Art-3_Jamilian

ORTHODONTICS                                                                                                                                                www.stomaeduj.com




CEPHALOMETRIC PARAMETERS IN CLEFT PALATE




                                                                                                                                                          Original Articles
PATIENTS WITH AND WITHOUT ORONASAL FISTULA
                         1a*                       2b                                       1c                         1d
Abdolreza Jamilian      , Ludovica Nucci  , Mohammadamin Narimani                                , Maziyar Farhadi          ,
                 1e                3                 4g
Mona Farahmand      , Zinat Kamali , Alireza Darnahal

1Department of Orthodontics, Faculty of Dentistry, Cranio Maxillofacial Research Center, Tehran Medical Sciences, Islamic Azad University, Tehran, Iran
2Multidisciplinary Department of Medical-Surgical and Dental Specialties, University of Campania Luigi Vanvitelli, Naples, Italy
3Faculty of Nutrition and Food Technology, National Nutrition and Food Technology, Research Institute Shahid Beheshti, University of Medical Science,
Tehran, Iran
4Department of Periodontics and Oral Medicine, School of Dentistry, University of Michigan, Ann Arbor, MI 48109 USA
a
  DDS, PhD, Professor; e-mail: info@jamilian.net; ORCIDiD: https://orcid.org/ 0000-0002-8841-0447
b
  DDS, PhD Student Orthodontic Program; e-mail: ludovica.nucci@virgilio.it; ORCIDiD: https://orcid.org/0000-0002-7174-7596
c
  DDS, PhD, Professor; e-mail: info@dr-narimani.com; ORCIDiD: https://orcid.org/0000-0001-6684-3121
d
  DDS; e-mail: maziyar.farhadi@yahoo.com; ORCIDiD: https://orcid.org/0000-0002-1912-8675
e
  DDS; e-mail: mona.farahmand87@gmail.com; ORCIDiD: https://orcid.org/0000-0002-0992-0263
f
 Deputy of Research &Technology; e-mail: Z.kamali@sbmu.ac.ir; ORCIDiD: https://orcid.org/0000-0003-1488-8971
6
  DDS, Post Doctorate Student of Periodontics; e-mail: alirezadarnahal@gmail.com; ORCIDiD: https://orcid.org/0000-0001-9629-731X

ABSTRACT                                                                               https://doi.org/10.25241/stomaeduj.2021.8(4).art.3

Introduction Failure in the fusion of medial nasal processes and maxillary prominences would result in the
development of a cleft lip while failure in merging the palatal shelves would result in the development of a
cleft palate (CP). At present, patients with a cleft palate (CP) have often undergone corrective surgery before
1 year of age. However, an oronasal fistula (ONF) often remains in the palate after surgery.
Methodology In this historical cohort, 40 patients with CP, 6-10 years old, who had undergone corrective
surgery at 1 year of age were selected by targeted sampling and assigned to two groups with ONF (N=20)
and without ONF (n=20). The two groups were matched in terms of age and gender. All patients had an
intraoral photograph of the maxilla and a lateral cephalogram. Twenty-three cephalometric parameters were
measured prior to the orthodontic treatment and compared between the two groups using a t-test.
Results CP patients with ONF had significantly higher Y-axis, gonial angle, Go1, Go2, GoGn-SN, U1-SN, and
lower lip to E line compared with those without ONF (P<0.05). The Jarabak index was significantly lower in
the CP patients with ONF compared with those without it (P<0.05). The difference in other cephalometric
parameters was not significant between the two groups (P>0.05).
Conclusion Patients with ONF had significantly greater growth in the vertical dimension compared with
those without ONF. Yet, the difference in the anteroposterior dimension was not significant between the two
groups.
KEYWORDS
Orthodontics and Dento-Facial Orthopedics; Oronasal Fistula (ONF); Cleft Lip; Cleft Palate; Cephalometric
Parameters.
1. INTRODUCTION                                                                  CP patients often experience restricted development
                                                                                 of craniofacial structures. Controversy exists regar-
Failure in the fusion of medial nasal processes                                  ding the causes of growth limitation in these
and maxillary prominences would result in the                                    patients. Some researchers [2] have attributed it
development of a cleft lip while failure in merging                              to the surgical repair while some other factors not
palatal shelves would result in the development of a                             related to the corrective surgery such as genetics
cleft palate (CP) [1]. At present, patients with a cleft                         (inherited trait), and adaptive changes due to the
palate (CP) often undergo corrective surgery before                              mechanical presence of the cleft. Growth limitations
1 year of age. However, a fistula often remains in the                           associated with CP may affect the growth pattern in
palate after surgery that causes complications for                               all three planes of vertical, sagittal and transverse
patients.                                                                        [3-5].
               OPEN ACCESS This is an Open Access article under the CC BY-NC 4.0 license.
               Peer-Reviewed Article
    Citation: Jamilian A, Nucci L, Narimani M, Farhadi M, Farahmand M, Kamali Z, Darnahal A. Cephalometric parameters in cleft palate patients with and
    without oronasal fistula. Stoma Edu J. 2021;8(4):247-252
    Received: October 11, 2021; Revised: 06 November; Accepted: November 11, 2021; Published: November 15, 2021
    *Corresponding author: Prof. Abdolreza Jamilian, DDS, PhD, Department of Orthodontics, Faculty of Dentistry, Cranio Maxillofacial Research Center,
    Tehran Medical Sciences, Islamic Azad University, Tehran, Iran
    Tel.: +98-22052228; Fax: +98-2122052228; e-mail: info@jamilian.net
    Copyright: © 2021 the Editorial Council for the Stomatology Edu Journal.




Stoma Edu J. 2021;8(4):247-252                                                                         pISSN 2360-2406; eISSN 2502-0285                    247
                    Jamilian A, et al.
www.stomaeduj.com



                    Oronasal fistula (ONF) is among the most common               Wits appraisal: The distance between AO line and BO line
Original Articles   complications of corrective surgery in CP patients            Facial angle: The angle at the intersection of Frankfurt
                    [6]. Its prevalence ranges from 4% to 35% following           plane and N-Pog line
                    primary palatoplasty [7]. Nasal regurgitation and             Y-axis: The angle at the intersection of SN line and N-Gn
                    speech problems (mainly hypernasality) are the main           line
                    symptoms associated with ONF. The location and                Gonial angle: The angle at the intersection of GO-Gn and
                    size of the fistula and its causes are highly variable.       Go-Ar lines
                    Primarily, it occurs as the result of repair under tension.   GO1: The angle at the intersection of Ar-Go line and
                    Postoperative infection may also be the cause [6].            Go-N line
                    The growth pattern of surgically-treated and untreated        GO2: The angle at the intersection of Go-N line and
                    CP patients has been extensively studied [8-12].              Go-Me line
                    However, information about the effect of presence/            GoGn-Sn: The angle at the intersection of Go-Gn and SN
                    absence of ONP after surgery on the growth pattern            line
                    is limited. Knowledge about the growth pattern of             N-Me: The distance between N point and Me point
                    CP patients can significantly affect the functional           (or posterior facial height)
                    and esthetic treatment outcomes [13]. Considering             S-Go: The distance between S point with Go point
                    the effect of surgical repair of the cleft on the growth      (or anterior facial height)
                    pattern of the jaw in CP patients and the gap of              Jarabak index: The ratio of the anterior facial height to
                    information about the effect of the presence/absence          the posterior facial height
                    of ONF after surgery on the growth pattern, this study        Inclination angle: The angle at the intersection of N’ line
                    aimed to assess the cephalometric parameters of 6 to          perpendicular to the palatal plane
                    10-year-olds with CP who had undergone corrective             U1-SN: The angle of upper incisors relative to SN line
                    surgery at 1 year of age prior to orthodontic surgery         U1-NA: The distance from upper incisors to NA line
                    in two groups with and without ONF.                           L1-mand.: The angle of lower incisors relative to the
                                                                                  mandibular plane
                                                                                  L1-NB: The distance between the lower incisors and NB
                    2. MATERIALS AND METHODS
                                                                                  line
                                                                                  Interincisal angle: The angle between the maxillary and
                    This historical cohort was conducted on 40 CP
                                                                                  mandibular incisors
                    patients, 6-10 years of age, (mean age of 8.25 years)
                                                                                  Nasolabial angle: The angle between the line tangent to
                    who had undergone corrective surgery at 1 year of
                                                                                  the nasal base and the line tangent to the upper lip
                    age and presented to a private office in Tehran. All
                                                                                  Upper lip to E-line: The distance between the upper lip to
                    patients had an intraoral photograph of the maxilla
                                                                                  the Pn-Pog’ or E-line
                    and a lateral cephalogram prior to their orthodontic
                                                                                  Lower lip to E-line: The distance between the lower lip to
                    treatment. The photographs and cephalograms had
                                                                                  Pn-Pog’ or E-line
                    been taken for treatment purposes not related to              For higher accuracy, all the above-mentioned
                    this study. All images were obtained in the habitual          parameters were measured twice by the same
                    centric occlusion, with teeth in contact, lips relaxed,       examiner with a 2-week interval to ensure adequate
                    and head in natural position. The parents or legal            intraobserver agreement. The intraclass correlation
                    guardians of all patients signed informed consent             coefficient (ICC) was calculated for this purpose.
                    forms prior to their participation in the study. The          The data were analyzed using SPSS version 22 (SPSS
                    study was approved by the ethics committee of                 Inc., IL, USA). The normal distribution of the data in each
                    School of Dentistry Islamic Azad University, Tehran,          group was analyzed using the Kolmogorov-Smirnov
                    Iran (Number 22710201961064).                                 test. The measured cephalometric parameters were
                    A total of 40 patients including 20 patients with             compared between the two groups using the t-test in
                    ONF and 20 patients without ONF were selected                 case of the normal distribution of the data and Mann-
                    and enrolled using targeted sampling. The patients            Whitney test in case of the non-normal distribution of
                    were matched in terms of age and gender in the two            the data. The level of significance was set at 0.05.
                    groups and there were 11 males and 9 females in each
                    group with a mean age of 8.4±1 years in the no fistula
                    and 8.1±1 years in the fistula group. Figure 1 and 2
                    show the occlusal view of patients with and without
                    ONF respectively. Their lateral cephalograms were
                    traced manually and the following cephalometric
                    parameters were measured on their lateral
                    cephalograms:
                    SN-FH: The angle between the SN line and the Frankfurt
                    plane
                    SNA: The angle at the intersection of SN line and NA line
                    SNB: The angle at the intersection of SN line and NB line
                    ANB: The angle at the intersection of NA line and NB line      Figure 1. Occlusal view with oronasal fistula.




 248                Stoma Edu J. 2021;8(4): 247-252                                                   pISSN 2360-2406; eISSN 2502-0285
Cephalometric parameters in cleft palate patients
                                                                                                                                                    www.stomaeduj.com




                                                                                                                                                  Original Articles
                                                                          Table 2. Cephalometric parameters in the vertical dimension in CP
                                                                         patients with and without ONF..

                                                                          Cephalometric            Absence         Presence        P value
                                                                          parameters               of ONF          of ONF          (t-test)
                                                                          Y-axis                   66.4±5.8        71.5±3.6        0.001
                                                                          Gonial angle             125.3±6.9 131.9±6.4 0.003
                                                                          Go1                      53.2±6.3        55.55±4.8 0.04
                                                                          Go2                      72.15±4.9 76.85±6.2 0.012
                                                                          Go-GN-SN                 33.4±4.1        35.9±3.5        0.038
                                                                          N-Me                     113±12.6        118.7±9.6 0.116
 Figure 4. Occlusal view without oronasal fistula.                        S-Go                     67.95±7.3 71.15±9.1 0.228
                                                                          Jarabak Index            63.15±6.2 59.5±4.2              0.035
3. RESULTS                                                                Inclination Angle 84.25±5                83.25±7.7 0.629

The ICC value indicated excellent intraobserver                          Table 3 presents the dental cephalometric parameters
agreement. The minimum ICC was 0.991 for the ANB                         in CP patients with and without ONF. The U1-SN in
while the maximum ICC was 1 for the facial angle,                        CP patients with ONF was significantly higher than
Wits appraisal, Inclination angle, U1-SN, L1-mand.,                      that in the group without fistula (P<0.005).
interincisal angle, gonial angle, Jarabak index, and                      Table 3. Dental cephalometric parameters in CP patients with and
nasolabial angle. The mean angular measurement                           without ONF.

error was 0.5°±0.5° and the mean linear measurement                       Cephalometric Absence                  Presence          P value
error was 0.5±0.5 mm.                                                     parameters    of ONF                   of ONF            (t-test)
Table 1 presents the cephalometric parameters in                          U1-Sn                99±14.3           74.2±17.5         0.001
the anteroposterior dimension in CP patients with                         U1-NA                5.05±2.6          5.80± 2.8         0.445
and without ONF. The t-test revealed no significant                       L1-Mand              90.85±8           89.4±8.7          0.289
difference between the two groups of CP patients
                                                                          L1-NB                4.05±1.8          4.00±1.8          0.947
with and without ONF in any of the cephalometric
                                                                          Interincisal         128.05±26.5       111.63±58.2 0.962
parameters in anteroposterior dimension (P>0.05).
                                                                          Angle
 Table 1. Cephalometric parameters in the anteroposterior dimension in
CP patients with and without ONF.                                        Table 4 presents the soft tissue cephalometric
 Cephalometric Absence                    Presence       P value         parameters in CP patients with and without ONF.
 parameters    of ONF                     of ONF         (t-test)        Among the soft tissue cephalometric parameters,
                                                                         the lower lip to E-line was significantly larger in CP
 SN-FH                    8.5±2.4         10.6±3.7       0.081           patients with fistula (P<0.005).
 SNA                      78.75±3         77.5±2.5       0.23
                                                                          Table 3. Soft tissue cephalometric parameters in CP patients with and
 SNB                      79.45±2.5 78.25±2.3            0.12            without ONF.
 ANB                      3.5±1.9         3.4±2.2        0.9              Cephalometric          Absence          Presence         P value
 Wits appraisal           -1.5±3.8        -2±4.8         0.72             parameters             of ONF           of ONF           (t-test)

 Facial angle             80.75±3.3 78.85±7.8            0.32             Nasolabial Angle 100.45±18.3 96.2±16.6                   0.446
                                                                          Up-lip to E-line       -3.25±3.4        -3.5±4.7         0.678
Table 2 presents the cephalometric parameters in the                      Low-lip to E-line      -1.25±2.6        1.6±4.2          0.015
vertical dimension in CP patients with and without
ONF. The results of the t-test revealed that the Y-axis                  4. DISCUSSION
was significantly greater in CP patients with ONF
                                                                         This study aimed to assess the cephalometric
compared with those without ONF (P<0.05). Also, the                      parameters prior to orthodontic treatment of
mean size of gonial angle was significantly larger in                    6-10-year-old CP patients who had undergone
CP patients with ONF compared with those without                         corrective surgery at 1 year of age in two groups
it (P<0.05). Go1 and Go2 in CP patients with ONF                         with and without ONF. The results showed that in the
were significantly greater than the corresponding                        anteroposterior dimension, no significant difference
values in patients without a fistula (P<0.05). The                       existed between the two groups. However, in
                                                                         the vertical dimension, patients with ONF had
Go-GN-SN parameter was significantly greater in
                                                                         significantly greater vertical growth. This stark
CP patients with ONF as well (P<0.05). The Jarabak                       difference in the vertical dimension may be due to
index was significantly lower in CP patients with ONF                    mouth breathing in children, causing a more vertical
compared with those without it (P<0.05).                                 growth pattern in patients with ONF.


Stoma Edu J. 2021;8(4):247-252                                                              pISSN 2360-2406; eISSN 2502-0285                       249
                    Jamilian A, et al.
www.stomaeduj.com



                    Naqvi et al. [13] reported short maxillary height in                between CLP patients and healthy controls in the
Original Articles   unoperated patients with ONF. However, the patients                 mandible (body, ramus, gonial angle, and mandibular
                    had a vertical growth pattern in their study. Their                 plane angle). The difference between their findings
                    results were in agreement with our findings. With                   and ours in the anteroposterior dimension may be
                    regards to the anterior growth however the results                  due to the fact that they had a healthy control group.
                    were in contrast to ours, which may be due to the                   Racial differences and number of lost teeth can also
                    presence of a healthy control group in their study.
                                                                                        affect the results.
                    Moreover, our study evaluated patients with and
                                                                                        Yoshida et al. [18] evaluated the morphology of
                    without ONF who had undergone corrective surgery.
                    Patients with CP have a lower anteroposterior growth                unoperated CLP patients using lateral cephalometry.
                    than healthy individuals [14,15].                                   They reported maxillary retrusion and a steeper
                    Xu et al, [8] in their case-control study on 106                    mandible in CLP patients, and the tendency for
                    cephalograms of non-syndromic CP patients and 102                   maxillary retrusion and a steeper mandible had a
                    healthy controls in three groups of samples reported                direct correlation with age. They also reported the
                    shortened cranial base length and reduced bony                      absence of labial proclination of incisor teeth in
                    nasopharyngeal height in patients with unrepaired                   CLP patients. However, 3 patients with CP had labial
                    isolated CP. The patients also had short maxillary                  proclination of the incisor teeth. Controversy in the
                    depth and height, and a posteriorly positioned                      results can be due to the difference in age range of
                    maxilla. The width of the nasal cavity, maxilla                     the patients, the presence of a healthy control group
                    and orbit had increased and they had shortened                      and the fact that they evaluated unoperated patients.
                    mandibular length and height. Adults had normal                     The sample size, age range of patients, technique of
                    nasopharyngeal and mandibular morphology.
                                                                                        surgery, and racial and environmental characteristics
                    Nonetheless, they still had shortened cranial base
                    length and short maxillary depth and anterior                       may explain the controversy in the results of studies.
                    height. The width of the nasal cavity, maxilla and                  Tooth loss and its time can also affect the results.
                    orbit had increased in them. They concluded that the                In general, the ONF presence is associated with
                    craniofacial growth and morphology in unrepaired                    a vertical growth pattern When diagnosing and
                    isolated CP patients is significantly affected by non-              treating CP patients with ONF, orthodontists should
                    surgical factors. The same results were reported by                 recognize the facial growth pattern in order to reduce
                    Ye et al [8]. The difference between our results and                undesirable instances of craniofacial development
                    theirs is due to the presence of a healthy control                  to the extent possible.
                    group in the aforementioned studies and the fact                    This study was somewhat limited by its sample size. In
                    that we evaluated patients who had undergone                        order to examine the efficacy of different corrective
                    corrective surgery while they studied unoperated                    surgical techniques and their timing on the cranial
                    patients.                                                           growth pattern of CP patients more conclusively,
                    In contrast to our study Costello et al. [16] reported
                                                                                        similar procedures with larger group of participants
                    that the maxillary growth was much lower in
                    unoperated patients while the posterior facial height               are recommended.
                    was higher in operated patients. Liao and Mars
                    [14] evaluated 39 patients with CP over 17 years of                 5. CONCLUSION
                    age in comparison with age- and gender-matched
                    controls. They reported that patients with unilateral               CP patients with ONF experienced a significantly
                    cleft lip and palate (CLP) had shorter height of the                greater growth in the vertical dimension compared
                    basal maxilla, shorter posterior length of the basal                with those without ONF. However, no significant
                    maxilla, and less protruded basal maxilla at the                    difference was noted in the anteroposterior
                    zygomatic level, compared with the control group.                   dimension between the two groups.
                    They found a significant correlation between the
                    posterior height of the basal maxilla and size of the               AUTHOR CONTRIBUTIONS
                    cleft maxillary segment [14]. The difference between                AJ: study concept, data interpretation, writing and revising the
                    their results and ours may be due to the fact that                  report and final approval of the article. LN: study design, literature
                    they only evaluated the maxilla while we assessed                   review. MN: administration, data interpretation, recruitment,
                    the cranial growth pattern.                                         statistical analysis. MAF: data gathering, data interpretation. MOF:
                    Capelozza et al. [17] evaluated the craniofacial                    critical revision and final approval of the article. ZK: statistical
                    morphology in CLP patients using lateral                            analysis and critical revision. AD: drafting, data interpretation,
                    cephalometry. They reported that CLP patients had                   critical revision and final approval of the article.
                    a smaller and more protruded maxilla. Also, the
                    anteroposterior facial height was much larger in                    CONFLICT OF INTEREST
                    CLP patients, and significant differences were noted                There is no conflict of interest.

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 250                Stoma Edu J. 2021;8(4): 247-252                                                         pISSN 2360-2406; eISSN 2502-0285
Cephalometric parameters in cleft palate patients
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                                                                                       Abdolreza JAMILIAN
                                                                                               DDS, PhD, Professor
                                                                                     Department of Orthodontics
                                                                                               Faculty of Dentistry
                                                                              Cranio Maxillofacial Research Center
                                                                                           Islamic Azad University
CV                                                                                                     Tehran, Iran

Professor Abdolreza Jamilian is a researcher and specialist in the field of Orthodontics. He received his D.D.S (1991), MSc
in Orthodontics (1998), and Fellowship of Orthognathic Surgery & Craniofacial Syndromes (2010) from the Shahid Beheshti
University in Tehran, Iran. He obtained his membership of the European Board of Orthodontics in 2013. Now he is professor at
the Islamic Azad University in Tehran. His practice is limited to orthodontics. He has lectured in several international congresses
and has been a consultant for various journals. He has published over 200 original, peer reviewed research and review articles,
15 book chapters and more than 300 scientific communications. He holds 3 patents in the United States Patent and Trademark
Office. Research interests: Class 3 malocclusion, Cleft lip and palate, Orthognathic surgery.




Stoma Edu J. 2021;8(4):247-252                                                              pISSN 2360-2406; eISSN 2502-0285                       251
                    Jamilian A, et al.
www.stomaeduj.com




                    Questions
Original Articles
                    1. What are the main symptoms associated with ONF?
                    qa. Hypernasality;
                    qb. Less growth in the vertical dimension;
                    qc. Maxillary protrusion;
                    qd. Mandibular retrusion.

                    2. According to this article, what is the difference between the growth patterns of
                    patients with fistula and patients without fistula?
                    qa. Greater growth in the anteroposterior dimension between the two groups;
                    qb. Less growth in the maxilla in patients with fistula compared with those without ONF;
                    qc. Greater growth in the vertical dimension in patient with fistula compared with those without ONF;
                    qd. No significant difference was noted between the two groups.

                    3. According to this article, at what age did patients with cleft palate perform surgery?
                    qa. At 6 years of age;
                    qb. At 10 years of age;
                    qc. Between 6 to 10 years of age;
                    qd. At 1 year of age.

                    4. According to this article, what causes a recurrence of a fistula after surgery?
                    qa. Genetic factors;
                    qb. Patient age at surgery;
                    qc. Patient gender;
                    qd. Penetration.




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 252                Stoma Edu J. 2021;8(4): 247-252                                      pISSN 2360-2406; eISSN 2502-0285