Eur J Plast Surg (2001) 24:118–122
DOI 10.1007/s002380100244
O R I G I N A L PA P E R
C. Lekkas · B.S. Latief · S.P.N. Ter Rahe
A.M. Kuijpers-Jagtman
The unoperated adult cleft patient: teeth in the cleft area
Received: 28 June 2000 / Accepted: 3 August 2000 / Published online: 30 March 2001
© Springer-Verlag 2001
Abstract In cleft lip and/or palate patients, anomalies of
the teeth in the cleft area are often found. The anomalies
consist of missing, supernumerary, or malformed teeth.
The studies, up to now, have been performed on patients
treated surgically in early childhood and therefore the influence of surgery on the number, size, and form of the
teeth in the cleft area cannot be excluded. Furthermore,
in the majority of publications the different types of
clefts are not analysed separately and the results are
therefore not reliable. In the present study, a sample of
267 adult cleft patients who had not undergone surgery
were investigated for variations in the size and form of
the teeth in the cleft area. Four different cleft groups
were examined: UCLA (n=174), UCLP (n=62), BCLA
(n=17) and BCLP (n=14). Absence of teeth was observed in all four groups, less so in the UCLA than in the
BCLA group. The absence of the most teeth was observed in the UCLP and BCLP groups. There was no difference in missing teeth between the unilateral and bilateral cleft lip and palate groups. When one single tooth
was found it was merely a peg-shaped one. Most of the
teeth were localised to the dorsal part of the cleft. Finally, combinations of more than one tooth were only incidentally found and only in the UCLA group. As the percentage of missing teeth in all four groups is clearly less
than that quoted in the literature for corresponding
groups of cleft individuals operated on early, it is suggested that surgical damage might be an additional factor
for absence of teeth in individuals operated on early.
C. Lekkas (✉)
Department of Oral and Maxillofacial Surgery,
University of Leiden, P.O. Box 9600, 2300 RC Leiden,
The Netherlands
Tel.: +31-71-5262372, Fax: +31-71-5266766
B.S. Latief
Department of Oral and Maxillofacial Surgery,
Universitas Indonesia, Jakarta, Indonesia
S.P.N. Ter Rahe · A.M. Kuijpers-Jagtman
Department of Orthodontics, Catholic University of Nijmegen,
The Netherlands
Keywords Surgically untreated cleft · Cleft · Permanent
dentition · Cleft lip and alveolus · Cleft lip, alveolus
and palate · Agenesis · Peg-shaped teeth ·
Supernumerary teeth
Introduction
Dental anomalies of the maxilla occur often in individuals with cleft lip and/or palate, who underwent surgery at
an early age [1, 2,13]. The most frequently observed
anomalies of the teeth are deviations in number, size, and
shape in the cleft area but also outside this area. In the
cleft area impacted teeth are rather common. Outside the
cleft area, the absence of teeth is frequently observed in
patients with not only a cleft of the lip and alveolus but
also of the palate. The numeric dental anomalies in the
cleft area vary from agenesis to supernumerary teeth;
variations in form and size in the area of the lateral incisor are often mentioned [10, 11, 15,16].
Concerning the anomalies in the cleft area, the various types of cleft are usually analysed as one single
group [9,10], although there are indications that the
quantity of anomalies depends on the type of cleft [7].
In the literature, the different types of cleft are only
separately analysed in a few papers [15, 16,17]. According to Tsai and King [17] the lateral incisor in the cleft
area of these individuals is mostly absent in unilateral
cleft lip, alveolus and palate patients. Ranta [14] comes
to almost the same conclusions in his review-paper concerning tooth formation in children with clefts.
Up to now all these studies were conducted on cleft
patients who underwent surgery in early childhood.
Therefore, damage of the tooth buds during the operation
could not be excluded. Ranta [14] suggests, in an attempt
to explain the absence of teeth in cleft patients in general, that “the surgical treatment of the cleft seems to be of
little importance as an etiological factor”. There are indications, however, that surgery as an etiological factor in
missing maxillary teeth outside or within the cleft area is
much more important than was previously assumed
119
[12,13]. Characteristic in this context is the observation
of Hellquist et al. [8] that “when the mucoperiosteal flap
is raised from the cleft border of the premaxilla during
the operation of the palate, the underlying tooth buds
were often seen as small blastulas not covered by bone”.
The idea that early surgery may cause absence of
teeth in the cleft is also supported by Brattstrom and
McWilliam[3]. In a study on bone grafting procedures in
clefts, they found a higher percentage of missing permanent lateral incisors and fewer supernumerary teeth in
the cleft area when the operation was performed within
the first year of life as compared to the group in which
bone graft surgery was performed after the eruption of
the permanent incisors. From a clinical point of view,
these observations are supported by the finding of
Broomell and Fischelis [5] concerning the development
of tooth buds and surrounding alveolar bone related to
the time the initial operation for the closure of the lip and
alveolus is performed.
Up to now, no study has been conducted to investigate the number and form of teeth in the cleft area in
adult patients who never underwent surgery. In such a
sample, a possible influence of surgery in early childhood can be excluded.
The aim of the present study is to investigate the numeric dental anomalies of the permanent dentition in the
cleft area in a group of adult individuals that did not undergo any kind of surgery to close the cleft. Moreover,
the study attempts to investigate whether there is a relation between numeric dental anomalies and the severity
of the cleft.
Material and methods
member. Individuals who mentioned extractions, or who showed
edentulous areas in the cleft area, but who could not remember
that extractions had taken place in the past were excluded from the
study.
The patients were divided into four groups:
● Unilateral cleft lip and alveolus (UCLA), n=174
● Unilateral cleft lip, alveolus and palate (UCLP), n=62
● Bilateral cleft lip and alveolus (BCLA), n=17
● Bilateral cleft lip, alveolus and palate (BCLP), n=14
All patients were collected and registered during expeditions in
the remote areas of Indonesia. After data collection, the patients
were treated surgically for closure of the cleft. The patients were
documented with dental casts, cephalograms, and often with standard intraoral and extraoral photographs, as described previously
[12].
Method
Two experienced investigators examined the dental casts independently. On the dental casts the cleft was identified and the teeth
adjacent to the cleft were recorded. The corresponding cephalograms were examined to discover possible tooth impactions in the
cleft area. The lateral cephalograms are presumed to be sufficient
for the localisation of impacted teeth in the precanine area of the
maxilla. In the literature, the terminology of the different types of
teeth in the cleft area is confusing. In this paper, the nomenclature
adopted by Bøhn (1963) is used. In this nomenclature, the form,
the position of the teeth in relation to the cleft, and the possible
numeric aberrances are indicated as follows:
● 0: no teeth in the cleft
● 12x: normally shaped lateral incisor ventral to the cleft
● 12y: normally shaped lateral incisor dorsal to the cleft
● 10: peg-shaped/canine-shaped tooth ventral to the cleft
● y: peg-shaped/canine shaped tooth dorsal to the cleft
In a previous study [2], there was no significant relationship between the anomalies and the sex of the patient; for that reason, the
sex of the patient was not taken into consideration in this review.
Results
Subjects
The sample consists of 267 individuals with cleft lip alveolus
and/or palate who did not undergo surgery. The selection criteria
were: no syndromes, no surgical procedures for cleft closure, no
extractions of teeth in the cleft area, as far as the patients could re-
The number of teeth in the single cleft area varies from
zero to two, and no patient had more than two. There
were no impacted teeth in the cleft area on dental cast or
lateral cephalogram examination.
Table 1 Group UCLA, n=174. Number of single tooth types or combinations of tooth types in a cleft and the percentage, which is the
number relative to the number of clefts (left-sided, right-sided and overall)
Tooth type or combination
No teeth
I2x
I2y
x
y
I2x + y
x+y
I2y + y
I2x + I2y
I2y + I2y
I2x + x
I2y + x
x+x
y+y
Right-sided clefts (n=40)
Left-sided clefts (n=134)
All clefts (n=174)
n
%
n
%
n
%
7
4
9
0
14
3
2
1
0
0
0
0
0
0
17.5
10
22.5
0
35.0
7.5
5.0
2.5
0
0
0
0
0
0
31
18
12
6
36
16
5
4
2
1
1
0
1
1
23.1
13.4
9.0
4.5
26.9
11.9
3.7
3.0
1.5
0.7
0.7
0
0.7
0.7
38
22
21
6
50
19
7
5
2
1
1
0
1
1
21.8
12.6
12.1
3.4
28.7
10.9
4.0
2.9
1.1
0.6
0.6
0
0.6
0.6
120
Table 2 Group UCLP, n=62. Number of single tooth types or combinations of tooth types in a cleft and the percentage, which is the
number relative to the number of clefts (left-sided, right-sided and overall)
Tooth type or combination
No teeth
I2x
I2y
x
y
I2x + y
x+y
I2y + y
I2x + I2y
I2y + I2y
I2x + x
I2y + x
x+x
y+y
Right-sided clefts (n=17)
Left-sided clefts (n=45)
All clefts (n=62)
n
%
n
%
n
%
8
2
0
0
6
0
0
0
1
0
0
0
0
0
47.1
11.8
0
0
35.3
0
0
0
5.9
0
0
0
0
0
16
0
9
0
17
0
0
1
0
0
0
1
0
1
35.6
0
20
0
37.8
0
0
2.2
0
0
0
2.2
0
2.2
24
2
9
0
23
0
0
1
1
0
0
1
0
1
38.7
3.2
14.5
0
37.1
0
0
1.6
1.6
0
0
1.6
0
1.6
Table 3 Group BCLA, n=17. Number of single tooth types or combinations of tooth types in a cleft and the percentage, which is the
number relative to the number of clefts (left-sided, right-sided and overall)
Tooth type or combination
No teeth
I2x
I2y
x
y
I2x + y
x+y
I2y + y
I2x + I2y
I2y + I2y
I2x + x
I2y + x
x+x
y+y
Right-sided clefts (n=17)
Left-sided clefts (n=17)
All clefts (n=34)
n
%
n
%
n
%
5
1
4
0
2
2
0
2
1
0
0
0
0
0
29.4
5.9
23.5
0
11.8
11.8
0
11.8
5.9
0
0
0
0
0
5
3
4
0
2
1
0
0
1
0
0
1
0
0
29.4
17.6
23.5
0
11.8
5.9
0
0
5.9
0
0
5.9
0
0
10
4
8
0
4
3
0
2
2
0
0
1
0
0
29.4
11.8
23.5
0
11.8
8.8
0
5.9
5.9
0
0
2.9
0
0
Besides variation in number, variation in shape was
also found. The shape varied from a normally shaped lateral incisor to a peg-shaped tooth. When more than one
tooth in the cleft area was present, combinations varied
from two normally shaped lateral incisors to two pegshaped teeth.
The results of the study are summarised in the tables.
Each contains information on a separate patient group
and starts with the most frequently observed pattern:
Table 1 =UCLA, Table 2 =UCLP, Table 3 =BCLA, and
Table 4 =BCLP.
Absence of teeth
In 21 8% of the patients in the UCLA group, an absence
of teeth is observed (Table 1). In the BCLA group, the
incidence of missing teeth is higher, being 29.4%
(Table 3). The absence of teeth was always observed bilaterally.
Absence of teeth in each cleft area is found in approximately 39% of clefts when besides lip and alveolus, unilaterally or bilaterally the palate was also cleft (Table 2
and Table 4). In both groups the percentage of missing
teeth was equal for a single cleft area regardless of
whether the cleft lip and palate were unilateral or bilateral.
Single normally shaped lateral incisor
A normal shaped lateral incisor was present in the cleft
area in all four groups. This normal incisor was localised
either to the dorsal part of the cleft near the canine or the
ventral part near the central incisor. In the UCLA group
the lateral incisor was positioned ventrally or dorsally in
relation to the cleft in equal numbers.
In the BCLA group, the number of dorsally localised
lateral incisors was twice as high as those localised ventrally. Finally, the dorsal localisation of the lateral incisor
121
Table 4 Group BCLP, n=14. Number of single tooth types or combinations of tooth types in a cleft and the percentage, which is the
number relative to the number of clefts (left-sided, right-sided and overall)
Tooth type or combination
No teeth
I2x
I2y
x
y
I2x + y
x+y
I2y + y
I2x + I2y
I2y + I2y
I2x + x
I2y + x
x+x
y+y
Right-sided clefts (n=14)
Left-sided clefts (n=14)
All clefts (n=28)
n
%
n
%
n
%
5
1
2
0
5
0
1
0
0
0
0
0
0
0
35.7
7.1
14.3
0
35.7
0
7.1
0
0
0
0
0
0
0
6
0
2
0
4
0
0
0
0
0
1
0
0
1
42.9
0
14.3
0
28.6
0
0
0
0
0
7.1
0
0
7.1
11
1
4
0
9
0
1
0
0
0
1
0
0
1
39.3
3.6
13.3
0
32.1
0
3.6
0
0
0
3.6
0
0
3.6
was four times higher in the UCLP and BCLP groups
compared to the ventrally located ones. Here, too, there
was no difference in the localisation of the lateral incisor
between the unilateral and bilateral cleft lip and alveolus
patients.
Single peg-shaped tooth
When one single peg-shaped tooth was found, this was
almost exclusively localised on the dorsal part of the
cleft near the cuspid. Only in the UCLA group a pegshaped tooth occasionally was found on the ventral part
of the cleft near the central incisor. Remarkably, this
ventral localisation of the single peg-shaped tooth was
exclusively observed in left-sided clefts.
More than one tooth
A comparison of the four tables shows that different
combinations of more than one tooth are possible; this is
only incidentally observed. Combinations of more than
one tooth are found in the cleft lip and alveolus groups,
rather than in the cleft lip and palate groups. The only
somewhat consistent combination of more than one tooth
in the cleft area is found in the UCLA group. It is a combination of a normal lateral incisor on the ventral part of
the cleft combined with a peg-shaped tooth located at the
dorsal part of the cleft.
Discussion
As far as we know, this is the first study on dental anomalies in the cleft area in a patient who has not undergone
surgery. Moreover, this is the first study analysing the
dental anomalies in the cleft area in all four main types
of cleft. Unfortunately, it is impossible to compare the
findings of the present study with the prevalence of dental anomalies of the non-cleft population of the same ethnic group. It would be unrealistic to expect a reliable epidemiological study on dental anomalies in a population
with such rudimentary medical and dental care that patients have reached adulthood without having had surgery on a cleft! For comparison, it could be assumed that
the prevalence of missing teeth of the noncleft population in our target area may be comparable to the prevalence in a population with roughly the same ethnic background as Malaysians and South Chinese. In a sample of
1,093 Hong Kong schoolchildren, only 0.64% showed an
absence of the upper lateral incisor [6]. In the Malaysian
population, Kong (cited by Brook in 1975 [4]) found that
invaginated permanent maxillary lateral incisors are
present twice as often in the Chinese as in the Malaysians.
Impacted teeth were not found in the cleft area of the
four groups of adult individuals in whom clefts had not
been surgically treated. This is in contrast to the frequently observed impaction of teeth in cleft lip and palate patients who underwent surgery at an early age. In
these patients, scar tissue, inward rotation of the maxillary segments, or bone grafting could be responsible for an
inhibiting effect on the growth of teeth in the cleft area.
The absence of teeth in the cleft area is correlated to
the severity of the cleft. Only 22% of the patients
showed absence of teeth in the UCLA group. A higher
percentage of missing teeth is found in the BCLA group.
The highest percentage of missing teeth is found in the
UCLP and BCLP groups. In this study, the percentage of
missing teeth is consistently lower than the percentage
reported in the literature concerning a corresponding
type of cleft in adult cleft patients who underwent surgery early in life. In a mixed sample of 33 cases of lip
and alveolar clefts (with or without associated cleft palate) surgically treated in early childhood, the author has
found 50% of teeth to be missing [1]. Because the sample consists of cleft groups with a lower and higher per-
122
centage of missing teeth altogether, it is reasonable to assume that in the UCLP and BCLP patients the percentage of missing teeth is higher than 50%.
The lowest number of missing teeth in our group is
most obvious in the BCLP group. Furthermore, in contrast with the literature [14], in our study there is no difference in the absence of teeth in a single cleft area between the UCLP and BCLP groups.
In conclusion, it should be emphasised that the substantial percentage of missing teeth in the adult cleft patients who underwent surgery in their early childhood
may be attributed to damage of tooth buds during surgery. It could also be assumed that the consistently higher percentage of missing teeth in the BCLP cases compared to the UCLA cases [14] may also be the result of
more difficult operations necessary for the closure of the
palate of the BCLP groups. The suggestion that surgical
damage might be an additional cause of missing teeth in
cleft patients who have undergone early surgery [3, 8,
12,13] seems to be justified by our findings.
In accordance with the literature, single and supernumerary teeth are also observed in the cleft area. The
twelve possibilities of normal or peg-shaped teeth registrated in the cleft area are summarised in the four tables.
Single tooth
Normal or peg-shaped teeth are the most frequently observed. In accordance with the literature [2, 15,16], the
single tooth was mostly localised at the dorsal fragment
near the canine.
Combinations of more than one tooth are only found
incidentally. Sometimes in non-cleft patients, supernumerary teeth are also observed in the area of the lateral
incisor; it is therefore difficult to say whether the combination of teeth observed in our cleft groups are attributed
to the presence of the cleft or should be considered as an
unexceptional although unusual aberrance of the normal.
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Benny Latief