|Year : 2014 | Volume
| Issue : 1 | Page : 43-47
Association of cervical vertrebra anomalies with cleft lip and palate
Mudita Srivastava1, Anshul Aggarwal2, Puneet Batra3, Sangeev Datana4, Prasanna Kumar5, Klara Agneta Macrcusson6
1 Senior lecturer; Postgraduate, Institute of Dental Sciences and Technology, Modinagar, Uttar Pradesh, India
2 Postgraduate, Institute of Dental Sciences and Technology, Modinagar, Uttar Pradesh, India
3 Postgraduate; Professor and Head, Institute of Dental Sciences and Technology, Modinagar, Uttar Pradesh, India
4 Associate Professor, Army Dental Centre, New Delhi, India
5 Professor, Army Dental Centre, New Delhi, India
6 Professor and Head, Maxillofacial Unit, University Hospital, Linkoping, Sweden
|Date of Web Publication||5-Feb-2014|
Department of Orthodontics and Dentofacial Orthopedics, Institute of Dental Science and Technology, Modinagar, Uttar Pradesh
Source of Support: None, Conflict of Interest: None
Introduction: The aim of the present study was to evaluate the developmental relationship in the vertebral column in the cervical region and to relate its association in patient's with cleft lip and palate. Materials and Methods: The sample consisted of 228 cleft patients in the experimental group and 225 non cleft samples in control group. Cleft patients are subdivided into cleft palate only (CPO), Unilateral cleft lip and palate (UCLP) and bilateral cleft lip and palate (BCLP). Cervical vertebrae were traced on lateral cephalogram in children with cleft lip and palate to determine the prevalence of vertebral anomalies and their categorization. The cervical vertebral anomalies were classified into two types: posterior arch deficiency (PAD) and fusion anomalies (FUS). Results and Conclusion: The study showed 19 percent of cleft patients with cervical vertebral anomalies and 4 percent of control group with cervical vertebral anomalies. Posterior arch deficiency occurred more frequently in unilateral cleft lip and palate(UCLP) and cleft palate only (CPO), Fusion occurred significantly more often in bilateral cleft lip and palate (BCLP).
Keywords: Cervical vertebral anomalies, cleft lip and palate, fusion
|How to cite this article:|
Srivastava M, Aggarwal A, Batra P, Datana S, Kumar P, Macrcusson KA. Association of cervical vertrebra anomalies with cleft lip and palate. J Cleft Lip Palate Craniofac Anomal 2014;1:43-7
|How to cite this URL:|
Srivastava M, Aggarwal A, Batra P, Datana S, Kumar P, Macrcusson KA. Association of cervical vertrebra anomalies with cleft lip and palate. J Cleft Lip Palate Craniofac Anomal [serial online] 2014 [cited 2021 Jan 25];1:43-7. Available from: https://www.jclpca.org/text.asp?2014/1/1/43/126561
| Introduction|| |
The cervical spine has been an interest area for the orthodontist to examine in routine lateral cephalograms. This is because of fact that the lateral cephalograms using the cervical spine as the reference structure  has been commonly used to assess natural head position. Skeletal age estimation can be made by the cervical spine method, which can be used in predicting growth potential for planning orthodontic treatment.  Anomalies of cervical vertebrae [fusions and posterior arch deficiency PAD] have been reported to occur more frequently in patients with cleft lip CL and palate. 
The cervical vertebrae develop from sclerotomes which surround the notochord and neural tube.  A series of cartilaginous rings appears within the sclerotomal paraxial mesodermal sheath at about 4 weeks intrauterine life.  Each cartilaginous ring is formed from adjacent halves caudal and cranial of the original somite; hence, vertebrae occupy intersegmental planes of the body wall. The cartilaginous arch fuses with the body at about 8 weeks  for most vertebrae, each ring ossifies from three centers to form the centrum and two halves of the neural arch of the vertebrae. Ossification commences by the 8 th week of fetal life and is completed at about three years. 
The development of the cleft palate (CP and PAD anomaly of cervical vertebral spine is from the same paraxial mesoderm. The mechanism involved in palatal shelf fusion during embryonic development also has an effect on the development and fusion of the posterior arch of first cervical vertebra.
The congenital anomalies of the vertebral spine may be one of the possible mechanisms for CP development. A short neck with vertebral synostosis or other vertebral anomalies may limit the fundamental head extension of the foetus, thus interfering with the anterior and descending development of the glossomandibular complex. This may lead to inadequate free space above the tongue, which is required to allow horizontal movement of the palatal shelves, and may induce the development of CP. 
Embryological development suggests that patients with CL and CP have a higher risk of having cervical spine anomalies than do patients in the general population.
It is possible to investigate these anomalies through lateral cephalometric radiographs as cervical vertebrae can be easily visualized with routine lateral cephalogram taken before orthodontic treatment.
The study was aimed to evaluate the developmental relationship in the vertebral column in the cervical region and to relate its association with patient's with CL and CP.
| Materials and Methods|| |
The sample consisted of 228 children chosen at random from Global Hospital and Research, Mount Abu and Armed Forces Dental Clinic (study group and 225 normal children at Institute of Dental Studies and Technologies, Modinagar (control group) who were undergoing orthodontic treatment. The cleft sample was subdivided into patients with CP only, and unilateral cleft lip and palate (UCLP) and bilateral cleft lip and palate (BCLP). The control group sample was not categorized.
Lateral cephalograms were collected for both the groups (control and study group). Since these patient's were already undergoing orthodontic treatment, pretreatment lateral cephalogram was collected for assessment.
In order to allow standardization of the sample, only radiographs of individuals between 10 and 22 years of age and without any craniofacial syndromes were included in study. Radiographs were examined on a film viewer by a single examiner. The profile of first four cervical vertebrae and atlanto occipital articulation were structurally traced on an acetate paper with 3H pencil under optimal illumination and cervical vertebral anomalies (CVA) were registered and categorized into posterior arch deficiencies (dehiscence and spina bifid) and fusion. 
The abnormalities of the cervical vertebrae were defined by visual assessment directly on the lateral cephalograms and structural tracings. They were classified according to the method of Sandham  and divided into two categories: PAD and fusion anomalies.
Although further categorization of these anomalies was done, (spina bifida and dehiscence under PAD, block fusion, and occipitalization under fusion anomalies) but for the assessment of sample, basic two types were considered as PAD and fusion [Figure 1], [Figure 2], [Figure 3], [Figure 4] and [Figure 5].
The PAD of the vertebrae was recorded when a uniform radioopacity without an internal cortical outline was observed at the distal margin of the posterior arch [Table 1].
Cervical vertebrae fusion were identified as osseous continuities without complete separation at the intervertebral disc or at the articular surfaces.
| Results|| |
Finding for the study group (N = 228) and the control group (N = 225) showed that 19.29% of cleft patients with the cervical vertebral anomalies, while only 4.4% of the normal population with cervical vertebral anomalies [Table 1], [Table 2], [Table 3] and [Table 4]
In study group
In UCLP PAD is more common that is 68% (20 out of 29 anomalies detected).
In CP group (cleft palate only) also the percentage of PAD were 66% (four out of six anomalies detected), as similar to UCLP.
In BCLP fusion anomalies were more common with 66% (six out of nine anomalies).
When comparing the overall study group, PAD was more with respect to any other anomalies.
UCLP group was more prone for cervical vertebral anomalies (65%) with respect to any other study sample group.
In the control group
A total of 10 patients had cervical vertebral anomalies (4.4%) and this was PADPAD and in eight patient with fusion.
| Discussion|| |
0Classification of cervical vertebral anomalies
These anomalies are commonly divided ,,
Posterior arch deficiencies
- Spina bifida-Incomplete ossification in the spinous process and generally occurs in the posterior arch of the vertebral unit.
- Dehiscence spinous process which implies incomplete development of the structures. Dehiscence in the atlas affects either the anterior arch or the posterior arch, posterior arch dehiscence being most common in the midline.
Fusions.  Fusion is bony union of one unit with another at the articulation facets, neural arch, or transverse processes of vertebrae.
- Block fusion. Fusion between two cervical vertebrae; in which the bony union includes the vertebral bodies.
- Occipitalization-the assimilation of the atlas to the base of the skull or atlantooccipital fusion. The essential point in the diagnosis of occipitalization is some degree of bony union between the skull and the atlas.
Prevalence of cervical vertebral anomalies in present study and comparing with other studies in literature
The prevalence of cervical vertebral anomalies in CL and UCLP was found to be higher in the present study as compared to other studies in literature [Table 5].
|Table 5: Comparison of sample types in different cleft studies with cervical vertebral anomalies|
Click here to view
The differences in frequency were attributed to the fact that the inclusion/exclusion criteria for the cleft type and ethnic variation of sample of the study. The prevalence of anomalies of the upper cervical vertebrae in orofacial cleft malformations has been studied comparing different types of clefts with a noncleft group.
A common finding is that children with CL or CP have more prevalence of cervical vertebral anomalies than the normal population [Table 5].
Fusion of the upper cervical vertebrae occurs most commonly between the second and the third (C2-C3) vertebrae. They are probably the result of a failure in normal embryological segmentation due to locally decreased blood supply during fetal development. Patients are generally asymptomatic, but increasing age or injury may precipitate symptoms.  The cervical spine anomalies often coexist with syndromes anomalies of the head and neck and emphasized that those anomalies should be detected by radiographic imaging as early as possible to optimize management and reduce the risk of neurologic injury. 
One of those syndromes is Klippel-Feil syndrome.  It characterized by congenital fusion of at least two of seven cervical vertebrae in the cervical spine, with limitations to movement of the head or neck, a short neck, and a low posterior hairline in patients with CL and palate.
Block fusion of cervical vertebrae is a general feature of the Klippel-Feil anomaly. Farman and Escobar, in there study,  74 cases of fusion of the cervical Vertebrae were found, 8 of which were block fusions. All of these were observed in CPO group McRae and Barnum  reported on 25 cases with occipitalization of the atlas. They noticed that in two of these patients, there were other fusions besides the occipitalization.
Farman and Escobar  described the radiographic appearance of congenital anomalies of the vertebral bodies due to defects in fusion or normal segmentation, occipitalization of the atlas, basilar impression, odontoid malformations, atlas malformations, spina bifida, and abnormal ossifications. These findings can be of great importance for patients in whom these anomalies assume clinical significance necessitating management or lifestyle changes to prevent or delay aggravation of the pathology. In the present study, the fusion was more common with UCLP group as compared with other study sample group. This is in contrast to previous findings in literature by Ross and Londsay  also examined lateral cephalograms of 342 children with CLP or CP and 800 noncleft controls and reported that the frequency of congenital vertebral fusion was 4.01% in children with CLP or CP and 0.75% in noncleft controls.
PAD was 11.8%, while Sandham  reported 10.5%. Shprintzen et al.,  indicated that CL and/or palate were associated with other anomalies in 63.4% of their sample.
Association of cervical vertebral anomalies with type of cleft
The degree of association of cervical vertebral anomalies differs  for the different cleft subtypes and CVA appear to be more closely associated with CP only and BCLP.
Lima et al.,  showed that the type of cleft and CVA and occurrence of cervical vertebrae anomalies are independent of the cleft type and gender.
Several studies have also report an association between UCLP and cervical vertebrae shortening, , forward position and decrease in the curvature of the cervical spine. Chen et al.,  suggested that the growth of the cervical vertebral width was almost completed during early cervical growth, and, cervical vertebrae were increased in height later in growth.
Clinical management of patients with cleft and CVA does not differ from that of other patients. The importance of CVA diagnosis is to allow a better understanding of the cleft development process.
Cervical vertebral anomalies may influence head posture or lifting of head and this could cause failure of palatal shelf fusion resulting in CL and palate.
There is high incidence of cervical vertebral anomalies with CLCP as shown by present study. Mechanism underlying the apparently altered development of cervical spine in CLCP patient is yet to be explored. However, it is said that development of cervical vertebrae from somites is susceptible to environmental disturbances during embryonic period.
Hence, the contribution of present study is to confirm an increased rate of CVA in patients with cleft. Since the groups of cervical vertebral anomalies are comprised of deficient as well as excessive osseous development, it was felt that a subclassification of these anomalies might provide more detailed information of cervical vertebral anomalies in various type of clefting.
| Conclusion|| |
Study showed a specific relation between the CP and cervical anomalies. The study showed 19% of cleft patients with cervical vertebral anomalies more frequent than only 4% of control group with cervical vertebral anomalies.
The vertebral anomalies followed a specific pattern in different types of cleft and was found to be PAD occurred more frequently in UCLP and CP only and fusion occurred significantly more often in BCLP.
| References|| |
|1.||Huggare J. Head posture and craniofacial morphology in adults from northern Finland. Proc Finn Dent Soc 1986;82:199-208. |
|2.||Baccetti T, Franchi L, McNamara JA Jr. An improved version of the cervical vertebral maturation (CVM) method for the assessment of mandibular growth. Angle Orthod 2002;72:316-23. |
|3.||Sonnesen L, Kjaer I. Cervical vertebral body fusions in patients with skeletal deep bite. Eur J Orthod 2007;29:464-70. |
|4.||Last R. Anatomy Regional and Applied. Edinburgh: Churchill Livingstone; 1978. p. 462, 477. |
|5.||Trotter M, Peterson RR. Morris's Human Anatomy. Vol. 12. New York: McGraw Hill Book Company; 1966. p. 143. |
|6.||Farman AG, Escobar V. Radiographic appearance of the cervical vertebrae in normal and abnormal development. Br J Oral Surg 1982;20:264-74. |
|7.||Vastardis H, Evans CA. Evaluation of cervical spine abnormalities on cephalometric radiographs. Am J Orthod 1996;109:581-8. |
|8.||Ross RB, Lindsay WK. The cervical vertebrae as a factor in the etiology of cleft palate. Cleft Palate J 1965;2:273-81. |
|9.||Sandham A. Cervical vertebral anomalies in cleft lip and palate. Cleft Palate J 1986;23:206-14. |
|10.||Osborne GS. The prevalence of anomalies of the upper cervical vertebrae in patient with cranio-facial malformations and their effects on osseous naso-pharyngeal depyh. Thesis for Ph.D. degree, Chicago: Center for Cranio-facial Anomalies, University of Illinois Medical Center; 1968. |
|11.||Ugar DA, Semb G. The prevalence of anomalies of the upper cervical vertebrae in subjects with cleft lip, cleft palate, or both. Cleft Palate Craniofac J 2001;38:498-503. |
|12.||Horswell BB. The incidence and relationship of cervical spine anomalies in patients with cleft lip and/or palate. J Oral Maxillofac Surg 1991;49:693-5. |
|13.||Lima MC, Franco EJ, Janson G, Carvalho IM, Santos CF, Capelozza AL. Prevalence of upper cervical vertebrae anomalies in patients with cleft lip and/or palate and noncleft patients. Cleft Palate Craniofac J 2009;46:481-6. |
|14.||Yochum TR, Rowe LJ. Essentials of skeletal radiology. Baltimore: Lippincott Williams & Wilkins; 2005. |
|15.||Manaligod JM, Bauman NM, Menezes AH, Smith RJ. Cervical vertebral anomalies in patients with anomalies of the head and neck. Ann Otol Rhinol Laryngol 1999;108:925-33 |
|16.||Yoshihara T, Suzukib J, Yasutaka Y. Anomaly of cervical vertebrae found on orthodontic examination: 8-year-old boy with cleft lip and palate diagnosed with Klippel-Feil syndrome. Angle Orthod 2010;80:975-80. |
|17.||Farman AG, Escobar V. Radiographic appearance of the cervical vertebrae in normal and abnormal development. Br J Oral Surg 1982;20:264-74. |
|18.||McRae DL, Barnum AS. Occipitalization of the atlas. Am J Roentgen Rad Ther Nucl Med 1953;70:23-46. |
|19.||Shprintzen RJ, Siegel-Sadewitz VL, Amato J, Goldberg RB. Anomalies associated with cleft lip, cleft palate, or both. Am J Med Genet 1985;20:585-95. |
|20.||Zuñiga C, Miralles R, Carvajal R, Ravera MJ, Contreras P, Cavada G. Comparative study between children with and without cleft lip and cleft palate, part 1: Cephalometric analysis. Cleft Palate Craniofac J 2000;37:281-5. |
|21.||Chen LL, Xu TM, Jiang JH, Zhang XZ, Lin JX. Quantitative cervical vertebral maturation assessment in adolescents with normal occlusion: A mixed longitudinal study Am J Orthod Dentofacial Orthop 2008;134:720. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]
[Table 1], [Table 2], [Table 3], [Table 4], [Table 5]
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