Introduction

Cleft lip and palate is one of the most common congenital anomaly that occurs in humans. In India, the incidence of cleft lip and or palate ranges from 0.25 to 1.56 per1000 live births.1 Non-syndromic orofacial clefts, which include cleft lip, cleft lip and palate, and cleft palate alone, comprise a range of disorders affecting the lips and oral cavity, the causes of which remain largely unknown. Effects on speech, hearing, appearance, and cognition can lead to long-lasting adverse outcomes for health and social integration. Affected children need multidisciplinary care from birth until adulthood and have higher morbidity and mortality throughout life than unaffected individuals. Although rehabilitation is possible with good quality care, orofacial clefts inevitably pose a burden to the individual, the family, and society, with substantial expenditure in terms of health and related services.

Care for children born with these defects is multidisciplinary and includes many disciplines such as nursing, plastic surgery, maxillofacial surgery, otolaryngology, speech therapy, audiology, counselling, psychology, genetics, orthodontics, and dentistry but it forms only a part of the clinical load of every area. This fragmentation of care has led to substantial variations in management, which continue to cause controversy. Furthermore, in both developing and developed countries, standards of care for patients with cleft lip, cleft lip and palate, or cleft palate alone remain a cause for concern. 2 Success in dento-facial orthopaedics is highly dependent on the patient’s maturational stage. Therefore, it is important to determine the period of accelerated growth of the relevant skeletal structure to achieve the most favourable response with the least potential morbidity.

Hassel and Farman in 1995 compiled a new Cervical Vertebral Maturation index (CVMI) which was based on the morphological changes in the lateral profile of C2, C3, and C4 cervical vertebrae as these areas can be visualized even in presence of a thyroid collar worn during radiation exposure. 3 The advantage of CVMI is that skeletal maturity can be assessed by lateral cephalometric radiograph taken as a part of routine investigation for orthodontic diagnosis and treatment planning. This study was conducted to evaluate differences in the cervical vertebrae skeletal maturation in cleft and non-cleft male and female individuals with respect to age using CVMI staging.

Materials and Methods

A retrospective study was conducted at Maulana Azad Institute of Dental Sciences, New Delhi for a period of eighteen months. Hundred and two lateral cephalograms of subjects within the age range of nine to eighteen years were examined to evaluate differences in the cervical vertebrae skeletal maturation. Patients with non-syndromic unilateral/bilateral cleft lip and palate and those without cleft and other systemic disorders were analysed. Lateral cephalograms taken in abnormal neck position, birth defects or those with growth abnormality were excluded from our study. Lateral cephalograms of the selected subjects were mounted on the X-Ray viewer and visual assessment of the skeletal maturity was performed with the Cervical Vertebral Maturation index given by Hassel and Farman.3 The Cervical Vertebral maturation method constitutes analysing the morphology of the bodies of the second (C2- the odontoid process), third (C3) and the fourth (C4) cervical vertebrae. This includes presence or absence of a concavity at the lower border of the body of C2, C3, and C4 and Shape of the body of C3 and C4. Cervical vertebral maturation stages are distinguished into six stages based on these parameters. (Figure 1)

The chronological age in whole years for each subject was calculated by subtracting date of birth from date on the radiograph. All the lateral cephalograms were divided into cleft and non cleft group. The non cleft group was considered as Group 1 and the cleft group was considered as Group 2. These groups were further subdivided into A and B based on the age range. Subjects from 9 to 13 years were considered as Group A and subjects from 14-18 years of age were considered as Group B. A number was allotted for the subdivided groups for further segregation of males and females (1 for males and 2 for females). (Figure 2)

Intra examiner reliability was tested by randomly selecting 35 lateral cephalometric radiographs and analysing them for skeletal maturation on two separate occasions after a two-week interval. Analysis was done using IBM SPSS Version 16.0. Mean and standard deviation was expressed for quantitative data and percentages for qualitative data. The comparison of CVMI stages for each group was done with Chi-square test. A p-value of less than 0.05 was considered as statistically significant.

Results

Fifty-one participants each with and without cleft-palate and lip were recruited in our study. Forty-nine (48%) participants were aged between 9 to 13 years, while 53 (52%) participants were aged between 14 to 18 years. Fifty-nine (58%) participants were male while 43 (42%) participants were female. The study participants were divided into groups based on age, gender and presence of cleft lip or palate.

At the age group of 9-13 years, comparison of CVMI status between cleft and non-cleft subjects revealed a statistically significant difference. Out of 22 cleft subjects only 1 subject was in CVMI 3 while the others were in CVMI i1 and 2. For the non-cleft group, there was equal distribution of subjects in CVMI 2 and 3. Few subjects were also found to be in CVMI 4 compared to no subjects in cleft group indicating a delay in skeletal maturity.

At the age group of 14-18 years, comparison of CVMI status between cleft and non-cleft subjects revealed a statistically significant difference. Out of 29 cleft subjects 6 subjects were in CVMI 3 while only 1 subject was found in CVMI 3 of the non-cleft group. Equal distribution was observed in the CVMI 4 and 5 of cleft and non-cleft groups. More disparity was found in CVMI 3.

Comparison of cleft and non-cleft males at the age range of 9-13 years showed that the skeletal maturation of non-cleft (Group 1 male) and cleft (Group2 male) subjects was statistically significantly different (p value <0.05). Six subjects of the cleft group (1A1) were found to be in initiation stage compared to no subject of the non-cleft group (2A1) in initiation. While nine subjects of the non-cleft group were found to be in transition stage compared to no subject of the cleft group. Comparison of cleft and non-cleft females at the age range of 9-13 years showed that the skeletal maturation of non-cleft (Group 1 male) and cleft (Group 2 male) subjects was statistically significantly different (p value <0.05). Three subjects of the non-cleft group were found to be in Deceleration stage compared to no subject of the cleft group in initiation. While six subjects of the cleft group were found to be in acceleration stage compared to three subjects were found in the non-cleft group.

An overall statistically significant difference in the CVMI stages of Group 1 and Group 2 subjects was observed between 9-18 years of age. There was a statistically significant difference in the CVMI stages of Group 1 and Group 2 subjects between 9-13 years of age. Results revealed that in the transition stage 91.7% of subjects were in the non-cleft group whereas only 8.3% of subjects were found in the cleft group showing a delay in skeletal maturation. The comparison of CVMI stages of Group 1 and Group 2 for both males and females between 14-18 years of age was statistically insignificant. The comparison of CVMI stages of Group 1 and Group 2 for males was statistically significantly different whereas the comparison for females was statistically insignificant. There was a statistically significant difference in the CVMI stages of Group 1 and Group 2 for males between 9-13 years of age. Transition stage 100% of subjects were in non-cleft group compared to no subjects in the cleft group. There was a statistically significant difference in the CVMI stages of Group 1 and Group 2 for females between 9-13 years of age. Results revealead that in deceleration stage 100% of subjects were in non-cleft group compared to no subjects in the cleft group. The difference in the CVMI stages of Group 1 and Group 2 males and females between 14-18 years of age was statistically insignificant.

Figure 1

Figure 2

Discussion

Assessment of skeletal age is part of the biological age assessment and is vital to orthodontic treatment planning. One of the most important issues related to cleft individuals is their skeletal maturation patterns. There are substantial growth impairments and skeletal disproportions associated with the CLP anomaly. 4, 5, 6 Maxillary retrusion, class III skeletal disproportion, and crossbites (anterior and/or posterior) are common findings in CLP patients. 5 In order to correct these discrepanices assessment of growth status is important to predict their growth potential. Only a few studies have addressed skeletal maturation in CLP participants in comparison with a control population and their findings are limited to the gender and ethnicity studied. 7, 8, 9 A study by Ross et al 1987 reported that the skeletal age is retarded in cleft children and the cleft children are shorter and lighter than that of control children. 10

The authors suggested the reason for height-weight retardation to be due to feeding problems and heightened frequency of infections. 11 Some investigators have suggested that this diminution resulted from events in adolescence (endocrine controls of maturation at puberty) and not the result of feeding difficulties, infections, or surgical interventions experienced in the months immediately following birth. 12 Rudman and Davis et al found that heights of cleft children to be below the 3rd percentile for that age group and suggests that children with cleft lip or cleft palate are forty times more likely to experience growth hormone deficiency than the non-cleft children. These studies emphasize the importance of treatment timing in cleft individuals. 13

In the current study, the collected sample consisted of age matched cleft and non-cleft subjects ranging from 9 to 18 years. This ensured equal distribution age wise, which helped accurately identify the pubertal growth spurt in both groups. The results of the present study showed that the cleft subjects had a delay in skeletal maturity compared to non-cleft subjects, irrespective of the gender. Bowers and Rosario et al found that specifically males with unilateral cleft lip and palate and isolated cleft palate were significantly shorter and thinner (reduced Body Mass Index) than normal, whereas females with isolated cleft palate differed from normal only in their shorter height. 14

At the age of 9-13 years, girls with UCLP are observed to attain skeletal maturation faster than boys with UCLP of similar age. Sun and Li reported similar significant difference in cleft boys where the authors observed that boys with cleft lip and or palate were at a higher risk of delayed growth period and retarded pubertal growth peak. 9 In another study conducted by Sun and Li assessed the skeletal maturation in Chinese females with cleft and noticed a delay in skeletal maturity in cleft patients.

Comparison of cleft and non-cleft males and females at the age range of 14-18 years showed that the skeletal maturation of non-cleft (Group 1 male) and cleft (Group 2 male) subjects was not statistically significantly different. The present study showed that children with UCLP of 14 to 18 years of age group, irrespective of the gender showed faster rate of skeletal maturation than non-cleft children, but the difference was statistically insignificant.

The findings of the study also suggest that in 9 to 13 years age group there is delay in skeletal maturation among children with cleft; where as in 14 to 18 years age group skeletal maturation was found to be comparable among children with cleft and non-cleft children. Possible reason could be the adolescent catch-up growth seen among the cleft children. The total growth period is longer among cleft children and thus they are able to catch up with the normal control group.

Our study included 9 to 18 year old participants with an outcome that indicated a highly statistically significant difference between the skeletal maturation of cleft and non-cleft subjects at the age range of 9-13 years confirming that participants with the cleft anomaly are more likely to have a decelerated pubertal spurt and a delayed pubertal peak. This proved the implication of cleft anomalies in growth patterns, which has been proposed by many investigators. 4, 15

Conclusion

Cleft patient treatment aims to address skeletal and dental disharmony through multidisciplinary care, where skeletal discrepancies in children with CLP may require orthopedic and/or surgical correction^. Generally, orthodontic treatment and intervention are timed to take place before or during the peak growth velocity or pubertal growth spurt to achieve favorable effects in correcting sagittal, transverse, and vertical plane disharmonies. In this study our aim was to evaluate differences in the cervical vertebrae skeletal maturation in cleft and non-cleft male and female individuals with respect to age. The conducted study indicates statistically significant differences between the skeletal age and chronological age in cleft and non-cleft subjects from the age group of 9-18 years.

Conflict of Interest

None.

Source of Funding

This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.