontia is the
complete absence of the entire dentition. Hypodontia can be
tion. Based on severity, it can also be classified into mild
(two or fewer missing teeth), moderate (3–5 missing teeth)
and severe (six or more missing teeth) forms (Hobkirk et al.,
Institute of Dentistry, Bart’s and The London School of Medicine and
Dentistry, Queen Mary University of London, London, UK
Corresponding author:
Ama Johal, Institute of Dentistry, Turner Street, Whitechapel, London
E1 2AD, UK.
Email: a.s.johal@qmul.ac.uk
2 Journal of Orthodontics 
1994). Polder et al. (2004) reported that 83% of individuals
with hypodontia have an absence of one or two permanent
teeth and only 0.14% show an absence of more than six per-
manent teeth. Alternatively, hypodontia can be regarded as
either syndromic or non-syndromic. Syndromic is more
commonly associated with severe hypodontia and can be
either sporadic or familial, within a family pedigree, whereas
non-syndromic is more commonly seen in the permanent
dentition and ranges in severity (Cobourne, 2007).
When attempting to understand the facial form of par-
ticipants with hypodontia, there are conflicting findings
concerning the relationship between the craniofacial struc-
tures in the anteroposterior (AP) and vertical planes, and
the severity of hypodontia. In the AP plane, hypodontia
may present with a skeletal I, II or III pattern (Bondarets
and McDonald, 2000; Celikoglu et al., 2010; Chung et al.,
2000). Others have reported this to be associated with
bimaxillary retrognathism (Ben-Bassat and Brin, 2003)
and/or maxillary hypoplasia (Sarnäs and Rune, 1983). In
terms of vertical changes, some observed an increased in
vertical dimension (Acharya et al., 2010; Wisth and
Thunold, 1974), while others have reported decreased ante-
rior facial height and maxillary-mandibular planes angle
(Dermaut et al., 1986; Nodal et al., 1994; Woodworth et al.,
1985). To date, evaluations have predominately been lim-
ited to using two-dimensional (2D) imaging techniques,
with the majority being conducted in those with severe
hypodontia, of limited sample sizes and the inclusion of
historic controls to quantify differences, which may explain
the observed heterogeneity among these studies.
The human face undoubtedly plays a key contribution to
an individual’s perceived physical attractiveness, with peo-
ple constantly being judged on their facial appearance
(Jørnung and Fardal, 2007; Ogaard et al., 1995; Riggio
et al., 1991). The facial soft tissues are integral to both diag-
nosis and treatment planning. In terms of the soft tissue
implications, there is insufficient literature to aid our under-
standing, as previous studies have involved the use of lat-
eral cephalograms, which are inherently limited in
visualising soft tissues (Ben-Bassat and Brin, 2003; Chan
et al., 2009; Ogaard and Krogstad, 1995; Sarnäs and Rune,
1983). A single study reported on the 3D soft tissue rela-
tionships in patients with ectodermal dysplasia, who had
some degree of hypodontia, and concluded that these par-
ticipants demonstrated a flat facial profile (Sforza et al.,
2006). However, the sample size was limited (n=35) and,
more importantly, included a very wide age range (age
range = 3–41 years) of participants (Sforza et al., 2006).
Several methods have been proposed for the evaluation
of facial soft tissue morphology. Among these, 3D optical
surface scanning has proven to be a reliable and reproduc-
ible technique in assessing soft tissue profile (Zecca et al.,
2016). It has the advantage of capturing images in a simple,
rapid manner and, most importantly, without any radiation
hazard (Aung et al., 1995).
Currently, information regarding the facial soft tissue
effects of hypodontia are lacking. Furthermore, no attempt
has been made to evaluate the influence of varying degrees
of hypodontia on the soft tissues. Thus, the aim of the pre-
sent study was to determine if there are differences in the
facial soft tissue morphology between patients with mild
(two or less) and severe (six or more) forms of hypodontia.
Materials and methods
Ethical approval was obtained for this prospective hospital-
based cohort study (Ref. 10/H0709/46) and written informed
consent was obtained from parents. Consequently, the find-
ings of the present study will be reported in line with the
STROBE guidelines (Vandenbroucke et al., 2007). Ninety-
two participants were recruited on the basis of the following
selection criteria: white children aged 11–16 years; and a con-
firmed diagnosis of non-syndromic mild (two or less) or
severe (six or more) hypodontia. Participants with a craniofa-
cial anomaly, significant infra-occlusion (3 mm or more), a
history of extraction or traumatic loss of permanent teeth and/
or orthodontic treatment were excluded. Participants were
recruited from those referred to and attending a hospital-
based multidisciplinary clinic between 2015 and 2019 for the
management of their hypodontia. Those who met the above
criteria were assigned to the following sub-groups based on
the extent and distribution of their missing teeth and age:
•• Sub-group A: two or fewer missing teeth in the ante-
rior (incisors and canines) region and categorised
into two groups: group 1 (11–13 years) and group 2
(14–16 years);
•• Sub-group B: six or more missing teeth in the ante-
rior and/or posterior region and categorised into two
groups: group 1 (11–13 years) and group 2 (14–16
years).
Optical surface scans
The technique used for data acquisition involved a vertical
laser beam that was emitted on to the participant’s face,
with two mirrors capturing the reflections and transmitting
the data to a coupled device camera while the patient was
rotated, in a specifically adapted and adjustable chair
between 0 and 220°, to capture a facial dataset that extended
from the left to right ears in a synchronised computer
(University College London, UK). A 3D facial scan was
obtained, based on the principle of optical triangulation
with a class 1 laser, with over 60,000 discrete coordinate
data points recorded for each scan, to a precision of 0.5
mm, which is then subject to surface analysis software
(Kau et al., 2005; Moss et al., 1989). A standardised posi-
tioning protocol was adopted to ensure comparability of
sequential images, in which the participant’s hair, where
necessary, was tied back to expose the forehead area, the
Johal et al. 3
Figure 1.  Frontal three-dimensional surface scan view with
landmarks.
eyes closed and the dentition in light occlusion. The surface
scanning system is calibrated on a weekly basis by a dental
meteorologist (LFZ), using a specially designed jig of
known dimensions. The calibration data are used to set up
an interpolation matrix to transform the subsequent digit-
ised input from the patient’s face into spatial coordinates.
Landmark-based analysis
A Cloud™ Windows compatible programme (University
College London, UK) was used allowing accurate place-
ment of 3D landmarks to measure distances and angles and
provide quantitative data for statistical analysis (Figures 1
and 2). The optical surface landmarks (n = 19) and inter-
landmarks (n = 5) used for measurement are defined in
Table 1. Inter-landmark distances were measured from the
reference landmark. Vertical and transverse inter-landmark
distances were measured between the points, in millimetres
(Table 1, Figures 1 and 2). Each surface landmark was
identified from the intersection of two projected surface
profiles in the horizontal (x-axis) and vertical (y-axis)
planes, at the corresponding point of interest. Thus, each
inter-landmark measurement corresponded to the actual
surface distance between the points, rather than a ‘simple’
linear 2D measurement. The following seven surface meas-
urements were undertaken in the following three planes of
space: AP (labio-mental fold and nasolabial angles); verti-
cal (lower and mid facial heights); and transverse (alar
base, intercanthal and transgonial widths).
Statistical analysis
Based on a previous pilot study the difference in optical sur-
face scans between matched pairs was normally ­distributed,
with a standard deviation of 0.34 mm (Chatterjee, 2008).
Figure 2.  Lateral three-dimensional surface scan view with
landmarks.
A minimum overall sample size of 42 pairs (in respect of the
mild and severe hypodontia groups) was proposed to offer
80% power, with a 95% confidence interval (CI) to demon-
strate a difference of 1 SD between the two groups, with a
type I error probability of 0.05. It was therefore decided to
enrol a minimum of 44 participants per group (n = 88) to
allow for drop-outs.
The measurements were statistically analysed using the
Statistical Package for Social Sciences program (SPSS, ver-
sion 20; SPSS Inc., New York, NY, USA). P < 0.05 was
considered statistically significant. In order to assess the dif-
ferences in the measurement of facial dimensions across
gender groups and severity categories and their interaction,
analysis of covariance (ANCOVA) was implemented. A
separate analysis was performed for each variable. Once the
overall significance of the F test was determined, a post-hoc
test was performed contrasting the different categories, cor-
rected by Tukey’s method. In order to assess the differences
in measurements between the categories of the independent
variables an ANCOVA was estimated for each. In these
models, severity and gender were introduced in a full facto-
rial fashion. Finally, post-hoc tests were performed by rank-
ing the least square means and performing a Tukey corrected
pairwise t-test and estimates for each category determined.
Error study
An interclass correlation (ICC) test was used to examine
intra-examiner reliability. This was assessed from repeat
measures of 10 randomly selected surface scans, in a
­random order, following a two-week intervening period.
A good level of agreement observed (ICC = 0.957, 95%
CI = 0.847–0.989).
4 Journal of Orthodontics 

Table 1.  Definition of optical surface landmarks (n = 19) and inter-landmarks (n = 5) used for measurement.

Assigned letter Landmark Definition of landmark

A Lateral canthus of the right eye Greatest concavity at the outer commissure of the right eye fissure

B Medial canthus of the right eye Greatest concavity at the inner commissure of the right eye fissure

C Medial canthus of the left eye Greatest concavity at the inner commissure of the left eye fissure

D Lateral canthus of the left eye Greatest concavity at the outer commissure of the left eye fissure

E Soft tissue nasion Greatest concavity in the facial midline between the forehead and the nose

F Subnasale Maximum depth of the concavity occurring at junction of columella base

and the upper lip

G Labarle superioris Most prominent point of the mucocutaneous border of the upper lip in
the mid-sagittal plane

H Labrale inferioris Most prominent point of the mucocutaneous border of the lower lip in
the mid-sagittal plane

I Labiomental fold Deepest concavity below the labrale inferioris

J Soft tissue pogonion Most prominent point of the soft tissue chin in the mid-sagittal plane

K Soft tissue menton Lowest point on the lower border of the chin in the mid sagittal plane

L Right gonion Point of greatest convexity at the lower border of the right gonial angle off
the mandible

M Left gonion Point of greatest convexity at the lower border of the left gonial angle off
the mandible

N Right alar base Most lateral point on the right alar base

O Pronasale Most prominent midline point of the nasal tip

P Left alar base Most lateral point on the left alar base

Q Right tragion Most superior and posterior tip of the right tragus

R Soft tissue ear point Anterior posterior aspect of the cartilage of the right ear

S Left tragion Most superior and posterior tip of the left tragus

Vertical inter-landmark points Corresponding facial dimension

E-F Mid facial height

F-K Lower facial height

Transverse inter-landmark points Corresponding facial dimension

B-C Intercanthal distance

N-P Alar base

L-M Transgonial width of face

Results (14–16 years) (Table 2). Nevertheless, the distributions of

Participant characteristics
The sample consisted of 92 participants, with an equal
distribution between the mild (n = 46) and severe (n =
46) hypodontia categories. Fifty-three (58%) participants
were female, with 50 (54%) being in the older age group
gender and age across the severity groups were not sig-
nificantly different (P = 0.83 and P = 0.40, respectively).
The gender distribution within the sample, revealed 25
(47%) girls were in the younger group and 28 (53%) were
in the older group. Regarding the boys, 17 (43%) were in
the younger group and 22 (56%) were in the older group,
Johal et al. 5

Table 2.  Distribution of gender and age with varying severity of hypodontia (n = 92).

Severity of hypodontia

Gender Age group Mild (n = 46, 50%) Severe (n = 46, 50%)

Female (n = 53, 58%) 11–13 12 (48) 13 (52)

  14–16 14 (50) 14 (50)

Male (n = 39, 42%) 11–13 7 (41) 10 (59)

  14–16 13 (59) 9 (41)

Values are given as n (%).

Table 3.  Distribution of absent teeth in the dental arches within the sample (n = 92).

Central Lateral First Second First Second

Tooth incisor incisor Canine premolar premolar molar molar

Maxilla 2 (0.33) 117 (19) 38 (6.41) 52 (8.78) 75 (12.6) 14 (2.3) 22 (3.7)

Mandible 74 (12.5) 41 (6.9) 14 (8.78) 29 (4.8) 78 (13.17) 8 (1.3) 28 (3)

Values are given as n (%).

Table 4.  Measurements by gender (n = 92).

Measurement Female (n = 53) Male (n = 39) Difference t ratio P value

Alar base 31.98 ± 3.21 34.7 ± 6.05 2.72 2.56 0.013

Intercanthus 33.14 ± 3.36 34.71 ± 5.78 1.57 1.52 0.134

Labio-mental fold angle 122.17 ± 25.62 117.9 ± 23.26 –4.27 –0.83 0.407

Lower face height 62.59 ± 5.79 65.75 ± 5.6 3.16 2.63 0.01

Mid face height 56.09 ± 3.85 57.46 ± 3.68 1.36 1.72 0.088

Nasolabial angle 116.36 ± 12.86 122.13 ± 12.79 5.77 2.13 0.036

Transgonial 108.91 ± 7.87 115.77 ± 8.45 6.86 3.96 <0.0001

Values are given as mean ± SD unless otherwise specified.

with no significant difference detected (P = 0.73) in this
parameter.
Table 3 shows the distribution of missing teeth (n =
592) within the dental arches for the whole sample (n =
92). The maxillary lateral incisor (19%) was the most fre-
quently occurring, followed by the mandibular second pre-
molar (13.2%), maxillary second premolar (12.6%) and
lower incisors (12.5%).
Surface anatomical measurements
In the comparison of gender, with the exception of the labio-
mental fold angle, all other surface measurements in the
girls were smaller than their male counterparts (Table 4).
In the comparison of surface measurements by severity
of hypodontia, with the exception of the labio-mental fold,
a general trend was observed towards smaller mean values
for those participants with severe hypodontia, with a statis-
tically significant difference (P = 0.023) observed only in
relation to the transgonial measurement (Table 5).
An ANCOVA was estimated to assess the difference in
measurements between the categories of the independent
variables. In these models, severity and gender were intro-
duced in a full factorial fashion. Utilising the 5% cut-off
point for each model (Table 6), significant differences for
alar base (F = 3.26, P = 0.025), lower face height (F = 3.46,
P = 0.025), nasolabial angle (F = 3.24, P = 0.026) and
transgonial distance (F = 7.74, P < 0.001) were observed.
6 Journal of Orthodontics 

Table 5.  Measurements by severity of hypodontia (n = 92).

Measurement Severe (n = 46) Mild (n = 46) Difference t ratio P value

Alar base 32.42 ± 3.32 33.85 ± 5.87 –1.43 –1.44 0.156

Intercanthus 33.36 ± 3.62 34.25 ± 5.39 –0.89 –0.93 0.354

Labio-mental fold angle 123.23 ± 20.58 117.5 ± 28 5.73 1.12 0.267

Lower face height 63.03 ± 5.58 64.83 ± 6.11 –1.8 –1.48 0.143

Mid face height 56.9 ± 3.92 56.44 ± 3.74 0.46 0.57 0.567

Nasolabial angle 118.62 ± 12.26 118.99 ± 13.98 –0.37 –0.13 0.893

Transgonial 109.75 ± 7.93 113.88 ± 9.15 –4.13 –2.31 0.023

Values are given as mean ± SD unless otherwise specified.

Table 6.  ANCOVA results for all measurements by severity

and gender (whole model). Figure 3.  Least squares means plot for alar base
measurement for severity of hypodontia and gender.
Dependent variable DF F P value n
38
Alar base 3 3.26 0.0252 92 37
Intercanthus 3 1.17 0.324 92 36
Labio-mental fold angle 3 1.25 0.2951 92 35

Lower face height 3 3.46 0.0196 92 34

33
Mid face height 3 1.12 0.344 92
32
Nasolabial angle 3 3.24 0.0259 92
31
Transgonial 3 7.74 0.0001 92 30
Each model includes severity, gender and the interaction between them 29
as covariates. Female Male Female Male
ANCOVA, analysis of covariance; DF, degrees of freedom, F, test Mild Severe
statistic, n, number of participants.
Severity(M/S) / Gender

Subsequent to ANCOVA, post-hoc tests were per-

formed by ranking the least square means and performing
a Tukey corrected pairwise t-test. For alar base, a signifi-
cant difference was found between mild-male (35.33 ±
1.03) and severe-female (31.28 ± 0.89) (Figure 3). A sig-
nificant difference in lower face height was found
between mild-male (67.35 ± 1.27) and severe-female
(62.3 ± 1.09) (Figure 4). Similarly, for the nasolabial
angle, a significant difference was found between mild-
male (125.6 ± 2.82) and mild-female (113.91 ± 2.48)
(Figure 5). With regards to the transgonial measurement,
two significant differences were found: mild-male
(118.42 ± 1.77) was found to be different from both
mild-female (110.39 ± 1.55) and severe-female (107.48
± 1.53) (Figure 6). In contrast, both mild-female and
severe-female were not significantly different.
Discussion
The present study is original in its attempt to determine
whether differences exist in the facial soft tissue morphol-
ogy of participants with mild (up to two) or severe (six or
more) non-syndromic hypodontia, using 3D optical surface
scanning as a reliable and reproducible technique for their
assessment. Such evaluation is of considerable clinical
value in the treatment planning of hypodontia, given that it
remains the most common inherited dental condition and
requires multidisciplinary care (Khalaf et al., 2014; Polder
et al., 2004). Previous attempts to address this question
have been limited by poor sample definition and sample
size or reliance on 2D radiographic imaging modalities to
assess the soft tissues.
Johal et al. 7

Figure 4.  Least squares means plot for lower face height Figure 6.  Least squares means plot for transgonial
measurement for severity of hypodontia and gender. measurement for severity of hypodontia and gender.

72 122
70 120
118
68
116
66 114
112
64
110
62 108
60 106
104
58
Female Male Female Male
Female Male Female Male
Mild Severe
Mild Severe
Severity(M/S) / Gender
Severity(M/S) / Gender

Figure 5.  Least squares means plot for nasolabial angle the fact that soft tissue maturation is more advanced in girls
(NLA) measurement for severity of hypodontia and gender.
than boys (Nanda and Ghosh, 1985). This, in turn, could
explain the differences found between genders within the
132 same age group in this current study. Furthermore, the
130
results of the present study are based on white children and
128
126
therefore not necessarily valid in their application to other
124 ethnic groups, who in turn may well demonstrate different
122 soft tissue features and this would be worthy of separate
120 investigation.
118 The most common congenitally missing tooth was the
116 maxillary lateral incisor (19%) followed by the mandibular
114 second premolars (13.2%). This finding was not consistent
112 with previous studies reporting prevalence levels, in which
110 they have found that the most common missing tooth was
108 the mandibular second premolar (Khalaf et al., 2014; Polder
Female Male Female Male
et al., 2004). This may reflect the fact that the participants
Mild Severe
were recruited from a multidisciplinary hypodontia clinic,
Severity(M/S) / Gender where they were seeking orthodontic treatment to address
their aesthetic concerns, which in turn are more apparent in
the anterior rather than the posterior region. The least com-
The STROBE statement was developed to help overcome mon missing tooth was the maxillary central incisor, in
either incomplete or inadequate research reporting and, as keeping with a previous meta-analysis (Khalaf et al., 2014).
such, not only assists authors when publishing analytical Similarly, hypodontia was more common in the maxillary
observational studies, but helps readers know what the study arch (54%), with the severe form most prevalent in females,
was designed to assess, how it was undertaken and what the both findings remaining consistent with the published lit-
findings mean. The STROBE guideline is a 22-item check- erature (Brook, 1974; Khalaf et al., 20142).
list that authors fulfil before submission and this in turn acts The present study utilised laser surface scanning to
to support editorial team and reviewers when considering acquire 3D images of the participant’s face. However, it is
such articles for publication (Vandenbroucke et al., 2007). recognised that the most commonly used technique to cap-
The sample consisted of 92 white participants, aged 11– ture a 3D image of the face is stereophotogrammetry, which
16 years. The number of girls was higher in both age offers a faster image capture time of 0.25 s, with image col-
groups, which is to be anticipated in hypodontia (Khalaf our and surface texture. While this imaging technique has
et al., 2014). There was no statistical difference in age become available more recently within our research centre,
among both genders. The sample was sub-grouped by age the current study continued 3D image capture using laser
to help reflect potential growth differences that exist and surface scanning. This decision was a reflection of the fact
8 Journal of Orthodontics 
that in view of the strict selection criteria applied to identify
the two distinct groupings, particularly the severe group
population given its overall lower prevalence and with the
study cohort being prospectively recruited over a number of
years, it was felt that this approach minimised any possible
error associated with using different imaging modalities.
In view of the lack of available studies assessing the
facial soft tissue dimensions of participants with hypodon-
tia, the findings of the present study will be compared with
reported skeletal differences, assessed from lateral cephalo-
metric analysis where possible. Overall, lower facial height
was reduced in the more severe hypodontia group and
between genders. This finding was consistent previous
studies, based on cephalometric analysis. Takahashi et al.
(2018), in their evaluation of the craniofacial morphology
of 106 non-syndromic hypodontia participants, reported a
smaller mandibular planes angle in patients with oligodon-
tia and they stated that skeletal patterns differed according
to the number of congenital missing teeth. One explanation
for this could be that the absence of posterior teeth in severe
hypodontia results in reduced vertical alveolar develop-
ment and corresponding condylar height. Similarly, Ben-
Bassat and Brin (2003), in their evaluation of a group of 28
children with at least 10 congenitally missing teeth (exclud-
ing third molars), using lateral cephalograms also observed
a reduced Frankfort mandibular planes angle in hypodontia
when compared to norms. Furthermore, they noted that the
location of missing teeth was related to the severity of skel-
etal discrepancy. However, in contrast to the findings of the
present study, they also reported that participants with ante-
rior missing teeth demonstrated a bigger reduction in facial
height, but this is likely to be explained by the fact that the
study sample all exhibited severe hypodontia and thus a
combination of anterior and posterior missing teeth (Ben-
Bassat and Brin, 2003). Bonderates and McDonald, in
their large sample (n = 1516) of patients aged 6–18 years,
found a strong correlation between severe hypodontia and
a reduced lower facial height, as determined from lateral
cephalograms. Their sample included patients with hypo-
hydrotic ectodermal dysplasia syndrome, with the latter
demonstrating significant reductions in mid face height
compared to non-syndromic patients. The present study
detected no differences in mid face height in any group.
This could be explained by the fact that those with hypo-
hydrotic ectodermal dysplasia syndrome are likely to
demonstrate deficient maxillary growth (Ben-Bassat and
Brin, 2003).
In terms of transverse surface inter-landmark analysis, in
the comparison by gender, larger measurements were
observed for males, with alar base and transgonial distances
reaching statistical significance, which serves to highlight
the importance analysing the genders separately. In the com-
parison by hypodontia severity, a statistically smaller
transgonial measurement was observed in the severe group,
which may suggest that the absence of posterior teeth and
their supporting alveolus imparts an underlying transverse
dimensional change.
In evaluating the results of all measurements in the trans-
verse plane by severity of hypodontia and gender, alar base
and transgonial distance revealed statistical significance,
while no significant difference in intercanthus width was
observed. This may again reflect the fact that non-­syndromic
hypodontia appears to have minimal mid face effects. It is
difficult to relate the findings of the current study to the lit-
erature, as previous studies are limited by their use of lateral
cephalometrics, designed to evaluate the sagittal and verti-
cal planes only. Although Sforza et al. (2006) used a 3D
computerised electromagnetic digitiser to measure soft tis-
sue morphology in patients with ectodermal dysplasia with
a different pattern and number of missing teeth, they did not
report any measurement of transgonial width.
In the AP plane, differences were observed in the nasola-
bial angle by severity of hypodontia and gender. This can be
explained by the fact that while the mild group demonstrated
up to two or less missing teeth in the anterior (incisors and
canines) region, the severe group could have demonstrated
this and more, not only in the incisor/canine region but in
the first and second premolar regions, thereby significantly
impacting on the upper lip position. These findings correlate
with earlier studies. Both Sfroza et al. (2006) and Bonderates
and McDonald (2000) reported that the nasolabial angle was
significantly reduced in patients with syndromic and non-
syndromic severe hypodontia. However, care must be taken
while interpreting their results as syndromes are associated
with phenotypic presentation influencing the soft tissue pro-
file. Furthermore, there are limited studies again using 2D
imaging that show that participants with non-syndromic
hypodontia demonstrate a straight facial profile compared to
controls associated with retroclination of incisors (Ben-
Bassat and Brin, 2003; Endo et al., 2006).
A potential limitation of the present study was selection
bias. Thus, in an attempt to reduce the risk, eligible partici-
pants were recruited prospectively and in a consecutive
manner from the hypodontia multidisciplinary clinic, help-
ing to minimise this risk. Notwithstanding this, it is possi-
ble that recruitment of participants from a hospital-based
clinic is less representative of the general population. The
sample size could have been larger to more optimally power
the study, particularly for the sub-group analysis, but it was
equally challenging recruiting sufficient numbers of par-
ticipants with severe hypodontia. In this respect, a formal
calculation of sample size was performed based on a pilot
study undertaken within the department, in view of the lack
of published data evaluating the soft tissue morphology in
hypodontia.
Conclusion
Significant reductions were seen in the 3D soft tissue mor-
phology of participants with severe hypodontia, in terms of
Johal et al. 9
the nasolabial angle, lower facial height, alar base and
transgonial widths, emphasising the importance of using
3D facial scanning as a relatively simple non-invasive
method of assessment.
Acknowledgements
We would like to thank all the staff involved with the multidisci-
plinary clinics for their help in recruitment. We would also like to
thank the patients for agreeing to partake in the study.
Declaration of conflicting interests
The author(s) declared no potential conflicts of interest with
respect to the research, authorship, and/or publication of this
article.
Funding
The author(s) received no financial support for the research,
authorship, and/or publication of this article.
ORCID iD
Ama Johal https://orcid.org/0000-0001-6841-227X
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