Received: 7 August 2020

| Accepted: 9 February 2021

DOI: 10.1111/eos.12781

ORIGINAL ARTICLE

Evaluation of masticatory function in patients with cleft lip and/

or palate

Diana Cassi1 | Alberto Di Blasio2 | Laura Di Benedetto3 | Corrado De Biase4 |

Giuseppe Pedrazzi5 | Maria Grazia Piancino4
1
Surgical, Medical and Dental
Department of Morphological Sciences Abstract
related to Transplant, Oncology and The aim of this study was to evaluate the masticatory pattern in children with cleft lip
Regenerative Medicine, University of
and/or palate (CL/P) through investigation of the prevalence of reverse sequencing
Modena and Reggio Emilia, Modena,
Italy chewing cycles. The study group included 18 patients with CL/P (mean age: 7.4 yr,
2
Orthodontic Division, Centro SD: 1.4 yr), 15 of whom had dental crossbite. The controls included a group of 18
Universitario di Odontoiatria, Department non-­CL/P children with the same types of crossbite as the study group (mean age:
of Medicine and Surgery, University of
Parma, Parma, Italy 7.2 yr, SD: 1.5 yr) and a group of 18 non-­CL/P subjects with normal occlusion (mean
3
Doctoral School in Life and Health age: 9.8 yr, SD: 1.9 yr). Mandibular movements during chewing of soft and hard
Science, University of Turin, Turin, Italy bolus were recorded with a kinesiograph. Kinematic signals were analysed using a
4
Orthodontic Division, Dental School, custom-­made software. A statistical analysis was performed to compare the degree
Department of Surgical Sciences,
University of Turin, Turin, Italy
of reverse-­sequencing chewing cycles between patients and controls (Kruskal–­Wallis
5
Unit of Neuroscience, Department of test with Dwass-­Steel-­Critchlow-­Fligner pairwise comparisons post hoc test). A sig-
Medicine and Surgery, Interdepartmental nificant difference between patients with CL/P and non-­CL/P subjects with normal
Centre of Robust Statistics, University of
occlusion was highlighted on the left side of mastication, which was the side with
Parma, Parma, Italy
the higher prevalence of crossbite with both types of bolus. No statistical differences
Correspondence were found between CL/P patients and healthy controls with crossbite. Cleft-­affected
Diana Cassi, Centro Universitario di
patients with posterior crossbite exhibited an anomalous masticatory pattern with in-
Odontoiatria, Via Gramsci 14 -­43125,
Parma, Italy. creased reverse chewing cycles on the crossbite side.
Email: diana.cassi@unipr.it
KEYWORDS
Funding information birth defect, chewing, congenital abnormalities, craniofacial abnormalities, malocclusion
This research did not receive any specific
grant from funding agencies in the public,
commercial, or not-­for-­profit sectors.

I N T RO D U C T ION arch relationship [3]. In particular, the inter-­arch discrep-

Orofacial clefts represent a heterogeneous group of con-
genital disorders involving the lips and the oral cavity. The
main categories are isolated cleft palate and cleft lip with or
without cleft palate (CL/P) [1]. Although these defects can
be surgically repaired in childhood, residual deformity due
to scarring of oral and facial structures results in a range of
continuing functional and aesthetic problems [2]. Maxillary
growth deficiency is usually observed in operated patients
born with CL/P, and it affects facial development and dental
ancy frequently results in dental crossbite occurring in the
early dentition [4]. Crossbite is a complex, asymmetric, and
worsening malocclusion that involves the teeth and all the
components of the masticatory system and their functions. It
results from dental and/or skeletal discrepancy between the
opposing arches and may lead to displacement or malposition
of the mandible [5].
It has long been known that children with unilateral pos-
terior crossbite display modified chewing patterns during
mastication using the crossbite side [6–­10]. This alteration

Eur J Oral Sci. 2021;00:e12781. wileyonlinelibrary.com/journal/eos © 2021 European Journal of Oral Sciences | 1 of 10
https://doi.org/10.1111/eos.12781
2 of 10
|    CASSI et al.
is associated with a significant increase in the frequency of
reverse-­sequence chewing pattern, which refers to the abnor-
mal movement of the mandible during the closing phase of
chewing. The definition of “reverse chewing cycle,” as the
inversion of the closing direction of the chewing cycle, was
provided by Lewin [6] and then adopted by other authors [8–­
10]. The direction of closure is represented by the vector of
the closing pattern in the last stage of the chewing cycle: it
represents a clinical indicator of chewing pattern characteris-
tics and balanced mastication and it does not change during
growth [6,11]. In cases of unilateral posterior crossbite,
during mastication on the crossbite side, the closing direc-
tion will be reversed with respect to the condition of normal
occlusion. According to the literature, the chewing pattern
during mastication on the healthy side maintains the same
features as physiological mastication regarding the closing
direction and the morphological and positional features (i.e.,
height, width, and spatial position that display no differences
from normal occlusion) [11]. The reverse sequence chewing
cycle is set and maintained by the automatisms of the central
nervous system's motor control, which is based on periph-
eral input arriving from the periodontal mechanoreceptors
[12,13].
Although the physiology of mastication has been thor-
oughly investigated, only a few studies have described the
chewing cycle in patients with congenital orofacial condi-
tions like CL/P. Understanding the influence of dentofacial
morphology on masticatory function might have a clinical
relevance in the management of cleft-­affected patients who
need prolonged, multidisciplinary care. The knowledge of al-
teration of the stomatognathic system may improve the com-
prehensive treatment of this complex condition.
The aim of the present study was therefore to evaluate the
masticatory function in children with CL/P through the inves-
tigation of the degree of reverse-­sequencing chewing cycles.
Specifically, we compared the occurrence of reverse chew-
ing patterns of patients with CL/P with an age and crossbite-­
matched group without CL/P, and a control group without
CL/P and crossbite. The null hypothesis was that there was
no significant difference between cleft-­affected subjects and
healthy controls with the same types of crossbite.
MAT E R IA L AN D ME T HOD S
Study participants
Fifty-­four children were included in this observational study.
The study group consisted of 18 consecutive patients with
CL/P (11 boys, seven girls; mean age [SD] = 7.4 [1.4] yr; age
range: 4.1–­12.9 yr), including unilateral CLP (UCLP), bilat-
eral CLP (BCLP), CP and CL. All patients were operated by
the same surgical team according to the following protocol:
(i) for complete clefts (unilateral and bilateral), a two-­stage
lip and palate repair procedure was performed that included
lip, nose, and soft palate closure at the age of 5–­6 months,
followed by hard palate closure and early gingival-­alveolar-­
plastic (GAP) surgery at the age of 16–­18 months; (ii) for
isolated CL or CP, a single-­stage lip or palate repair at the
age of 5–­6 months was performed (i.e., the lip or hard and
soft palates were closed in a single operation). Based on the
occlusal diagnosis, which was performed by two trained op-
erators on the dental casts, patients were classified according
to the presence and type of crossbite as follows: (i) anterior,
(ii) right or left unilateral posterior, (iii) unilateral posterior
and anterior, (iv) bilateral posterior, or (v) bilateral posterior
and anterior (Figure 1). For the crossbite classification, the
operators adopted the following definitions: anterior cross-
bite is a condition where one or more primary or permanent
maxillary teeth in the canine and/or incisor region occlude
lingual to their antagonist; posterior crossbite corresponds to
the condition of one or more primary or permanent maxillary
teeth in the molar and/or premolar regions occluding in an
abnormal buccolingual relation with their antagonists (i.e.,
the buccal cusps of the maxillary posterior teeth contact or
occlude lingually to the buccal cusps of the corresponding
mandibular teeth).
In addition to the CL/P group, the following two groups
were recruited: an age and crossbite-­matched group without
CL/P (18 children: 11 boys, seven girls; mean age [SD] = 7.2
[1.5] yr; age range: 5.1–­10 yr) and a control group with-
out CL/P and crossbite (18 children: nine boys, nine girls;
mean age [SD] = 9.8 [1.9] yr; age range 7.4–­16.8 yr). They
were selected among patients referred to the Department of
Orthodontics, University of Turin (Italy).
Before participating in the study, informed consent was
obtained from the parents and the study was approved by
the Institutional Review Board of the University Hospital
“Health and Science Complex Turin -­Italy” no. CS/246, in
accordance with the Code of Ethics of the World Medical
Association (Declaration of Helsinki) for experiments in-
volving humans.
Inclusion criteria for the cleft group were the presence of
repaired CL/P; inclusion criteria for the control group with
crossbite were absence of congenital malformation and pres-
ence of crossbite involving any dental region (anterior and/
or posterior, unilateral, and bilateral). The inclusion criteria
for the control group with normal occlusion were absence of
congenital malformation and absence of crossbite. Exclusion
criteria for all participants were: signs or symptoms of myo-
facial disorders causing discomfort or pain of the masticatory
musculature; previous orthodontic therapy; presence of mobile
exfoliating primary teeth; presence of caries or pain; presence
of any prosthesis; presence of diabetes and/or celiac disease, as
the boluses used in the present study contained sugars that are
contraindicated in subjects with these medical conditions.
EXPLORING CHEWING PATTERN IN CHILDREN WITH CL/P
F I G U R E 1 Crossbite classification in (A)
cleft lip and/or palate (CL/P) patients: (A)
anterior; (B) right or left unilateral posterior;
(C) unilateral posterior and anterior; (D)
bilateral posterior; (E) bilateral posterior
and anterior
(C)
Recording of chewing cycles
Recordings of the chewing cycles were performed for all sub-
jects by the same operator, following a standardized protocol, in
a silent and comfortable environment [14]. Patients were com-
fortably seated in a chair and they were asked to fix the eyes
on a target placed 90 cm away directly in front of their seating
position, and to avoid movements of the head. Each record-
ing began in maximal intercuspation. Patients were asked to
find the starting position by lightly tapping their teeth together
and clenching. They were then asked to hold this position with
a test bolus on the tongue before recording. Participants were
subsequently instructed to chew a soft bolus and then a hard
bolus: each bolus was chewed first on the right side, then on
the left side and, eventually, in a free mode. The duration of
each test was 10 s and the test was repeated three times. The
side of mastication was visually checked by an operator and,
at the end of the recording, participants were asked to spit the
bolus in a glass to assess whether it had actually been chewed.
The soft and hard boluses used were a chewing gum and a wine
gum, respectively. Both gums had the same size (20 mm in
length, 1.2 mm in height, and 0.5 mm in width) but different
weights (2 g for the soft bolus and 3 g for the hard bolus). The
wine gum was chosen to provide a rubber-­like texture without
sticking to the teeth.
Mandibular movements were measured with a kinesio-
graph (K7; Myotronics), which measures jaw movements
with an accuracy of 0.1 mm. Multiple Hall effect sensors in
a light-­weight array (113 g) tracked the motion of a mag-
net attached to the midpoint of the lower incisors [15]. The
  
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(B)
(D)
(E)
kinesiograph was interfaced with a computer for data storage
and analysis. The kinematic signals were analysed using a
custom-­made software (Department of Orthognatodontics,
University of Turin, Italy). The first cycle, during which the
bolus was transferred from the tongue to the dental arches,
was excluded from the analysis. The chewing cycles were
divided into non-­reverse and reverse, based on the vector di-
rection of closure.
Repeatability of measurements was assessed on 10 partic-
ipants, through two series of soft bolus chewing recordings
performed by the same operator. The interval between the
two recordings was 15 min.
Differences in both right and left chewing sequences were
evaluated among the three groups.
For CL/P patients and controls with crossbite, differences
between pathologic and normal side of mastication were fur-
ther evaluated as follows: (i) in patients with unilateral pos-
terior crossbite with or without anterior crossbite, right and
left chewing sequences were pooled according to “affected”
and “not affected” side; (ii) in patients with bilateral posterior
crossbite with or without anterior crossbite, right and left se-
quences were pooled as “affected” side; (iii) in patients with
anterior crossbite or without crossbite, right and left chewing
cycles were pooled as “not affected” side.
Sample size calculation and statistical analysis
Normal distribution of the data was tested using
Kolmogorov-­Smirnov test and Shapiro-­Wilk test. Previous
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TABLE 1 Masticatory function data of the study group according to age, gender, type of cleft, and location of crossbite

Chewing on the right side Chewing on the left side

Soft bolus Hard bolus Soft bolus Hard bolus

Age (yr,
Patient Gender months) Cleft Crossbite TOT REV % TOT REV % TOT REV % TOT REV %
1 M 5, 5 BCLP Ant 37 2 5 38 0 0 37 10 27 41 1 2
2 M 5 UCLP Unilateral post 28 4 14 42 0 0 30 29 97 40 40 100
3 F 12, 9 CL Unilateral post 35 4 11 46 21 46 30 2 7 46 21 46
4 M 9, 8 CP No CRB 39 9 23 39 10 26 38 1 3 42 1 2
5 M 5 UCLP Unilateral post/ant 24 1 4 34 13 38 40 2 5 20 0 0
6 F 8, 9 UCLP Unilateral post/ant 41 4 10 50 2 4 38 2 5 46 4 9
7 F 6, 10 UCLP Unilateral post/ant 30 30 100 22 22 100 31 0 0 23 1 4
8 F 7, 7 UCLP Unilateral post/ant 17 3 18 20 1 5 25 15 60 24 23 96
9 M 7, 1 BCLP Bilateral post/ant 33 15 45 41 34 83 36 15 42 40 11 28
10 F 5, 7 BCLP Unilateral post/ant 22 1 5 30 0 0 22 14 64 27 25 93
11 M 6, 4 UCLP Unilateral post/ant 25 0 0 27 6 22 24 4 17 28 14 50
12 M 7, 10 BCLP Ant 28 7 25 36 5 14 35 9 26 40 6 15
13 F 4, 11 BCLP Bilateral post Not recorded 25 16 64 Not recorded 25 11 56
14 F 5, 9 BCLP Unilateral post/ant 34 13 38 31 11 35 38 13 34 32 20 63
15 M 9, 7 CL No CRB 40 13 33 38 6 16 44 6 14 40 3 8
16 M 9, 5 CL No CRB 42 10 24 35 2 6 45 24 53 37 0 0
17 M 5, 6 BCLP Bilateral post/ant 11 0 0 6 5 83 12 12 100 12 11 92
18 M 9, 0 CL Ant 36 11 31 43 1 2 43 4 9 41 2 5
Abbreviations: %, percentage of reverse chewing cycles; ANT, Anterior; BCLP, Bilateral Cleft Lip and Palate; CL, Cleft Lip; CP, Cleft Palate; CRB, Crossbite; F, Female; M, Male; POST, Posterior; REV, Reverse chewing
cycles; TOT, Total number of cycles; UCLP, Unilateral Cleft Lip and Palate.
CASSI et al.
EXPLORING CHEWING PATTERN IN CHILDREN WITH CL/P
TABLE 2 Repeated measurements of reverse chewing cycles
1st meas. 2nd meas.
Chewing Mean Mean Median difference
Side (%) (median) (median) (95% CI) Bias
RIGHT 2.3 (1.0) 2.0 (0.0) 0.5 (−2.0; 3.0)
LEFT 22.8 (8.50) 26.4 (13.0) −2.43 (−16.0; 3.0)
Note: Median difference, Bland-­Altman analysis, and Lin's concordance correlation coefficient (CCC). The median of paired differences, 95% confidence interval and
p-­values were obtained from the Wilcoxon's signed rank test.
Abbreviation: LoA, limits of agreement.
investigations on reverse-­sequencing chewing patterns in
patients with unilateral posterior crossbite [9] suggested
that the effect size—­ defined as the difference between
two group means divided by their pooled standard devia-
tion (Cohen's d), or as the standard deviation of several
population means (three or more groups) divided by the
common within-­ population standard deviation obtained
from an overall ANOVA F test (Cohen's f)—­could be large
(Cohen's d > 1.1 and Cohen's f > 0.5), leading us to use
an f estimate equal to f = 0.53. We then used G*Power
v.3.1.9.2 [16,17] to provide a rough estimation of the re-
quired sample size. Assuming a comparison of three groups
(patients with CL/P, controls with crossbite, controls with
normal occlusion) at α = 0.05 (5%), power = 0.8 (80%),
and f = 0.53, the required sample size was estimated to be
39 units (13 per group). Assuming a stratification for bolus
hardness (soft, hard) and then comparing six groups with
the same alfa, power, and effect size, the required sample
size was estimated to be 54 units (18 per group).
To assess the repeatability of the recording procedure, the
differences in percentages of reverse chewing cycles between
the first and second set of measurements were evaluated
using Bland-­Altman analysis and plot, Lin's concordance co-
efficient (CCC), intraclass correlation coefficient (ICC), and
the Wilcoxon's test for paired comparisons. It is commonly
accepted that pairs of measurements have an excellent repro-
ducibility if Lin's CCC and ICC are higher than 0.9, good if
higher than 0.8, and acceptable if higher than 0.7.
Given that continuous variables were not normally dis-
tributed, Kruskal–­Wallis test with Dwass-­Steel-­Critchlow-­
Fligner pairwise comparisons post hoc test were used to
compare frequencies of reverse chewing cycles between the
control and patient groups. The relevant variable was the pro-
portion of reverse chewing cycles, which was computed by
dividing the number of reverse chewing cycles by the number
of recorded chewing cycles in each set of recordings. Given
that the denominator value was similar for each participant,
the uncertainty of measurements was roughly the same; thus,
the proportion of reverse chewing cycles was considered a
continuous variable.
Descriptive plots were used to represent the distribution
of reverse chewing cycles in each group.
  
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Bland-­Altman analysis (95% CI)
LoA-­lower LoA-­upper CCC
0.3 (−1.0; 1.60) −3.28 (−5.6; −0.96) 3.88 (1.57; 6.20) 0.87 (0.61; 0.96)
−3.6 (−10.4; 3.29) −22.5 (−34.7; −10.3) 15.3 (3.1; 27.5) 0.93 (0.76; 0.98)
The statistical analyses were performed with the statistical
software jamovi version 1.6.13 [18] and r version 4.0.3 [19].
The results were considered statistically significant for a p-­
value <5% (p < 0.05).
RESULTS
Demographic details and masticatory function data of pa-
tients with CL/P are reported in Table 1. The distribution
of CL/P was: six patients with UCLP, seven patients with
BCLP, one patient with CP, and four patients with CL. When
considering occlusion, seven (39%) patients showed unilat-
eral posterior and anterior crossbite (two on the right and five
on the left side), two (11%) had unilateral posterior crossbite
(one on either side); thus, three (17%) patients had crossbite
on the right side and six (33%) on the left side. Two (11%)
patients showed bilateral posterior and anterior crossbite, one
(5%) had bilateral posterior crossbite, three (17%) had an-
terior crossbite, and three (17%) had no crossbite. Occlusal
discrepancies were related to the type of cleft as follows: uni-
lateral posterior and anterior crossbite in five patients with
UCLP and in two patients with BCLP; the two cases with bi-
lateral posterior and anterior crossbite had BCLP; unilateral
posterior crossbite was observed in one patient with UCLP
and in one patient with CL; the single case with bilateral pos-
terior crossbite had BCLP; anterior crossbite was observed
in two patients with BCLP and one patient with CL; one pa-
tient with CP and two patients with CL had no crossbite. The
distribution of types of crossbite was the same in the control
group with crossbite as in the CL/P group.
The results of the assessment of recording protocol re-
peatability are shown in Table 2 and Figure 2. The differ-
ences in the prevalence of reverse chewing cycles between
the first and second evaluations were not statistically signif-
icant (p > 0.05). The medians of paired difference between
the first and second measurements were 0.5 (−2.0; 3.0) and
−2.43 (−16.0; 3.0) for the right and left side, respectively.
Lin's CCC was 0.87 (0.61; 0.96) for the right side and 0.93
(0.76; 0.98) for the left side.
A statistically significant difference in the proportion
of reverse chewing cycles between patients with CL/P and
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|    CASSI et al.

FIGURE 2 Bland-­Altman plots for the repeated measurements of reverse chewing cycles

T A B L E 3 The proportion of reverse chewing cycles in cleft patients (CL/P), control subjects with normal occlusion (Control), and non-­CL/P
patients with crossbite (CRB) when exposed to a soft or a hard bolus

CL/P Control CRB CL/P versus control CRB versus control CL/P versus CRB

Median Median Median Median difference Median difference Median difference

(Q1; Q3) (Q1; Q3) (Q1; Q3) (95% CI) p-­value (95% CI) p-­value (95% CI) p-­value
Chewing on the right side (%)
Soft bolus 18 (5; 31) 3.5 (0; 15.3) 9.5 (3; 21.8) 14.5 (−2; 30.8) 6 (−6.5; 25.3) 8.5 (−11; 24)
0.059 0.495 0.421
Hard bolus 19 (4.24; 44) 4 (0; 9.5) 15 (4; 51.8) 15 (0.5; 33) 11 (0; 39) 4 (−32.7; 27)
0.046 0.057 0.965
Chewing on the left side (%)
Soft bolus 26 (7; 53) 3.5 (2.25; 15.8) 34.5 (4.5; 88) 22.5 (3; 50) 31 (1.5; 76.5) −8.5 (−64; 32.6)
0.013 0.012 0.896
Hard bolus 21.5 (4.25; 61.3) 3 (0.5; 7) 29.5 (3; 85.3) 18.5 (1; 53) 26.5 (0; 67) −8 (−71; 40.5)
0.015 0.029 0.973
Note: Median values, 25th percentile (1st quartile, Q1), 75th percentiles (3rd quartile, Q3), median difference with 95% CI and pairwise comparisons between the
groups. 95% confidence intervals for median differences were determined by bootstrap (BCa) technique (R packages “boot” and “simpleboot”). p-­values were obtained
by the Kruskal–­Wallis test followed by the Dwass-­Steel-­Critchlow-­Fligner post hoc test.

controls was found on the left side of mastication, which was
the side with the higher prevalence of crossbite (soft bolus
–­median difference [95% CI] = 22.5 [3; 50], p = 0.013;
hard bolus –­median difference [95% CI] = 18.5 [1; 53],
p = 0.015); on the right side, the differences were less pro-
nounced and the p-­value was higher, being just statistically
significant for the hard bolus (15 [0.5; 33], p = 0.046) and
barely insignificant for the soft bolus (14.5 [−2; 30.8],
p = 0.059; Table 3). Similarly, a statistically significant
higher proportion of reverse chewing cycles was found in
non-­CL/P controls with dental crossbite than in healthy sub-
jects with normal occlusion on the left side of mastication
(soft bolus –­median difference [95% CI] = 31 [1.5; 76.5],
p = 0.012; hard bolus –­median difference [95% CI] = 26.5
[0; 67], p = 0.029). On the right side, however, there was no
statistically significant difference between the two groups for
soft bolus (6 [−6.5; 25.3], p = 0.495), while the difference
was barely insignificant for hard bolus (11 [0; 39], p = 0.057;
Table 3). No statistically significant difference between pa-
tients with CL/P and non-­CL/P controls with crossbite was
detected, whether on the right or left side of mastication
(Table 3); this result was confirmed when comparing the per-
centages of reverse-­sequencing chewing patterns between the
crossbite and non-­crossbite sides (Table 4).
Eventually, the difference observed between the affected
and not affected side within each group was significant for non-­-
CL/P controls (soft bolus –­median difference [95% CI] = 64.0
[24.0; 86.0], p = 0.002; hard bolus –­median difference [95%
CI] = 69 [51.5; 85], p < 0.001) and significant for hard bolus
in patients with CL/P (soft bolus –­median difference [95%
CI] = 18.0 [−12.0; 48.0], p = 0.844; hard bolus –­median dif-
ference [95% CI] = 58.5 [35.5; 88.0], p < 0.001; Table 4).
Masticatory function and chewing pattern analysis of
CL/P group showed that 11 patients (61%) had reverse cycles
EXPLORING CHEWING PATTERN IN CHILDREN WITH CL/P   
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T A B L E 4 The proportion of reverse

Patients with Controls with CL/P patients –­Controls with
chewing cycles according to side of
CL/P CRB CRB
crossbite given for CL/P patients and
controls with crossbite (CRB) Median Median Median difference
(Q1; Q3) (Q1; Q3) (95% CI) p-­value
Crossbite affected side
Soft bolus 34.0 (8.0; 62.0) 68.0 (29.5; 90.5) −34.0 (−73.0; 14.0) 0.957
Hard bolus 63.0 (38; 92.0) 75.0 (38.0; 91.8) −12.0 (−37.5; 22.0) 0.986
Non affected side
Soft bolus 16.0 (5.5; 26.8) 4.0 (3.0; 11.0) 12.0 (−3.0; 24.0) 0.270
Hard bolus 4.5 (0.5; 14.8) 6.0 (1.5; 13.5) −1.5 (−9.0; 7.0) 1.000

Median difference Median difference

(95% CI) (95% CI)
p-­value p-­value
Affected side –­non affected side
Soft bolus 18.0 (−12.0; 48.0) 64.0 (24.0; 86.0)
0.844 0.002
Hard bolus 58.5 (35.5; 88.0) 69 (51.5; 85)
<0.001 <0.001
Note: Median values, 25th percentile (1st quartile, Q1), 75th percentiles (3rd quartile, Q3), median difference
with 95% CI. Statistical comparisons were performed with the Kruskal–­Wallis test followed by the Dwass-­
Steel-­Critchlow-­Fligner pairwise comparisons post hoc test. 95% CI for the median difference were obtained
by the bias corrected and accelerated (BCa) bootstrap technique using the R packages “boot” version 1.3-­25
and “simpleboot” version 1.1-­7.

F I G U R E 3 Bilateral chewing patterns of patient #11 with right unilateral posterior and anterior crossbite, during chewing on the right crossbite
side and on the left non-­crossbite side. Green tracing: opening; red tracing: closing; the green arrow indicates the normal closure direction (anti-­
clockwise with the left-­hand bolus); the red arrow indicates the reverse closure direction (anti-­clockwise with the right-­hand bolus)

during chewing on the crossbite side. Of the seven (39%) pa-
tients not showing reverse cycles, three had no crossbite and
three had crossbite only in the anterior inter-­canine region.
One patient with UCLP, unilateral posterior and anterior
crossbite, did not present reverse cycles.
An example of pathological chewing pattern in the frontal
plane is shown in Figure 3. The reverse sequence chewing
cycle occurs only on the crossbite side (right) whilst, on the
healthy side, the chewing cycle displays normal physiologi-
cal closing direction (left).
DISCUSSION
In the present study, the analysis of masticatory function was
performed in a group of growing subjects affected by CL/P
8 of 10
|    CASSI et al.
with different types of dental crossbites; particularly, we in-
vestigated the prevalence of reverse-­sequencing chewing cy-
cles during chewing of soft and hard boluses. According to
our preliminary results, cleft patients with posterior crossbite
(independently from the type of congenital malformation)
exhibit an anomalous masticatory pattern with an increased
frequency of reverse chewing cycles on the crossbite side.
Data were compared with the chewing patterns recorded in
an age and crossbite-­matched group without CL/P and in
a control group without CL/P and crossbite; we found that
mandibular kinetics in children with CL/P during chewing
are like those reported in non-­CL/P patients with the same
type of malocclusion, but different from controls without
malocclusion.
Only a few studies have been performed to evaluate mas-
ticatory function in patients born with a cleft condition, and
these have principally focused on masticatory muscle ac-
tivity [20,21] or bite force [22]. Recently, MONTES et al.
[23] investigated the capacity of 8-­to 10-­yr-­old children with
UCLP to comminute a test food and showed that these pa-
tients have poor masticatory performance. The effect of pos-
terior crossbite on the chewing pattern has previously been
described in three patients with UCLP by HIDESHIMA [24],
who found that reversed patterns were seen more frequently
during chewing on the crossbite side. A case was published
by MIYAWAKI and TAKADA [25] reporting the changes in
jaw movement and temporalis muscle activity of a patient
with UCLP before and after edgewise treatment: the reversed
crossing jaw-­ movement pattern before treatment changed
to a normal grinding after treatment. PIANCINO et al. [26]
described reverse-­sequencing chewing patterns in a patient
with BCLP and bilateral posterior crossbite. A recent study
investigated masticatory muscle activity in 25 children with
UCLP compared to a sample with no cleft abnormalities: the
authors found that the presence of unilateral posterior cross-
bite affects the temporal muscle activity in cleft patients, who
showed significantly increased electromyographical poten-
tials during rest position [27].
To the best of our knowledge, no studies have been pub-
lished so far reporting on mandibular kinetic movements
during chewing in a series of patients affected by cleft.
Our study has shown a higher prevalence of reverse-­
sequencing chewing cycles during chewing on the crossbite
side than in the non-­crossbite side. Interestingly, the number
of reverse sequence cycles was higher during mastication of
hard boluses compared to soft boluses, as it is represented in
Table 4. It is remarkable that the reverse sequence chewing
cycle occurred predominantly on the crossbite side whilst,
on the healthy side, the chewing cycle displayed physiologi-
cal closing direction. Our results are consistent with previous
findings on non-­cleft patients, showing that the percentage
of reverse sequence chewing cycles during mastication on
the crossbite side is extremely high (around 60%–­70% on
average), depending on the severity of the malocclusion and
the bolus type [9,11,28,29].
Functional asymmetry and anomalous chewing pattern
occurred in all subjects (except one) with posterior crossbite.
In contrast, patients with anterior crossbite displayed a chew-
ing pattern with no increased frequency of reverse sequence
chewing cycles. These findings are in accordance with those
observed in non-­cleft patients, in whom functional asymme-
try is typical of unilateral posterior crossbite, but is not pres-
ent in anterior crossbite [14]. Thus, the effect of crossbite on
chewing patterns during masticatory function differs accord-
ing to the dental region involved. The impact of malocclusion
on masticatory function might depend on the functional role
of the teeth involved, as the anterior teeth exert a major influ-
ence on postural control of the mandible rather than on the
chewing cycle in the frontal plane.
The similarity of masticatory function between children with
and without CL/P but with the same crossbite malocclusion is
not surprising, since it is related to the neuromuscular motor
control of mastication. Masticatory movements are character-
ized by rhythmicity and a diversity of patterns of jaw posture.
YOSHINO et al. [30] suggested that the primary motor cortex
may be involved in the initiation and control of jaw movements
and that the ventral pre-­motor cortex may be involved in motor
planning, therefore playing a role as a higher order motor area
related to the initiation and control of jaw movements [31,32].
The cortex sends signals to the brainstem, which elaborates
the patterns of mastication in response to the inputs from the
masticatory cortex and from the periphery [33,34]. Sensory
receptors, such as muscle spindles, periodontal, and even in-
tradental pressure receptors, exert strong influences on the
chewing pattern being generated by the central pattern genera-
tor, eventually modifying both the frequency of motor neuron
bursts and their intensity [35]. Inputs from mechanoreceptors
are critical, not only for various trigeminal reflexes (such as
the jaw-­opening reflex or the periodontal-­masseter reflex), but
also for masticatory control [36,37]. These observations could
explain the results of this study: conceivably, the birth defect is
not responsible for masticatory disorders per se, but it affects
the morphological characteristics of the orofacial structures,
generating the malocclusion to which, in turn, the altered man-
dibular kinetics are linked. Investigations of the electromyo-
graphic activity during reverse and non-­reverse chewing cycles
in unilateral posterior crossbite showed that the asymmetry of
the kinetic pattern results in unbalanced neuromuscular acti-
vation [28,38]. Given that the unilateral posterior crossbite
usually occurs at a very early age, functional asymmetry may
disturb the craniofacial development, leading to an asymmetric
growth of anatomical structures at the end of growth that can
no longer be corrected by orthodontic therapy alone [8,39–­42].
According to the literature, the orthodontic correction
of unilateral posterior crossbite through appropriate appli-
ances leads a significant reduction in the reverse sequencing
EXPLORING CHEWING PATTERN IN CHILDREN WITH CL/P
of masticatory cycles on the affected side [9,29]. Thus, the
resolution of the dental malocclusion might be advisable to
restore the masticatory function and improve the muscular
coordination.
The main limitation of this study is the heterogeneity of
the cleft group in terms of the type of congenital malforma-
tion and malocclusion. Orofacial clefts show a broad spec-
trum of clinical presentations due to a complex aetiology
with both genetic and environmental contributions; the level
of expression and severity ranges from minor defects to im-
portant craniofacial abnormalities and they include CL alone,
CL plus CP, and CP alone. This represents a particular chal-
lenge and results in variability of the occlusal traits. In the
present study, we chose to include consecutive patients with
phenotypic diversity and to detail their chewing patterns,
considering that masticatory function is poorly investigated
in this population.
The resulting dental occlusion in CL/P cases is also
influenced by the surgical treatments performed: notably,
the frequency and severity of crossbite are consequences
of the maxillary arch constriction caused by surgery.
Moreover, surgical protocols widely differ with regard to
timing and techniques, and they may well affect interarch
discrepancy differently, thus impacting chewing pattern.
Therefore, the results of the present study should also be
considered in the light of the surgical approach adopted.
Particularly, it should be borne in mind that an intermedi-
ate bone grafting is considered the gold standard in many
centers; however, such an approach may be associated
with a residual alveolar cleft and a lower prevalence of
anterior crossbite, which can have a significant influence
on chewing patterns.
Our preliminary results should be considered in the context
that further investigations involving a larger number of pa-
tients with more a homogeneous phenotype would be needed
to reduce the influence of interfering factors. Additionally,
prospective studies with long-­term follow-­up are necessary
to investigate the functional changes after orthodontic correc-
tion of posterior cross-­bite in cleft-­affected subjects.
CONFLICT OF INTEREST
The authors declare no conflicts of interest.
AUTHOR CONTRIBUTIONS
Diana Cassi: writing -­original draft preparation (lead),
visualization (lead), review and editing (equal); Alberto Di
Blasio: resources (lead), supervision (lead), review and edit-
ing (equal); Laura Di Benedetto: validation (lead), investi-
gation (supporting), review and editing (equal); Corrado De
Biase: investigation (lead), data curation (lead), review and
editing (equal); Giuseppe Pedrazzi: formal analysis (lead),
visualization (supporting), review and editing (equal); Maria
Grazia Piancino: conceptualization (lead), methodology
  
| 9 of 10
(lead), project administration (lead), writing -­original draft
preparation (supporting), review and editing (equal).
ORCID
Diana Cassi https://orcid.org/0000-0001-9076-1337
Alberto Di Blasio https://orcid.org/0000-0002-0219-6946
Laura Di Benedetto https://orcid.
org/0000-0002-1712-6780
Corrado De Biase https://orcid.
org/0000-0002-7129-1971
Giuseppe Pedrazzi https://orcid.
org/0000-0002-5971-2040
Maria Grazia Piancino https://orcid.
org/0000-0002-0220-5283
R E F E R E NC E S
1. Mossey PA, Little J, Munger RG, Dixon MJ, Shaw WC. Cleft lip
and palate. Lancet. 2009;374:1773–­85.
2. Hartsfield JK. Clinical genetics for the dental practitioner. In: Dean
JA editor. McDonald and Avery's dentistry for the child and ado-
lescent, 10th edn. St. Louis, MO: Mosby; 2015. p. 87–­109.
3. Vig KWL, Mercado AM. Overview of orthodontic care for chil-
dren with cleft lip and palate, 1915–­2015. Am J Orthod Dentofac
Orthop. 2015;148:543–­56.
4. Cassi D, di Blasio A, Gandolfini M, Magnifico M, Pellegrino
F, Piancino MG. Dentoalveolar effects of early orthodontic
treatment in patients with cleft lip and palate. J Craniofac Surg.
2017;28:2021–­6.
5. Magnifico M, Di Blasio A, Cassi D, Di Blasio C, Gandolfini M.
Asymmetric expansion with a modified quad helix for treatment of
isolated crossbite. Case Rep Dent. 2017;2017:1–­5.
6. Lewin A. Electrognathographics: Atlas of diagnostic procedures
and interpretation. Berlin: Quintessence, 1985; p. 82–­5. ISBN13
978-­0867151565.
7. Ben-­Bassat Y, Yaffe A, Brin I, Freeman J, Ehrlich Y. Functional
and morphological-­occlusal aspects in children treated for unilat-
eral posterior cross-­bite. Eur J Orthod. 1993;15:57–­63.
8. Throckmorton GS, Buschang PH, Hayasaki H, Pinto AS. Changes
in the masticatory cycle following treatment of posterior uni-
lateral crossbite in children. Am J Orthod Dentofac Orthop.
2001;120:521–­9.
9. Piancino MG, Talpone F, Dalmasso P, Debernardi C, Lewin A,
Bracco P. Reverse-­sequencing chewing patterns before and after
treatment of children with a unilateral posterior crossbite. Eur J
Orthod. 2006;28:480–­4.
10. Sever E, Marion L, Ovsenik M. Relationship between masticatory
cycle morphology and unilateral crossbite in the primary dentition.
Eur J Orthod. 2011;33:620–­7.
11. Piancino MG, Kyrkanides S. Understanding masticatory function
in unilateral crossbites. Iowa: John Wiley & Sons; 2016. https://
onlin​elibr​ary.wiley.com/doi/book/10.1002/97811​18971901
12. Lund JP, Kolta A. Generation of the central masticatory pat-
tern and its modification by sensory feedback. Dysphagia.
2006;21:167–­74.
13. Morquette P, Lavoie R, Fhima M-­D, Lamoureux X, Verdier D,
Kolta A. Generation of the masticatory central pattern and its mod-
ulation by sensory feedback. Prog. Neurogibol. 2012;96:340–­55.
10 of 10
|    CASSI et al.
14. Piancino MG, Comino E, Talpone F, Vallelonga T, Frongia G,
Bracco P. Reverse-­sequencing chewing patterns evaluation in an-
terior versus posterior unilateral crossbite patients. Eur J Orthod.
2012;34:536–­41.
15. Jankelson B. Measurement accuracy of the mandibular kinesio-
graph -­a computerized study. J Prosthet Dent. 1980;44:656–­66.
16. Faul F, Erdfelder E, Lang A-­G, Buchner A. G*Power 3: a flexible
statistical power analysis program for the social, behavioral, and
biomedical sciences. Behav Res Methods. 2007;39:175–­91.
17. Faul F, Erdfelder E, Buchner A, Lang A-­G. Statistical power anal-
yses using G*Power 3.1: tests for correlation and regression analy-
ses. Behav Res Methods. 2009;41:1149–­60.
18. The jamovi project. jamovi. (Version 1.6) [Computer Software];
2020. [cited 2021 Jan 20]. Available from https://www.jamovi.org.
19. R Core Team. R: a Language and environment for statistical com-
puting. (Version 4.0) [Computer software]; 2020. [cited 2021Jan
20]. Available from https://cran.r-­ proje​
ct.org. (R packages re-
trieved from MRAN).
20. Li W, Lin J, Fu M. Electromyographic investigation of mastica-
tory muscles in unilateral cleft lip and palate patients with anterior
crossbite. Cleft Palate-­Craniofacial J. 1998;35:415–­8.
21. Sakamoto T, Ohtsuka K, Harazaki M, Isshiki Y. An electromyo-
gram study on mandibular movement in unilateral cleft lip and
palate patients before and after orthodontic treatment. Bull Tokyo
Dent Coll. 1999;40:195–­202.
22. Sipert CR, Sampaio AC, Trindade IE, Trindade ASJR. Bite force
evaluation in subjects with cleft lip and palate. J Appl Oral Sci.
2009;17:136–­9.
23. Montes ABM, de Oliveira TM, Gavião MBD, de Souza BT.
Occlusal, chewing, and tasting characteristics associated with oro-
facial dysfunctions in children with unilateral cleft lip and palate: a
case-­control study. Clin Oral Investig. 2018;22:941–­50.
24. Hideshima M. Masticatory movements in cleft lip and palate pa-
tients with unilateral buccal cross-­bite. The effect of different buc-
cal overlap on pattern of chewing. Nihon Hotetsu Shika Gakkai
Zasshi. 1989;33:1168–­82.
25. Miyawaki S, Takada K. Incisor crossbite and repaired unilateral
cleft lip and palate: changes in jaw movement and temporalis mus-
cle activity before and after edgewise treatment -­case report. Cleft
Palate Craniofac J. 1997;34:533–­7.
26. Piancino MG, Talpone F, Servo C, Bracco P. Valutazione del pat-
tern masticatorio di una paziente con esiti di labiopalatoschisi e
crossbite bilaterale. Ortodon Clin. 2005;1:53–­60.
27. Szyszka-­ Sommerfeld L, Woźniak K, Matthews-­ Brzozowska T,
Kawala B, Mikulewicz M, Machoy M. The electrical activity of
the masticatory muscles in children with cleft lip and palate. Int J
Paediatr Dent. 2018;28:257–­65.
28. Piancino MG, Farina D, Talpone F, Merlo A, Bracco P. Muscular
activation during reverse and non-­reverse chewing cycles in unilat-
eral posterior crossbite. Eur J Oral Sci. 2009;117:122–­8.
29. Piancino MG, Cordero-­Ricardo M, Cannavale R, Vallelonga T,
Garagiola U, Merlo A. Improvement of masticatory kinematic
parameters after correction of unilateral posterior crossbite: rea-
sons for functional retention. Angle Orthod. 2017;87:871–­7.
30. Yoshino K, Kawagishi S, Takatsuki Y, Amano N. Different roles of
the primary motor and ventral premotor cortex in jaw movements.
In: Nakamura Y, Sessle GJ, editors. Neurobiology of mastication.
From molecular to system approach. Cambridge, MA: Elsevier
BV; 1999. p. 515–­7.
31. Onozuka M, Fujita M, Watanabe K, Hirano Y, Niwa M,
Nishiyama K, et al. Mapping brain region activity during
chewing: a functional magnetic resonance imaging study. J
Dent Res. 2002;81:743–­6.
32. Takada T, Miyamoto T. A fronto-­parietal network for chewing of
gum: a study on human subjects with functional magnetic reso-
nance imaging. Neurosci Lett. 2004;360:137–­40.
33. Katoh M, Taira M, Katakura N, Nakamura Y. Cortically in-
duced effects on trigeminal motoneurons after transection of the
brainstem at the pontobulbar junction in the cat. Neurosci Lett.
1982;33:141–­6.
34. Enomoto S, Schwartz G, Lund JP. The effects of cortical ablation
on mastication in the rabbit. Neurosci Lett. 1987;82:162–­6.
35. Lund J. Patterning of rhythmical feeding behaviour by brainstem
neurons and sensory afferents. Acta Physiol Scand. 1999;167:A14.
36. Ishii H, Kang Y. Molecular basis underlying GABA(A) re-
sponses in rat mesencephalic trigeminal neurons. NeuroReport.
2002;13:2265–­9.
37. Johnsen SE, Trulsson M. Receptive field properties of human
periodontal afferents responding to loading of premolar and molar
teeth. J Neurophysiol. 2003;89:1478–­87.
38. Piancino MG, Falla D, Merlo A, Vallelonga T, de Biase C,
Dalessandri D, et al. Effects of therapy on masseter activity and
chewing kinematics in patients with unilateral posterior crossbite.
Arch Oral Biol. 2016;67:61–­7.
39. Pirttiniemi P, Kantomaa T, Lahtela P. Relationship between cra-
niofacial and condyle path asymmetry in unilateral cross-­bite pa-
tients. Eur J Orthod. 1990;12:408–­13.
40. Poikela A, Kantomaa T, Pirttiniemi P. Craniofacial growth after a
period of unilateral masticatory function in young rabbits. Eur J
Oral Sci. 1997;105:331–­7.
41. Sonnesen L, Bakke M, Solow B. Bite force in pre-­orthodontic chil-
dren with unilateral crossbite. Eur J Orthod. 2001;23:741–­9.
42. Thilander B, Bjerklin K. Posterior crossbite and temporoman-
dibular disorders (TMDs): need for orthodontic treatment? Eur J
Orthod. 2012;34:667–­73.
How to cite this article: Cassi D, Di Blasio A, Di
Benedetto L, De Biase C, Pedrazzi G, Piancino MG.
Evaluation of masticatory function in patients with
cleft lip and/or palate. Eur J Oral Sci.
2021;00:e12781. https://doi.org/10.1111/eos.12781