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-

ancy frequently results in dental crossbite occurring in the
Orofacial clefts represent a heterogeneous group of con- early dentition [4]. Crossbite is a complex, asymmetric, and
genital disorders involving the lips and the oral cavity. The worsening malocclusion that involves the teeth and all the
main categories are isolated cleft palate and cleft lip with or components of the masticatory system and their functions. It
without cleft palate (CL/P) [1]. Although these defects can results from dental and/or skeletal discrepancy between the
be surgically repaired in childhood, residual deformity due opposing arches and may lead to displacement or malposition
to scarring of oral and facial structures results in a range of of the mandible [5].
continuing functional and aesthetic problems [2]. Maxillary It has long been known that children with unilateral pos-
growth deficiency is usually observed in operated patients terior crossbite display modified chewing patterns during
born with CL/P, and it affects facial development and dental 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
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is associated with a significant increase in the frequency of (i) for complete clefts (unilateral and bilateral), a two-­stage
reverse-­sequence chewing pattern, which refers to the abnor- lip and palate repair procedure was performed that included
mal movement of the mandible during the closing phase of lip, nose, and soft palate closure at the age of 5–­6 months,
chewing. The definition of “reverse chewing cycle,” as the followed by hard palate closure and early gingival-­alveolar-­
inversion of the closing direction of the chewing cycle, was plastic (GAP) surgery at the age of 16–­18 months; (ii) for
provided by Lewin [6] and then adopted by other authors [8–­ isolated CL or CP, a single-­stage lip or palate repair at the
10]. The direction of closure is represented by the vector of age of 5–­6 months was performed (i.e., the lip or hard and
the closing pattern in the last stage of the chewing cycle: it soft palates were closed in a single operation). Based on the
represents a clinical indicator of chewing pattern characteris- occlusal diagnosis, which was performed by two trained op-
tics and balanced mastication and it does not change during erators on the dental casts, patients were classified according
growth [6,11]. In cases of unilateral posterior crossbite, to the presence and type of crossbite as follows: (i) anterior,
during mastication on the crossbite side, the closing direc- (ii) right or left unilateral posterior, (iii) unilateral posterior
tion will be reversed with respect to the condition of normal and anterior, (iv) bilateral posterior, or (v) bilateral posterior
occlusion. According to the literature, the chewing pattern and anterior (Figure 1). For the crossbite classification, the
during mastication on the healthy side maintains the same operators adopted the following definitions: anterior cross-
features as physiological mastication regarding the closing bite is a condition where one or more primary or permanent
direction and the morphological and positional features (i.e., maxillary teeth in the canine and/or incisor region occlude
height, width, and spatial position that display no differences lingual to their antagonist; posterior crossbite corresponds to
from normal occlusion) [11]. The reverse sequence chewing the condition of one or more primary or permanent maxillary
cycle is set and maintained by the automatisms of the central teeth in the molar and/or premolar regions occluding in an
nervous system's motor control, which is based on periph- abnormal buccolingual relation with their antagonists (i.e.,
eral input arriving from the periodontal mechanoreceptors the buccal cusps of the maxillary posterior teeth contact or
[12,13]. occlude lingually to the buccal cusps of the corresponding
Although the physiology of mastication has been thor- mandibular teeth).
oughly investigated, only a few studies have described the In addition to the CL/P group, the following two groups
chewing cycle in patients with congenital orofacial condi- were recruited: an age and crossbite-­matched group without
tions like CL/P. Understanding the influence of dentofacial CL/P (18 children: 11 boys, seven girls; mean age [SD] = 7.2
morphology on masticatory function might have a clinical [1.5] yr; age range: 5.1–­10 yr) and a control group with-
relevance in the management of cleft-­affected patients who out CL/P and crossbite (18 children: nine boys, nine girls;
need prolonged, multidisciplinary care. The knowledge of al- mean age [SD] = 9.8 [1.9] yr; age range 7.4–­16.8 yr). They
teration of the stomatognathic system may improve the com- were selected among patients referred to the Department of
prehensive treatment of this complex condition. Orthodontics, University of Turin (Italy).
The aim of the present study was therefore to evaluate the Before participating in the study, informed consent was
masticatory function in children with CL/P through the inves- obtained from the parents and the study was approved by
tigation of the degree of reverse-­sequencing chewing cycles. the Institutional Review Board of the University Hospital
Specifically, we compared the occurrence of reverse chew- “Health and Science Complex Turin -­Italy” no. CS/246, in
ing patterns of patients with CL/P with an age and crossbite-­ accordance with the Code of Ethics of the World Medical
matched group without CL/P, and a control group without Association (Declaration of Helsinki) for experiments in-
CL/P and crossbite. The null hypothesis was that there was volving humans.
no significant difference between cleft-­affected subjects and Inclusion criteria for the cleft group were the presence of
healthy controls with the same types of crossbite. 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/
MAT E R IA L AN D ME T HOD S or posterior, unilateral, and bilateral). The inclusion criteria
for the control group with normal occlusion were absence of
Study participants congenital malformation and absence of crossbite. Exclusion
criteria for all participants were: signs or symptoms of myo-
Fifty-­four children were included in this observational study. facial disorders causing discomfort or pain of the masticatory
The study group consisted of 18 consecutive patients with musculature; previous orthodontic therapy; presence of mobile
CL/P (11 boys, seven girls; mean age [SD] = 7.4 [1.4] yr; age exfoliating primary teeth; presence of caries or pain; presence
range: 4.1–­12.9 yr), including unilateral CLP (UCLP), bilat- of any prosthesis; presence of diabetes and/or celiac disease, as
eral CLP (BCLP), CP and CL. All patients were operated by the boluses used in the present study contained sugars that are
the same surgical team according to the following protocol: contraindicated in subjects with these medical conditions.
EXPLORING CHEWING PATTERN IN CHILDREN WITH CL/P   
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F I G U R E 1 Crossbite classification in (A) (B)

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) (D)

(E)

Recording of chewing cycles kinesiograph was interfaced with a computer for data storage
and analysis. The kinematic signals were analysed using a
Recordings of the chewing cycles were performed for all sub- custom-­made software (Department of Orthognatodontics,
jects by the same operator, following a standardized protocol, in University of Turin, Italy). The first cycle, during which the
a silent and comfortable environment [14]. Patients were com- bolus was transferred from the tongue to the dental arches,
fortably seated in a chair and they were asked to fix the eyes was excluded from the analysis. The chewing cycles were
on a target placed 90 cm away directly in front of their seating divided into non-­reverse and reverse, based on the vector di-
position, and to avoid movements of the head. Each record- rection of closure.
ing began in maximal intercuspation. Patients were asked to Repeatability of measurements was assessed on 10 partic-
find the starting position by lightly tapping their teeth together ipants, through two series of soft bolus chewing recordings
and clenching. They were then asked to hold this position with performed by the same operator. The interval between the
a test bolus on the tongue before recording. Participants were two recordings was 15 min.
subsequently instructed to chew a soft bolus and then a hard Differences in both right and left chewing sequences were
bolus: each bolus was chewed first on the right side, then on evaluated among the three groups.
the left side and, eventually, in a free mode. The duration of For CL/P patients and controls with crossbite, differences
each test was 10 s and the test was repeated three times. The between pathologic and normal side of mastication were fur-
side of mastication was visually checked by an operator and, ther evaluated as follows: (i) in patients with unilateral pos-
at the end of the recording, participants were asked to spit the terior crossbite with or without anterior crossbite, right and
bolus in a glass to assess whether it had actually been chewed. left chewing sequences were pooled according to “affected”
The soft and hard boluses used were a chewing gum and a wine and “not affected” side; (ii) in patients with bilateral posterior
gum, respectively. Both gums had the same size (20 mm in crossbite with or without anterior crossbite, right and left se-
length, 1.2 mm in height, and 0.5 mm in width) but different quences were pooled as “affected” side; (iii) in patients with
weights (2 g for the soft bolus and 3 g for the hard bolus). The anterior crossbite or without crossbite, right and left chewing
wine gum was chosen to provide a rubber-­like texture without cycles were pooled as “not affected” side.
sticking to the teeth.
Mandibular movements were measured with a kinesio-
graph (K7; Myotronics), which measures jaw movements Sample size calculation and statistical analysis
with an accuracy of 0.1 mm. Multiple Hall effect sensors in
a light-­weight array (113 g) tracked the motion of a mag- Normal distribution of the data was tested using
net attached to the midpoint of the lower incisors [15]. The 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.
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TABLE 2 Repeated measurements of reverse chewing cycles

1st meas. 2nd meas. Bland-­Altman analysis (95% CI)

Chewing Mean Mean Median difference

Side (%) (median) (median) (95% CI) Bias LoA-­lower LoA-­upper CCC
RIGHT 2.3 (1.0) 2.0 (0.0) 0.5 (−2.0; 3.0) 0.3 (−1.0; 1.60) −3.28 (−5.6; −0.96) 3.88 (1.57; 6.20) 0.87 (0.61; 0.96)
LEFT 22.8 (8.50) 26.4 (13.0) −2.43 (−16.0; 3.0) −3.6 (−10.4; 3.29) −22.5 (−34.7; −10.3) 15.3 (3.1; 27.5) 0.93 (0.76; 0.98)
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 The statistical analyses were performed with the statistical
patients with unilateral posterior crossbite [9] suggested software jamovi version 1.6.13 [18] and r version 4.0.3 [19].
that the effect size—­ defined as the difference between The results were considered statistically significant for a p-­
two group means divided by their pooled standard devia- value <5% (p < 0.05).
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 RESULTS
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 Demographic details and masticatory function data of pa-
an f estimate equal to f = 0.53. We then used G*Power tients with CL/P are reported in Table 1. The distribution
v.3.1.9.2 [16,17] to provide a rough estimation of the re- of CL/P was: six patients with UCLP, seven patients with
quired sample size. Assuming a comparison of three groups BCLP, one patient with CP, and four patients with CL. When
(patients with CL/P, controls with crossbite, controls with considering occlusion, seven (39%) patients showed unilat-
normal occlusion) at α = 0.05 (5%), power = 0.8 (80%), eral posterior and anterior crossbite (two on the right and five
and f = 0.53, the required sample size was estimated to be on the left side), two (11%) had unilateral posterior crossbite
39 units (13 per group). Assuming a stratification for bolus (one on either side); thus, three (17%) patients had crossbite
hardness (soft, hard) and then comparing six groups with on the right side and six (33%) on the left side. Two (11%)
the same alfa, power, and effect size, the required sample patients showed bilateral posterior and anterior crossbite, one
size was estimated to be 54 units (18 per group). (5%) had bilateral posterior crossbite, three (17%) had an-
To assess the repeatability of the recording procedure, the terior crossbite, and three (17%) had no crossbite. Occlusal
differences in percentages of reverse chewing cycles between discrepancies were related to the type of cleft as follows: uni-
the first and second set of measurements were evaluated lateral posterior and anterior crossbite in five patients with
using Bland-­Altman analysis and plot, Lin's concordance co- UCLP and in two patients with BCLP; the two cases with bi-
efficient (CCC), intraclass correlation coefficient (ICC), and lateral posterior and anterior crossbite had BCLP; unilateral
the Wilcoxon's test for paired comparisons. It is commonly posterior crossbite was observed in one patient with UCLP
accepted that pairs of measurements have an excellent repro- and in one patient with CL; the single case with bilateral pos-
ducibility if Lin's CCC and ICC are higher than 0.9, good if terior crossbite had BCLP; anterior crossbite was observed
higher than 0.8, and acceptable if higher than 0.7. in two patients with BCLP and one patient with CL; one pa-
Given that continuous variables were not normally dis- tient with CP and two patients with CL had no crossbite. The
tributed, Kruskal–­Wallis test with Dwass-­Steel-­Critchlow-­ distribution of types of crossbite was the same in the control
Fligner pairwise comparisons post hoc test were used to group with crossbite as in the CL/P group.
compare frequencies of reverse chewing cycles between the The results of the assessment of recording protocol re-
control and patient groups. The relevant variable was the pro- peatability are shown in Table 2 and Figure 2. The differ-
portion of reverse chewing cycles, which was computed by ences in the prevalence of reverse chewing cycles between
dividing the number of reverse chewing cycles by the number the first and second evaluations were not statistically signif-
of recorded chewing cycles in each set of recordings. Given icant (p > 0.05). The medians of paired difference between
that the denominator value was similar for each participant, the first and second measurements were 0.5 (−2.0; 3.0) and
the uncertainty of measurements was roughly the same; thus, −2.43 (−16.0; 3.0) for the right and left side, respectively.
the proportion of reverse chewing cycles was considered a Lin's CCC was 0.87 (0.61; 0.96) for the right side and 0.93
continuous variable. (0.76; 0.98) for the left side.
Descriptive plots were used to represent the distribution A statistically significant difference in the proportion
of reverse chewing cycles in each group. of reverse chewing cycles between patients with CL/P and
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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 was barely insignificant for hard bolus (11 [0; 39], p = 0.057;
the side with the higher prevalence of crossbite (soft bolus Table 3). No statistically significant difference between pa-
–­median difference [95% CI] = 22.5 [3; 50], p = 0.013; tients with CL/P and non-­CL/P controls with crossbite was
hard bolus –­median difference [95% CI] = 18.5 [1; 53], detected, whether on the right or left side of mastication
p = 0.015); on the right side, the differences were less pro- (Table 3); this result was confirmed when comparing the per-
nounced and the p-­value was higher, being just statistically centages of reverse-­sequencing chewing patterns between the
significant for the hard bolus (15 [0.5; 33], p = 0.046) and crossbite and non-­crossbite sides (Table 4).
barely insignificant for the soft bolus (14.5 [−2; 30.8], Eventually, the difference observed between the affected
p = 0.059; Table 3). Similarly, a statistically significant and not affected side within each group was significant for non-­-
higher proportion of reverse chewing cycles was found in CL/P controls (soft bolus –­median difference [95% CI] = 64.0
non-­CL/P controls with dental crossbite than in healthy sub- [24.0; 86.0], p = 0.002; hard bolus –­median difference [95%
jects with normal occlusion on the left side of mastication CI] = 69 [51.5; 85], p < 0.001) and significant for hard bolus
(soft bolus –­median difference [95% CI] = 31 [1.5; 76.5], in patients with CL/P (soft bolus –­median difference [95%
p = 0.012; hard bolus –­median difference [95% CI] = 26.5 CI] = 18.0 [−12.0; 48.0], p = 0.844; hard bolus –­median dif-
[0; 67], p = 0.029). On the right side, however, there was no ference [95% CI] = 58.5 [35.5; 88.0], p < 0.001; Table 4).
statistically significant difference between the two groups for Masticatory function and chewing pattern analysis of
soft bolus (6 [−6.5; 25.3], p = 0.495), while the difference 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- healthy side, the chewing cycle displays normal physiologi-
tients not showing reverse cycles, three had no crossbite and cal closing direction (left).
three had crossbite only in the anterior inter-­canine region.
One patient with UCLP, unilateral posterior and anterior
crossbite, did not present reverse cycles. DISCUSSION
An example of pathological chewing pattern in the frontal
plane is shown in Figure 3. The reverse sequence chewing In the present study, the analysis of masticatory function was
cycle occurs only on the crossbite side (right) whilst, on the performed in a group of growing subjects affected by CL/P
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|    CASSI et al.

with different types of dental crossbites; particularly, we in- average), depending on the severity of the malocclusion and
vestigated the prevalence of reverse-­sequencing chewing cy- the bolus type [9,11,28,29].
cles during chewing of soft and hard boluses. According to Functional asymmetry and anomalous chewing pattern
our preliminary results, cleft patients with posterior crossbite occurred in all subjects (except one) with posterior crossbite.
(independently from the type of congenital malformation) In contrast, patients with anterior crossbite displayed a chew-
exhibit an anomalous masticatory pattern with an increased ing pattern with no increased frequency of reverse sequence
frequency of reverse chewing cycles on the crossbite side. chewing cycles. These findings are in accordance with those
Data were compared with the chewing patterns recorded in observed in non-­cleft patients, in whom functional asymme-
an age and crossbite-­matched group without CL/P and in try is typical of unilateral posterior crossbite, but is not pres-
a control group without CL/P and crossbite; we found that ent in anterior crossbite [14]. Thus, the effect of crossbite on
mandibular kinetics in children with CL/P during chewing chewing patterns during masticatory function differs accord-
are like those reported in non-­CL/P patients with the same ing to the dental region involved. The impact of malocclusion
type of malocclusion, but different from controls without on masticatory function might depend on the functional role
malocclusion. of the teeth involved, as the anterior teeth exert a major influ-
Only a few studies have been performed to evaluate mas- ence on postural control of the mandible rather than on the
ticatory function in patients born with a cleft condition, and chewing cycle in the frontal plane.
these have principally focused on masticatory muscle ac- The similarity of masticatory function between children with
tivity [20,21] or bite force [22]. Recently, MONTES et al. and without CL/P but with the same crossbite malocclusion is
[23] investigated the capacity of 8-­to 10-­yr-­old children with not surprising, since it is related to the neuromuscular motor
UCLP to comminute a test food and showed that these pa- control of mastication. Masticatory movements are character-
tients have poor masticatory performance. The effect of pos- ized by rhythmicity and a diversity of patterns of jaw posture.
terior crossbite on the chewing pattern has previously been YOSHINO et al. [30] suggested that the primary motor cortex
described in three patients with UCLP by HIDESHIMA [24], may be involved in the initiation and control of jaw movements
who found that reversed patterns were seen more frequently and that the ventral pre-­motor cortex may be involved in motor
during chewing on the crossbite side. A case was published planning, therefore playing a role as a higher order motor area
by MIYAWAKI and TAKADA [25] reporting the changes in related to the initiation and control of jaw movements [31,32].
jaw movement and temporalis muscle activity of a patient The cortex sends signals to the brainstem, which elaborates
with UCLP before and after edgewise treatment: the reversed the patterns of mastication in response to the inputs from the
crossing jaw-­ movement pattern before treatment changed masticatory cortex and from the periphery [33,34]. Sensory
to a normal grinding after treatment. PIANCINO et al. [26] receptors, such as muscle spindles, periodontal, and even in-
described reverse-­sequencing chewing patterns in a patient tradental pressure receptors, exert strong influences on the
with BCLP and bilateral posterior crossbite. A recent study chewing pattern being generated by the central pattern genera-
investigated masticatory muscle activity in 25 children with tor, eventually modifying both the frequency of motor neuron
UCLP compared to a sample with no cleft abnormalities: the bursts and their intensity [35]. Inputs from mechanoreceptors
authors found that the presence of unilateral posterior cross- are critical, not only for various trigeminal reflexes (such as
bite affects the temporal muscle activity in cleft patients, who the jaw-­opening reflex or the periodontal-­masseter reflex), but
showed significantly increased electromyographical poten- also for masticatory control [36,37]. These observations could
tials during rest position [27]. explain the results of this study: conceivably, the birth defect is
To the best of our knowledge, no studies have been pub- not responsible for masticatory disorders per se, but it affects
lished so far reporting on mandibular kinetic movements the morphological characteristics of the orofacial structures,
during chewing in a series of patients affected by cleft. generating the malocclusion to which, in turn, the altered man-
Our study has shown a higher prevalence of reverse-­ dibular kinetics are linked. Investigations of the electromyo-
sequencing chewing cycles during chewing on the crossbite graphic activity during reverse and non-­reverse chewing cycles
side than in the non-­crossbite side. Interestingly, the number in unilateral posterior crossbite showed that the asymmetry of
of reverse sequence cycles was higher during mastication of the kinetic pattern results in unbalanced neuromuscular acti-
hard boluses compared to soft boluses, as it is represented in vation [28,38]. Given that the unilateral posterior crossbite
Table 4. It is remarkable that the reverse sequence chewing usually occurs at a very early age, functional asymmetry may
cycle occurred predominantly on the crossbite side whilst, disturb the craniofacial development, leading to an asymmetric
on the healthy side, the chewing cycle displayed physiologi- growth of anatomical structures at the end of growth that can
cal closing direction. Our results are consistent with previous no longer be corrected by orthodontic therapy alone [8,39–­42].
findings on non-­cleft patients, showing that the percentage According to the literature, the orthodontic correction
of reverse sequence chewing cycles during mastication on of unilateral posterior crossbite through appropriate appli-
the crossbite side is extremely high (around 60%–­70% on ances leads a significant reduction in the reverse sequencing
EXPLORING CHEWING PATTERN IN CHILDREN WITH CL/P   
| 9 of 10

of masticatory cycles on the affected side [9,29]. Thus, the (lead), project administration (lead), writing -­original draft
resolution of the dental malocclusion might be advisable to preparation (supporting), review and editing (equal).
restore the masticatory function and improve the muscular
coordination. ORCID
The main limitation of this study is the heterogeneity of Diana Cassi https://orcid.org/0000-0001-9076-1337
the cleft group in terms of the type of congenital malforma- Alberto Di Blasio https://orcid.org/0000-0002-0219-6946
tion and malocclusion. Orofacial clefts show a broad spec- Laura Di Benedetto https://orcid.
trum of clinical presentations due to a complex aetiology org/0000-0002-1712-6780
with both genetic and environmental contributions; the level Corrado De Biase https://orcid.
of expression and severity ranges from minor defects to im- org/0000-0002-7129-1971
portant craniofacial abnormalities and they include CL alone, Giuseppe Pedrazzi https://orcid.
CL plus CP, and CP alone. This represents a particular chal- org/0000-0002-5971-2040
lenge and results in variability of the occlusal traits. In the Maria Grazia Piancino https://orcid.
present study, we chose to include consecutive patients with org/0000-0002-0220-5283
phenotypic diversity and to detail their chewing patterns,
considering that masticatory function is poorly investigated R E F E R E NC E S
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AUTHOR CONTRIBUTIONS
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Diana Cassi: writing -­original draft preparation (lead), 11. Piancino MG, Kyrkanides S. Understanding masticatory function
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Blasio: resources (lead), supervision (lead), review and edit- onlin​elibr​ary.wiley.com/doi/book/10.1002/97811​18971901
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Biase: investigation (lead), data curation (lead), review and 2006;21:167–­74.
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