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Annals of African Medicine Vol. 12, January-March, 2013

Epidemiology of adult cleft patients in North‑western Nigeria: Our experience

Sunday O. Ajike, Rafel A. Adebola, Akinwale Efunkoya, Joshua Adeoye, Olumide Akitoye, Ngutu Veror

Grassroot Smile Initiative, Magaji Rumfa Street, Kano, Kano State, Nigeria

Correspondence to: Prof. S. O. Ajike, Maxillofacial Unit, Ahmadu Bello University Teaching Hospital, Zaria, Kaduna State, Nigeria. E‑mail: mfujiks@gmail.com

Abstract

Background/Objective: To review cleft lip and palate procedures over a three-year (2008-2010) partnership between the smile train and our organization, the Grasssroot Smile Initiative (GSI). Method: A three-year retrospective study (2008-2010) involving 79 adult patients with clefts. Results: Seventy nine (14.4%) of 550 patients with orofacial clefts seen and treated within a three-year period were adults with age range of 17 to 81 years; mean 31.45 ± 13.09. Majority were between 20 and 39 years. There were 54 (68.4%) males and 25 (31.6%) females, with the male:female ratio of 2.2:1. Analysis of the cleft types/site revealed 35 (44.3%) lip alone, 22 (27.8%) lip and alveolus, 7 (8.9%) lip and palate and 15 (19%) palate alone. Seven (8.9%) of these patients had other relatives with clefts. Sources of information were friends and relatives; 33 (41.8%), radio; 18 (22.8%), charity organization/NGO; 13 (16.5%), hospitals/physicians; 5 (6.3%), and others; 10 (12.7%). 57 patients with lip clefts had surgery under local anesthesia while the remaining 22 patients were done under general anesthesia. All clefts of the lip were repaired using the Millard advancement rotational flap for complete cleft, simple straight line closure for incomplete and double layer closure for the palate. Conclusion: The incidence of adult patients with orofacial cleft is not rare in our community, probably due to limited access to specialized health care facilities, poverty and ignorance. Furthermore, some of these patients are not aware that these facial defects can be repaired. The advent of the smile train organization and free services has resulted in this harvesting phenomenon.

Keywords: Adults, clefts, lip, palate

Résumé

Contexte/objectif: À une fente labiale et palatine des procédures d'examen sur trois ans (2008-2010) de partenariat entre le train de sourire et de notre organisation, l'Initiative de Smile Grasssroot (GSI).Méthode: Une triennale étude rétrospective portant sur 79 patients adultes avec fentes (2008-2010).Résultats: Soixante-dix neuf (14,4%) de 550 patients avec bucco-faciale fentes vus et traités dans un délai de trois ans étaient des adultes âgés de 17 à 81 ans ; moyenne ± 31.45 13,09. Majorité étaient âgés de 20 à 39 ans. Il y avait 54 (68,4%) hommes et 25 femmes (31,6%), avec le mâle: Femelle rapport de 2.2:1. Analyse du site fente types/a révélé 35 lèvre (44,3%) seulement, 22 (27,8%) lèvre et alvéole, 7 lèvre (8,9 %) et palais et 15 Palais (19%) seulement. Sept (8,9%) de ces patients avaient des autres parents avec fentes. Sources d'information étaient des amis et des parents ; 33 (41,8%), radio ; 18 (22,8%), organisation de bienfaisance/ONG ; 13 (16,5%), hôpitaux/médecins; 5 (6,3%) et autres ; 10 (12,7%). 57 patients avec fentes de lèvre avaient chirurgie sous anesthésie locale, tandis que les autres 22 patients ont été effectuées sous anesthésie générale. Toutes les fentes de la lèvre ont été réparées à l'aide de la Millard avancement rotation Rabat pour fissure complète, la fermeture de la ligne droite simple fermeture incomplète et double couche pour le palais. Conclusion: L'incidence des patients adultes atteints de bucco-faciale une fente n'est pas rare dans notre

Annals of African Medicine Vol. 12, No. 1; 2013Access this article online

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DOI: 10.4103/1596-3519.108243

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Original Article

Vol. 12, January-March, 2013 Annals of African Medicine Annals of African Medicine

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Ajike, et al.: Adult clefts in north‑wetern Nigeria

Introduction

Clefts of the lip and palate are the most common congenital anomalies of the orofacial region.[1] Reports of adult clefts are rare in the developed countries. An extensive search of the English literature with the key words cleft, lip, palate, adult revealed only few documented cases, all from the developing world.[2,3] In the developed world, early childhood treatment with well laid out protocol is the rule whereas in our community this is an exception probably due to ignorance, poverty, limited access to specialized healthcare facilities and non‑inclusion of clefts in the National health scheme. Acceptable protocol includes repair of cleft lip at 10 to 12 weeks, primary palatoplasty at 9 to 12 months.[3] This protocol aims to address both the aesthetic and speech defects respectively while preventing sociopsychological effects on the patients. In our environment, the patients were not boarded psychosocially.

The persistence of this anomaly to adulthood in our environment has resulted in some surgical challenges because of the wide width of the clefts; hence, there is a need to document our experience in the management of these adults.

Materials and Methods

We carried out a retrospective study of 79 adults with orofacial cleft seen and managed by the Grassroots Smile Initiative in conjunction with Smile Train Organization between January 2008 and December 2010. The case files of these 79 patients were analyzed for the types/site of clefts, laterality, age, sex, method of anesthesia and the surgical repairs. The cleft deformities were classified using anatomical descriptions: Cleft lip, cleft lip and alveolus, cleft lip and palate, cleft palate (hard or soft), and laterality. All clefts of the lip were repaired using the Millard advancement rotational flap for complete cleft, simple straight line closure for incomplete and double layer closure for the palate. The data extracted are presented.

Results

Of the 550 orofacial cleft patients seen during the period of study, 79 patients (23 (29.1%) in 2008,

31 (39.1%) in 2009 and 25 (31.6%) in 2010) were adults with an age range of 17‑81 years old. The mean age was 31.45 13.09 and a median of 29. The age range for females was 17‑81 years, mean of 35.79 3.54 while that of the males was 17‑71 years with a mean of 29.79 11.72. The majority 52 (65.8%) were in the 3rd and 4th decades of life. There were 54 (68.4%) males and 25 (31.6%) females with a male:female ratio of 2.2:1 [Table 1]. The oldest and youngest were both females.

There were 35 (44.3%) cleft lip alone, 22 (27.8%) cleft lip and alveolus, 7 (8.9 %) cleft lip and palate and 15 (19 %) cleft palate alone [Table 2]. Regarding laterality, the left‑sided cleft (68.6%) was the most common [Table 3]. Seven (8.9%) of these patients had other relatives with clefts. Sources of information were friends and relatives; 33 (41.8%), radio; 18 (22.8%), charity organization/NGO; 13 (16.5%), hospitals/physicians; 5 (6.3%),

communauté, probablement en raison d'un accès limité aux établissements de soins spécialisés, la pauvreté et l'ignorance. En outre, certains de ces patients ne sont pas au courant que ces défauts du visage peuvent être réparés. L'avènement de l'organisation de train de sourire et services gratuits a entraîné ce phénomène récolte.

Mots clés: Adultes, fentes, lèvres, bouche

Table 2: Types of clefts as seen in 79 adult patients

Types of clefts No %Lip alone 35 44.3Unilateral 28Complete 21Incomplete 7Bilateral 7Complete 6Incomplete 1Lip and alveolus 22 27.8Unilateral 19Bilateral 3Lip and palate 7 8.9Unilateral lip and palate 4Bilateral lip and palate 3Palate alone 15 19.0Hard 11Soft 4Total 79 100

Table 1: Age and gender distribution of 79 patients

Age Gender Total

M F17‑19 9 3 1220‑29 21 8 2930‑39 16 7 2340‑49 3 2 550‑59 3 3 660‑69 1 1 270‑79 1 0 180‑89 0 1 1Total 54 25 79

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Ajike, et al.: Adult clefts in north‑wetern Nigeria

and others; 10 (12.7%) [Table 4]. Fifty seven patients with lip clefts had surgery under local anesthesia while the remaining 22 patients were done under general anesthesia. All clefts of the lip were repaired using the Millard advancement rotational flap for complete cleft, simple straight line closure for incomplete and double layer closure for the palate.

Seven of these patients had patients with clefts, 2 females had boys, another 2 females had nephews and the remaining 3 men had 2 children with various forms of clefts.

Discussion

Adult clefts are very rare. An extensive search of the English literature with the keywords clefts, lip, and palate, adult revealed only few reports, all from the developing world.[2‑4] Our study had revealed a high incidence of adult clefts when compared to the reports from other studies.[2,3,5] Obuekwe and Akpata[2] from Benin, Nigeria and Orkar, et al.[5] from Jos, Nigeria reported an adult incidence of 4.9% with the age range of 18‑34 years, mean of 23.3 and 3 (2.8%) age range 18‑26 years, mean 22.3 years respectively compared to our study which documented 14.4%. However, our study is similar to that of Iregbulum[4] who reported 14% as adults. The high incidence of adult clefts in our study is due to poverty, ignorance and lack of access to medical specialist, and the harvesting phenomenon within the North west and North central of Nigeria, also the government of Nigeria and most African countries do not give clefts priority in their health scheme/program coupled with the fact that this surgical outreaches were free with several public announcements. Other Nigerian authors[2,4] had earlier implicated ignorance and financial constraints and underreporting.

Most studies have favored a male predominance,[6,7] but some have found a female predominance.[1,2] In our study, a male predominance in all the age groups was observed. A family history of clefts was reported in 8 (8.9%) of our patients while Karmee, et al.[6] and Eshete, et al.[1] documented 23 and 3 (3.1%) cases respectively. In consistence with most studies,[2,3,6,‑8] this report found majority (68.8%) to be left sided, a left‑sided clefting and a right‑sided clefting at a ratio 2.2:1 [Table 3]. In this study, only 8.9% of the patients had relatives with cleft, whereas Rajabian and Sherkat[7] reported 20.1%; 147 (8.8%) in first degree and 189 (11.3%) for the second degree relatives with clefts but similar to Iregbulum’s[4] report of 6%.

Regarding the source of information [Table 4], we had the initial belief that most of the people in the rural areas at least have radio as a source of information in the northern part of our country, but unfortunately only 22.8% of our patients heard through the medium compared with 41.8% that heard through and had seen friends and relatives operated. This is in contrast to the report of Aziz, et al.[3] who recruited their patients through newspapers and words of mouth. These people were actually convinced by seeing other operated cases before turning up for the surgery. The apathy to this surgery was that some of the patients wandered how and where tissues for closure would be got from as some even believe that these tissues were from the dead.

Different surgical options for the repair of clefts have been proposed and no option is applicable to all of the patients. Each patient should be assessed properly and the surgical approach be individualized. However, regardless of the surgical approach, the surgical purpose of the cleft lip repair is mainly for aesthetics while that of the palate is for adequate velopharyngeal function and development of normal speech. Patients with both lip and palatal clefts had two stage procedures, while some authors have practiced a simple stage. Complete cleft lip was repaired using the Millard’s rotation advancement flap [Figure 1] with repair of the nasal floor. For the incomplete, a simple straight line closure was performed [Figure 2], while bilateral clefts were repaired using the two‑flap procedures with satisfactory result for the broad African nose type when compared with the Caucasian pointed tip. However, no patient reported back for secondary repair of the nose. They were probably satisfied; further strengthening the acceptability of this anomaly in our local environment, as this was used as means of begging for alms in the public. It is common practice for some of the patients to refuse surgery.

Table 3: Laterality of cleft lips

Laterality of lip defects Right (%) Left (%)Lip alone

Complete 5 (29.4) 6 (11.8)Incomplete 5 (9.8) 2 (3.9)Lip and alveolus 13 (25.5) 6 (11.8)Lip and palate 2 (3.9) 2 (3.9)Total 35 (68.6) 16 (31.4)

Table 4: Sources of information

Source of information No (%)Charity organization 13 (16.5)Friends and relatives 33 (41.8)Radio 18 (22.8)Hospital/physician 5 (6.3)Others (newspaper, flyers, etc.,) 10 (12.6)Total 79 (100)

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Ajike, et al.: Adult clefts in north‑wetern Nigeria

Regarding the palatal clefts, the complete unilateral had the 2‑flap palatoplasty [Figure 3], two of the incomplete clefts (soft palate) had a V‑Y push back palatoplasty as described by Kilner and Wardill,[9] while the remaining two incomplete clefts had the Von Langenbeck palatoplasty. Sale[10] in 2010 reported the successful use of u‑shaped flaps for the incomplete clefts in 34 patients. Our experience compares favorably with the Bangladesh study.[3] Based on our experience and reports by other authors, adult clefts are usually wider due to maxillary growth; however, there are more bulky soft tissues to handle. For the lips, adequate separation into the skin, muscles and mucosa allowed easy layered closure coupled with back cut of the c‑flap in some cases to achieve coverage of wide clefts with little tension and satisfactory aesthetic result [Figure 1b]. For the palatoplasties, adequate closure was achieved by fracturing the pterygoid hamulus in eight cases and dissection of the greater palatine vessels [Figure 3] from its canal combined with osteotomy of the canal medially to free the palatal fingers. In addition to the hamulus fracture, some authors[3] have used biomaterials as adjuncts to facilitate movement of the palatal finger mucosa, while Randall, et al.[11] used the Furlow double‑reverse Z‑plasty and Tan, et al.[12] combined the Von Langenbeck procedure with a Z‑plasty to achieve closure.

Jeffery and Borman[13] reported that the incidence of palatal fistulae following primary repair of the palate ranges from 18% to 34%, in our study there were only 2 (13.3%) cases. This low incidence is a result of our surgical method. Haapanen[14] started that the method of palatal repair and age at surgery has a major effect on the speech outcome. Normal speech was attained in our patients over a period of time.

All the 57 cases done under local anesthesia with 5% dextrose saline had cleft lip defects while the remaining 22 with palatal clefts had surgery under general anesthesia. Cost of surgery under local anesthesia was a third the cost of general anesthesia. Hence, this afforded us more surgical funding for more patients. However, only surgical procedures were done, there is a need for multidisciplinary team of specialists. Existence of this team would provide optimal treatment both in aesthetics and function. We have tried to achieve this, despite limited resources of manpower.

In conclusion, the incidence of adult patients with orofacial clefts is not rare in our community probably due to limited access to specialized health care facilities, poverty and ignorance. Furthermore, some of these patients are not aware that these facial defects can be repaired. The advent of the smile train organization and free services has resulted in this harvesting phenomenon. This report demonstrated the obstacles to the surgical procedures done in a poor resource country. There is the need for the inclusion of the cleft management in the national health scheme.

References

1. Eshete M, Gravenm PE, Topstad T, Befikadu S. The incidence of c; left lip and palate in Addis Ababa., Ethiopia. Ethiop Med J 2011;49:1-5.

2. Obuekwe O, Akpata O. Pattern of cleft lip and palate (corrected) in Benin City, Nigeeria. Centr Afri J Med 2004;50:65-9.

3. Aziz SR, Rhee ST, Redai I. Cleft sutgery in rural bangladesh: Reflections and exeperiences. J oral Maxillofac Surg 2009;67:1581-8.

4. Iregbulem LM. The incidence of cleft lip and palate in Nigeria. Cleft Palate J 1982;19:201-5.

5. Okar KS, Ugwu BT, Momoh JT. Cleft lip and palate: The Jos experience. East Afr Med J 2002;79:510-3.

Figure 1: (a) Preoperative unilateral cleft lip. (b) Post operative unilateral cleft lip

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Figure 2: (a) Pre operative bilateral cleft of the upper lip. (b) Post operative bilateral cleft of the upper lip

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Figure 3: (a) Pre operative cleft of the hard and soft palate. (b) Post operative cleft of the hard and soft palate

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Ajike, et al.: Adult clefts in north‑wetern Nigeria

6. Karimee YA, Saedi B, Sazegar AA, Mehdipour P. Epidermiological aspects of cleft lip and palate in Iran. Acta Med Iran 2011;49:54-8.

7. Rajabian MM, Sherkat M. Epidermiological study of ral cefts in Iran: An ananlysis of 1669 cases. Cleft Palate Craniofac J 2000;37:191-6.

8. Yi NN, Yeow VK, Lee ST. Epidermiology of cleft lip and palate in Singapore- a 10 year hospital based study. Ann Acad Med Singapore 1999;28:655-9.

9. Wardill EM. The tecnique of operation for cleft palate. Br J Surg 1937;25:117.

10. Sale A. Correction of incomplete cleft palate by u-shaped flap palatoplasty. Afr J Paediatr Surg 2010;7:185‑7.

11. Randall P, La Rossa D, Solomon M, Cohen M. Experience with the Furlow double-reversing z-plasty for cleft palate repair. Plas Reconstr Surg 1986;77:569-76.

12. Tan WQ, Xu JH, Yao JM. The single Z-plasty for cleft repair: A preliminary report. Cleft Palat Craniofac J 2012;49:635-9.

13. Jeffery SL, Boorman JG, Dive DC. The use of cartilage grafts for closure of cleft palate fistula. Br J Plast Surg 2000;53:551-4.

14. Happanen ML. Effect of method of cleft palate repair on the quality of speech at the age of 6 years. Scad J Plat Reconstr Surg Hand Surg 1995;29:245-50.

Cite this article as: Ajike SO, Adebola RA, Efunkoya A, Adeoye J, Akitoye O, Veror N. Epidemiology of adult cleft patients in North-western Nigeria: Our experience. Ann Afr Med 2013;12:11-5.Source of Support: Smile train organization, Conflict of Interest: None declared.