|Year : 2019 | Volume
| Issue : 1 | Page : 11-16
Correlating causative factors in cleft lip and palate patients: An epidemiological study
Bhagyashree B Desai, Dolly P Patel, Surina V Sinha, Mahesh Jain, Roopal N Patel, Sheron T Bhanat
Department of Orthodontics and Dentofacial Orthopaedics, AMC Dental College and Hospital, Ahmedabad, Gujarat, India
|Date of Web Publication||4-Feb-2019|
Dr. Bhagyashree B Desai
Department of Orthodontics and Dentofacial Orthopaedics, AMC Dental College and Hospital, Bhalakia Mill Compound, Opp Anupam Cinema, Khokhara, Ahmedabad - 380 008, Gujarat
Source of Support: None, Conflict of Interest: None
Aims: The primary aim of this study was to correlate the incidence of cleft lip and palate (CLP) with possible etiological factors such as consanguinity, history of abortions or miscarriages, type of delivery, oral destructive habits, and family history. Materials and Methods: This retrospective study was completed using a standardized questionnaire which was prepared to investigate the information of 166 patients referred to/undergoing orthodontic rehabilitation treatment of CLP at the hospital. The variables assessed were consanguinity, affected members in family and relatives, gestational history, past abortions and/or miscarriages, medications taken by mother during pregnancy, and parental habits of tobacco chewing and/or smoking. Statistical Analysis Used: Correlation of the above-mentioned variables as well as any mutual effects of gender and cleft type was assessed statistically by Pearson's Chi-square test and Fisher's exact test. Results: The results of the present study provide demographic details of cleft patients in Gujarat, which show that cleft deformity is seen more in male patients (n = 98; 59%). Unilateral CL and palate of left side (CL + PUL) occurs most frequently (n = 62; 37.3%) both in male (n = 40; 40.8%) and female (n = 22; 32.4%) patients. Occurrence of CL + PUL is followed by bilateral CL and palate (CL + PB) (n = 45; 27.1%) and unilateral CL and palate of right side (n = 27; 16.3%). The oral destructive habits of parents (n = 82; 49.4%), past abortions and miscarriage (n = 47; 28.3%), family history (n = 26; 15.7%), and consanguinity (n = 24; 14.5%) could be correlated with the occurrence of CLP. Conclusion: This can serve as a guide for future reference to health workers so that they can take measures to create awareness among the people to avoid consanguineous marriages, use of tobacco, abortions, and create awareness about a family history. The outcome could also help government/public health sector workers to develop strategies for awareness, identification, and treatment of cleft deformities.
Keywords: Cleft lip, cleft palate, consanguinity, tobacco consumption
|How to cite this article:|
Desai BB, Patel DP, Sinha SV, Jain M, Patel RN, Bhanat ST. Correlating causative factors in cleft lip and palate patients: An epidemiological study. J Cleft Lip Palate Craniofac Anomal 2019;6:11-6
|How to cite this URL:|
Desai BB, Patel DP, Sinha SV, Jain M, Patel RN, Bhanat ST. Correlating causative factors in cleft lip and palate patients: An epidemiological study. J Cleft Lip Palate Craniofac Anomal [serial online] 2019 [cited 2021 Jul 27];6:11-6. Available from: https://www.jclpca.org/text.asp?2019/6/1/11/251463
| Introduction|| |
Cleft lip (CL), with or without cleft palate (CL/+P), is the most common congenital deformity of the face and mouth which exhibits variable phenotypes. The affected individuals may have CL, cleft palate (CP), or both (CL + P) and their prevalence varies by ethnic group and geographic location.
Global surveys have shown that the frequency of CLP varies greatly from country to country. Africans have the lowest prevalence rates (1/2500) and North American-Indians and Orientals have the highest prevalence rates (1/500). The frequency of cleft is higher in Asian people than that in other races., In India alone, the number of infants born every year with CL + P is 28,600, which means 78 affected infants are born every day or 3 infants with clefts born every hour. Approximately 70% of the CLP cases are of nonsyndromic variety and occur as an isolated condition, but 30% of oral clefts are syndromic and are associated with some anomalies.
The etiology and mode of transmission of CLP is very complex because of the congenital anomalies that are associated with it. The etiological factors include heredity, fetal environment, and environmental risk factors for CL/P such as stress, consanguinity, smoking, alcohol ingestion, use of medication during pregnancy, insufficient ingestion of folic acid in the pregestational period and in the first quarter of pregnancy, a past of miscarriage and/or stillbirth, maternal diseases, and a family history of clefts., CLP is polygenic and multifactorial involving both genetic and environmental influences. A lot has been done to improve the diagnosis and treatment as well as for reduction of incidence of CLP with team approach. Patients with CLP present complex biological, sociological, and psychological problems and their rehabilitation involves several disciplines. Earlier studies reported a drastic decline in the quality of life and psychosocial performance in children with clefts. Hence, the present study is a humble effort to retrospectively analyze the data on the spectrum of CLP in Gujarati Indian population and correlating it to the etiological factors for future awareness and prevention.
| Materials and Methods|| |
This retrospective study was completed using the information and history of 166 patients referred for or undergoing orthodontic rehabilitation as part of treatment for CLP from 2015 until 2017. A sample size of 166 achieves 80% power to detect an effect size (W) of 0.2410 using Chi-square test with a significance level (alpha) of 0.05000.
As this was a hospital-based study, purposive sample collection was done. All the patients with CL/+P irrespective of the type; who were referred for orthodontic rehabilitation and/ or being treated at Department of orthodontics at AMC Dental College and Hospital were included in the study with the due consent of their parents.
Any patient, who had syndromic CLP, was excluded from the study.
An ethics and research committee approval was also obtained from the institutional review board (ref. 312/21.09.2016). Patients' distribution according to gender is shown in [Graph 1] as the final sample of the 166 patients included 98 males and 68 females. All patients with oral clefts were screened and examined for clinical condition. Cases were analyzed according to the variables such as consanguinity, history of cleft deformity in siblings/parents and grandparents, gestational history and history of abortions or miscarriages, details of medications/drugs taken during pregnancy, and habits such as tobacco chewing and smoking by a single principal investigator [Annexure 1]
The data collected were analyzed using the SPSS software (IBM Version 20.0, Chicago, IL, USA). For descriptive purposes, results are presented as frequency and percentages. Fisher's exact test was applied to assess correlation between cleft types and gender. The significance level was predetermined at 10% (P < 0.1). And, for the distribution of associated causative factors, Pearson's Chi-square test was applied (P < 0.001).
| Results|| |
In the present study, a total of 166 cleft patients were included. The results of the present study provide demographic details of cleft patients in Gujarat. The most frequently observed cleft type was unilateral CL+P of left side (CL + PUL) (n = 62; 37.3%), followed by bilateral CL + P and palate (CL + PB) (n = 45; 27.1%) and CLP of the right side (n = 27; 16.3%). On the other hand, the least frequent cleft types were unilateral CL+CP of left side (n = 1, 0.6%) and CL of the left side with primary cleft palate (n = 1; 0.6%) [Table 1].
A higher incidence of CL + PUL was observed in males (n = 40 [40.8%]) compared to females (n = 22 [32.4%]), this could be attributed to the total number of male patients being higher. The overall association between gender and cleft type was statistically significant (P = 0.09) [Graph 2].
Distribution of causative factors
The overall distribution of causative factors is presented in [Table 2]. The oral destructive habits of parents including tobacco chewing and smoking (n = 82; 49.4%), history of abortions and miscarriage (n = 47; 28.3%), family history (n = 26; 15.7%), and consanguinity (n = 24; 14.5%) could be correlated with the occurrence of CLP.
The association between the incidence of cleft and all presented causative factors including medications taken during pregnancy (n = 25, 15.1%) was statistically significant with P < 0.001; except for habits (P = 0.877), though significantly a high number of parents (82 [49.4%]) presented with habits.
| Discussion|| |
The prevalence or incidence of orofacial clefts differs among different populations. In the present study, it is evident that distribution of oral clefts was more in males (59%) than in females. Similarly, facial clefts were more commonly seen in males with a male-to-female ratio., And, for American-Indian population, males outnumbered females in both CLP and CL.
Many studies report a male predominance in the sex ratio in CLP patients and a female predominance in patients with cleft palate defects in accordance with our results. According to our study, the CLP was more frequent than CL and CP. Other studies conducted in South Indian population showed similar results, stating CLP being the most frequently occurring cleft anomaly., However, unlike our study, Khajanchi et al. found that CL was the most common among the studied cleft abnormalities, followed by CLP.
It is also widely accepted that left-sided unilateral clefts are more common than right-sided unilateral CLs,,, which is supported by our study.
Not much is known about the influence of consanguinity on craniofacial anomalies or CLP in Indian population. In the present study, parents of 14.5% (n = 24) patients had consanguineous marriage; likewise, previous studies have also found that there was a significant correlation of children with clefts being born to parents who shared a consanguineous relationship and consanguinity is a risk factor for clefts in the craniofacial region.
Our study showed that 15.7% of patients had a positive family history of clefts, which was in accordance with previous Brazilian and Saudi Arabian studies which stated that a history of oral clefts either in the father's or in the mother's family was strongly associated with clefts.
In the present study, 49.4% of patients' parents had a habit of chewing tobacco and/or smoking. Similarly, according to a study done by Chowdhury et al. in 23.2% of their cases, The father had habit of (smoking and/or chewing) tobacco; before the time of gestation. A study by Leite et al. showed that maternal passive smoking during pregnancy was associated with CL/P. In addition, according to Nehra et al.'s study, active and passive smoking during the first trimester of pregnancy resulted in an increased frequency of CLP.
Our study showed that 28.3% of patients' mothers had a history of abortion and miscarriage. Furthermore, 15.1% of mothers had a history of using medications during pregnancy. However, a detailed study regarding the use of particular drug/drugs can be carried out. It is stressed upon by previous studies to focus among environmental risk factors for CL/P, including consanguinity, smoking, alcohol ingestion, use of medication during pregnancy, insufficient ingestion of folic acid in the pregestational period and in the first quarter of pregnancy, a past history of miscarriage and/or stillbirth, maternal diseases, and family history of clefts.
| Conclusion|| |
This study will provide baseline information on the status of these cleft patients focusing on the significant association between the above-mentioned variables and cleft deformities among different communities. This can serve as a guide for future reference to health workers so that they can take measures to create awareness among the people to avoid consanguineous marriage, habits, and abortions. This is a humble attempt to identify and associate causative factors as a study of this sort has not been carried out for Gujarati population previously. Government (health sector) could also develop strategies in health sector for awareness, identification, and treatment of cleft deformities. A further population-based study and educational intervention is required so that the policy can be developed.
We would like to thank:
- The Department of Orthodontics and Dentofacial Orthopaedics, AMC Dental College and Hospital, Khokhara, Ahmedabad
- Dr. Hemen Jaju MS Plastic Surgery and
- Dr. N. M. Shaikh. MS Plastic Surgery.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Auslander M, Brown AS, Dalston RM, Elmendorf EN, Elster BA, Jones MC, et al
. Parameters for evaluation and treatment of patients with cleft lip/palate or other cranofacial anomalies. Cleft Palate-Craniofacial Journal 1993;30 (suppl).
Kim NY, Baek SH. Cleft sidedness and congenitally missing or malformed permanent maxillary lateral incisors in Korean patients with unilateral cleft lip and alveolus or unilateral cleft lip and palate. Am J Orthod Dentofacial Orthop 2006;130:752-8.
Jamilian A, Nayeri F, Babayan A. Incidence of cleft lip and palate in Tehran. J Indian Soc Pedod Prev Dent 2007;25:174-6.
] [Full text]
Mossey P, Little J. Addressing the challenges of cleft lip and palate research in India. Indian J Plast Surg 2009;42 (suppl 1).
Schutte BC, Murray JC. The many faces and factors of orofacial clefts. Hum Mol Genet 1999;8:1853-9.
Murthy J, Venkatesh Babu G, Bhaskar L. Clinical and demographic factors associated with cleft lip and palate in South India: A hospital based study. Int J Latest Res Sci Technol 2004;3:80-3.
Paranaíba LM, Miranda RT, Martelli DR, Bonan PR, Almeida HD, Orsi Júnior JM, et al.
Cleft lip and palate: Series of unusual clinical cases. Braz J Otorhinolaryngol 2010;76:649-53.
Murthy J, Bhaskar L. Current concepts in genetics of nonsyndromic clefts. Indian J Plast Surg 2009;42:68-81.
] [Full text]
Beluci ML, Genaro KF. Quality of life of individuals with cleft lip and palate pre- and post-surgical correction of dentofacial deformity. Rev Esc Enferm USP 2016;50:217-23.
Khajanchi MU, Shah H, Thakkar P, Gerdin M, Roy N. Unmet burden of cleft lip and palate in rural Gujarat, India: A population-based study. World J Surg 2015;39:41-6.
Vanderas AP. Incidence of cleft lip, cleft palate, and cleft lip and palate among races: A review. Cleft Palate J 1987;24:216-25.
Reddy SG, Reddy RR, Bronkhorst EM, Prasad R, Ettema AM, Sailer HF, et al.
Incidence of cleft lip and palate in the state of Andhra Pradesh, South India. Indian J Plast Surg 2010;43:184-9.
] [Full text]
Jamilian A, Lucchese A, Darnahal A, Kamali Z, Perillo L. Cleft sidedness and congenitally missing teeth in patients with cleft lip and palate patients. Prog Orthod 2016;17:14.
Jose BA, Subramani SA, Mokhasi V, Jayan M. Consanguinity and clefts in the craniofacial region: A retrospective case-control study. J Cleft Lip Palate Craniofac Anomal 2015;2:113-7. [Full text]
Leite IC, Koifman S. Oral clefts, consanguinity, parental tobacco and alcohol use: A case-control study in Rio de Janeiro, Brazil. Braz Oral Res 2009;23:31-7.
Ravichandran K, Shoukri M, Aljohar A, Shazia NS, Al-Twaijri Y, Al Jarba I, et al.
Consanguinity and occurrence of cleft lip/palate: A hospital-based registry study in Riyadh. Am J Med Genet A 2012;158A:541-6.
Chowdhury CR, Khijmatgar S, Kishore NP, Shetty V. Oral health status among cleft lip and palate patients in South India: A profile. J Cleft Lip Palate Craniofac Anomal 2017;4:S152-9.
Nehra R, Tafweez R, Mobeen N, Akhtar S. Maternal exposure to active, passive smoking and tobacco chewing and risk of oral cleft in new born. SZPGMI 2015;29:97-9.
[Table 1], [Table 2]
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