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Table of Contents   
ORIGINAL RESEARCH  
Year : 2021  |  Volume : 32  |  Issue : 4  |  Page : 472-479
Comparison of dentofacial morphology between parents of children with and without craniofacial anomalies using cephalogram imaging


1 Department of Orthodontics and Dentofacial Orthopedics, Institute of Dental Studies and Technologies, Uttar Pradesh, India
2 Department of Orthodontics and Dentofacial Orthopedics, Manav Rachna Dental College, Faridabad, Haryana, India
3 Department of Plastic Surgery, Sant Parmanand Hospital, Civil Lines, Delhi, India
4 Reader, Department of Orthodontics and Dentofacial Orthopaedics, Institute of Dental Studies and Technologies, Modinagar, Uttar Pradesh, India
5 Department of Paediatrics, Vardhaman Mahavir Medical College and Safdarjung Hospital, Delhi, India

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Date of Submission25-Jun-2021
Date of Decision13-Jan-2022
Date of Acceptance17-Feb-2022
Date of Web Publication18-May-2022
 

   Abstract 


Objective: To compare cephalometric parameters in parents of children with bilateral cleft lip and palate (BCLP) and parents of children without any craniofacial anomaly. Methods: A sample of 100 sets of parents were segregated into two groups of 50 parents each. The two groups included parents of children with BCLP and the parents of children with no craniofacial anomaly. Lateral cephalograms and postero-anterior (PA) cephalograms were examined for 100 parents of both the BCLP and control group, respectively. Tracings were scrutinized for 34 linear and angular parameters of dentocraniofacial skeleton. Data were analysed using unpaired Student's t-test. Result: Evaluated lateral cephalograms revealed 15 significant parameters in parents of children with BCLP including a significant increase in Ptm-S, maxillary length, SNA, articular angle and gonial angle. A significant decrease was noted in SN length, cranial base flexion, mandibular length, mandibular body length, total facial height, upper facial height, posterior facial height, Y-axis length and soft tissue total anterior facial height. Upon evaluation of PA cephalograms, four significant parameters were found in parents of children with BCLP. Cranial width, bizygomatic width, maxillary width and lateral interorbital width were significantly decreased in the parents of children with BCLP. Conclusion: Distinct dentocraniofacial morphological features have been found in parents of children with BCLP. Evaluation of these features might be the key to predicting the occurrence of clefts in their children and also planning for future linkage analysis studies amongst them.

Keywords: Bilateral cleft lip and palate (BCLP), cleft, craniofacial anomaly, dentocraniofacial morphology, parent

How to cite this article:
Arora A, Batra P, Sood SC, Raghavan S, Sood A. Comparison of dentofacial morphology between parents of children with and without craniofacial anomalies using cephalogram imaging. Indian J Dent Res 2021;32:472-9

How to cite this URL:
Arora A, Batra P, Sood SC, Raghavan S, Sood A. Comparison of dentofacial morphology between parents of children with and without craniofacial anomalies using cephalogram imaging. Indian J Dent Res [serial online] 2021 [cited 2022 Dec 7];32:472-9. Available from: https://www.ijdr.in/text.asp?2021/32/4/472/345428



   Introduction Top


Birth defects are one of the leading causes of child disability and mortality worldwide. Complete bilateral cleft lip and palate (BCLP) is the most severe amongst the orofacial cleft subtypes. Before any surgical correction, these infants are characterized by a prominent premaxilla, reduced posterior maxillary height and a small, retruded mandible.[1] Currently, a multifactorial model of inheritance is favoured in which genetic risk factors interact with environmental covariates[2],[3],[4] to produce cleft phenotypes.

A range of cleft lip and/or cleft palate (CL/P) microform features have been proposed in siblings as well as in non-cleft parents (e.g. absence of maxillary lateral incisor, formes frustes, nasal deformity, high palatal vault, etc.) as cleft phenotypic presentations. Identification of the cleft microforms can improve the prediction of relative risks, assist in identifying individual cleft type etiopathogenesis and decode the hereditary contribution to the orofacial cleft.[5],[6]

Cephalometric analysis has been used previously for analysing and comparing parental features (affected and unaffected). In addition, literature exists upon orofacial clefting and phenotypic presentations within relatives of patients with orofacial clefts, based on a heterogenous sample. Hence, the present study is based on a homogenous sample of parents of children with BCLP. The study aims to assess the dentocraniofacial morphological features within parents of children with BCLP and the parents of children with no craniofacial anomaly.


   Materials and Methods Top


The study was conducted in the orthodontic department of a dental college in North India in collaboration with a secondary hospital. Before the start of the study, approval was obtained from the institutional ethical committee (ref. IDST/ERBC/2011/04) and informed consent was taken from the parents. The sample was calculated to the power of 90% and α of 0.05 with an effect size of 0.6 factoring in the prevalence of BCLP with almost 20% of the total cleft population (Incidence 1:800). A total of 100 sets of parents were selected which were segregated into two groups each (as given below) following application of inclusion and exclusion criteria:

Group 1 (Experimental group): Sets of 50 parents (50 mothers and 50 fathers) of children with BCLP (Source data - secondary hospital).

Group 2 (Control group): Sets of 50 parents (50 mothers and 50 fathers) of children with no craniofacial anomaly (Source data - orthodontic department of a dental college).

The inclusion and exclusion criteria were as follows:

Inclusion criteria

  1. Biological parents.
  2. Both mother and father to be available.
  3. Parents of non-syndromic complete bilateral cleft lip palate children.
  4. Control group - Parents of children with no craniofacial deformity.


Exclusion criteria

  1. No subjects are to be affected by any medical condition that is recognized to influence tooth size and morphology.
  2. No subject should have a predisposition to clefting (i.e. chromosomal anomalies, single-gene causes, teratogenic causes).
  3. No pregnant women to be included.
  4. No previous history of surgical or orthodontic treatment.


A questionnaire was administered to the parents of both groups for excluding extrinsic factors of a cleft in the respective offspring. Patients with syndromes were not included in this sample. Cephalograms were taken for parents of both the BCLP and control group using the cephalostat (Kodak Dental Systems, France, Model: 8000 C, Type: CG 810, 06/2006) with an exposure time of 1 s, set at 70–80 kvp and 12 mA, on a laminate film 15 × 30 cm (Kodak, T-MAT, Resende, Brazil). Cephalometric tracings were done on lead acetate paper and were analysed by a single examiner to avoid inter-examiner variations.

Twenty-seven parameters on lateral cephalogram and seven parameters on postero-anterior (PA) cephalogram [Table 1] and [Table 2]; [Figure 1]a, [Figure 1]b, [Figure 1]c were analysed and compared. Fifty randomly selected radiographs were re-evaluated after 1 week by the same examiner for assessment of intra-examiner reliability using paired t-test and intraclass coefficient (ICC). The value ranged from 0.88 to 0.96 with 0.93 being reported as the average ICC value using a two-way mixed model [Table 3]. Data were statistically analysed via software SPSS (statistical package for social sciences) version 21.0. The unpaired Student's t-test was applied to verify possible differences between dentocraniofacial morphology of parents of children with BCLP and parents of children with no craniofacial anomaly. As it is known that the use of multiple t-tests with cephalometrics is known to result in false positives. To address this concern, the Bonferroni test was applied to adjust for the same.
Table 1: Skeletal, dental and soft tissue parameters studied on lateral cephalogram

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Table 2: Comparison of parameters between parents of children with BCLP and parents of children without any craniofacial anomaly

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Table 3: The intraclass correlation coefficient for reliability

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Figure 1: (a) Skeletal linear parameters; (b) Skeletal angular parameters; (c) Dental and soft tissue parameters; (d) Landmarks of PA cephalogram

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Adjustment of data (Mossey et al., 1997)[3]

The data collected was in pairs (mother and father) in both groups. Before analysis, it was necessary to adjust the variables to remove the gender bias. It was calculated by taking a mean of the values of the cephalometric parameters for the two specific parental groups and by taking the difference between means of mother and father of one group and adding it to the mother values. The mid-parent value, i.e., the average of both mother and father was then calculated. This was done for both groups. Both groups were compared over the mid-parent value of each.


   Results Top


The mean age of the parents of the two groups was as follows: Experimental group - 33.47 + 3.78 years; Control group - 34.03 + 3.91 years.

Comparison of the lateral cephalometric parameters between the two groups revealed a significant increase in Ptm-S distance, maxillary length, SNA angle, articular angle, gonial angle in the experimental group (p < 0.05). We also found a significant decrease in anterior cranial base length, cranial base flexion angle, mandibular length, mandibular body length, ramus flexion angle, total facial height, upper facial height, posterior facial height, Y-axis length, soft tissue total and lower anterior facial height (p < 0.05).

The mean and standard deviation of the linear and angular lateral cephalometric variables of parents of children with BCLP and parents of children without any craniofacial anomaly are presented in [Table 2], [Figure 1]a, [Figure 1]b, [Figure 1]c, [Figure 1]d. [Table 4] shows the comparison of results between previous studies of lateral cephalogram data on parental findings with the present study.
Table 4: Comparison of results from previous studies of lateral cephalogram data on parental findings with present study

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On comparison of the PA cephalometric parameters between the two groups, we found a significant decrease in the cranial width, bizygomatic width, maxillary width and lateral interorbital width in the experimental group (p < 0.05).

The mean and standard deviation of the linear PA cephalometric variables of both study groups are presented in [Table 2]. [Table 4] shows the comparison of results between previous studies on PA cephalogram data (parental findings) and the present study.


   Discussion Top


Numerous investigators over the years have reported that the dentocraniofacial phenotype in CL/P is different from that of non-cleft individuals.[7],[8] Cleft phenotypes are compounded by disordered muscle insertions, functional disturbances and various surgical and non-surgical manipulations.[9] Cleft-specific phenotypic features are also demonstrated in regions of the craniofacial complex that are remote from the cleft site. In addition, abnormalities of number, size, shape, development, eruption and enamel of teeth are present more frequently in children affected with cleft lip with or without cleft palate (CL ± P) or cleft palate (CP) than in the general population[10] which may be inherited.

In our study, the anterior cranial base length (SN length) was found to be significantly decreased in the experimental group. This finding is similar to as reported by Perkiomaki et al.[11] In contrast, an increase in anterior cranial base length has also been reported by other studies.[5],[12]

Cranial base flexion angle was found to be reduced in the experimental group in our study. Coccaro et al.[13] have reported similar results in their study. In contrast, other studies show opposing results in their experimental non-homogeneous sample.[14],[15]

The perpendicular distance between Ptm and S and maxillary length was found to be significantly increased in our experimental group. Nakasima and Ichinose[14] also found them to be increased in their samples. These findings indicate an abnormally placed and large-sized maxilla causing a maxillary protrusion in the experimental group leading to an increased SNA angle in the experimental sample. This is in agreement with the observations of Raghavan et al.[15] reflecting a protruded maxilla. Thus, features of the abnormal maxilla in parents could lower the threshold for expression of a cleft in their offspring. Melsen[16] speculated that the anterior sliding movements between the vomer and the ethmoid bone, and between the vomer and the premaxilla/maxilla are quite likely to occur to allow the pronounced forward growth of the maxillary complex relative to the cranial base which takes place during development.

On lateral cephalogram, the total facial height and the posterior facial height were found to be significantly decreased in the experimental group compared to the control group; findings which are in agreement with other studies.[14],[15] This was attributed to the fact that the upper part of the face of parents (of children with cleft lip and palate group) had smaller outer dimensions.

It was also observed that upper facial height was found to be significantly decreased in the experimental group. The same has been reported by a majority of studies.[13],[14],[15],[17] This decrease in total anterior face height, upper anterior face height and posterior face height points towards a generalized tendency for a smaller face in the experimental group. In coordination with this skeletal finding, the soft tissue total and lower anterior facial height were also found to be decreased in the experimental group.

The protruded maxilla and short maxillary height cause the maxilla to be placed more upward and forward. Thereby, tipping the palatal plane down posteriorly and causing the mandible to rotate clockwise. We also found an articular angle to be increased in the experimental group. These three factors together contribute to the clockwise rotation of the mandible. Hence, an increased gonial angle was found to be significantly increased in the experimental group of our study. This increase has also been observed in other studies.[15],[17] Similar to the gonial angle, the Y-axis angle was also found to be significantly increased in the experimental group. This finding is again in coordination with an increase in gonial angle.

The mandibular length was found to be significantly decreased in our experimental group. Lewin HS in his cephalometric study of analysis of the middle third of the face found smaller mandibles in patients affected with cleft lip and palate.[18] The similarity between findings in parents and children could be an influencing factor in decreasing the threshold for expression of a cleft in their children. If we consider relating mandibular growth to the descent of the tongue for palatal fusion to occur; in our study, the mandibular length that is short would cause overall shortening of the mandible. This disproportion in mandibular size and tongue may hinder the normal descent of the tongue and hence the fusion of palatal shelves. In our study, ramus flexion angle and Y-axis length were found to be significantly decreased in our experimental group.

PA cephalograms were used in this study because this image demonstrates transverse and vertical dimensions of the cranial vault and the upper and middle thirds of the face. In our study, cranial width was found to be significantly decreased in parents of children with BCLP as compared to parents of children without any craniofacial anomaly. It was also found to be significantly smaller in other studies.[14],[15] This could be a manifestation of the smaller size of the cranial base in their parents found in our study and can be considered to be a genetic predisposition leading to the production of a potential cleft lip and palate anomaly.

Bizygomatic width was found to be significantly decreased in parents of children with BCLP as compared to parents of children with no craniofacial anomaly. Nakasima and Ichinose[14] with their large sample size have also reported significantly narrower bizygomatic width in this sample of parents of children with cleft lip and palate. It was also reported to be smaller by Raghavan et al.[15]

Interorbital width (lateral border of the orbit) has been shown to be increased in the parental groups by Sato.[17] However, in our study, it was found to be significantly decreased in parents of children with BCLP as compared to parents of children with no craniofacial anomaly.

Maxillary width was found to be decreased but not statistically significant in the experimental group of our study. Athanasiou, et al.[19] in their longitudinal study of dental arch dimensions in BCLP patients found decreased transverse dimension in their experimental sample of parents of BCLP patients. Hence, a similar occurrence in our experimental group of parents of children suggests a genetic predisposition for the development of cleft in children. This statistically significant difference in facial dimensions of parents of children with BCLP could represent the fact that BCLP patients have smaller supero-lateral dimensions. This morphological feature of disproportion in the relative size of the cranium, maxilla and mandible could lead to failure of the tongue to descend down and palatal shelves to fuse as the tongue might get trapped. This intrinsic tissue deficiency could be attributed to a possible predisposing factor to clefting in their offspring.

Nasal width was assessed in our study and no statistically significant difference was observed in parents of children with BCLP as compared to parents of children with no craniofacial anomaly. According to several authors, a larger nasal width has been reported which is in contrast to the results of this study.[12],[14],[15],[17]

Bigonial width in the present study was found to be decreased, but not significant statistically in parents of children with BCLP as compared to parents of children with no craniofacial anomaly. Other studies also found a significant decrease in bigonial width in the experimental group of parents of children with cleft.[12],[15] This could be due to a generalized decrease in the craniofacial dimensions.

Total craniofacial height was found to be decreased in parents of children with BCLP as compared to parents of children with no craniofacial anomaly, though not statistically significant. It was also found to be shorter in their various experimental groups of cleft types by Nakasima and Ichinose.[14]

The present study boasts of a homogenous sample of BCLP in the North-Indian population. However, the study has limitations in terms of its small sample size. Questions may also be raised regarding the ethical justification of using lateral cephalograms in the parents. While it is acknowledged that the radiographic exposure is to be kept to the minimum, previous research trends in this particular area[6],[20] indicate that a voluntary informed consent justifies the usage of lateral cephalograms for a better understanding of the dentocraniofacial features that may predispose certain set of parents to produce offsprings with cleft deformities. It may also be noted that the usage of digital radiographic methods has further allowed for a reduction of the effective radiation dose compared to the methods used in the previous studies. In future studies, non-radiographic 3-D imaging techniques may be considered for further elimination of radiographic exposure.


   Conclusion Top


Significant differences between parents of children with BCLP and parents of children with no craniofacial anomaly exist in the cephalometric parameters, thereby, suggesting a possibility of dentocraniofacial features contributing to BCLP in their children. Therefore, these dentocraniofacial morphological features in parents of children with BCLP could act as prediction factors like risk of occurrence of BCLP in their offspring. However, further research is required to come to a definite conclusion.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Dahl E, Kreiborg S, Jensen BL. Roentgencephalometric Studies of Infants with Untreated Cleft Lip and Palate. What Is a Cleft Lip and Palate? A Multidisciplinary Update. Vol 7. Stuttgart: Georg Thieme Verlag; 1989. p. 113-5.  Back to cited text no. 1
    
2.
Jugessur A, Shi M, Gjessing HK, Lie RT, Wilcox AJ, Weinberg CR, et al. Genetic determinants of facial clefting: Analysis of 357 candidate genes using two national cleft studies from Scandinavia. PLoS One 2009;4:e5385.  Back to cited text no. 2
    
3.
Mossey PA, McColl JH, Stirrups DR. Differentiation between cleft lip with or without cleft palate and isolated cleft palate using parental cephalometric parameters. Cleft Palate Craniofac J 1997;4:27-35.  Back to cited text no. 3
    
4.
Rahimov F, Marazita ML, Visel A, Cooper ME, Hitchler MJ, Rubini M, et al. Disruption of an AP-2alpha binding site in an IRF6 enhancer is associated with cleft lip. Nat Genet 2008;40:1341-7.  Back to cited text no. 4
    
5.
Mossey PA, Arngrimsson R, McColl J, Vintiner GM, Connor JM. Prediction of liability to orofacial clefting using genetic and craniofacial data from parents. J Med Genet 1998;35:371-8.  Back to cited text no. 5
    
6.
Mossey PA, Batra P, McIntyre GT. The parental dentocraniofacial phenotype-an orofacial clefting microform. Cleft Palate Craniofacial J 2010;47:23-34.  Back to cited text no. 6
    
7.
da Silva Filho OG, Carvalho Lauris RC, Capelozza Filho L, Semb G. Craniofacial morphology in adult patients with unoperated complete bilateral cleft lip and palate. Cleft Palate Craniofac J 1998;35:111-9.  Back to cited text no. 7
    
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Foster TD, Lavelle CL. The size of the dentition in complete cleft lip and palate. Cleft Palate J 1971;8:177-84.  Back to cited text no. 8
    
9.
Liao Y-F, Mars M. Hard palate repair timing and facial morphology in unilateral cleft lip and palate: Before versus after pubertal peak velocity age. Cleft Palate Craniofac J 2006;43:259-65.  Back to cited text no. 9
    
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Ranta R. A review of tooth formation in children with cleft lip/palate. Am J Orthod Dentofacial Orthop 1986;90:11-8.  Back to cited text no. 10
    
11.
Perkiomaki MR, Yoon Y-J, Tallents RH, Barillas I, Herrera-Guido R, Moss ME, et al. Association of distinct craniofacial features in nonsyndromic cleft lip and palate family members. Cleft Palate Craniofac J 2003;40:397-402.  Back to cited text no. 11
    
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AlEmran SE, Fatani E, Hassanain JE. Craniofacial variability in parents of children with cleft lip and cleft palate. J Clin Pediatr Dent 1999;23:337-41.  Back to cited text no. 12
    
13.
Coccaro PJ, D'Amico R, Chavoor A. Craniofacial morphology of parents with and without cleft lip and palate children. Cleft Palate J 1972;9:28-38.  Back to cited text no. 13
    
14.
Nakasima A, Ichinose M. Size of the cranium in parents and their children with cleft lip. Cleft Palate J 1984;21:193-203.  Back to cited text no. 14
    
15.
Raghavan R, Sidhu SS, Kharbanda OP. Craniofacial pattern of parents of children having cleft lip and/or cleft palate anomaly. Angle Orthod 1994;64:137-44.  Back to cited text no. 15
    
16.
Melsen B. Histological analysis of the postnatal development of the nasal septum. Angle Orthod 1977;47:83-96.  Back to cited text no. 16
    
17.
Sato T. [Craniofacial morphology of parents with cleft lip and palate children]. Shikwa Gakuho 1989;89:1479-506.  Back to cited text no. 17
    
18.
Levin HS. A cephalometric analysis of cleft palate deficiencies in the middle third of the face. Angle Orthod 1963;33:186-94.  Back to cited text no. 18
    
19.
Athanasiou AE, Maaheri M, Zarrinnia K. Longitudinal study of the dental arch dimensions in bilateral cleft lip and palate patients. J Pedod 1987;11:253-68.  Back to cited text no. 19
    
20.
Mossey PA, McColl J, O'Hara M. Cephalometric features in the parents of children with orofacial clefting. Br J Oral Maxillofac Surg 1998;36:202-12.  Back to cited text no. 20
    

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Correspondence Address:
Dr. Anubhuti Sood
Clinical Research Officer, Room No-352, O.P.D Block, Department of Paediatrics, Vardhaman Mahavir Medical College and Safdarjung Hospital, Delhi - 110 029
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijdr.ijdr_623_21

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