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 Table of Contents  
ORIGINAL ARTICLE
Year : 2020  |  Volume : 7  |  Issue : 2  |  Page : 89-98

Cephalometric evaluation of skeletal base relationship in patients with cleft lip and palate in a tertiary hospital in South India


1 Department of Orthodontics and Dentofacial Orthopedics, Amrita School of Dentistry, Kochi, Kerala, India
2 Department of Oral and Maxillofacial Surgery, Amrita School of Dentistry, Kochi, Kerala, India

Date of Submission22-Jan-2020
Date of Acceptance04-Jun-2020
Date of Web Publication31-Jul-2020

Correspondence Address:
Dr. Meguja K Johnson
Department of Orthodontics and Dentofacial Orthopedics, Amrita School of Dentistry, Kochi - 682 041, Kerala
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jclpca.jclpca_2_20

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  Abstract 


Objective: The study is aimed at assessing the cephalometric characteristics and distribution of skeletal base patterns in patients with cleft lip and palate (CLP) in a cohort of patients reporting to a tertiary hospital in South India. Materials and Methods: A retrospective observational study was carried out on 44 consecutive patients with CLP and 43 age- and sex-matched control orthodontic patients. Linear and angular measurements were obtained from the digitally traced lateral cephalograms using Dolphin software, and the data were analyzed. Results: Patients with CLP exhibit reduction in maxillary as well as mandibular length. Maxilla and mandible are both retropositioned and retrognathic. However, the maxillomandibular relationship shows the prevalence of Class I skeletal pattern (47.73%). Conclusion: Patients with CLP demonstrated smaller, retrognathic and retropositioned jaws, however with Class I skeletal relationship. The present data would be beneficial in determining the orthodontic requirements of patients with cleft in South India.

Keywords: Cephalometry, cleft lip, cleft lip and palate, cleft palate, craniofacial growth, craniofacial morphology, maxillary retroposition


How to cite this article:
Johnson MK, Kuriakose MJ, Varma N K, Ajith V V, Subash P. Cephalometric evaluation of skeletal base relationship in patients with cleft lip and palate in a tertiary hospital in South India. J Cleft Lip Palate Craniofac Anomal 2020;7:89-98

How to cite this URL:
Johnson MK, Kuriakose MJ, Varma N K, Ajith V V, Subash P. Cephalometric evaluation of skeletal base relationship in patients with cleft lip and palate in a tertiary hospital in South India. J Cleft Lip Palate Craniofac Anomal [serial online] 2020 [cited 2020 Dec 1];7:89-98. Available from: https://www.jclpca.org/text.asp?2020/7/2/89/291137




  Introduction Top


Cleft lip and palate (CLP) is the second-most common developmental disorder, which constitutes 11%–15% of all congenital anomalies.[1] It is the most common congenital disability of the head-and-neck region with a highly variable incidence among populations, demonstrating the highest incidence in Asians and Native Americans (1/700 live births) and the lowest incidence in Africans (1/2500 live births).[2]

Midface deficiency has been frequently reported in patients with CLP.[3] Gesch et al. (Germany, 2006) and Goyenc et al. (Turkey, 2008) stated that patients with unilateral CLP present maxillary retroposition and mandibular deficiency.[4],[5] A study in the Mexican population (2015) revealed that most patients with CLP presented a skeletal Class III maxillomandibular relation, a large mandibular body length, an increase in the vertical growth of the maxilla.[6] Ana Lopez-Gimenez et al. who studied the Spanish population also observed the prevalence of skeletal Class III pattern in the patients with cleft.[7] Chaisrisookumporn et al. (Texas, 1995) showed that there was no statistically significant difference in skeletal parameters between patients with unilateral and bilateral cleft.[8] Ethnic variation is seen in the craniofacial morphologies of patients with CLP across the globe.[3],[4],[5],[6],[7] Khanna et al. (Uttar Pradesh, 2020), while studying growth patterns in patients with an operated cleft in two age groups (mean age 9 and 15), found that in both groups, the skeletal relationship was Class III.[9] However, no cephalometric study could be traced in the literature on South Indian patients with cleft, which could provide insight into their skeletal base relationship and characteristics. The multiracial and multiethnic population in India makes the cephalometric norms of North India incomparable with that of South India.[10] The success of the various and prolonged treatment modalities which the patient undergoes is influenced by the skeletal pattern of the patients.

This study is, hence, aimed at assessing cephalometric characteristics and the distribution of skeletal base pattern (Class I, Class II, and Class III) in a cohort of patients with cleft reporting to a tertiary health Centre in South India.


  Materials and Methods Top


This was a retrospective observational study carried out in a tertiary hospital in South India. The data were obtained from prospectively collected electronic medical records of a CLP clinic. Patients who met the following strict inclusion and exclusion criteria were included in the study.

Inclusion criteria

  • Patients with CLP
  • Patients with South Indian ancestors for at least one previous generation
  • Patients who visited the tertiary health center from 2010 to 2018
  • Patients aged between 9 and 16.


Exclusion criteria

  • Patients with cleft lip or cleft palate alone
  • Patients with other known congenital syndromes or anomalies (ascertained by a clinical geneticist in the same hospital)
  • Patients of non-South Indian origin
  • Patients who have had orthodontic treatment initiated or completed
  • Patients with poor quality lateral radiographs.


Forty-four patients who met the eligibility criteria were included in the study. This consisted of 22 females and 22 males, including 11 bilateral CLP patients and 33 unilateral CLP patients. The mean age of the patients with cleft included in the study was 12.18 ± 2.77 years (male 12.09 ± 2.89 and female 12.27 ± 2.71). All the patients had undergone lip and palate surgery at the appropriate age. In addition, 43 age- and sex-matched patients (without cleft) who have undergone lateral cephalometric study were included as control group. This included 22 females and 21 males. The mean age of the control group was 13.35 ± 1.99 years (male 13.09 ± 2.09 and female 13.59 ± 1.89). All the lateral cephalograms of both study and control groups were taken before the initiation of any orthodontic treatment.

Cephalometric analysis

Fifteen cephalometric landmarks were used for the study. Six angular measurements and 12 linear measurements [Table 1] were obtained using these landmarks on the Dolphin imaging system (version 11.95, Chatsworth, Los Angeles, CA, USA), and the data were analyzed.
Table 1: Maxilla-Cranial base relationship

Click here to view


Cephalometric analysis was divided into three parts as follows: (1) Maxilla to cranial base relationship (2) mandible to cranial base relationship and (3) maxilla to mandible relationship. The angular and linear measurements were taken from different analyses such as Wits analysis, Steiner's analysis, McNamara analysis, Bjork analysis, Rakosi-Jarabak analysis, Cephalometrics for Orthognathic Surgery analysis, and Downs analysis designed for the Caucasian population but commonly used for the Indian population. Significant statistical differences between the norm and the measurements obtained were ascertained. Furthermore, comparison of the measurements for patients with cleft and the control patients was made to determine if there was a statistically significant difference. The lateral cephalogram of each patient was redigitized at a 1 month interval to avoid any significant observer bias. The mean value of the two measurements was taken for analysis.

Statistical analysis

Statistical analysis was performed using IBM SPSS version 20.0 software (IBM SPSS, Chicago, Illinois, USA). Categorical variables are expressed using frequency and percentage. Numerical variables are presented using mean and standard deviation. To find out the percentage distribution of skeletal base patterns in CLP patients, descriptive statistics were performed. Independent sample t-test was used to study the statistical significance of all clinical parameters among both the groups. A value of P < 0.05 is considered to be statistically significant.


  Results Top


Each cephalometric parameter of all the patients within the study group was tabulated and compared with that of the control group. The data obtained were separately categorized into different cephalometric subunits and relationship.

Maxilla-cranial base relationship

Eight parameters were assessed for both the study and the control group to evaluate the relationship of maxilla to cranial base [Table 2]A and [Table 2]B. About 61.36% of the total population (patients with cleft) exhibited maxillary retrognathism. About 56.82% of the patients showed reduced N Perp-Pt A value. The retroposition of the maxilla was observed in 95.45% (Basion-Posterior Nasal Spine (Ba-PNS)) to 100% (Basion-Anterior nasal spine (Ba-ANS)) of the cases. 100% of subjects showed a reduction in effective midfacial length. Midfacial height deficiency was observed in 100% of the studied population. The maxillary plane was tipped up in 45.45% and tipped down in 15.91% of the subjects. The effective maxillary length was reduced in 95.45% subjects.
Table 2:

Click here to view


In the control group, only 2.33% exhibited maxillary retrognathism. 27.91% of the patients showed reduced N Perp-Pt A value. Retroposition of the maxilla was observed in 88.37% (Ba-PNS) to 97.67% (Ba-ANS) of the controls. 93.02% of subjects showed a reduction in effective midfacial length. Midfacial height deficiency was observed in 97.67% of the control group. The maxillary plane was tipped up in 76.74%. The effective maxillary length was reduced in 95.35% subjects.

Mandible-cranial base relationship

Seven parameters were assessed for each patient to study the relationship of the mandible to cranial base [Table 3]A and [Table 3]B. In the study group, 47.73% (NPog-FH) to 63.64% (Sella-Nasion-Point B) of the patients exhibited mandibular retrognathism. Reduction in the mandibular length was observed in 100% of the subjects. About 93.18% of the patients with cleft exhibited a reduction in ramal length, while 97.73% exhibited reduced mandibular body length. 45.45% of the patients with cleft showed steep mandibular plane and backward rotation, while only 11.36% showed forward rotation of the mandible.
Table 3:

Click here to view


In the control group, 27.91% (NPog–FH) to 39.53% (N Perp-Pog) exhibited mandibular retrognathism. Reduction in the mandibular length was observed in 74.42% of the subjects. 95.35% of the control group exhibited a reduction in ramal length, while 81.39% exhibited reduced mandibular body length. 18.60% of the control patients showed steep mandibular plane and backward rotation, while only 23.26% showed forward rotation of the mandible.

Maxilla-mandible relationship

Three parameters (ANB, AO-BO, [Co-A] – [Co-Gn]) were assessed for each patient to find out the maxillomandibular relationship [Table 4]A and [Table 4]B. When two of these parameters fell in the same category (class I/II/III), the patient was categorized under that particular skeletal base pattern.
Table 4:

Click here to view


Overall, the study group had a prevalence of Class I Skeletal base by 47.73%, followed by Class III (29.55%) and Class II (22.73%) [Table 5] and [Graph 1]. Within the patients with cleft, Male patients showed the prevalence of Class I skeletal base at 54.55%, while female patients showed the prevalence of Class I skeletal base at 40.91%.
Table 5: Skeletal base relationship

Click here to view



The control group had a prevalence of Class II Skeletal base by 53.49%, followed by Class I (30.23%) and Class III (16.28%) [Table 5] and [Graph 2]. Within the control group, while Male patients showed the prevalence of Class I and Class III skeletal base by 42.86%, female patients showed the prevalence of Class II skeletal base by 63.64%. Percentage distribution of Class I/II/III according to ANB, Wits appraisal and McNamara value are plotted in [Graph 2], [Graph 3], [Graph 4], [Graph 5], [Graph 6], [Graph 7], [Graph 8].



Comparing the measurements between the study group and the control group revealed a statistically significant difference between the two groups in many of the parameters [Table 6].
Table 6: Comparison of various craniofacial morphology parameters

Click here to view



  Discussion Top


The objective of this study was to describe and compare the craniofacial cephalometric morphologies and to find out the distribution of skeletal base relationship in patients with cleft (CLP) reporting to a tertiary hospital in South India. Since it has been shown that no significant difference exists in the skeletal base parameters between the patients with bilateral and unilateral cleft, the data from bilateral clefts were pooled with that of the patients with unilateral cleft.[8] Naduwinmani et al. did a cephalometric comparison between patients with unilateral and bilateral cleft in a south Indian population. All their patients were above 15 years, and they also found no statistically significant difference between unilateral and bilateral patients when comparing the skeletal parameters. Their study showed maxillary deficiency in patients with cleft, but maxillary and mandibular length ratio was not statistically different from normal.[11]

Ross stated that patients with CLP show a delay in maxillary growth, which results in maxillary retrusion.[12] Moss and Coupe and Subtelny suggested that as an inherent consequence of the initial malformation, there was maxillary deficiency.[13],[14] Substantiating the findings of Moss and Coupe and Subtelny, the present study also proved the retroposition of the maxilla in patients with CLP. The present study also observed decreased midfacial length (100%) and overall maxillary retrognathism (61.36%).

Bishara and Dahl et al. reported the tendency towards bimaxillary retrognathia in patients with cleft.[15],[16] However, in 1982 Long et al. hypothesized that the mandibular size increases in patients with CLP to compensate for the maxillary deficiency.[17] Contradicting it, Eriksen, et al. has observed significantly shorter mandibular length in patients with more extensive fissures.[18] In the current study, reduction in the mandibular length was observed in 100% of the patients, and overall mandibular retrognathism was observed in 47.73% to 63.64% of the patients with CLP.

A Mexican study (2015) revealed that most of the subjects with cleft presented a skeletal Class III maxillomandibular relation.[6] A Spanish study (2018) also observed the prevalence of skeletal Class III patterns in patients with Unilateral CLP and skeletal Class I in patients with Bilateral CLP.[7] In contrast to those studies, this study in South Indian patients with cleft showed prevalence of Class I skeletal base pattern, followed by Class III and Class II skeletal base.

The control group also showed a reduction in all the cephalometric values compared to the standard norms. This could be due to the ethnic variation, as proved by the previous literature.[19],[20] It could also be the result of selecting the control group from patients who have undergone lateral cephalometric study. Even though the control group exhibited decreased values from the standard norms, there was a statistically significant difference between the two groups in many of the parameters studied. The control patients showed the prevalence of Class II skeletal base pattern, followed by Class I and Class III skeletal base.


  Conclusion Top


  • Maxillary and mandibular lengths are significantly reduced in the patients with CLP
  • Maxilla (61.36%) and mandible (63.64%) are retrognathic and are retropositioned (95.45%–100%) in most of the cases
  • Even though the maxilla and mandible show reduction in size and are retrognathic and retropositioned, the maxillomandibular relationship shows a prevalence of 47.73% Class I skeletal pattern.


The prognosis for the treatment outcome of patients with cleft with a Class I skeletal base relationship is good. Since the skeletal base relationship is Class I, relapse tendency will be less at the completion of treatment. With both maxilla and mandible being retropositioned and retrognathic, but in a Class I skeletal base relation, the management of patients with cleft could involve a bi-jaw surgery when carrying out the skeletal correction. This information will be helpful in planning the potential treatment requirements in patients with cleft in South India.

Scope for future studies

A similar study can be carried out on a larger sample size. Setting up cephalometric standards for patients with cleft in India would enable us to accumulate information to redefine the treatment protocols and help treat every person according to the set standards.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Holst AI, Holst S, Nkenke E, Fenner M, Hirschfelder U. Vertical and sagittal growth in patients with unilateral and bilateral cleft lip and palate-a retrospective cephalometric evaluation. Cleft Palate Craniofac J 2009;46:512-20.  Back to cited text no. 1
    
2.
Toro-Ibacache V, Araya JC, Muñoz AD, Soto GM. Morphologic variability of nonsyndromic operated patients affected by cleft lip and palate: A geometric morphometric study. Am J Orthod Dentofacial Orthop 2014;146:346-54.  Back to cited text no. 2
    
3.
Liu R, Lu D, Wamalwa P, Li C, Hu H, Zou S. Craniofacial morphology characteristics of operated unilateral complete cleft lip and palate patients in mixed dentition. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2011;112:e16-25.  Back to cited text no. 3
    
4.
Gesch D, Kirbschus A, Mack F, Gedrange T. Comparison of craniofacial morphology in patients with unilateral cleft lip, alveolus and palate with and without secondary osteoplasty. J Craniomaxillofac Surg 2006;34 Suppl 2:62-6.  Back to cited text no. 4
    
5.
Goyenc YB, Gurel HG, Memili B. Craniofacial morphology in children with operated complete unilateral cleft lip and palate. J Craniofac Surg 2008;19:1396-401.  Back to cited text no. 5
    
6.
Burak MY, Ponglertnapakorn A, Calderón EG. Analysis of the cephalometric skeletal and dental characteristics of adult patients with cleft lip and palate who received orthopedic, orthodontic and/or surgical treatment during their childhood and adolescence. Rev Mex Ortod 2015;3:e22-32.  Back to cited text no. 6
    
7.
Lo×Pez-Gime×Nez A, Silvestre-Rangil J, Silvestre FJ, Paredes-Gallardo V. Craniofacial cephalometric morphologies in different cleft types: A retrospective cross-sectional study of 212 patients. Oral Radiol 2018;34:127-35.  Back to cited text no. 7
    
8.
Chaisrisookumporn N, Stella JP, Epker BN. Cephalometric profile evaluations in patients with cleft lip and palate. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1995;80:137-44.  Back to cited text no. 8
    
9.
Khanna R, Tikku T, Verma SL, Verma G, Dwivedi S. Comparison of maxillofacial growth characteristics in patients with and without cleft lip and palate. J Cleft Lip Palate Craniofacial Anomalies 2020;7:30-42.  Back to cited text no. 9
    
10.
Kharbanda OP. Epidemiology of malocclusion and orthodontic treatment needs. In: Orthodontics: Diagnosis of and Management of Malocclusion and Dentofacial Deformities. 2nd ed. 2013: Elsevier: India, New Delhi; p. 18-31.  Back to cited text no. 10
    
11.
Naduwinmani JP, Hallolli C, Naduwinmani SL. Cephalometric profile evaluation in patients with cleft lip and palate. Int J Contemp Dent 2011;2:63-9.  Back to cited text no. 11
    
12.
Ross RB. Cranial base in children with lip and palate clefts. Cleft Palate J 1965;31:157-66.  Back to cited text no. 12
    
13.
Moss ML. Malformations of the skull base associated with cleft palate deformity. Plast Reconstr Surg 1956;17:226-34.  Back to cited text no. 13
    
14.
Coupe T, Subtelny J. Cleft palate-deficiency or displacement of tissue? Plast Reconstr Surg 1960;26:600-12.  Back to cited text no. 14
    
15.
Bishara SE. Cephalometric evaluation of facial growth in operated and non-operated individuals with isolated clefts of the palate. Cleft Palate J 1973;10:239-46.  Back to cited text no. 15
    
16.
Dahl E, Kreiborg S, Jensen BL, Fogh-Andersen P. Comparison of craniofacial morphology in infants with incomplete cleft lip and infants with isolated cleft palate. Cleft Palate J 1982;19:258-66.  Back to cited text no. 16
    
17.
Long RE Jr., Jain RB, Krogman WM. Possible sex-discriminant variables in craniofacial growth in clefting. Am J Orthod 1982;82:392-402.  Back to cited text no. 17
    
18.
Eriksen J, Hermann NV, Darvann TA, Kreiborg S. Early postnatal development of the mandible in children with isolated cleft palate and children with nonsyndromic Robin sequence. Cleft Palate Craniofac J 2006;43:160-7.  Back to cited text no. 18
    
19.
Govinakovi PS, Al-Busaidi I, Senguttuvan V. Cephalometric norms in an Omani adult patients of Arab Descent. Sultan Qaboos University Med J 2018;18:182-9.  Back to cited text no. 19
    
20.
Wu J, Hägg U, Rabie AB. Chinese Norms of McNamara's cephalometric analysis. Angle Orthodont 2007;77:12-20.  Back to cited text no. 20
    



 
 
    Tables

  [Table 1], [Table 2], [Table 3], [Table 4], [Table 5], [Table 6]



 

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