|Year : 2014 | Volume
| Issue : 1 | Page : 38-42
Cleft data from surgical camps on rails: A doorstep health care delivery
Sameek Bhattacharya1, Ashish Rai1, Prabhat Shrivastava2
1 Department of Burns, Plastic, Maxillofacial and Microvascular Surgery, Dr. RML Hospital and PGIMER, New Delhi, India
2 Department of Burns, Plastic, Maxillofacial and Microvascular Surgery, Lok Nayak Hospital and Associated Maulana Azad Medical College, New Delhi, India
|Date of Web Publication||5-Feb-2014|
271, Abhinav Apartment, Vasundhara Enclave, New Delhi - 110 096
Source of Support: None, Conflict of Interest: None
Aim: The aim of the following study is to investigate the epidemiology of cleft lip and palate (CL/P) patients in a given population. Patients and Methods: Data from cleft camps conducted at Life Line Express Hospital-on-train in Madhya Pradesh, India from year 2003 to 2007 was analyzed. A total of 839 patients of non-syndromic CL/P were included in the study. The data was analyzed for age of the patients, type of cleft, laterality of the defect, their relation to the gender of the patient and presence of cleft in first degree relatives. Result: Nearly 59.8% of the patients were in the age group of 5-18 years. There was an overall male predominance. Unilateral clefts were recorded in 83.3% and bilateral clefts in 14.7% of cases. Only 2% of the patients had isolated cleft palate (CPO) and females had 2.3 times higher risk for CPO than males. Of unilateral cleft lip, left side was more common (67.3%). Nearly 4.9% patients had history of familial inheritance among first degree relatives. Conclusion: In India, vast territories lack basic medical amenities and all modern medical facilities are limited to cities. "Cleft surgery at doorstep" is an extremely effective strategy in treating the "hidden" cleft population and collecting a meaningful epidemiological data from a "virgin" territory. We also stress upon the need to include facial clefts as a notifiable disease in our country to understand the gender, regional and ethnic variations of the disease.
Keywords: Cleft camp, cleft lip, cleft palate, epidemiology cleft lip and palate, life-line express
|How to cite this article:|
Bhattacharya S, Rai A, Shrivastava P. Cleft data from surgical camps on rails: A doorstep health care delivery. J Cleft Lip Palate Craniofac Anomal 2014;1:38-42
|How to cite this URL:|
Bhattacharya S, Rai A, Shrivastava P. Cleft data from surgical camps on rails: A doorstep health care delivery. J Cleft Lip Palate Craniofac Anomal [serial online] 2014 [cited 2020 May 25];1:38-42. Available from: http://www.jclpca.org/text.asp?2014/1/1/38/126557
| Introduction|| |
Cleft lip and palate (CL/P) is not a notifiable disease in India. All published data from Indian subcontinent pertaining to CL/P are from major hospital or tertiary care centers, which are located in big cities and are not accessible to poor patients living in remote areas. Life Line Express, operated by Impact India Foundation  is a "Hospital-on-train" fully equipped with operation theatre, pre-operative and post-operative ward. The train utilizes the extensive network of Indian Railways which has access to far flung areas of the country. Cleft surgery camps are organized by taking the train to these remote areas, which have no access to specialized medical facilities. In addition to the unique experience of operating patients "on-train," the cleft camps gave us an opportunity to peep into the community for a meaningful cleft data. The camps were preceded by extensive publicity regarding free of cost cleft surgery in a "hospital-at-doorstep." The patients were provided in-house boarding and lodging at the camp site and free transport to and fro from their homes. All meals and medicines were also provided by the organizers. This way it was possible to tap almost the entire cleft population of the region and even the poorest of the poor got registered in the camp.
| Patients and Methods|| |
Five cleft surgery camps were held consecutively from 2003 to 2007, in three adjoining remote rural districts of the state of Madhya Pradesh in India; covering a population of approximately 3 million over 22,000 km 2 [Table 1]. The camps were preceded by intense publicity campaign in all the villages of the district informing about the dates, venue and the nature of surgery that would be performed. Existing network of health care personnel at primary health care centers were utilized for this purpose. The outpatient screening was conducted at the local primary school or community center. The same facility was also utilized as a make shift ward for pre and post-operative admission.
|Table 1: Number of CL/P: Cleft lip + Cleft lip and Palate|
+ Cleft patients in each camp
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The camps were of 7 days duration. On the 1 st day patients were examined and screened for operation. As these were remote areas with no hospital backup, patients <5 years of age and syndromic clefts were not operated. For the same reason no palatal repair was performed. These patients were referred to the parent hospitals of the surgical team. After screening the eligible candidates for surgery, they were investigated for hemoglobin level, bleeding and clotting time. The operating schedule of next 5 days was prepared. Usually 10-12 cases were operated per day. Two to three surgeons performed surgeries simultaneously. All lip repairs were done by either Millard's or modified Randall-Tennison's technique. Patients <15 years of age were operated under general anesthesia and those above this age group were operated under local anesthesia. All patients were admitted and received parenteral antibiotics and analgesics. Sutures were removed on the 6 th post-operative day.
The clinical record of all the patients attending the camp was maintained in a standard proforma. The parameters recorded were age at presentation, sex, type of cleft and laterality. The clefts were classified as cleft lip (CL), CLP, cleft palate only (CPO) and cleft lip with alveolus (CL + A). Pre-operative and post-operative photographic records of all the operated patients were maintained. The epidemiological data so obtained from five camps over 5 years was analyzed.
| Observations and Results|| |
The total number of cleft patients who presented in five camps, over 5 successive years were 839 [Table 2]. Reconstructive surgery was performed in 256 patients.
Patients of all age groups presented to the camp. They were classified in three age groups of <5 years, 5-18 years and more than 18 years. [Figure 1] shows the number of cleft patients according to the age group. Majority (59.8%) of the patients were in the age group of 5-18 years while 33.3% of patients were in age group of <5 years. A very significant observation in the study was that 58 (6.9%) patients were of the age group of >18 years [Figure 1]. Out of them 10 were married and 7 of them were grandparents of more than 60 years of age. There were two couples wherein both husband and wife had CLs. The youngest patient was a 24-day-old male child while the oldest patient was 70-year-old.
There was an overall male predominance with 606 male patients to 233 female patients (2.6:1). The male predominance among unilateral and bilateral group was in the tune of 2.7:1 and 2.5:1 respectively. Highest male predominance was noticed in CLP group with male to female ratio of 4.4:1 in unilateral clefts and 5:1 in bilateral clefts. The lowest male to female ratio was seen in cleft palate (CPO) group (1.1:1).
Type of cleft
The patients were categorized as unilateral or bilateral CL with or without CP and CPO. Out of 839 patients, unilateral cleft was recorded in 83.3%, bilateral cleft in 14.7% and CPO in 2% of cases respectively. The ratio of unilateral to bilateral cleft was 5.8:1. In unilateral clefts, CL was the most common form of cleft accounting for 60.1% of the cases followed by CLP (26.3%) and CL + A (13.6%). Among bilateral clefts, CL comprised 43.1% followed by CL + A (32.5%) and CLP (24.4%).
The occurrence of left sided cleft was most common (61.7%) followed by right sided cleft (21.5%) and bilateral cleft (14.7%). This pattern of occurrence was almost similar in male and female patients [Figure 2].
|Figure 2: Comparison between type of clefts amongst male and female|
Click here to view
41 patients (4.9%) had family history of clefts in first degree relatives.
| Discussion|| |
At the outset, it is imperative to discuss the limitation of the study. All patients <5 years in age and patients with involvement of palate were excluded from surgical management on logistic grounds. This surgical preference tends to introduce bias in the epidemiological nature of study. However, in our opinion this bias is limited in nature. As the camps were held in different districts each year and with wide publicity, not many were aware of the exclusion criteria and therefore patient participation was in large number. The data of all the patients presenting in out-patient department was recorded irrespective of patients getting operated or being referred. Therefore in spite of bias and in absence of any cleft epidemiology from this region, we found it relevant to present this data.
We have analyzed the epidemiological data from five consecutive camps held from 2003 to 2007, at three remote rural districts in Madhya Pradesh, India. The total number of non-syndromic cleft patients was 839. There were 502 patients in the age group of 5-18 years and 58 in the age group of >18 years. Together they accounted for 66.7% of the total patients. This depicts the level of ignorance and poor health infrastructure in these remote areas. In the study by Kumar et al.  from Riyadh, Saudi Arabia only 7.8% of the patients presented for the first time at the age of 10 year and above. Onyango and Noah  in their study from Nairobi, Kenya had reported only 25% of patients who presented to the hospital were in the age group of >5 years.
There was an overall male predominance with male to female ratio of 2.6:1. This predominance existed in all groups of CL/P with highest male to female ratio in CLP gp (4.4:1) and lowest in CPO gp (1.1:1). The trend of overall male predominance is seen in all major studies. Calzolari et al.  in their large study of 5449 patients in Eurocat registry have reported overall male to female ratio of 1.92:1. Similarly Reddy et al.  (1.9:1), Cooper et al.  (1.4:1) and Yi et al.  have reported high male to female ratio. In regards to gender ratio among various groups of clefts, the results are varied. Owens et al.  have reported highest male predominance in the CLP group of 1.98:1 and in cases of CL of 1.52:1. Kumar et al.,  Derijcke et al.  in their study have reported higher male preponderance in all type of clefts whereas Rajabian and Sherkat  has reported female predominance in cases of CL with male to female ratio of 1:1.16.
Most of the studies show that isolated CP is more common in female (Yi et al.,  Theogaraj et al.,  Derijcke et al.  ). Although in our study isolated CP was overall more common in males then females (1.1:1) but it affected only 1.5% of male patients when compared with 3.4% of females. Thus female patients had 2.3 times higher risk of having isolated CP than males. Vallino-Napoli et al.  also have reported similar risk pattern for males and females.
CL was the most common cleft and accounted for 72.5% the patients followed by CLP which was 25.5% of the patients. This is in contrast to the findings of Reddy et al.  from Andhra Pradesh, India who had reported that CLP (64%) was more common than CL (33%). Similar predominance of complete clefts (63.4%) has also been reported by Calzolari et al.  in their study from 14 European countries and also by Rajabian and Sherkat,  Onyango and Noah,  McLeod et al.  and Kumar et al. 
A surprising finding in our study was very few patients with isolated CP (2%).
A review of literature showed varied incidence of CPO in various studies. Womersley and Stone  in their study in 247 children have reported CPO as the most common cleft and Hagberg et al.  reported similar incidence of cleft of the lip, lip and palate and palate alone. Rajabian and Sherkat  has reported it to be 17.4%, Kumar et al.  as 22.4%. In the study by Reddy et al.  from India, the incidence of CPO was 2%, similar to our finding. One reason of low turnout of CPO patients in our camp was the policy of not operating CPs in camp. This policy must have deterred patients of CPO from visiting the camps.
The most commonly affected side was left followed by right and bilateral cleft, both in males and females. Onyango and Noah,  Yi et al.,  Czeizel,  Tolarovα  also reported similar findings. In the study by Kumar et al.  left sided clefts were most common followed by bilateral clefts. Theogaraj et al.  have reported that left sided clefts occurred in half of the patients with right side and bilateral clefts constituting 25% each. A study of 6423 clefts at Chang Gung Craniofacial Centre showed 41% as left sided, 20% as right sided, 17% as bilateral and 22% isolated CP respectively. 
41 patients (4.9%) in our study had positive family history of clefts in first degree relatives. Wyszynski et al.  have reported 4% risk of familial recurrence in first degree relatives. In another Indian study by Theogaraj et al.,  there was 12.1% incidence of clefts in family and Kumar et al.  from Saudi Arabia reported a positive family history in 26.8% of cases.
| Conclusion|| |
The facility of "hospital on wheels" allowed us to operate and gather data from areas still deprived of basic medical facilities. A large number of cleft populations were "discovered" in these camps. As majority of the population in these areas are farmers for generation and belong to poor socio-economic status, surgery for esthetic correction is not their priority. Moreover unavailability of medical facility in the entire district further dissuades them from seeking medical help until it's a life-threatening emergency. It was surprising to know that majority of adult patients were not even aware that correction of these cleft deformities is possible by surgery.
Considering the fact that there are large numbers of districts with similar socio-economic-cultural profile in our country, this study may only be the tip of an iceberg. By extrapolating the data from the present study even by conservative estimate, the actual quantum of the affected yet untreated cleft individuals may be considerably alarming. We feel that a similar situation might be prevalent in many other third world countries. "Door step targeting" of this undiscovered cleft population world-wide can be an extremely effective strategy in realizing the dream of a cleft free society. We also stress upon the need to include facial clefts as a notifiable disease in our country to understand the gender, regional and ethnic variation of the disease.
To further improve patients participation and to provide complete treatment, palatoplasty can be included in the future programs. For this, collaboration with local hospitals for pediatric intensive care unit may be worked out. The camp must also provide speech therapy services in order to sensitize and popularize the importance of the therapy in cleft patients.
| Acknowledgment|| |
We thank Impact India Foundation, which operates Life Line Express for providing us the opportunity to serve people in remote areas of our country and permitting us to publish this data.
| References|| |
|1.||Available from: http://www.impactindia.org/ [Last accessed on 2013 Nov 11]. |
|2.||Kumar P, Hussain MT, Cardoso E, Hawary MB, Hassanain J. Facial clefts in Saudi Arabia: An epidemiologic analysis in 179 patients. Plast Reconstr Surg 1991;88:955-8. |
|3.||Onyango JF, Noah S. Pattern of clefts of the lip and palate managed over a three year period at a Nairobi hospital in Kenya. East Afr Med J 2005;82:649-51. |
|4.||Calzolari E, Pierini A, Astolfi G, Bianchi F, Neville AJ, Rivieri F. Associated anomalies in multi-malformed infants with cleft lip and palate: An epidemiologic study of nearly 6 million births in 23 EUROCAT registries. Am J Med Genet A 2007;143:528-37. |
|5.||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. |
|6.||Cooper ME, Stone RA, Liu Y, Hu DN, Melnick M, Marazita ML. Descriptive epidemiology of nonsyndromic cleft lip with or without cleft palate in Shanghai, China, from 1980 to 1989. Cleft Palate Craniofac J 2000;37:274-80. |
|7.||Yi NN, Yeow VK, Lee ST. Epidemiology of cleft lip and palate in Singapore - A 10-year hospital-based study. Ann Acad Med Singapore 1999;28:655-9. |
|8.||Owens JR, Jones JW, Harris F. Epidemiology of facial clefting. Arch Dis Child 1985;60:521-4. |
|9.||Derijcke A, Eerens A, Carels C. The incidence of oral clefts: A review. Br J Oral Maxillofac Surg 1996;34:488-94. |
|10.||Rajabian MH, Sherkat M. An epidemiologic study of oral clefts in Iran: Analysis of 1,669 cases. Cleft Palate Craniofac J 2000;37:191-6. |
|11.||Theogaraj SD, Joseph LB, Mani M. Statistical analysis of 750 cleft lip and palate patients. Indian J Plast Surg 2007;40:70-4. |
|12.||Vallino-Napoli LD, Riley MM, Halliday JL. An epidemiologic study of orofacial clefts with other birth defects in Victoria, Australia. Cleft Palate Craniofac J 2006;43:571-6. |
|13.||McLeod NM, Urioste ML, Saeed NR. Birth prevalence of cleft lip and palate in Sucre, Bolivia. Cleft Palate Craniofac J 2004;41:195-8. |
|14.||Womersley J, Stone DH. Epidemiology of facial clefts. Arch Dis Child 1987;62:717-20. |
|15.||Hagberg C, Larson O, Milerad J. Incidence of cleft lip and palate and risks of additional malformations. Cleft Palate Craniofac J 1998;35:40-5. |
|16.||Czeizel A. Studies of cleft lip and cleft palate in east European populations. Prog Clin Biol Res 1980;46:249-96. |
|17.||Tolarová M. Orofacial clefts in Czechoslovakia. Incidence, genetics and prevention of cleft lip and palate over a 19-year period. Scand J Plast Reconstr Surg Hand Surg 1987;21:19-25. |
|18.||Noordhoff MS, Chen PK. Unilateral cheilopalsty. In: Mathes SJ, editor. Plastic Surgery. 2 nd ed. Vol. IV. Philadelphia: Saunders Elsevier; 2006. p. 167. |
|19.||Wyszynski DF, Beaty TH, Maestri NE. Genetics of nonsyndromic oral clefts revisited. Cleft Palate Craniofac J 1996;33:406-17. |
[Figure 1], [Figure 2]
[Table 1], [Table 2]