Methods
The present study was cross sectional in design based on medical records and key informant interviews. The interviewed mothers were consecutively selected because the cleft lip and cleft palate are rare conditions in the general population. The medical workers who routinely participate in the management of delivering mothers and new born babies were purposively recruited and interviewed as key informants as they are perceived to have interacted with mothers of children with cleft lip and palate. Thus, the findings may not represent the general population from which the respondents were drawn.
We excluded still births because they are routinely whisked away immediately for burial by relatives [
15] before proper examination. Furthermore, interviewing a mother about a previous still birth could be very traumatizing. Inevitably, this exclusion could have led to loss of data. It should be noted that medical records used in the present study were for the mothers who delivered babies in the two hospitals. Although the records bore detailed physical conditions including any deformities of the baby, they lacked any identifiers. In order to avoid double documentation, records of children in the pediatric units were excluded in the study, which could have led to missing data.
We employed key informant interviews, which have the advantage of getting the information in detail. The exhaustive discussion of the questions was supplemented by translating the interview guide of the mothers into the local language (Rufumbira), which is understood by the respondents. The voice recorder was instrumental in reviewing the whole interview process and to pick some information that had not been recorded on paper during the face to face discussion.
Findings
The overall period prevalence of oro-facial clefts in Kisoro District was 0.77/1,000 live births. This value was comparable to findings in previous study in Uganda [
15] and Malawi [
17] with approximately equal sample size (Table
4).
Table 4
The comparison of occurrence of cleft lip and cleft palate in previous and the present study
| British | 13 years | 325,727 | 1.4/1000 |
| Iraqis | 3 years | 229,992 | 0.8/1000 |
| Ghanaians | 4 years | 4,000 | 6.3/1000 |
| Nigerians | 1 year | 5,037 | 1.35/1000 |
| Malawians | 1 year | 25, 562 | 0.67/1000 |
| Ugandans | 1 year | 26,286 | 0.73/1000 |
Present study | Ugandans | 6 years | 25,985 | 0.77/1000 |
Cleft lip with cleft palate was the most common variation of the oro-facial clefts in the present study. This was in support of previous studies [
1,
7,
18]. On the other hand, cleft palate alone was the least common in the two hospitals in Kisoro District (Table
1), similar to what was previously reported [
1,
2]. However, the previous studies had larger sample sizes and broader inclusion criteria suggestive of cautious comparison with the present study.
In the present study, the left side of the face was the most commonly affected with oro-facial clefts which is in agreement with previous findings [
9]. The majority (60%) of the children with oro-facial clefts were delivered in Kisoro Hospital as compared to 40% in St. Francis Hospital, Mutolere, despite the two hospitals having approximately the same number of deliveries in the study period. The explanation for this difference is not obvious. Moreover, based on the number of deliveries in the two hospitals, the ownership of St. Francis Hospital, Mutolere (private) vesus Kisoro Hospital (public) seem to have no influence on the patient attendance.
About a third (n = 7) of the mothers admitted having relatives with cleft lip and cleft palate (Table
2), which was also echoed by some medical staff members (n = 5) who stated that oro-facial clefts are inherited conditions (Table
3) suggestive of the role of heredity [
19,
20]. Similar findings had previously been reported [
1,
3,
18,
21] (Table
4).
In the present study, most mothers had preterm births and infants of low birth weight (less than 2.5 kg, Table
1), which is in agreement with Pantaloni and Byrd [
2]. Hagberg and co-workers [
22] reported that children with oro-facial clefts and additional malformations had lower birth weight and were born earlier than children with only oro-facial clefts. Although none of any other developmental anomaly was recorded in the present study, the majority of the medical staff members (n = 21) believed there are variable frequencies of such deformities in children with oro-facial clefts (Table
3).
Pantaloni and Byrd [
2] reported an association between a high incidence of oro-facial clefts and low socio-economic status, presumably due to poor nutrition in the lower end of the economic scale. However, we were not able determine the socio-economic status of the families of these children.
We noted a higher frequency of boys (n = 13) with oro-facial clefts as compared to girls (n = 7; Table
1), which is in agreement with previous studies [
1,
6,
15,
18,
23].
Regarding birth rank, most children were in the 4
th and 5
th rank, implying that multi-para could be a risk factor of oro-facial clefts (Table
1). However, Pantaloni and Byrd [
2] indicated that the birth order of children with oro-facial clefts is not significantly different from that of their normal counterparts.
The trend in seasonal variation of oro-facial clefts seems not to be a clear cut. In the present study most of the children were born in the months of April to June (Table
3) which corroborates a previous study [
24]. Edwards [
24] reported that most of the babies in Birmingham with oro-facial clefts were born in the first half of the year with a peak in the month of March. On the other hand, Pantaloni and Byrd [
2] reported a higher incidence of oro-facial clefts in January and February while Owens et al. [
18] indicated an association of a significant increase in the frequency of the clefts with conception in the second half of the year in Liverpool, United Kingdom.
In the present study threatened abortion was reported by 20% of the mothers. Although, Saxén [
25] reported a significant association between oro-facial clefts and threatened abortion during the second trimester, he concluded that threatened abortion might be a symptom of an already malformed embryo rather than a cause of the clefting. Penicillin derivatives were the most common drugs consumed during pregnancy (Table
1). This was contrary to Niebyl [
26] who indicated that penicillin derivatives are safe during pregnancy. Our findings may be coincidental bearing in mind that retrospective reporting of drug histories is prone to recall bias with no reliable evidence of compliance [
18].
Most of the mothers reported that they live on the hill sides, which obviates the fact that Kisoro District is generally hilly and implies that they could have been subjected to chronic hypoxia. Castilla et al. [
27] hypothesized the association of teratogenic effect of altitude on the development of the cleft lip and cleft palate, presumably due to hypertonic hypoxia.
In the present study, the majority of the mothers reported the maternal age of less than 30 years (Table
1). although we may not have established paternal age with certainty because of relying on information from third party. Pantaloni and Byrd [
2] indicated that the risk of developing oro-facial clefts increases if both parents are over 30 years of age.
In support of previous findings [
11], the majority of the medical staff indicated that feeding and recurrent infections were the most common problems encountered by children with oro-facial clefts (Table
3). The majority of the mothers were hurt on realizing that their children had oro-facial clefts and also thought that the cause of the clefts was a supernatural phenomenon (Table
2), which is a similar belief reported in Ghana [
5]; Kenya, Russia, Cambodia, India, Egypt and Peru [
28] and Nigeria [
29]. Furthermore, the medical workers reported that most mothers were disappointed and depressed on realizing that they had given birth to a child with an oro-facial cleft (Table
3). Moreover, the children with cleft lip and/or cleft palate were not readily accepted in the communities and were regarded as outcasts. This finding was similarly expressed by Agbernorku and co-workers in South East Ghana [
5]. The negative attitude tends to affect the child’s psychosocial development and has previously been reported [
5,
12‐
14,
30,
31].
In the present study, surgical intervention and psychosocial support were the recommended modalities for managing a child with cleft lip and cleft palate (Tables
2 and
3). This recommendation corroborates previous authors [
28] who reported that surgical care alone is insufficient if harmful beliefs continue to victimize the affected individual. Mednick et al. [
28] pointed out that care of the entire person includes providing scientific explanations and understanding of cultural beliefs that may continue to traumatize individuals with CL and/or CP even after surgical repair. However, the two hospitals in Kisoro District lacked cleft lip and cleft palate surgical teams of their own, but relied on surgical interventions under the auspices of “Flying Doctors” sponsored by African Medical and Research Foundation (St. Francis Hospital, Mutolere, Medical Superintendent, personal communication).