Orofacial Clefts: A Clinical Community Study in a Developing Country

Aim. e study aimed at �nding the types of orofacial cles, reasons for delay in the repair of the deformity and medium through which patients and/or their relatives heard about the outreach program. Methods. is study was from 2009 to 2011 at two diﬀerent study sites. e team visited various media houses and health facilities at each study site to disseminate information concerning the cle outreach program. Patients and/or their parents who visited the hospitals were interviewed using a questionnaire designed for the study to retrieve needed information for the study. Patients were then examined and booked for cle repair by specialists in the team. Results. A total of 61 patients were recorded from both study sites, with ages ranging from 3/12 to 54 years old, with a mean age of 6.7 ± 2.7 (SD) years; while, male:female ratio was 1.05:0.5. Cle lip only (52.5 % , 𝑛𝑛 𝑛 𝑛𝑛 ) was the highest type of orofacial cle recorded in the study. In terms of occurrence, complete cle (63.9 % , 𝑛𝑛 𝑛 𝑛𝑛 ) was majority; while, position-wise, unilateral cle right (32.8 % , 𝑛𝑛 𝑛 𝑛𝑛 ) was the highest. A signi�cant ( 𝑃𝑃 𝑛 𝑛𝑃𝑛𝑃𝑃 ) association between males and unilateral right cle lips was identi�ed. Lack of �nance (47.5 % , 𝑛𝑛 𝑛 𝑛𝑛 ) was the main reason for delaying in deformity repair. Also, information from physician (41.0 % , 𝑛𝑛 𝑛 𝑛𝑛 ) was the major medium for information dissemination. Conclusion. Unilateral cle lip constitutes most of the orofacial cles in this study, with lack of �nance being the commonest reason why patients delay in the repair of their deformity.


Introduction
Most Cle Lip (CL) and Palate (CP) studies are based on hospital or birth registry statistics which may not re�ect the true incidence of the disease. Even though community-based surveys may re�ect close to true incidence, they are di�cult to conduct. It requires a large knowledgeable work force and is time consuming and expensive. In developing countries such as Ghana, untreated CLs and CPs are found with increasing frequency, and patients oen present to the surgeon far past the optimal time for closure of the cle deformities.
Orofacial cles are common congenital malformations whose etiology is complex and likely to have both genetic and environmental etiology [1][2][3][4]. Although numerous techniques and protocols are employed in the repair of this congenital deformity, the surgical literature is consistent in recommending early repair, typically completing primary repair of the lip and palate by the age of two years [5]. A commonly accepted protocol in many surgical textbooks is repair of a cle lip at 10 to 12 weeks of age, followed by primary palatoplasty at nine to 12 months of age, before development of speech [1,[5][6][7]. It is well known that timely closure of palatal defect is associated with improved speech outcome [6,8] and late closure of the palate has been clearly demonstrated to give a poor outcome [7,9]. In addition, delayed repair of the cle can lead to impaired family and societal relationships with potential long-term psychological effects on the child [10,11].
In Ghana, the �rst community-based study on cle was reported by Agbenorku et al. (2010), where they reported a cle lip/cle palate prevalence of 5.0 per 1000 people in the Wudoaba communities in the southeastern border of Ghana [12]. A retrospective review of cle lip and palate operations carried out at the Komfo Anokye Teaching Hospital (KATH) in Kumasi, Ghana, reported that a total of 344 patients were operated on for cle lip with or without palate (CL +/P) during the 5-year period (2001)(2002)(2003)(2004)(2005), that is, 69 operations per year [13]. Again in KATH, a four-years (2006-2009) review of cles repaired cases by Agbenorku et al. (2011) reveals a higher incidence of congenital cle abnormalities repaired during the period under review (i.e., 132 surgeries per year). Most prevalence reports on CL +/P had their data from hospitals, with relatively few studies done in communities [14].
e model of this study was different. Information about CL and CP were disseminated through radio stations announcements and Churches, hospitals, schools, and community centers by the education team of the outreach and volunteers in both areas of the study. Two hospitals were selected for the cle repair surgery aer the community educations. is study aimed at �nding the types of orofacial cles in the study areas, reasons for delay in the repair of the deformity, and the commonest information medium through which patients and/or their relatives heard about the outreach programs.

Study Settings. e Global Evangelical Mission
Hospital (GEMH) located at Apromase, a village about 10 km southwest of Ejisu, the capital of the Ejisu-Juaben Municipal and also about 12 km southeast of Kumasi. e hospital has thirty-�ve beds and is well known in the region for its expertise in skin ulcer and orofacial cle management. Patients from different facilities have been referred to GEMH from all over the country. A total of 2153 patients (out-and in-patients) visited the hospital in the year 2011 (Biostatistics Unit, 2011). e surgical department of the hospital has a standard surgical theater with a recovery ward.
e Sogakope study in the Volta Region was the second community-based study where cle patients were also mostly grown-ups. Aer the dissemination of the cle repair information through various media, patients came voluntarily to meet the team, where they were scheduled for surgery at the South Tongu District Hospital at Sogakope. Cases were reported from many towns and villages including Wudoaba (a very prone area for orofacial cles).

Data Collection and Analysis.
Ethical clearance for the study was approved by the Ethics Committee of the School of Medical Sciences, Kwame Nkrumah University of Science and Technology, Kumasi, Ghana. Demographic data for this prospective study from January 2009 till December 2011 were obtained from the patients or relatives of patients aer giving their consent for the study. Information related to the surgical procedures were also recorded at the hospital prior to and aer the surgery. Information collected included demographic features of the patients, medium through which the information about the team was heard, types of cle lip/palate, number of cases seen, and surgeries performed. e data were then analyzed, and results, displayed in tables and graphs by using SPSS version 12.0 (SPSS, Inc, Chicago, IL).

Patient Management
Technically, the surgery can be performed at any time aer the child is born. In general, the rule of 10 was adopted. at is, the baby was operated on at about 10 weeks old, with body weight about 10 lb, that is, 4.5 kg, and hemoglobin level of about 10.0 g/dL. e risks in general anesthesia are much reduced when these parameters are attained. For babies, cle palates were customarily repaired at about 12 months of age.

Surgery for Unilateral Cle
Lip. e aim of the surgery is to repair the defect and realign the deranged muscle back to the normal anatomic position. e end result should be a lip with relatively normal look both aesthetically and dynamically. Millard's rotation-advancement repair method was the surgical technique used (Figures 1 and 2).

Surgery for Bilateral Cle
Lip. e preoperative formalities and the aim of the operation are the same as for unilateral cle lip. e technique used was the straight-line procedure ( Figure 3).

Surgery for Cle
Palate. e purpose of cle palate operations is to produce anatomic closure and, eventually, normal speech, as well as to minimize maxillary growth retardation and dental alveolar deformity. It is important that the palatal defect be repaired before the child initially attempts to speak. e technique usually used was Langenbeck variant (Figure 4).

Demographic Characteristics of Patients.
A total of 61 patients were recorded from both study sites, with ages ranging from 3/12 to 54 years old, with a mean age of 6.7±2.7 (SD) years ( Figure 5). In terms of sex, males outnumbered females in the ratio 1.0:0.48.

Types of Orofacial Cles among Patients.
Cle lip and/or alveolus only (52.5%, 2) was the highest type of orofacial cle recorded in the study, followed by cle palate only (29.5%, ) as shown in Table 1.

Occurrence and Position of Orofacial Cles among
Patients. Orofacial cles were categorized and taken in consideration as it occurs and positions on the face of patients.  ) was the highest, as shown in Table 2.

Reasons for Delay in Presentation of
Deformity. e most common reason for delay in deformity repair was lack of �nance (4�.5%, ). Others included lack of awareness of treatment availability, superstition, fear of death from surgery, and long distance to health facilities (Table 3).

Information Dissemination Medium in Communities.
Information from physicians/nurses (41.0%, ) were the major medium through which patients and/or their parents heard about the cle outreach program. Other recorded media included TV/radio (FM) stations. Friends/relatives, charity organizations such as churches and mosques were also important media ( Figure 6).

Analysis of Data.
Logical Regression analysis was used to determine possible signi�cant association between patients� demographic features and cle deformity in the study. A probability value ( -value) of less than 0.05 was considered to be statistically signi�cant at 95% con�dence interval.
Demographic features used were age and sex; characteristics of cles used included type, occurrence, and position. e analysis of the results reveals a signi�cant ( ) association between males and unilateral right cle lips (Table 4).

Discussion
In developed countries, with the advancement of medical services and awareness among patients, it is rare to �nd adults  with unoperated CL and CP. However, the scenario is totally different in developing countries. e report of more CL in males and more CP in females from the African studies is consistent with the literature [15,16]. e overall report of CL/CP being higher in males in the African studies is also consistent with the literature [15]. e wide range in the con�dence intervals in most of the studies suggests that the precision is low, which may be a result of poor ascertainment of cases. e results of this study show a high number of males as compared to females. Furthermore, unilateral CL   was revealed to be the most prevalent type of orofacial cle among the patients. To buttress the �ndings of the study, an analysis to determine the signi�cant association between demographic features and orofacial cle was performed. e regression analysis reveals that being a male is a risk factor for unilateral right CL ( )� no other signi�cant was determined. In cle endemic areas in developing countries such as Ghana, early sonography investigation of foetus should also take into consideration unilateral CL deformity, especially in male foetus so as to educate and counsel involved parents how to feed and care for the unborn baby before surgical repair of the cle.  Schwarz and Khadka (2004) reported in their studies that late presentation of patients with cle deformities in developing countries is still very common [5,6]. Unfortunately in developing countries untreated CL/CPs are found with increasing frequency, and patients oen present to the surgeon far past the optimal time for closure of the cle deformities [13,17]. is study was not different from the already reported ones in terms of late presentation of patients with cle deformities. Even as old as 21 years and above patients constituting 29.5% ( ) of the patients had their cle deformities repaired. Due to the constant reporting of such situation in many orofacial cle studies, an essential objective of this study was to determine reasons attributing to such occurrences in developing countries such as Ghana. e results of the study reveal that, lack of �nance (money), unawareness of treatment availability, and superstition (the belief that it is a spiritual affair) were the three major reasons given by patients and/or parents. Lack of money being the commonest reason is not unexpected because Ghana is a developing country, where the majority of its citizens live on less than one US dollar a day; and on average US$600 is required for cle lip surgery. Notwithstanding this, all patients in the study were treated for free on the provisions of the National Health Insurance Scheme (NHIS). e problem of lack of �nances as a reason for late presentation of medical problems is certainly endemic throughout developing countries [6]. e problem is compounded for rural people, as they must pay for travelling and accommodation expenses in addition to the medical services at the health care center, where such surgery can be provided [6]. Adeyemo et al., in a similar study in Nigeria, reported lack of money (56.7%), distance to health facilities (18.4%), and a lack of knowledge of the availability of surgical repair services (13.3%) as the three most common reasons for late presentations of CL/CP for repair [18]. Schwarz and Khadka [6] reported that the main reasons for late presentations of CL/CP in Nepal were lack of knowledge of availability of services (31%), lack of services near at hand (29%), lack of �nance (24%), and lack of time (13%). e former had a similar most common reason as this study, while the latter had lack of �nance as the third factor. e difference in most common reason may be due to differences in geographical location, since, Ghana, Nigeria, and Nepal are all developing countries.
Free outreach cle repair programs in Ghana and other developing countries will go a long way to alleviate the late presentations of CL/CP. is approach would also lessen much of the �nancial burden and time constraints associated with travelling to a regional center by bringing the health care closer to home. To solve the problem of lack of knowledge of the availability of surgical repair services in Ghana, increased access to primary health care services combined with providing primary health care workers with information regarding the availability of CL/CP surgery would also reduce the rate of late presentation. is approach had been con�rmed by the study as the appropriate procedure to disseminate information to patients and/or their relatives. Most (41.0%) of the patients/relatives claimed to have had information about the outreach program via the health care worker (physician/nurse). Radio advertising and the involvement of charity organizations in community outreach program are also suitable means of informing communities of the availability of orofacial cle management.
Several challenges are encountered in the management of patients with delayed presentation of cle lip and palate especially in adulthood and adolescence. ese include surgical, orthodontic, speech, anesthetic, and psychological challenges. Although sound knowledge of the fundamentals of cle surgery will certainly allow any surgeon to repair an adult cle lip or palate adequately, obtaining the most favorable surgical outcome may require creative modi�cation to the design of the cle secondary to various factors, including the increased size of the adult cle [19]. Surgeons should be prepared to repair adult patients with cle that are signi�cantly enlarged in three dimensions. Closure will require signi�cant so-tissue dissection, as well as the possible use of biomaterials to repair wide palatal cles [20].

Conclusion
Unilateral cle lip constitutes most of the orofacial cles in this study, with lack of �nance been the commonest reason why patients delay in the repair of their deformity. e promotion of health education on orofacial cle in our various health institutions, especially in endemic areas, and fully funded, well organized outreach programs, will go a long way to curve down the number of unrepaired cles and reduce stigmatization in Ghana and other developing countries with similar conditions.