Article_7_3_4-1

www.stomaeduj.com   ORTHODONTICS
                    EVALUATION OF UPPER ARCH CHANGES IN PATIENTS
Original Articles
                    WITH UNILATERAL CLEFT LIP AND PALATE AFTER
                    MAXILLARY EXPANSION USING DIGITAL DENTAL CASTS
                    Fabrizia d’Apuzzo1a , Ludovica Nucci1b , Abdolreza Jamilian2c , Rosario Rullo1d , Vincenzo Grassia1e , Letizia Perillo1f*
                    1
                        Multidisciplinary Department of Medical-Surgical and Dental Specialties, University of Campania Luigi Vanvitelli, Naples, Italy
                    2
                        Department of Orthodontics, Islamic Azad University, Tehran, Iran

                    a
                      DDS, MS, PhD, Research fellow; e-mail: fabriziadapuzzo@gmail.com; ORCIDiD: https://orcid.org/0000-0003-0291-9339
                    b
                      DDS, PhD student; e-mail: ludortho@gmail.com; ORCIDiD: https://orcid.org/0000-0002-7174-7596
                    c
                      DDS, MSc, OMFS, Full professor; e-mail: info@jamilian.net; ORCIDiD: https://orcid.org/0000-0002-8841-0447
                    d
                      MD, MS, Associate professor; e-mail: rosario.rullo@unicampania.it; ORCIDiD: https://orcid.org/0000-0002-5418-8171
                    e
                      DDS, MS, PhD, Lecturer; e-mail: grassiavincenzo@libero.it; ORCIDiD: https://orcid.org/0000-0002-6671-2380
                    f
                      MD, MS, PhD, Professor, Head, Dean; e-mail: letizia.perillo@unicampania.it; ORCIDiD: https://orcid.org/0000-0001-6175-7363


                    ABSTRACT                                                                               https://doi.org/10.25241/stomaeduj.2020.7(3).art.4

                    Objective To compare the upper arch dimensions of young patients with unilateral cleft lip and palate
                    (UCLP) before and after treatment with bonded maxillary expander and hybrid activation protocol using
                    digital dental casts.
                    Methods Sixteen subjects with UCLP, aged between 7 and 14 years (mean age 10.9 ± 2.7 years) consecutively
                    treated with bonded maxillary expander and hybrid activation were included. The dental casts before and
                    after treatment were digitalized using a 3Shape scanner. Intercanine, interpremolar and intermolar widths
                    (at cusp and gingival levels) and arch perimeters were measured. The significance level for statistical analyses
                    was set as p < 0.05.
                    Results The total treatment time using the hybrid expansion protocol lasted 12 ± 1.9 months while the active
                    expansion time lasted 4 ± 0.2 months. Patients with UCLP showed significant differences in all transverse
                    upper arch dimensions both at cusp and gingival level.
                    Conclusion The use of a bonded maxillary expander with a hybrid activation protocol during growth may be
                    efficient to improve all transverse upper arch widths in patients affected by UCLP.
                    KEYWORDS
                    Cleft Lip and Palate; Maxillary Expansion; Bonded Expander; Hybrid Expansion Protocol; Digital Dental Casts.



                    1. INTRODUCTION                                                                        morbidity and mortality throughout life compared
                                                                                                           to noncleft subjects [5]. Oral rehabilitations usually
                    Cleft Lip and Palate (CLP) is one of the most common                                   require complex and challenging interdisciplinary
                    types of congenital craniofacial birth anomalies due                                   treatments since the first post-birth weeks do not
                    to environmental, genetic and epigenetic risk factors                                  allow the most adequate early recovery of vital
                    interacting among them [1-3].                                                          functions [5,6].
                    They can occur between the fourth and twelfth week                                     Craniofacial growth harmony in patients with
                    of gestation due to the failure of fusion between                                      unilateral or bilateral CLP is affected in all 3
                    the medial nasal and maxillary processes in the                                        dimensions (3D). Previous studies showed that it
                    primary palate or the palatal units in the secondary                                   can be only partially improved after surgery during
                    palate [4]. Affected patients need interdisciplinary                                   childhood [7-9]. Lip surgery is usually performed at
                    care since birth until adulthood and evince higher                                     6 months of age using the Delaire technique, soft


                                    OPEN ACCESS This is an Open Access article under the CC BY-NC 4.0 license.
                                    Peer-Reviewed Article
                         Citation: d’Apuzzo F, Nucci L, Jamilian A, Rullo R, Grassia V, Perillo L., Evaluation of upper arch changes in patients with unilateral cleft lip and palate
                         after maxillary expansion using digital dental casts. Stoma Edu J. 2020;7(3):184-190.
                         Received: May 29, 2020; Revised: June 05, 2020; Accepted: June 07, 2020; Published: June 09, 2020
                         *Corresponding author: Prof. Letizia Perillo; Multidisciplinary Department of Medical-Surgical and Dental Specialties, University of Campania Luigi
                         Vanvitelli, via L. De Crecchio 6, 80138 Naples, Italy
                         Tel./Fax: +39 3334027273; e-mail: letizia.perillo@unicampania.it
                         Copyright: © 2020 the Editorial Council for the Stomatology Edu Journal.




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Upper arch changes in patients with CLP after maxillary expansion
                                                                                                                                 www.stomaeduj.com




                                                                                                                                 Original Articles
 Table 1. Age, treatment time and sex ratio of the UCLP patients.

 Variable                                                                           Mean                         SD
 Initial age (years)                                                                 10,9                        2,7
 Total Treatment time (months)                                                       12,0                        1,9
 Active Treatment time (months)                                                      4,0                         0,2
 Sex                     Female                                                       10

                         Male                                                         6


 Table 2. Random and systematic errors of the measurements performed on the digital dental models.

                                                          T0                                                T1
                                                                    Systematic                                   Systematic
 Measurements                            D Dahlberg                                           D Dahlberg
                                                                    error P level                                error P level
 3-3 cusp                                    1.30                        NS                          1.55              NS

 3-3 gingival                                1.24                        NS                          0.78              NS

 4-4 cusp                                    0,89                        NS                          1.56              NS
 4-4 gingival                                1.50                        NS                          1.34              NS
 5-5 cusp                                    1.45                        NS                          2,01              NS
 5-5 gingival                                1.67                        NS                          1.34              NS
 6-6 cusp                                    1.07                        NS                          1.67              NS
 6-6 gingival                                0.98                        NS                          0.87              NS
 AP                                          4.06                        NS                          1.81              NS

*p < 0.05, **p < 0.005

palate surgery at 12 months and hard palate surgery                     formation, as reported in normal patients [23].
at 18 months, whereas secondary alveolar bone                           However, to the best of our knowledge, there are
graft is usually planned when the patient is around                     no studies treating subjects affected by CLP using a
9 to 12 years old [10-12]. When the surgical protocol                   bonded maxillary expander with a hybrid activation
for the hard palate closure is delayed, the occlusion                   [24-27]. The bonded expander seems to have some
is even more affected [12].                                             advantages such as better control of vertical growth,
During the early mixed dentition, the maxillary arch                    reduced interocclusal interferences and less dental
linear dimensions significantly differ between the                      tipping due to acrylic splints covering both occlusal,
unilateral cleft lip and palate (UCLP) group and the                    buccal and palatal surfaces of teeth [28-30]. Many
noncleft group, [13] while the intercanine width                        papers conducted the palatal analysis of patients
seems to be more reduced in bilateral complete                          with CLP as compared to controls using novel three-
clefts due to scars resulting from palatal surgery [14].                dimensional (3D) technologies [31-33]. Nowadays,
It has been also demonstrated that children with                        the use of digital models is considered useful for
UCLP usually suffer from a nasomaxillary complex                        treatment planning and outcome evaluations and
deficiency which continues worsening until the end                      the reliability of measurements from 3D dental casts
of their growth and have maxillary dental arches                        as compared to those obtained directly from plaster
constricted in width and length [15].                                   models is evidence-based [34]. Thus, the purpose of
Thus, it is suggested to ideally start to perform an                    this study was to evaluate upper arch dimensions
early dentofacial orthopedic treatment protocol                         on digital dental casts in young patients with UCLP
during the mixed dentition [16]. In the light of these                  before and after treatment using a bonded maxillary
findings, an interceptive treatment with a palatal                      expander with a hybrid activation.
expander is usually proposed as a first approach [17-
19]. The palatal expander allows to correct transverse                  2. METHODOLOGY
skeletal maxillary constriction and dental posterior
crossbite [20]. There are several different maxillary                   This retrospective observational study was based on
expansion modalities based on the appliance design                      the analysis of data collected from patients with UCLP
[21] or the type of activation and no differences were                  who had completed an orthodontic treatment with
found between slow or rapid expansion protocols in                      maxillary expansion at the Orthodontic Program of
children with bilateral CLP [17,22]. Thus, the positive                 the University of Campania Luigi Vanvitelli, Naples,
effects on the occlusion are undeniable in patients                     Italy. The research was carried out in accordance
with CLP modifying the upper arch diameters                             with the Declaration of Helsinki and approved by
and stimulating the transversal skeletal growth,                        the ethics committee of the University of Campania
even without leading to the same amount of bone                         Luigi Vanvitelli, Naples, Italy (Prot. N°147). An



Stoma Edu J. 2020;7(3): 184-190                                                             pISSN 2360-2406; eISSN 2502-0285     185
                    d’Apuzzo F, et al.
www.stomaeduj.com

Original Articles



                     Figure 1. Intraoral photograph of bonded maxillary expander in a    Figure 2. Assessment on the digital model of arch widths: at the cusp
                    patient with UCLP.                                                  (yellow lines) and gingival (orange lines) level.


                    informed consent was obtained from the parents of                   arch were considered: transverse intercanine (3-3),
                    each patient. Here are the inclusion criteria: patients             interpremolar or interdeciduous molar (4-4 and 5-5)
                    affected by UCLP, age ranging between 7 and 14                      and intermolar (6-6) distances at both cusp tips and
                    years, constricted upper arch, cervical vertebral                   gingival levels, and arch perimeter (AP) calculated
                    maturation between CS1 and CS4 and treatment                        as circumference of the dental arch, measured from
                    protocol including a bonded maxillary expander with                 the distal surface of the maxillary permanent first
                    hybrid activation [24,26]. Subjects with other cranio-              molars on the right side to the distal surface of the
                    facial syndromes, previous orthodontic treatment or                 contralateral ones.
                    incomplete documentation were excluded.
                    All patients had been previously treated at the                     2.1. Statistical analysis
                    Division of Maxillofacial Surgery at the University of              The reliability of the measurements was determined
                    Campania Luigi Vanvitelli, Naples, Italy, by the same               by randomly selecting five dental casts before and
                    surgeon, using the same protocol and method as                      after the expansion treatment. They were measured
                    follows: lip surgery at 6 months, soft palate surgery at            twice after a two-week interval. Dahlberg’s formula
                    12 months and hard palate surgery at 18 months. No                  was used to quantify the measurement error. An
                    infant orthopaedics was carried out prior to surgery.               Intraclass Correlation Coefficient was also calculated
                    The initial (T0) and after expansion (T1) plaster                   to assess test/retest reliability. An unpaired t-test and
                    models of each patient were collected to be digita-                 a Wilcoxon signed-rank test were utilized to evaluate
                    lized and then analyzed [35].                                       any significant difference before and after treatment.
                    The treatment was performed with a bonded palatal                   The statistical significance was set at p <0.05.
                    expander (Fig.1) [16] applied on the upper lateral                  All statistical analyses were performed with SPSS
                    teeth. The activation started after the appliance                   software (Statistical Package for the Social Sciences,
                    onset and ended when overcorrection was achieved.                   SPSS, Version 12, Chicago, IL, USA).
                    The protocol used had two phases: the initial phase
                    started at the chair side and included three steps                  3. RESULTS
                    with four, two and one turn, respectively; the second
                    one followed at home with one turn every 3 days [24,                The total sample included 16 patients, 10 females
                    26]. During the active expansion phase depending                    and 6 males. The initial mean age was 10.9 ± 2.7 years.
                    on the degree of maxillary constriction, the patients               The total treatment phase with the bonded expander
                    were monitored every 2 weeks.                                       (T0-T1) lasted 12.0 ± 1.9 months while the active
                    After the active expansion phase, the screw of the                  expansion phase was 4.0 ± 0.2 months (Table 1).
                    appliance was closed with acrylic composite and                     The statistical analysis confirmed that there were
                    the expander was used as a retainer for about 8                     no systematic measurement errors before and after
                    months. The expander was removed after about                        treatment, as shown in (Table 2).
                    1 year and a second plaster model was realized.                     Comparing measurements at T0 and T1 showed
                    Both dental casts were scanned using the 3shape                     statistically significant differences in all transverse
                    TRIOS®, with a reported manufacturing accuracy                      arch widths while arch perimeter values revealed no
                    of less than 20 microns (www.3shape.com), by the                    differences (Table 3).
                    same trained operators. The digital models were                     As to the difference arch diameters at both cusp tips
                    exported in STL surface mesh then imported in                       and gingival levels (Table 4), data showed statistically
                    Viewbox 4 (dHal Software, Kifissia, Greece). A total                significant differences only for the diameter 3-3
                    of nine linear measurements of the upper dental                     measured at cusp level.



  186               Stoma Edu J. 2020;7(3): 184-190                                                         pISSN 2360-2406; eISSN 2502-0285
Upper arch changes in patients with CLP after maxillary expansion
                                                                                                                                     www.stomaeduj.com




                                                                                                                                     Original Articles
 Table 3. Measurements before (T0) and after maxillary expansion (T1).

                                        T0 mean value                  T1 mean value
           Variable                                                                        STD Error          t            S
                                            (mm)                           (mm)
 3-3 cusp                                     26,72                        31,36             1,59           0,01*       0,01*
 3-3 gingival                                 21,72                        24,81             1,38           0,04*       0,04*
 4-4 cusp                                     34,18                        40,05             1,73         0,004**       0,006*
 4-4 gingival                                 23,08                        28,43             1,51         0,003**       0,007*
 5-5 cusp                                     40,68                        46,40             1,92          0,009*       0,01*
 5-5 gingival                                 26,94                        33,16             1,82         0,004**      0,005**
 6-6 cusp                                     49,90                        55,79             1,65         0,003**      0,002*
 6-6 gingival                                 32,76                        38,65             1,55         0,002**      0,003**
 AP                                           88,76                        88,41             3,18           0,91         0,61
*p < 0.05, **p < 0.005

 Table 4. Mean differences between measurements at the cusp and            slow and rapid activation through Quad-helix and
gingival level of arch widths, before and after maxillary expansion.       Hyrax expanders, respectively, in children with
                                                                           bilateral CLP, showed differences only with respect
                    Mean
  Variable       difference        STD Error          t           S        to the treatment time. Specifically, the expansion
                 T1-T0 (mm)                                                active phase ranged from 4 to 21 months in the
     3-3             - 1,54           0,41        0,002**      0,004**     slow protocol and from 7 to 14 days in the rapid
                                                                           one [17,22]. Façanha et al. [20] assessing the effects
     4-4             - 0,52           0,48          0,29        0,53
                                                                           achieved with the Haas and Hyrax expanders using
     5-5              0,50            0,59          0,41        0,77
                                                                           a rapid protocol in patients with UCLP, highlighted
     6-6              0,00            0,39          0,98        0,95       similar increases of the transverse dimensions. Ayub
*p < 0.05, **p < 0.005
                                                                           et al. [39] performing the expansion with a rapid
                                                                           protocol, found significant increases of all maxillary
4. DISCUSSION                                                              measurements including arch perimeter probably
                                                                           due to the sample selection based on patients with
Several previous studies showed significant different                      a posterior crossbite. Conversely, in our study, this
maxillary morphologies in patients with CLP in mixed                       parameter did not change significantly. However, a
or permanent dentition when compared to matched                            direct comparison of our findings with other studies
controls before any orthopedic/orthodontic                                 was only partially possible due to the differences
treatment [31-33,36]. In particular, the upper inter-                      in cleft features and size, patients’ age, dentition
canine widths significantly decreased both at cusp                         period, expansion appliance and protocol as well
and gingival levels [13-15,37]. Therefore, there is                        as the data analyses performed. Nevertheless, our
a complete agreement on the need to expand                                 treatment outcomes may add a missing piece in the
the maxillary arch in patients affected by CLP. The                        puzzle about maxillary arch changes after expansion
reasons to use an expander in the orthodontic treat-                       treatment, underlying the possibility to achieve
ment plan are manifold. Iwasaki et al. [38] found that                     similar effects in a reduced time with a different type
the nasal airway flow significantly improved after                         of expander and activation [16,24-26]. Our results
rapid maxillary expansion in patients with UCLP                            confirmed the need for orthodontic treatment
increasing both the quantity of airflow and nasal                          during the development of the permanent dental
cross-sectional area on the cleft side. Also, speech                       occlusion and showed increases in all transverse
and deglutition enhance after expansion due to                             upper arch dimensions. Moreover, the bonded
wider space for the tongue, [6,33] but, above all, the                     expander seemed to exert a more homogeneous
occlusal relationships result improved by widening                         lateral pressure on the two halves of the maxilla
the palatal dimensions [18].                                               avoiding any possible interference of the lower
Many investigations focused on dental and alveolar                         arch due to the smooth acrylic splints covering
changes in patients with BCL and UCLP using diffe-                         the occlusal surface. Thus, the bonded expander
rent appliances and protocols. Our results have                            along with the hybrid activation protocol should
shed light on a different expansion appliance and                          be suggested in UCLP as a procedure to achieve
protocol in patients with UCLP. Pugliese et al. [21]                       an efficient maxillary expansion with arch widths
found that the maxillary expansion with Hyrax,                             homogeneous increase avoiding buccal tipping
Quad-helix and Expander with Differential Opening                          of premolars and molars maintaining an adequate
(EDO), in bilateral CLP patients, had similar changes                      active treatment time, of about 4 months, in the
in arch size, whereas only Quad-helix expander                             middle between the slow and rapid protocol [17,22].
and EDO were associated with a greater shape at                            This investigation, of course, had some limitations
the intercanine area. Other researchers, comparing                         such as the retrospective nature of the study design



Stoma Edu J. 2020;7(3): 184-190                                                           pISSN 2360-2406; eISSN 2502-0285           187
                     d’Apuzzo F, et al.
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                     with short-term outcomes, currently lacking long-                        CONFLICT OF INTEREST
 Original Articles   term data [23]. Moreover, some subjects with UCLP
                                                                                              The authors declare no conflict of interest.
                     had some permanent teeth missing in the dental
                     arch on one or both sides.                                               AUTHOR CONTRIBUTIONS
                     The elimination of these patients, however, may
                     have introduced higher selection bias eliminating                        Made substantial contributions to conception and design of the
                     more severely affected subjects with smaller arch                        study and performed data analysis and interpretation: FdA, VG, LP.
                     widths which is actually a common clinical frame of                      Performed data acquisition, as well as provided administrative,
                     CLP anomalies.                                                           technical, and material support: LN, AJ, RR.
                     Future studies should be planned with an increased
                                                                                              FINANCIAL SUPPORT AND SPONSORSHIP
                     sample size, even to compare, with multicentric
                                                                                              None.
                     randomized clinical trials, this novel approach with
                     the other conventional maxillary expansion protocol                      ETHICAL APPROVAL AND CONSENT TO PARTICIPATE
                     in patients with UCLP.
                                                                                              Research was performed in accordance with the Declaration of
                     5. CONCLUSION                                                            Helsinki and approved by the ethics committee of the University
                                                                                              of Campania Luigi Vanvitelli, Naples, Italy (Prot. N° 147).
                                                                                              An informed consent to participate in the study was obtained.
                     Growing patients with UCLP showed significant
                     differences in all transverse upper arch dimensions                      CONSENT FOR PUBLICATION
                     after orthodontic treatment with a bonded maxillary
                     expander and a hybrid activation protocol.                               A written informed consent for publication was obtained.




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 188                 Stoma Edu J. 2020;7(3): 184-190                                                             pISSN 2360-2406; eISSN 2502-0285
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                                                                                        Fabrizia D’APUZZO
                                                                            DDS, MS, PhD, Research Fellow
                                   Multidisciplinary Department of Medical-Surgical and Dental Specialties
                                                             University of Campania Luigi Vanvitelli Naples
                                                                                             Naples, Italy



CV
Fabrizia d’Apuzzo is a graduate of Dental Medicine (October 2012 with honors) from the Second University of Naples (Italy). She
attended the “Université Lille II Droit et Santé" in Lille (France) between September 2010 and April 2011 as an “Erasmus” student,
and the University of Trieste (Italy) on a 3-month travel grant from December 2011 to perform her experimental thesis. She was
awarded her Postgraduate Diploma in Orthodontics in February 2017 and her PhD in Biomedical and Biotechnological Sciences
in December 2019. And she is currently a Research Fellow at the University of Campania Luigi Vanvitelli, Naples (Italy).
Dr. d’Apuzzo has authored and co-authored more about 30 articles, 3 book chapters and over 40 posters. She has been a speaker
at international conferences and she received several awards and distinctions.



Stoma Edu J. 2020;7(3): 184-190                                                            pISSN 2360-2406; eISSN 2502-0285                     189
                     d’Apuzzo F, et al.
www.stomaeduj.com




                     Questions
 Original Articles
                     1. Cleft lip and palate is a?
                     qa. Congenital craniofacial birth anomaly;
                     qb. Congenital dental disorder;
                     qc. Nosocomial infection;
                     qd. Acquired disorder.

                     2. What is the etiology of cleft lip and palate?
                     qa. Environmental;
                     qb. Genetic;
                     qc. Epigenetic;
                     qd. All of the aforementioned.

                     3. What is the first surgery performed in CLP patients?
                     qa. Alveolar bone graft;
                     qb. Lip;
                     qc. Soft palate;
                     qd. Hard palate.

                     4. Patients with cleft lip and palate can have
                     qa. Skeletal maxillary constriction;
                     qb. Dental posterior crossbite;
                     qc. Reduced intercanine widths;
                     qd. All of the aforementioned.




 190                 Stoma Edu J. 2020;7(3): 184-190                           pISSN 2360-2406; eISSN 2502-0285