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Oral health status and behaviour of Greek patients
with cleft lip and palate
V. Parapanisiou *, S. Gizani *, M. Makou **, L. Papagiannoulis *
Depts. of *Paediatric Dentistry and ** Orthodontics, Dental School, University of Athens, Greece.
Abstract
Aim: This was to record the oral health profile and more specifically the prevalence of carious and hypoplastic lesions
in children and adolescents with cleft lip and palate (CLP).
Study Design and Methods: The study group consisted of 41 children and adolescents (4-18 years-old) with
CLP group while a similar number of non-cleft persons (HLP)
matched for sex, age and orthodontic treatment (75.6%),
was used as the control group. Information regarding the oral
health habits, medical and dental history were collected using a questionnaire. Stimulated saliva was collected to evaluate the flow rate and buffering capacity as well as the levels
of mutans streptococci and lactobacilli using the chair-test
CRT® (Ivoclar –Vivadent). Oral hygiene (OH) was assessed
using the index of Silness and Loe [1964]. The prevalence
of initial/white spot and cavitated carious lesions as well as
hypoplasia was evaluated based on the criteria by ICDAS
[2005] and Nyvad et al [2008] as well as Koch et al [1987]
respectively. The statistical analysis was carried out using
the t-test and the chi- square test. RESULTS: Approximately
half of the CLP and HLP subjects were brushing their teeth
2 x per day. Both groups reported an average of 3 main and
2 snack meals per day. Levels of mutans streptococci and
lactobacilli as well as the quality of the saliva were similar for
both groups. The plaque index score was significantly higher
in the CLP than in the control group (p=0.0003). The prevalence of cavitated carious lesions was similar in both groups
but that of the initial/white spot lesions, especially at the
area of maxillary incisors, was higher in the CLP group (85%,
p=0.000). Conclusions: The increased numbers of initial/
white spot lesions combined with poor OH found in the CLP
group predispose for an increased risk of further development to carious cavitated lesions. Taking into consideration
that the majority of those patients were under orthodontic
treatment, the application of an intensive individualized oral
health preventive program, focused on remineralisation of
the initial caries, is imperative.
Introduction
Cleft lip and palate (CLP) is one of the most common congenital malformations. Epidemiological studies have shown
great variation in its occurrence and its prevalence is reported to range from 0.91 to 2.69 per 1,000 in Caucasians and
from 0.85 to 2.68 per 1,000 in Japanese [Vanderas, 1987]. An
epidemiologic study in Greece showed that the total prevalence for the years 1985-1988 and 1989 was estimated to be
0.47/1000 live births for CLP and 0.34/1,000 live births for
cleft palate only [Nasika, 1997].
Most studies have shown higher caries prevalence [Johnsen
and Dixon, 1984; Dahllof et al, 1989; Bokhout et al., 1996a]
and poorer oral hygiene (OH) in CLP children than in healthy
children of the same age while other studies have not been
able to confirm these findings [Al-Wahadi et al., 2005; Bokhout et al., 1996b]. These contradictory results have been
attributed to factors such as the small size and the large
age-range of the sample, the lack of control group and the
absence of differentiation between the various types of cleft
[Hasslof and Twetman, 2007].
The aim of this study was to a) register the oral hygiene and
dietary habits, b) to assess the OH status including caries
and dental anomalies and c) to evaluate the levels of cariogenic micro organisms and the quality of saliva in Greek
patients with CLP. The null hypothesis was that OH status
of CLP children and adolescents does not differ from that of
the HLP group.
Materials and Methods
Subjects. The cleft group (CLP) consisted of 41 children with
CLP (23 boys and 18 girls) aged 4-18 years (mean age 10.54,
SD±=3.37 years). Those children with multiple abnormalities
or recognized syndromes were excluded. The study group
consisted of 26 children and adolescents with unilateral CLP,
10 with bilateral CLP and 5 with cleft palate; 75.6% of the patients were under orthodontic treatment. The control group
(HLP) consisted of 41 healthy children and adolescents with
mean age of 10.7 years (SD±3.03 years) matched for age,
gender and orthodontic treatment. Both groups were selected from the patients of the Postgraduate Paediatric and
Orthodontic Dental Clinic of the University of Athens.
Questionnaire. Using a questionnaire completed by the
parents/patients, information was obtained concerning the
OH habits (tooth brushing frequency, parental help during
toothbrushing, use of dental floss, age of the first dental visit,
frequency of dental attendance), the fluoride exposure (application of topical fluoride and the use of fluoride supplements, such as fluoride tablets) as well as the dietary habits
Key words: cleft lip; cleft palate; oral health; caries; children and adolescents.
Postal address: Prof. L. Papagiannoulis. Dept of Paediatric Dentistry,
University of Athens, Thivon 2, Goudi, Athens, 11527,Greece.
Email: [email protected]
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Parapanisiou et al.
(frequency of daily consumption of juices and refreshments,
the number of main meals and snacks as well as the number
of meals containing sugars per day) of the study participants.
Saliva evaluation and assessment of cariogenic microbiota.
Stimulated saliva was collected after chewing a paraffin
gum for 5 minutes and expectorating into a cup. The salivary secretion rate was evaluated in ml/min. The number of
lactobacilli (LB) and mutans streptococci (MS) in saliva was
detected using the CRT® chair-side tests (Ivoclar-Vivadent).
The buffering capacity of the saliva was determined using
the CRT® buffer test (Ivoclar-Vivadent).
Evaluation of oral health. All the children had a clinical examination by two trained and calibrated paediatric dentists.
The intra- and inter-examiner reliability tests for caries, initial/
white spot lesions and hypoplasia were performed prior to
the study and showed kappa values greater than 0.8.
OH was assessed using the plaque index (PI) of Silness and
Loe [1964] on the six Ramfjord teeth (16,12,24,36,32,44 proximal, buccal, lingual) with the use of a disclosing agent
(score 0=no dental plaque in the gingiva area, score 1= a film
of dental plaque adhering to the free gingival margin (only
upon probing), score 2=moderate accumulation of dental
plaque at the gingival margin (seen by naked eye), score
3=abundant dental plaque in the gingival margin).
Dental caries was assessed after professional cleaning of
the tooth surfaces and both cavitated and initial/white spot
lesions were registered. Cavitated carious lesions were diagnosed after visual examination using a blunt probe and a
dental mirror according to the criteria by Koch et al. [1967]:
score 1= the explorer sticks to the tooth surface and demands effort in order to be removed, score 2= loss of dental
tissue with cavitation. The dmf/DMF indices were used to
express caries experience [Klein, 1939]. The evaluation of
the caries was also based on bite-wings radiographs.
Initial/white spot lesions were defined as opacities of white,
or brownish colour with oval or round shape following the
gingival margin with decreased enamel translucency and
clearly defined from the adjacent enamel. They usually appeared on single teeth and predominantly on the incisal two
thirds of the crown. [ICDAS, 2005; Nyvad et al., 2008].
Hypoplasia was defined as when a tooth surface was determined as showing that its colour was white, yellow or brown
and its surface was disintegrated, abraded, or even having an atypical restoration (extensive, irregular and atypical
restoration replacing broken and hypomineralized enamel)
[Koch et al., 1987].
Anomalies of the teeth and evaluation of the occlusion was
noted when there was presence of crowding, and of posterior or anterior crossbite as well as the type of malocclusion were recorded. Developmental anomalies related to the
number, size and morphology of the teeth were clinically and
radiographically evaluated by panoramic radiograph.
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Statistical analysis. Descriptive statistics, including mean
value and standard deviation of each clinical entity, was
carried out for all the parameters examined. The level of
significance was set at p<0.05. The t-test and Chi-Square
test were used to examine the difference between the two
groups concerning all the parameters registered. The same
tests were also used to investigate the differences in the OH
condition and microbiological parameters between children
from the CLP group and HLP group that were undergoing
orthodontic treatment.
Results
Questionnaire. Based on the questionnaire, toothbrushing
frequency was found to be similar in both groups (cleft and
control) with approximately half of the children brushing their
teeth 2 x per day (Figure 1a). From the cleft group, 53.7%
of the parents reported that they supervised their children’s
toothbrushing on a daily basis while in the control group,
39% of the parents did so (Figure 1b). Significantly (p<0.05)
more patients with CLP were taking fluoride supplements
(29%) than the ones in the control group (5%). The majority of
patients from both groups had had at least one professional
fluoride application in the last 12 months (80.4% of the CLP
and 90.2% of HLP group, p>0.05). Data on the frequency of
daily consumption of juices and refreshments showed that
few patients from both groups consumed drinks more than
3 times daily (7.3% and 4.9% of CLP and HLP respectively,
p>0.05) while 61% from both groups consumed drinks 1 to
3 times daily. Children of both groups had an average of 3
main and 2 snack meals per day, while, on an average, one
and a half of those meals contained sugars.
Saliva evaluation and assessment of cariogenic microbiota.
More than 70% of children from both CLP and HLP had high
levels of mutans streptococci and lactobacilli (>105 cfu/ml).
Normal salivary flow rate (>0.7ml/min) was found in 55.2% of
CLP children and 65.9% of HLP children. High buffering capacity was found in 91.9% of children with CLP and 92.6%
of HLP children. Differences were not statistically significant
(p>0.05). Patients from both CLP and HLP group being under orthodontic treatment had high levels of cariogenic bacteria (Table 1).
Oral health evaluation. The plaque index of the total dentition was significantly higher in the CLP than in the control
group (CLP group, mean score: 1.03, SD: 0.35 vs HLP group,
mean score: 0.76, SD±0.29, p=0.0003) and the same applied
for the teeth adjacent to the cleft (CLP group, mean score:
1.11, SD±0.52 vs HLP group, mean score: 0.82, SD±0.40,
p=0.0027). The tendency was similar when the factor of orthodontic treatment was taken into consideration. CLP subjects under orthodontic treatment had worse OH than those
in the HLP group (Table 1).
No significant differences were found in the prevalence of
cavitated carious lesions between patients with cleft and
without. The mean dmfs score was found to be 7.24 for the
Oral health of patients with cleft lip and palate
Figure 1a
Figure 1b
Figure 1. Bar graphs showing percentage of: a. Toothbrushing frequency in the CLP and the HLP group; b. Parental supervision
during toothbrushing in the CLP and the HLP group.
Figure 2. Caries experience in the primary dentition in the CLP and HLP groups.
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Parapanisiou et al.
CLP and 8.38 for the HLP group (Figure 2). For both groups,
the filled surfaces component was the largest of the dmfs
index mean score. A similar picture was seen for the permanent dentition (CLP: mean score: DMFS 3.00 and FS 2.39
vs HLP: mean score: DMFS 3.40 and FS 2.05). The situation
was also similar between CLP and HLP subjects under orthodontic treatment (Table 1).
The majority of initial/white spot lesions in the permanent
dentition were found in the CLP group (85%, p=0.000). Most
of patients in the CLP group had 1 to 3 initial/white spot
lesions, mainly on the maxillary front teeth (Table 2). One
should take into consideration that most of children in CLP
group were under orthodontic treatment.
The percentage of children with enamel hypoplasia was
higher in the CLP group than in the HLP group (29.3% versus
17% respectively). However this difference was not statistically significant (p>0.05).
Supernumerary teeth were not found in the control group
while 9.8% of children from CLP group had at least one supernumerary tooth (p<0.05). Rotated and missing teeth were
found significantly more often in the CLP group than in the
HLP group (68.3% and 43.9% vs 29.3% and 5% respectively, p=0.000). Anterior and posterior crossbites were found
more often in the CLP group than in the HLP group (58.5%
and 39% vs 9.8% and 0%, respectively, p<0.001)
Discussion
This was a comparative cross-sectional study of children
with and without CLP matched for sex, age and orthodontic
treatment. The results from the questionnaire showed that
the majority of the study subjects with and without CLP
brushed their teeth at least once a day without parental supervision in most cases. This is in agreement with the results
of the study by Dahllof et al. [1989], which is the only study,
as far as we know, that compared the OH habits between
CLP and HLP children and adolescents.
Table 1. Oral health status in CLP and HLP in a group of
Greek children going under orthodontic treatment.
CLP (mean value) HLP (mean value)
P1I *
1.04
0.80
PlIs **
1.19
0.85
Dmft
3.00
4.16
DMFT
2.61
1.25
SM >105 cfu/ml
78.57%
85.7%
LB >10 cfu/ml
75%
64.2%
5
CLP: patients with cleft; HLP: healthy patients; PlI: plaque index on Ramfjord teeth, respectively; PlIs: plaque index on teeth situated near the cleft, respectively; dmft: caries index in the deciduous dentition; DMFT: caries index
in the permanent dentition; MS: levels of mutans streptococci; LB: levels of
lactobacilli; *p=0.004; **p=0.000
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Table 2. Distribution of initial/white spot lesions in the CLP
and HLP in a group of Greek children under going orthodontic treatment.
Number of initial/white spot lesions
0
1
2
3
4
5
6
8
10
CLP
17
7
4
5
3
2
1
1
1
HLP
35
3
-
2
1
-
-
-
-
CLP: patients with cleft; HLP: healthy patients
Regarding the use of fluoride supplements, the CLP group
received fluoride tablets more often than that of HLP group
(29.2% vs 4.8%). Although this difference was statistically
significant, this result should be evaluated with caution as
this finding was based on a reported behaviour.
In assessing the prevalence of cavitated carious lesions in
both the CLP and the HLP group, the findings in our study
are in agreement with those of other studies [Lauterstein and
Mendelsohn, 1964; Lucas et al., 2000]. In a recent systematic review, Hasslof and Twetman [2007] concluded that the
literature has failed to prove that there is difference in the
caries status between patients with and without CLP, up to
now. In our study, caries diagnosis was based on both clinical and radiographic examination. While no differences were
found in the prevalence of cavitated carious lesions, significantly more initial/white spot lesions were found in CLP than
HLP group (85% vs 15%). Our finding is in agreement with
the study by Bokhout et al. [1996]. It seems that patients
with CLP may be at greater risk for progression of initial/
white spot lesions to cavitated caries. Primarily the poor OH
in CLP patients might explain this finding.
Possible reasons for the poorer OH hygiene in CLP group
are the following: the difficulty in tooth cleaning because of
the presence of residual scar tissue as a result of the multiple
surgical procedures carried out at the cleft region; the lack of
interest for oral hygiene due to many other health problems
such as otitis media, difficulty in speech and the fear that
children often have when they brush their teeth at the cleft
area. All the above factors make the importance of the application of individualized preventive oral health programs in
CLP patients, imperative.
The majority of children from CLP and HLP group had high
levels of mutans streptococci and lactobacilli. This can be
partially explained by the fact that both groups had poor OH
habits and similar caries experience as they were all patients
of the paediatric dental clinic where mostly high caries risk
patients are seeking oral health care.
Concerning the dental anomalies, supernumerary, missing
and rotated teeth were found to be significantly more frequent among the children with cleft (p<0.05). Furthermore
Oral health of patients with cleft lip and palate
the prevalence of enamel hypoplasia was higher in children
with CLP than those without, but the difference was not statistically significant. These findings are in agreement with the
literature [Dahllof et al., 1989; Vichi and Franchi, 1995; Slayton et al., 2003; Aizenbud et al., 2005]. The differences in the
prevalence of the dental anomalies in our study, as well as in
the literature, may be due to differences in the size and the
method of sampling plus the fact that most of the studies did
not have a control group [Aizenbud et al., 2005].
The design of the present study took into consideration the
criteria introduced by Twetman and Hasslof [2007]. However
due to the small size and the wide age-range of the sample,
it should be considered as a pilot study. Nevertheless this
research is the only study of its kind that has ever been carried out in Greece.
Conclusion
The increased numbers of initial/white spot lesions combined with poor OH found in a Greek group of children with
CLP predisposes to an increased risk for further development of carious cavitated lesions. As the majority of CLP
patients in the study were under orthodontic treatment, the
application of an intensive individualized OH preventive program focused on the improvement of OH and remineralisation of initial/white spot carious lesions is imperative.
Acknowledgements
Approval for the study was obtained from the Ethics Committee of the Dental
School of the University of Athens; a written consent from all the participants/
parents was obtained.
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