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A PROSPECTIVE STUDY OF CLEFT LIP AND PALATE AMONGST CHILDREN AND ADULTS IN NORTH-EASTERN NIGERIA.

Byline: ADESINA OA, EFUNKOYA AA, OMEJE KU and SULEIMAN AR

Abstract

The aim of this study was to review the peculiarities in the presentation and management of children and adults with orofacial clefts attending a tertiary care facility in North Eastern Nigeria. Sixty-seven consecutive patients; both children and adults with cleft lip and palate were studied at the maxillo-facial surgery clinic of University of Maiduguri Teaching Hospital, Maiduguri over a period of one year, with a self-administered questionnaire. Fifty-one (76.1%) patients below age 12 years regarded as children were seen and sixteen (23.9%) were adults (13 years and above).

Isolated cleft lip was the most common (62.7%) type of cleft in both groups of patients. Congenital anomalies were about six times more common in children (25.4%) compared with adult patients (4.5%). Associated medical conditions (25.4%) were only noticed in children. Five out of six wound breakdown at the vermilion was noticed in children. Out of the ten cases of hypertrophic scar seen, seven were observed in children. About 54% of the cleft lips repaired in children were carried out under local anesthesia (LA) with conscious sedation, while the remaining patients were treated under general anaesthesia. All the cleft lips repairs in adult were carried out under local anaesthesia with conscious sedation. Vermilion notching was noticed in five adults and one child.

Two cases of stitch marks were reported in adults while only one case was seen in children. Study revealed similarity in cleft pattern in both children and adults. However, the low incidence in adults may be attributed to effectiveness of the free surgery services in this region. Higher number of children operated under general anesthesia reflects the peculiarity of children in terms of increased chances of not cooperating with treatment under LA.

Key Words: Cleft lip, cleft palate, management, children, adults.

INTRODUCTION

The presence of facial clefts has been associated with psychological and emotional trauma, probably due to obvious gross cosmetic deformity seen in such patients.1 These patients also suffer undernourishment, speech difficulties, otology disorders and impaired facial growth among other problems.2,3

The management of cleft lip and palate aims at restoring a normal facial appearance, speech and hearing. It also eliminates the difficulties encountered in feeding of such patients. This is best achieved under a multidisciplinary team usually composed of a maxillofacial surgeon, plastic surgeon, speech pathologist, orthodontist, social worker, audiologist and nutritionist.1

Most surgeons favour early repair due to positive psychological influence on parents and siblings, the moulding action of the united lip on the maxillary arch and the greater healing potential of the very young tissues.4 However anecdotal reports reveal that late presentation of cleft lip and palate patients may be attributed to financial constraints and ignorance of the patients about free cleft services among other factors. This study aimed to determine the peculiarities in the presentation and management of children and adults with orofacial clefts attending a tertiary health care facility in North Eastern Nigeria.

METHODOLOGY

This was a hospital-based study conducted at the University of Maiduguri Teaching Hospital (UMTH), for a period of one year. UMTH is the main tertiary health care centre in North Eastern Nigeria where free cleft lip and palate services sponsored by a non-governmental organisation are being offered. The study protocol was approved by the research and ethics committee of the hospital and; informed consent forms were obtained from the adult patients and the parents of participating children. Sixty-seven patients consisting of children and adults with orofacial cleft who presented between October 2013 and September 2014 were included in the study. Patients previously operated requiring secondary surgery were excluded. Relevant clinical history was collected from the mothers of the child patients and directly from the adult patients.

Patients who were 12 years and below were regarded as children and those above 12 years as adults. This categorization was only based on the patients' ability to tolerate surgical repair under local anaesthesia.

All patients were reviewed and evaluated by the various specialists in the hospital cleft lip and palate team. The cleft deformities were classified using Kernahan and Stark classification (1958).5 Data collected included age, gender, type of cleft, associated medical and congenital anomalies, and surgical complications.

Data analysis was conducted using statistical package for social sciences (SPSS) version 13.06. Statistical significance was inferred at P<0.05

RESULTS

Sixty-seven patients, 51 children (76.1%) and 16 adults (23.9%) with cleft lip and palate were evaluated during the study period. Their ages ranged from a day old to thirty five years. The median age was 1.75 years for children and 18.5 years for adults. There was significant statistical difference between the number of children and adult patients seen. (P = 0.003). The number of adult males seen was twice the number of their female counterparts whilst gender distribution was almost equal in children. Type of cleft seen were distributed as cleft lip only (42 cases, 62.7%), cleft lip and palate (22 cases, 32.8%) and isolated cleft palate (3 cases, 4.5%). Table 1 shows distribution of the various cleft types in children and adults by gender.

Unilateral clefts of the lip were repaired using Millard's technique7 and bilateral clefts of lip by Millard's myoplastic technique.8 Clefts of the palate were repaired using von Langenbeck's technique.4

TABLE 1: GENDER DISTRIBUTION AND TYPE OF CLEFT SEEN IN CHILDREN AND ADULTS

Type of cleft###Children###Adult###Total (%)

###Male (%)###Female (%)###Male (%)###Female (%)

Cleft lip only###12 (17.9)###18 (27.0)###8 (11.9)###4 (5.9)###42 (62.7)

Cleft lip and palate###12 (17.9)###7 (10.4)###3 (4.5)###0 (0.0)###22 (32.8)

Cleft palate only###0 (0.0)###2 (3.0)###1 (1.5)###0 (0.0)###3 (4.5)

Total###24 (35.8)###27 (40.4)###12 (17.9)###4 (5.9)###3 (4.5)

TABLE 2: ASSOCIATED CONGENITAL ANOMALIES IN CHILDREN AND ADULT WITH CLEFT LIP AND PALATE

Anomalies###Children###Adult###Total (%)

###CL###CL/P###CP###CL###CL/P###CP

Strabismus###1###1###1###0###0###0###3 (15.0%)

Anopthalmia###0###0###0###0###0###2###2 (10.0%)

Proptosis###0###1###0###0###0###0###1 (5.0%)

Low set ears###0###1###0###0###0###0###1 (5.0%)

Malformed ears###1###1###0###0###0###0###2 (10.0%)

Flattened nasal bridge###1###2###0###1###0###0###4 (20.0%)

Frontal bossing###1###1###0###1###0###0###3 (15.0%)

Polydactyl###0###0###0###1###0###0###1 (5.0%)

Lower lip pits###0###2###0###0###0###0###2 (10.0%)

Ankyloglossia###0###1###0###0###0###0###1 (5.0%)

Total###6###10###1###3###0###0###20(100.0)

TABLE 3: ASSOCIATED MEDICAL CONDITIONS SEEN IN CHILDREN WITH CLEFTS (N=17)

Medical Condition###Male Children###Female Children###Total (%)

###CL###CL/P###CP###CL###CL/P###CP

Anaemia###1###2###0###0###1###1###5 (29.4)

Malnutrition###0###3###0###0###2###0###5 (29.4)

Respiratory tract infection###1###1###0###0###2###0###4 (23.5)

Otitis media###1###0###0###0###0###1###2 (11.8)

Gastroenteritis###1###0###0###0###0###0###1 (5.9)

Total###4###6###0###0###5###2###17 (100.0)

TABLE 4: TYPE OF ANAESTHESIA USED FOR CLEFT REPAIRS IN CHILDREN AND ADULTS

Type of anaesthesia###Children###Adult###Total

###Lip repair###Palate repair###Lip repair###Palate repair

Local anaesthesia with conscious###13###0###14###0###27

sedation

General anaesthesia###24###9###0###5###38

Total###37###9###14###5###65

TABLE 5: COMPARISON OF SURGICAL COMPLICATIONS FOLLOWING LIP REPAIR IN CHILDREN AND ADULT

Type of complication###Adult###Children###Total (%)

###Male (%)###Female (%)###Male (%)###Female (%)

Wound dehiscence###1 (3.8)###0 (0.0)###2 (7.7)###3 (11.5)###6(23.1)

Stitch marks###1 (3.8)###1 (3.8)###0 (0.0)###1 (3.8)###3(11.5)

Hypertrophic scar###2 (7.7)###1 (3.8)###3 (11.5)###4 (15.4)###10(38.5)

Vermilion notching###3 (11.5)###2 (7.7)###2 (7.7)###0 (0.0)###7(26.9)

Total (%)###7 (26.9)###4 (15.4)###7 ( 26.9)###8 (30.8)###26(100.0)

All cleft palates in both children and adults were repaired under general anaesthesia while all cleft lip in adult patients were repaired under local anaesthesia and conscious sedation (using appropriate dose of intravenous Diazepam for age).

Twenty patients (17 children and 3 adults) had congenital anomalies, that included atrial septal defects, ventricular septal defects, polydactylism and low set ears. These anomalies occurred more in male then female children (Table 2). Systemic medical compromise was noticed in 17 patients who were all children (Table 3). The most common health problems were anaemia (29.4%) and malnutrition (29.4%) followed by respiratory tract infection (23.5%). Table 4 shows the type of anaesthesia used for the surgical repair of both cleft lip and palate. All the palatal repairs in both children and adults were performed under general anaesthesia. All the cleft lips in adults were repaired under local anaesthesia with conscious sedation whereas in children only 13 out of the 27 repaired were repaired under local anaesthesia with conscious sedation.

During the period of this study, 65 patients had surgical repair of their cleft defects (Table 4). Out of fifty-one cases of lip repair done, thirty-seven were children while fourteen were in adults. Fourteen cases of palatal closure were done; nine of these were in children while five were in adults. Fourteen patients (8 children and 6 adults) had post-operative complications (Table 5). Most (57.1%) of these patients were children. Five of the six wound dehiscence observed occurred in children. Seven out of ten cases of hypertrophic scar also occurred in children. Vermilion notching and stitch marks were more common in adults. Vermilion notching was noticed in five adults compared with only one case among children. All cases of wound dehiscence were successfully treated with antibiotics and only one case needed a repeat closure of the wound.

Two out of the fourteen repaired palatal cleft had anterior palatal fistula and nasal regurgitation, one each in a child and an adult patient.

DISCUSSION

Health practitioners working in the field of cleft deformity believe that early treatment improves the overall outcome of the patient's speech, cosmetics, and psychological perspectives. However, in developing countries a significant number of patients with cleft lip and palate have been observed to present for treatment in either late childhood or in some instances as adults.9 This study revealed that about one-fourth of the patients presented as adults. Adekeye10 in a study from Kaduna, North-West Nigeria reported a relatively large number of patients with cleft lip and palate presenting for treatment in late childhood. The study suggested that limited surgical facilities, long distances patients travelled to treatment centres coupled with inability to afford transportation cost and ignorance especially among rural community dwellers may be partly responsible for the late presentation of cleft patients.

Based on these prior observations, it may be prudent to increase access to primary health care services combined with the provision of primary health workers with adequate information on the availability of cleft care services. These steps may probably reduce the incidence of late presentations of patients with cleft lip and palate in North-Eastern Nigeria.

In the present study, there was a slight predominance of males over females amongst the children studied with a male to female ratio of 1.2:1. This is similar to the findings of Oldfield in 1985.11 Adekeye12 in a study from Kaduna, Nigeria also reported a male preponderance. However, in their study clefts of the lip alone were more frequently diagnosed in females. This contrasts with the findings of Fogh-Anderson13, and Oldfield11, but agrees with those of Oluwasanmi and Adekunle14, and Ugboko et al.15 Cleft lip and palate in the present report was found to be twice as common in males as females. This was similarly reported by Adekeye.12 Sowemimo16 however recorded only a slight male preponderance.

In agreement with some African and Asian studies1, isolated cleft lip was the most common type of cleft (62.7%) in the present study, followed by combined cleft lip and palate (32.8%) and isolated cleft palate (4.5%). However, the incidence obtained for cleft palate in the present report differs from 13.2% and 12.7% reported by Adekeye10 and Olasoji1 respectively. This may be attributed to the relatively large sample size of their studies compared with the present report. More clefts were seen on the left side than the right side in both children and adults patients. This is in consonance with the findings of Oluwasanmi and Adekunle14 and Sowemimo.16

In the present study 30% of the cases presented with associated congenital anomalies, a figure that is similar to the 28.3% reported by Ademiluyi et 17 and Olasoji et al.18 Patients with combined cleft lip and palate had the highest incidence of congenital anomalies, followed by patients with isolated cleft lip and patient with isolated cleft palate. This contrasts with the findings of Oluwasanmi and Adekunle14, Sowemimo16 and Bixker 19, who all reported the highest associated anomaly rate in isolated cleft palate.

Anaemia, malnutrition and respiratory tract infection were the commonest ailments that they presented with similar to the findings recorded by other workers from developing countries.16,20 Patients with combined lip and palate were found to have more of such conditions. Malnutrition and anaemia in such patients is possibly due to their inability to develop appropriate negative intra-oral pressure for breast-sucking2 while recurrent respiratory tract infections in these patients have been attributed to frequent nasal regurgitation as a result of the palatal defect.21

The fourteen cases of cleft lip repair in adult were carried out under local anaesthesia with conscious sedation so as to remove these patients from our long operation waiting lists. Thirteen of the thirty-seven cleft lips repaired in children were done under local anaesthesia with conscious sedation for similar reasons, the remaining were done under general anaesthesia. Those children who were treated under local anaesthesia with conscious sedation were carefully selected and did not have any associated congenital anomalies. The successful repair of cleft lip using local anaesthesia has being reported in previous studies.

Although, the repair of cleft lip and palate is generally performed with the use of endotracheal anaesthesia; limited medical facilities, dearth of skilled and qualified anaesthetists and the inability of patients to afford surgery under general anaesthesia has necessitated the use of local anaesthesia with conscious sedation as an alternative particularly in adult patients. This approach is safe, cost effective and does not affect aesthetic and functional outcome of the surgical repair.15 In all patients with bilateral cleft lip and palate with prominent and proclined prolabium, the non-availability of an orthodontic specialist, necessitated repair of cleft lip without pre-surgical orthodontic treatments.22

The strong development of the lip on each side of the cleft, especially in the older age groups allows sufficient mobilisation of the labial flaps especially when labial and buccal relieving incisions were made on the periosteum on both sides of the lip.1 This is similar to the findings of Adekeye10 and Olasoji et al.1

The postoperative course for most of the patients was uneventful. In five of the thirty seven (13.5%) children who had cleft lip repair, wound breakdown was observed at the reconstructed nasal floor and vermillion. Amongst the fourteen adult patients treated for cleft lip, only one (2%) had wound dehiscence at the vermilion. Contamination of surgical site in clefts from mucous secretion has been reported to be common in children.4

Adekeye10, however, attributed the wound dehiscence observed in his study to contamination of surgical site by the attending mothers. Sowemimo16 opined that wound dehiscence in cleft surgery may sometimes be due to nosocomial infections from attending medical team. In the present study contamination of surgical site by mucous secretion was also highly implicated as a major cause of wound breakdown.

Slight notching at the vermilion of the lip occurred in five adult patients that had lip repair, while only two cases of post-operative lip notching was recorded in children. The reason for this difference is not known. McCarthy and Court21 attributed vermillion notching after cleft lip repair to excessive sacrifice of vermillion during primary repair or inadequate approximation of orbicularis muscle fibres. Kernahan and Bauer22 emphasized the importance of orientation of the orbicularis oris muscle in the cleft lip repair for better lip function and appearance. Similar to previous reports in Nigeria and some African countries1,16 hypertrophic scar was noticed at 2 weeks postoperative review in seven children and three adults respectively.

Although the development of hypertrophic scar and keloids at incision sites following surgery have been reported to be relatively common among Blacks when compared with Caucasians, the etiology of this condition is still not clear.23 Satisfactory improvement of some of the scars was noticed in majority of children at 3 months post-operative review. This may probably be due to the greater healing potential commonly observed in the very young.

All cleft palates repaired in the present study were done under general anaesthesia. The anterior hard palate was closed with combination of lateral mucosa advancement flap and a vomer flap from the anterior septum as described by Von Lagenbeck.4 Similar technique was employed in both children and adult patients. One case each of oro-nasal fistula development following palatoplasty was noticed in a child and an adult patient. The anterior fistula was probably due to breakdown resulting from tension at the suture line.

Although there was no speech therapist to access patient's speech outcome following cleft palate repair, anecdoal assessment showed slight improvement of speech in most of the adult patients after surgery. Otis-monasterio et al24 observed that speech outcomes are usually unacceptable in delayed palatal closure.

CONCLUSION

The study revealed similarity in cleft pattern in both children and adult. However, the low number of adults in the present study has buttressed the effectiveness of the free cleft treatment services in our region. Successful repair of orofacial cleft in adult patients and selected cases of children under local anaesthesia can serve as a guide for centres with paucity of anaesthetists and reduce financial implications of treatment under general anaesthesia. However a careful case-by-case selection of best treatment option is advocated.

REFERENCES

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3 McDonald R. E, Avery D.: Dentistry for the child and adolescent, 7th Edition pg 101-105, 742-45.

4 Herbert J. B: A textbook of Oral and Maxillofacial Surgery, Surgical Repair of the cleft lip pg 1831-35.

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7 Arotiba GT, Olasoji HO.: Modern objectives of Unilateral Cleft Lip Repair. Nig Qt J. Hosp. Med vol 7 (4) Oct-Dec '97.

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10 Adekeye E. O Lavery K M.: Cleft lip and palate in Nigerian children and Adults: A comparative study. Br J Oral Maxillofac Surg 1985; 23: 398-403.

11 Oldfield MC.: Some observations of the causes and treatment of harelip and cleft palate based on the treatment of 1,041 patients. Br J Surg 1958; 46: 311-15.

12 Adekeye EO.: Occurrence of cleft lips and palates in Kaduna Nigeria. Nig Med J 1982; 3: 19-25.

13 Fogh - Anderson P.: Incidence and aetiology of cleft lip, alveolus and palate in humans. In: Scher Chardt ed. Introductory Surgery Stuttgert: 1966: 4-8.

14 Oluwasanmi J. O Adekunle OO.: Congenital Cleft of the face in Nigeria. Plast Reconstruct Surg 1970 46: 245-48.

15 Ugboko V, Owotade F, Otuyemi O, Adejuyigbe O.: Experience with cleft lip and palate patients seen in Nigeria Teaching Hospital. Paed Dent J 1997; 7: 41-44.

16 Sowemimo GOA.: Cleft lip and palate in Nigerian. Nig Med J 1976; 6: 410-416.Ademuluyi S. A, Oyemeyin J. O. Sowemimo G. O. A.: Associated congenital abnormalities in Nigerian children with cleft lip and palate. W Afr Med J 1989; 8: 135-37.

17 Olasoji H. O, Dogo D, Obiano K, Yawe T.: Cleft lip and palate in North Eastern Nigeria. Nig Qt J Hosp Med 1987; 7: 209-13.

18 Bixler D.: Genetics and clefting. Cleft Palate J 1981; 18: 10-18.

19 Nwoku AL.: Experiences on the surgical repair of unoperated adult cleft patients in Nigeria. Nig Med J 1976; 4: 417-21.

20 McCarthy J. G. Court 3c. Secondary deformities of cleft lip and palate. In: Georgrade N. G. Rietkonh R., Bermick W. J. (eds). Textbook of plastic. Maxillofacial and Reconstructive surgery; Baltimore, Williams and Wilkins 1992: 307-19.

21 Kernehan DA, Bauer BS.: Functional cleft lip repairs a sequential layered closure with orbicularis muscle realignment. Plast Reconstruct Surg 1983; 72: 459.

22 Maurice H.: The aesthetic treatment of hare lip with a description of a new operation for the more scientific remedy of the deformity. Dublin J. Med. Sci. 1868; 45: 292-93.

23 Ortiz-Monasterio F, Serrano RA. Cultural aspects of cleft lip and palate treatment. In: Grabb W C, Rosenstein W, Bzoch K R (eds) Cleft Lip and Palate. Little Brown, Boston, 1971.
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Publication:Pakistan Oral and Dental Journal
Article Type:Report
Geographic Code:6NIGR
Date:Sep 30, 2016
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