Art-1-Jamilian

ORTHODONTICS                                                                                                                                                           www.stomaeduj.com




                                                                                                                                                                     Original Articles
MAXILLARY SINUS VOLUME IN CLEFT LIP
AND PALATE PATIENTS WITH AND WITHOUT
AN ORONASAL FISTULA USING CBCT
               1a                     2b                          1c                     1d                               1e*
Bita Kiaee          , Ladan Hafezi          , Mahshid Karani           , Faezeh Amiri          , Abdolreza Jamilian

¹Department of Orthodontic, Dental School, Tehran University of Medical Sciences, Tehran, Iran
²Maxillofacial Radiology Department, Faculty of Dentistry, Tehran Medical Sciences, Islamic Azad University, Tehran, Iran
a
  DDS, MS, Assistant Professor; e-mail: dr.bitakia@gmail.com; ORCIDiD: https://orcid.org/0000-0002-9117-3593
b
  DDS, MS, Assistant Professor; e-mail: Lhafezzi@yahoo.com; ORCIDiD: https://orcid.org/0000-0002-1727-2665
c
 DDS; e-mail: dr.mahkm96@gmail.com; ORCIDiD: https://orcid.org/0000-0002-8586-3846
d
  DDS; e-mail: dr.faezehamiri1989@gmail.com; ORCIDiD: https://orcid.org/ 0000-0003-0184-3061
e
  DDS, MS, Professor; e-mail: info@jamilian.net; ORCIDiD: https://orcid.org/0000-0002-8841-0447
ABSTRACT                                                                                https://doi.org/10.25241/stomaeduj.2021.8(3).art.1

Introduction Formation of oronasal fistula is a common complication after surgical closure of cleft lip and
palate (CLP). This study aimed to compare the maxillary sinus volume in 9-12-year-old CLP patients with and
without an oronasal fistula who had undergone surgical closure of the cleft at 1 year of age, using cone-
beam computed tomography (CBCT).
Methodology This descriptive, cross-sectional, analytical study was conducted on CBCT scans of 50 patients
with unilateral CLP who were between 9-12 years and had undergone surgical closure of the cleft at 1
year of age in two groups with and without an oronasal fistula (n=50). The patients were selected among
those presenting to a private orthodontic office between 2001-2009 and already had CBCT scans taken for
orthodontic treatment. The 3D CBCT scans were reconstructed with Mimics software, and the volume of the
maxillary sinuses was measured on the images. Data were compared using t-test.
Results The maxillary sinus volume was significantly smaller in patients with oronasal fistula compared with
those without it (9510.7±492 mm3 vs. 10278.2±512 mm3, P<0.000). The maxillary sinus of the affected side
was smaller than that of the unaffected side in both groups of patients with and without an oronasal fistula
(P<0.05).
Conclusion IImmature patients with unilateral CLP and oronasal fistula have a smaller maxillary sinus than
unilateral CLP patients without an oronasal fistula, and may be at higher risk of respiratory infections.
KEYWORDS
Cone-Beam Computed Tomography; Orofacial Cleft; Fistula; Maxillary Sinus.
1. INTRODUCTION                                                                     Development of an oronasal fistula indicates failure
                                                                                    of the surgical closure of the palate to obstruct the
The failed fusion of the medial nasal and maxillary                                 communication between the oral and the nasal
processes would result in the occurrence of cleft                                   cavity.
lip while failed fusion of the palatine prominences                                 According to the classification by Pittsburg, seven
would lead to the formation of a cleft palate [1,2].                                types of fistula are present [8], which can be
Cleft lip and palate (CLP) has a prevalence of 1 per                                symptomatic or asymptomatic. Symptomatic fistula
500 live births [3]. Surgical management of CLP was                                 can cause several complications such as leakage
first performed approximately 150 years ago [4]. At                                 of foods and drinks from the oral cavity into the
present, CLP patients often undergo surgery before                                  nasal cavity, bad odor, rhinitis, impaired hearing,
the 1st year of age. However, an oronasal fistula may                               hypernasality, infection, and speech problems
develop postoperatively due to the infection of the                                 [7,9,10].
palate or tension of the flap, and cause problems                                   CLP patients often suffer from decreased maxillary
for the patient [5]. The oronasal fistula is a common                               sinus volume and significant esthetic impairments
complication of surgical management of CLP with a                                   due to the maxillary deficiency at the midface, where
prevalence rate of 9-50%. The rate of recurrence of                                 the maxillary sinuses are located. These patients
the fistula after surgery is as high as 35-70% [6,7].                               often develop recurrent sinusitis for no clear reason.
              OPEN ACCESS This is an Open Access article under the CC BY-NC 4.0 license.
              Peer-Reviewed Article
    Citation: Kiaee B, Hafezi L, Karani M, Amiri F, Jamilian A. Maxillary sinus volume in cleft lip and palate patients with and without an oronasal fistula using
    CBCT Stoma Edu J. 2021;8(3):157-162.
    Received: July 15, 2021; Revised: July 23, 2021; Accepted: July 27, 2021; Published: August 03, 2021
    *Corresponding author: Abdolreza Jamilian, DDS,MS
    Department of Orthodontics, Dental School, Cranio Maxillofacial Research Center, Tehran Medical Sciences, Islamic Azad University, Tehran, Iran.
    Tel: +982122011892; Fax: +982122052228; e-mail: info@jamilian.net
    Copyright: © 2021 the Editorial Council for the Stomatology Edu Journal.




Stoma Edu J. 2021;8(3): 157-162                                                                            pISSN 2360-2406; eISSN 2502-0285                           157
                    Kiaee B, et al.
www.stomaeduj.com



                    Also, they have smaller sinuses due to a different         (on sagittal CBCT scans). The CVS of each patient
Original Articles
                    developmental process during the embryonic stage,          was determined by two examiners after reaching a
                    surgical scars, recurrence of fistula, leakage of foods    consensus.
                    and liquids into the nasal cavity, frequent infections,    The exclusion criteria were history of previous
                    and different pattern of air circulation in the nose and   orthodontic treatment, orthognathic surgery,
                    sinuses [1,8,11-14].                                       trauma, syndromes, frequent colds (more than 6
                    Several studies have been conducted on CLP patients        times in 1 year), medication intake at the time of
                    using 2D lateral cephalometry, which has high              CBCT, inflammatory diseases of the upper airways at
                    diagnostic accuracy for clinical applications, despite     the time of CBCT, and systemic conditions.
                    simplicity and low cost [15-20]. However, cone-            All CBCT images had been obtained in standard
                    beam computed tomography (CBCT) has become                 upright position with maximum intercuspation.
                    increasingly popular in the recent years due to its        Also, all images had been taken with NewTom 5G
                    higher accuracy at a comparable cost.                      CBCT scanner (NEWTOM | CEFLA S.C., Imola, Italy)
                    Controversy exists regarding the maxillary sinus           with a total scanning time of 14-18 s, 3.4 s exposure
                    volume in CLP patients such that some studies              time, and 0.3 mm3 voxel size. Three-dimensional
                    reported a significantly smaller volume of the             reconstruction of images was performed according
                    maxillary sinuses in CLP patients compared with            to the Demirtas method [11].
                    normal individuals [15,21-23] while some others            After standardization of images, the maxillary sinus
                    found no significant difference in this respect            volume was quantified. For this purpose, first the
                    [8,11,24-26]. Considering the existing controversy in      skeletal borders adjacent to the sinus structure were
                    this respect, and the gap of information regarding the     were traced.
                    maxillary sinus volume in patients who developed
                    an oronasal fistula after surgery compared with
                    those who did not, this study aimed to compare the
                    maxillary sinus volume in 9-12-year-old CLP patients
                    with and without an oronasal fistula who had
                    undergone the surgical closure of the cleft at 1 year
                    of age using CBCT. The null hypothesis was that no
                    significant difference would be found in the maxillary
                    sinus volume between CLP patients with and without
                    oronasal fistula.

                    2. MATERIALS AND METHODS

                    This descriptive, cross-sectional, analytical study was
                    conducted on 50 unilateral CLP patients between
                    9-12 years who had undergone surgical closure
                    of the cleft at 1 year of age in two groups with and
                    without oronasal fistula (n=50). The patients were
                    selected from among those presenting to a private
                    orthodontic office in Tehran between 2010-2019 and
                    who already had CBCT scans taken for orthodontic
                    treatment. The study was approved by the ethics
                    committee of School of Dentistry (IR.IAU.DENTAL.
                    REC.1399.19).
                    The sample size was calculated to be 25 for each
                    group according to the results of a pilot study on 10
                    patients from each group considering α=0.05, β=0.2,
                    mean maxillary sinus volume of 10671 mm3 and
                    10081 mm3 in the two groups and standard deviation
                    of 715 mm3 using two-sample t-test, assuming equal
                    variances in PASS 15.
                    The CBCT scans of unilateral CLP patients between
                    9-12 years who had undergone surgical closure of the
                    cleft at 1 year of age were retrieved from the archives
                    of a private orthodontic office from 2010-2019 by
                    convenience sampling, and assigned to two groups
                    with and without oronasal fistula.
                    The inclusion criteria were age between 9-12 years,
                    history of surgical closure of the cleft before 1 year      Figure 1. Quantification of the maxillary sinus volume on axial,
                    of age, and cervical vertebral stage (CVS) 2 or 3          sagittal and coronal CBCT sections using the Mimics software.




 158                Stoma Edu J. 2021;8(3): 157-162                                              pISSN 2360-2406; eISSN 2502-0285
Maxillary Sinus Volume in Cleft Lip and Palate
                                                                                                                                     www.stomaeduj.com




                                                                                                                                   Original Articles
 Figure 2. Identifying the maxillary sinus at the cleft
and non-cleft sides.
Next, the sinus area between the bones and the area                     4. DISCUSSION
between the infundibulum and the uncinate process                       This study compared the maxillary sinus volume
was measured (Figs 1 and 2). Then, a 3D model was                       in 9-12-year-old CLP patients with and without
prepared to assess the sinus volume. After image                        oronasal fistula who had undergone surgical closure
reconstruction and standardization of orientation                       of the cleft at 1 year of age using CBCT. The null
in axial, coronal and sagittal planes, the Mimics                       hypothesis was that no significant difference would
software suite-20 (Materialise, 3001 Leuven, Belgium)                   be found in the maxillary sinus volume between
was used for the measurements.                                          CLP patients with and without oronasal fistula. The
The data were analyzed by SPSS version 22 using                         results showed that the maxillary sinus volume was
t-test. All measurements were repeated on 20                            significantly smaller in patients with oronasal fistula
randomly selected CBCT scans after a 2-week interval                    compared with those without it. Also, the maxillary
by another examiner, and the reliability of the                         sinus volume in the cleft side was significantly smaller
measurements was ensured by test-retest reliability.                    than that in the non-cleft side in both groups. Thus,
Since R was found to be >0.8, the results were found                    the null hypothesis of the study was rejected. The
to be adequately reliable.                                              smaller size of the maxillary sinus at the cleft side
                                                                        can be due to different developmental processes
3. RESULTS
                                                                        during the embryonic stage, maxillary deficiency,
                                                                        surgical scars, recurrence of fistula, leakage of
This study evaluated 50 patients including 25 with
                                                                        foods and liquids into the nasal cavity and frequent
and 25 without oronasal fistula. The group with
                                                                        infections, and different patterns of air circulation
oronasal fistula included 17 females (66%) and 8
                                                                        in the nose and sinuses. The present results were
males (34%) with a mean age of 10±1 years. Of all,
30 patients (80%) were in CVS II (15 from each group)                   in agreement with those of Demirtas et al [11]. Our
and 10 (20%) were in CVS III (5 from each group). The                   methodology was also similar to that of Demirtas
control groups were matched with the test group in                      et al, [11] although they did not assess the effect
terms of age, CVS and gender.                                           of the presence of oronasal fistula on the maxillary
Table 1 presents the mean maxillary sinus volume in                     sinus volume; however, they assessed patients
the two groups of patients. As shown, the maxillary                     with a mean age of 13.5 years while we evaluated
sinus volume at the cleft side of patients with                         patients between 9-12 years. The assessment of
oronasal fistula was significantly lower than that at                   patients in this age range was an advantage since
the cleft side of patients without fistula (P<0.000).                   evidence shows airway growth and development in
The maxillary sinus volume at the non-cleft side of                     two periods of 6-9 and 12-15 years, with an interval
patients with oronasal fistula was also significantly                   between 9-12 years [27]. Also, CBCT images are not
lower than that at the non-cleft side of patients                       often obtained from patients younger than 9 years
without fistula (P<0.000). The maxillary sinus volume                   of age (CBCT is often first requested for grafting
at the cleft side was significantly smaller than that                   prior to canine eruption). Moreover, the soft tissue
at the non-cleft side in patients with (P<0.000) and                    becomes more stable after 9 years of age. Erdura
without (P<0.000) oronasal fistula.                                     et al. [8] evaluated the maxillary sinus volume of
Table 1. Mean maxillary sinus volume in the two groups of patients with and without oronasal fistula.

 Maxillary sinus volume                 Sinus volume at the               Cinus volume at the
                                        cleft side                        non-cleft side                P value
 Oronasal fistula                       Mean± std. deviation              Mean± std. deviation
 Present (n=5)                          9510.78±492                       10282.8±483                   p<0.0001
 Absent (n=5)                           10278.2±512                       10932.9±554                   p<0.0001
 FP value                               Fp<0.0001                         Fp<0.0001




Stoma Edu J. 2021;8(3): 157-162                                                             pISSN 2360-2406; eISSN 2502-0285        159
 Original Articles
                     Kiaee B, et al.
www.stomaeduj.com



                     unilateral CLP patients of approximately 13 years                     operated by different surgeons at 1 year of age, and
                     of age. They found no significant difference in the                   different techniques had been used for cleft closure,
                     maxillary sinus volume between the cleft and non-                     which could have different effects. Due to the small
                     cleft sides, which was different from the present                     number of patients, they could not be standardized
                     results, and may be attributed to the different age
                                                                                           in this respect. Future studies should address this
                     range of patients. Hikosaka et al. [18] measured the
                     maxillary sinus volume in patients with CLP. They                     topic and preferably enroll patients operated by the
                     assessed the computed tomography (CT) scans of                        same surgeon and with the same surgical technique
                     109 CLP and 100 non-CLP patients and found no                         for cleft closure. Moreover, the effects of the surgical
                     significant difference between the two groups, which                  technique and time of surgery on the recurrence
                     was in contrast to our findings. Also, they found no                  of the fistula are important topics that need to be
                     significant difference in the maxillary sinus volume                  scrutinized in further studies.
                     of the cleft and non-cleft sides, which was different
                     from the present results. The difference between
                     the two studies may be due to the use of different                    5. CONCLUSION
                     imaging modalities (CBCT vs. CT). Also, they did not
                     specify the patients’ age. Agarwal et al. [16] evaluated              Immature patients with unilateral CLP and oronasal
                     the differences in maxillary sinuses of the cleft and                 fistula have a smaller maxillary sinus than unilateral
                     non-cleft sides. They made the measurements on                        CLP patients without an oronasal fistula, and may
                     the maxilla using a combination of reconstructed
                                                                                           be at higher risk of respiratory infections. Also, the
                     axial, coronal and lateral CT images. The maxillary
                     sinus volume was calculated on 3D reconstructed                       maxillary sinus at the cleft side is smaller than that at
                     images. They found significant reduction of length,                   the non-cleft side in unilateral CLP patients.
                     width, height, depth and volume of the maxillary
                     sinus at the cleft side, which supported the present                  ACKNOWLEDGMENTS
                     results. However, they did not mention the patients’                  None.
                     age range.
                     The assessment of patients between 9-12 years was
                                                                                           AUTHOR CONTRIBUTIONS
                     a strength of this study since this age range is ideal
                                                                                           AJ: Study concept and design; critical revision of the manuscript
                     for assessment of the size of maxillary sinuses [27].
                     The use of CBCT was another strength of this study                    for important intellectual content; administrative, technical, and
                     since it provides highly accurate data regarding                      material support; study supervision. LH: Acquisition of data.
                     the dimensions of anatomical structures, and has                      BK, AJ: Analysis and interpretation of data. FA: Drafting of the
                     high measurement accuracy. Also, the effect of                        manuscript. MK: Statistical analysis.
                     the presence of the fistula on the maxillary sinus
                     dimensions was evaluated in this study, which has                     FUNDING
                     not been addressed before.
                                                                                           This research did not receive any specific grant from funding
                     Not evaluating bilateral CLP patients and small
                                                                                           agencies in the public, commercial, or not-for-profit sectors.
                     sample size were among the limitations of this study.
                     Future studies with a larger sample size are required
                     on bilateral CLP patients. Another limitation was                     COMPETING INTERESTS
                     that, patients evaluated in this study had been                       The authors declare that they have no competing interests.


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 160                 Stoma Edu J. 2021;8(3):157-162                                                           pISSN 2360-2406; eISSN 2502-0285
Maxillary Sinus Volume in Cleft Lip and Palate
                                                                                                                                                  www.stomaeduj.com



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                                                                                                       Bita KIAEE
                                                                                   DDS, MSc, Assistant Professor
                                                                                   Department of Orthodontics
                                                                                                  Dental School
                                                                           Tehran University of Medical Sciences
                                                                                                    Tehran, Iran

CV
Dr. Kiaee received her DDS (2013) and MSc in Orthodontics (2017) from Tehran University in Tehran, Iran. Since 2017, she has
been working as an Assistant Professor at the Department of Orthodontics within the Faculty of Dentistry of Tehran University.
Her research areas of interest are mostly clinical projects.




Stoma Edu J. 2021;8(3):157-162                                                             pISSN 2360-2406; eISSN 2502-0285                      161
                    Kiaee B, et al.
www.stomaeduj.com




                    Questions
Original Articles
                    1. Which item was among the exclusion criteria stated in this article?
                    qa. History of surgical closure of the cleft before 1 year of age;
                    qb. Orthognathic surgery;
                    qc. Cervical vertebral stage (CVS) 2 or 3;
                    qd. Age between 9-12 years.

                    2. Which statement is NOT TRUE based on the findings of this article?
                    qa. The maxillary sinus volume at the cleft side of patients with oronasal fistula was significantly lower than
                    that at the cleft side of patients without fistula;
                    qb. The maxillary sinus volume at the non-cleft side of patients with oronasal fistula was significantly lower
                    than that at the non-cleft side of patients without fistula;
                    qc. The maxillary sinus volume at the cleft side was significantly smaller than that at the non-cleft side in
                    patients with and without oronasal fistula;
                    qd. The maxillary sinus volume at the cleft side of patients without oronasal fistula was significantly lower
                    than that at the cleft side of patients with fistula.

                    3. Which of the following statements is True based on the findings of this article?
                    qa. Immature patients with unilateral CLP and oronasal fistula have a smaller maxillary sinus than unilateral
                    CLP patients without an oronasal fistula;
                    qb. Immature patients with unilateral CLP and oronasal fistula may be at lower risk of respiratory infections;
                    qc. The maxillary sinus at the non-cleft side is smaller than that at the cleft side in unilateral CLP patients;
                    qd. Adult patients with bilateral CLP and oronasal fistula have a smaller maxillary sinus than unilateral CLP
                    patients without an oronasal fistula.

                    4. Which one is the prevalence of cleft lip and palate patients based on the present study?
                    qa. 1 per 700 live births;
                    qb. 1 per 1000 live births;
                    qc. 1 per 500 live births;
                    qd. 2.3 per 1000 live births.




 162                Stoma Edu J. 2021;8(3): 157-162                                         pISSN 2360-2406; eISSN 2502-0285