|Year : 2017 | Volume
| Issue : 3 | Page : 126-131
Epidemiologic indices of cleft lip and palate as seen among Igbos in Enugu, Southeastern Nigeria
Chukwunonso Azubuike Jac-Okereke, Ifeanyi Igwilo Onah
Department of Surgery, Enugu State University of Science and Technology, Park Lane; Department of Plastic Surgery, National Orthopaedic Hospital, Enugu, Nigeria
|Date of Web Publication||21-Nov-2017|
Chukwunonso Azubuike Jac-Okereke
Department of Surgery, Enugu State University of Science and Teaching Hospital, Parklane, Enugu 400001
Source of Support: None, Conflict of Interest: None
Objectives: The objective of the study was to provide more recent local data, and an estimate of the incidence of cleft lip and/or palate among Igbos. Design: The study design was a descriptive transversal study of birth records and case notes between January 2007 and December 2011 and records of clinical attendance and interviews with patients' mothers. Names and state of origin were used as inclusion criteria for Igbos. Setting: Previous local data on epidemiology are over three decades old. The National Orthopaedic Hospital, Enugu (NOHE) is an apex center for plastic surgery in southeastern Nigeria. Enugu State University of Science and Technology Teaching Hospital (ESUTTH) is the largest state-owned health facility. There are ethnoracial variations in the epidemiology of clefts. Materials and Methods: All patients born with clefts at ESUTTH and all records of clefts in the period available at NOHE hospital in the period. Within the period, 262 cleft patients were studied: 139 males and 123 females; male:female 1.13:1. Main Outcome Measure: Main outcome measures were the live birth incidence of clefts, the most frequently occurring birth order, and socioeconomic class affected. Results: There were 5810 live births with a 0.00103270223752151 birth incidence. That among Igbos was 0.0010700909577314. Infants in the first birth order and of low socioeconomic class accounted for the highest incidence. The socioeconomic status was found to be statistically significant (P = 0.0001, confidence interval: 95%) but birth order was not (P = 0.932). There was a positive family history in 9.5% with first-degree relatives accounting for 6%. The mean maternal age was 28.88 years (range: 17–47 years), and mean paternal age was 38.55 years (range: 23–58 years). Conclusion: The epidemiologic indices differ from data published over three decades ago. There are differences in cleft epidemiology within Nigeria.
Keywords: Clefts, epidemiology, Igbos, Southeastern Nigeria
|How to cite this article:|
Jac-Okereke CA, Onah II. Epidemiologic indices of cleft lip and palate as seen among Igbos in Enugu, Southeastern Nigeria. J Cleft Lip Palate Craniofac Anomal 2017;4, Suppl S1:126-31
|How to cite this URL:|
Jac-Okereke CA, Onah II. Epidemiologic indices of cleft lip and palate as seen among Igbos in Enugu, Southeastern Nigeria. J Cleft Lip Palate Craniofac Anomal [serial online] 2017 [cited 2022 Jan 27];4, Suppl S1:126-31. Available from: https://www.jclpca.org/text.asp?2017/4/3/126/218875
| Introduction|| |
Cleft lip and/or palate (CLP) is the world's most common major congenital craniofacial anomaly. Its genetic factors are now well established through segregation analysis and a multifactorial mode of inheritance is suggested by most studies., One main reason for the difficulties in determining etiology in nonsyndromic clefts is that it's polygenic multifactorial, with a significant genetic predisposition to environmental factors. Ethnoracial variations exist in the epidemiology of clefts., Reports show that the incidence among Africans is low., This aligns with studies done in southeastern Nigeria over three decades ago. This was previously the most recent study of clefts in this region. More recent studies in incidence and epidemiology are due.
The National Orthopaedic Hospital, Enugu (NOHE) is an apex center for plastic surgery in the region. From 2007, there was a notable increase in the volume of cleft patients presenting to the center due to the inception of an ongoing free treatment program there. The Enugu State University of Science and Technology Teaching Hospital (ESUTTH) is the largest state-owned health facility. With free maternal and child health care program from 2007, there was a remarkable increase in the number of annual births recorded in the hospital. Newborn babies are examined by the midwife and doctor before transfer to the mother. Congenital anomalies are noted in the birth register. There are no known previous data specific for Igbos in Nigeria.
There is a recent prevalence study involving over six ethnic groups in Benin city, and a nationwide study of the prevalence of clefts in Nigeria, but we are unaware of any study of its epidemiology targeting a single indigenous Nigerian ethnic group or a recent Nigerian study indicating the incidence of clefts among live births.
Nigeria is the most populous African nation, with an estimated population of 140,431,790 in 2006 when the last national census was conducted. However, more recent estimates indicate over 180,000,000. With over 300 ethnic groups, the nation is a mix of various cultures. The Igbos account for approximately 18% of her population and are the predominant inhabitants of Enugu, the third most populous of the five southeastern states. Hence, this study aims to provide more recent data on the epidemiology of clefts and estimates its incidence among Igbos.
| Materials and Methods|| |
This is a descriptive transversal study done between January 2007 and December 2011. Patients receiving free treatment at NOHE have their records prospectively uploaded on Smile Train Express which has a secure database. This was accessed, as well as birth records at ESUTTH within this period. All newborns were examined by the doctor/midwife on duty and congenital malformations noted. Subsequently, the data were entered into the birth register. Five cleft births were thus identified. The sixth (cleft palate) had been missed at birth and was noted by one of the authors on presentation at the surgical outpatient clinic within the period.
Furthermore, case notes of all patients presenting at NOHE were reviewed. Information was also gathered from records of clinical attendance and interviews with patients' mothers. The gender, birth order, maternal and paternal ages at time of birth, type of cleft, and presence of family history of cleft, and an evaluation of the socioeconomic status (SES) of the subjects were obtained from the studied materials. SES is usually a categorization based on education, profession, and perceived economic status of the family, and this is strongly hinged on the qualities of the family head or chief income earner. Two of these factors, income and educational status,, were used in some instances to assess SES. There is no consensus on various socioeconomic classifications in Nigeria because of the unstructured nature of the society. We adopted the Kuppuswamy scale using the education and occupation of the family head or chief income earner as independent determinants since the education and occupation of head of family are not prone to change significantly over time, and that the income ranges in the scale lose their relevance following the depreciation in the value of the naira. The modified weighted scale [Table 1] was applied and patients assigned to the upper, upper middle, lower middle, lower, and poor class based on their score ranging from 2 to 17.
|Table 1: Socioeconomic Class Scale, modeled after Kuppuswamy socioeconomic scale (Urban, 1976)|
Click here to view
Names were used as criteria to classify patients as Igbo, and those with non-Igbo names being excluded in arriving at the ethnic incidence. This was a limitation of the study. Data collected were entered into Excel (Microsoft, Raymond, WA), and statistical analysis performed using the Statistical Package for the Social Sciences version 22.0 (SPSS Inc., Chicago, IL, USA).
| Results|| |
A total of six clefts (all Igbos) were noted among the 5810 live births recorded over the five-year period, giving an incidence of 1:968 (1.03: 1000). Two hundred and three patients with non-Igbo names were excluded leaving 5607 live births (96.5% of all live births); and an incidence for Igbos derived as 1:934 (1.07: 1000). All live births at ESUTTH were recorded, and the data were taken from the hospital's birth record for the period.
A total of 317 cleft surgeries were recorded in the period, of which 262 were performed for Igbo patients. The distribution of clefts was as follows: isolated cleft lip (ICL) 58%, cleft lip and palate (CLP) 32%, and isolated cleft palate (ICP) 10%. Left-sided clefts (LCs) were most predominant, with a ratio of 1.75:1.1:1 compared with bilateral (BLC) and right-sided clefts (RCs), respectively [Figure 1].
The gender distribution was male 139 and females 123. This reflects a slightly higher male incidence with a male/female ratio of 1.13:1. In the different types of cleft, there was also variance in the gender distribution with male:female for ICL 1.3:1, CLP 1.1:1, and ICP 1:1.8 [Figure 2]. The age distribution of patients was 110 infants, 101 children (2–18), 51 adults (≥19), and the oldest being 60 years old. Mean age for females was 9.5 ± 13.0 and for males 9.0 ± 12.1. The age distribution of clefts showed that, in all age groups, ICL and ICP had the highest and least frequency, respectively [Table 2].
Parental age at the time of birth for infants and children were analyzed. Mean maternal age was 28.88 years ± 5.81 (range: 17–47) and mean paternal age was 38.55 years ± 6.92 (range: 23–58). In 9.5%, there was a positive family history of clefting, with first-degree relative accounting for 6% and ICL being most predominant.
In the birth order, in all the types of clefting, the first position was the most common (overall 24%) but not significant (P = 0.932). The highest birth order recorded was 13th. There was a pattern of decreasing frequency of clefting as the birth order increased [Figure 3].
Over 80% belong to the poor and low socioeconomic group. As the SES worsened, there was increasing clefting [Figure 4]. The total live birth at ESUTTH rose from 1233 in the year before the inception of the free maternal and child care program to 2252 in its second year but then declined sharply to 397 with redefinition of the program to involve only referrals from primary and secondary institutions. SES was shown to be a significant determinant of clefting (P = 0.0001, confidence interval: 95%). Most of the patients treated (and 78% of all adult patients) were from Enugu and Ebonyi state [Table 3] and [Table 4].
| Discussion|| |
The methodology follows Iregbulem's study. The incidence here is higher than his. His study was also a decade after the civil war which killed over 3 million Igbos; hence the full potential for cleft may not have been expressed at that time in Enugu. Our rates are, however, close to other studies in Ibadan, and international estimates but less than recent studies from Benin city and Ghana. It may be from a hypothesis by Iregbulem of an increase in the incidence of CLP in Nigeria due to the onward propagation of the genetic trait and increased survival of the newborn. The NigeriaCRAN prevalence survey/study was a nationwide multicenter one which pooled various ethnic groups together but was reported based on geopolitical zone. Although the study in Benin pooled over six ethnicities, peculiar differences were observed in the pattern of clefting among the Urhobo and Bini. Such ethnic and regional differences in cleft and its birth incidences are well recognized.,,, The methodology of the NigeriaCRAN and Benin reports differ from ours in that they are prevalence studies, while we focused on incidence based on hospital birth cleft records. Iregbulem collected data from a tertiary hospital like we did, but there was no free treatment available in that hospital then. As a result, women in the lower socioeconomic group, who have been noted to give birth to most of the cleft children,, were most likely much more represented in our series.
The finding of >80% of parents belonging to a low socioeconomic class aligns with a previous report in Enugu, but differs from reports from Benin and Abraka. The sharp decline in recorded births following the redefinition of the free treatment in our view indicates the lower socioeconomic class who benefit most from free treatment programs had largely stopped coming. The pattern of higher prevalence with lower SES has been reported in other countries.,, This decline may have also affected the study results. With a reduction in patronage by pregnant women from whom majority of cleft births are expected, there may have been a lower incidence obtained than would have been if the free treatment had continued unchanged. Further studies on congenital anomalies from this and other geographic locations should involve multiple centers. This should include all tiers of health-care birth institutions, as well as the traditional birthplaces, this will prevent the inadvertent exclusion of birth anomalies more likely to be found among people who patronize differing tiers of health-care facilities. Such studies should have a physician examine all live births as with Iregbulem. Our findings also differ from that found among African Americans. We believe that the African Americans have a better socioeconomic standing than Igbos living in Nigeria where more people live below the poverty line; estimated to be about 63% in 2010 and is expected to increase significantly with the growing population. It is noteworthy that most patients were from Enugu and Ebonyi states which are ranked in the bottom of the five states in the southeastern region based on socioeconomic indices. Ebonyi state is in the bottom five of all the states in Nigeria. The other four are from northern Nigeria. The volume of cleft repairs done in northern Nigeria, which has the worst poverty indices, is greater than the combined number done in the southwestern and southeastern part which have much better socioeconomic indices.
It is possible our study missed out submucous clefts and bifid uvula since not all births were personally examined by the authors as Iregbulem did. However, these are rarer forms and improvements in birth registries, and clinical examination of all births by surgeons should pick such up.
In our study, ICL was most frequent, as with the study done by Iregbulem and the Ghana study. It differs from studies done in Enugu, Brazil, Benin, and Iran where CLP was the most common. In all studies, as in ours, ICP was the least frequent and more frequent among female patients.,, Our slight male preponderance agrees with other works from Nigeria, and other countries.,,, As with previous studies, most of the clefts were left sided.,,, The ratio of laterality (LC > BLC > RC) and the tendency for males to have more LC and females RCs have been reported in a previous study.
This study had a positive family history in 9.5%. This result is close to the Nigeria-wide study which reports 10.4%, lower than figures from the Benin, and the Ghana studies, but higher than that from Iregbulem's study where there was a positive family history of 6%. Higher percentages of positive family history in other studies were attributable to consanguineous marriages, which is very uncommon in southeastern Nigerian. The low incidence of familiarity may stem from people's reluctance to divulge information on such anomalies out of fear. A study indicates the Igbos belong to a set of ethnic groups in Nigeria that attribute the etiology to Spiritism, the mother, or the child. However, it may point to a greater role of environmental rather than genetic factors in cleft deformities seen among Igbos. Studies have shown variations in the genetic factors seen in Nigerians as against previously reported literature. The environmental trigger in Nigeria is yet to be elucidated, but alcohol and herbal medications in pregnancy have been implicated in Nigeria and other studies.,,,
As with Iregbulem's study, most of the subjects belonged to the first birth order. Albeit this has not been proven to be significant, the finding of a low socioeconomic group of parents may suggest a high role of nurture in the multifactorial etiology of clefts among Igbos; a view shared by Iregbulem.
| Conclusion|| |
The estimated incidence of clefts in Enugu and its demographics differs from previous reports among other ethnicities in Nigeria. This data will be useful in advocacy and planning for care of cleft patients in the region. Similar studies among other groups are encouraged and will be facilitated by the establishment of a national register for clefts.
| Acknowledgements|| |
We appreciate Drs Michael Chime, Charles Ilogebe, and Francis Ani for their assistance with data collection; and SmileTrain for funding cleft care at NOHE.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Butali A, Mossey PA, Adeyemo WL, Jezewski PA, Onwuamah CK, Ogunlewe MO, et al.
Genetic studies in the Nigerian population implicate an MSX1 mutation in complex oral facial clefting disorders. Cleft Palate Craniofac J 2011;48:646-53.
Marazita ML, Goldstein AM, Smalley SL, Spence MA. Cleft lip with or without cleft palate: Reanalysis of a three-generation family study from England. Genet Epidemiol 1986;3:335-42.
Rajabian MH, Aghaei S. Cleft lip and palate in Southwestern Iran: An epidemiologic study of live births. Ann Saudi Med 2005;25:385-8.
] [Full text]
Mossey P, Little J. Addressing the challenges of cleft lip and palate research in India. Indian J Plast Surg. 2009;42(3):S9-S18.
Vanderas AP. Incidence of cleft lip, cleft palate, and cleft lip and palate among races: A review. Cleft Palate J 1987;24:216-25.
Cobourne MT. The complex genetics of cleft lip and palate. Eur J Orthod 2004;26:7-16.
Iregbulem LM. The incidence of cleft lip and palate in Nigeria. Cleft Palate J 1982;19:201-5.
Omo-Aghoja VW, Omo-Aghoja LO, Ugboko VI, Obuekwe ON, Saheeb BD, Feyi-Waboso P, et al.
Antenatal determinants of oro-facial clefts in Southern Nigeria. Afr Health Sci 2010;10:31-9.
Butali A, Adeyemo WL, Mossey PA, Olasoji HO, Onah II, Adebola A, et al.
Prevalence of orofacial clefts in Nigeria. Cleft Palate Craniofac J 2014;51:320-5.
National Population Commission of Nigeria. 2006 Population and Housing Census of the Federal Republic of Nigeria: National and State Population and Housing Tables, Federal Republic of Nigeria - Abuja: National Population Commission (NPC), 2009.
United Nations, Department of Economic and Social Affairs, Population Division (2015). World Population Prospects: The 2015 Revision, Key Findings and Advance Tables. ESA/P/WP.241.
Yoon YS, Oh SW, Park HS. Socioeconomic status in relation to obesity and abdominal obesity in Korean adults: A focus on sex differences. Obesity (Silver Spring) 2006;14:909-19.
Chukwuonye II, Chuku A, Okpechi IG, Onyeonoro UU, Madukwe OO, Okafor GO, et al.
Socioeconomic status and obesity in Abia State, South East Nigeria. Diabetes Metab Syndr Obes 2013;6:371-8.
Bairwa M, Rajput M, Sachdeva S. Modified kuppuswamy's socioeconomic scale: Social researcher should include updated income criteria, 2012. Indian J Community Med 2013;38:185-6.
] [Full text]
Gupta B, Antia AU. Incidence of congenital heart disease in Nigerian children. Br Heart J 1967;29:906-9.
Agbenorku P, Yore M, Danso KA, Turpin C. Incidence of orofacial clefts in Kumasi, Ghana. ISRN Plast Surg 2013;2013:1-6.
Croen LA, Shaw GM, Wasserman CR, Tolarová MM. Racial and ethnic variations in the prevalence of orofacial clefts in California, 1983-1992. Am J Med Genet 1998;79:42-7.
Onah II, Opara KO, Olaitan PB, Ogbonnaya IS. Cleft lip and palate repair: The experience from two West African sub-regional centres. J Plast Reconstr Aesthet Surg 2008;61:879-82.
Acuña-González G, Medina-Solís CE, Maupomé G, Escoffie-Ramírez M, Hernández-Romano J, Márquez-Corona Mde L, et al.
Family history and socioeconomic risk factors for non-syndromic cleft lip and palate: A matched case-control study in a less developed country. Biomedica 2011;31:381-91.
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]
Litwack J. Nigeria economic report. Nigeria economic report ; no. 1. Washington DC ; World Bank, 2013.
Martelli-Júnior H, Bonan PR, Santos RC, Barbosa DR, Swerts MS, Coletta RD, et al.
An epidemiologic study of lip and palate clefts from a Brazilian reference hospital. Quintessence Int 2008;39:749-52.
Magdalenić-Mestrović M, Bagatin M. An epidemiological study of orofacial clefts in Croatia 1988-1998. J Craniomaxillofac Surg 2005;33:85-90.
Shapira Y, Lubit E, Kuftinec MM, Borell G. The distribution of clefts of the primary and secondary palates by sex, type, and location. Angle Orthod 1999;69:523-8.
Gregg T, Boyd D, Richardson A. The incidence of cleft lip and palate in Northern Ireland from 1980-1990. Br J Orthod 1994;21:387-92.
Fadeyibi IO, Adeniyi AA, Jewo PI, Saalu LC, Fasawe AA, Ademiluyi SA, et al.
Current pattern of cleft lip and palate deformities in Lagos, Nigeria. Cleft Palate Craniofac J 2012;49:730-5.
Elahi MM, Jackson IT, Elahi O, Khan AH, Mubarak F, Tariq GB, et al.
Epidemiology of cleft lip and cleft palate in Pakistan. Plast Reconstr Surg 2004;113:1548-55.
Farina A, Wyszynski DF, Pezzetti F, Scapoli L, Martinelli M, Carinci F, et al.
Classification of oral clefts by affection site and laterality: A genotype-phenotype correlation study. Orthod Craniofac Res 2002;5:185-91.
Oginni FO, Asuku ME, Oladele AO, Obuekwe ON, Nnabuko RE. Knowledge and cultural beliefs about the etiology and management of orofacial clefts in Nigeria's major ethnic groups. Cleft Palate Craniofac J 2010;47:327-34.
Butali A, Mossey PA. Epidemiology of orofacial clefts in Africa: Methodological challenges in ascertainment. Pan Afr Med J 2009;2:5.
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.
DeRoo LA, Wilcox AJ, Drevon CA, Lie RT. First-trimester maternal alcohol consumption and the risk of infant oral clefts in Norway: A population-based case-control study. Am J Epidemiol 2008;168:638-46.
Lorente C, Cordier S, Goujard J, Aymé S, Bianchi F, Calzolari E, et al.
Tobacco and alcohol use during pregnancy and risk of oral clefts. Occupational Exposure and Congenital Malformation Working Group. Am J Public Health 2000;90:415-9.
[Figure 1], [Figure 2], [Figure 3], [Figure 4]
[Table 1], [Table 2], [Table 3], [Table 4]