|Year : 2020 | Volume
| Issue : 2 | Page : 61-66
Oral health-related quality of life of children with repaired cleft lip and palate in Yaounde, Cameroon: A cross-sectional study
Atanga Léonel Christophe1, Ngaba Mambo Olive Nicole2, Edouma Bohimbo Jacques Gérard3, Ndjolo Alexis1
1 Department of Ophthalmology-ENT Stomatology, Faculty of Medicine and Biomedical Sciences/University of Yaounde, Yaounde, Cameroon
2 Department of Ophthalmology-ENT Stomatology, Faculty of Medicine and Biomedical Sciences/University of Yaounde; Yaounde Central Hospital, Yaounde, Cameroon
3 Yaounde Central Hospital, Yaounde, Cameroon
|Date of Submission||19-May-2020|
|Date of Decision||02-Aug-2020|
|Date of Acceptance||18-Oct-2020|
|Date of Web Publication||30-Dec-2020|
Dr. Atanga Léonel Christophe
P.O. Box 4362, Yaounde
Source of Support: None, Conflict of Interest: None
Background: Further care needs and impact of repaired cleft lip and palate (CLP) on the daily life of children are unknown in our setting, since many of them are lost to follow-up after surgery. Aim: To assess the oral health-related quality of life (OHRQoL) of children with repaired CLP and to identify the main issues that deserve additional care. Methods: We carried out a single-center, cross-sectional study of 27 children (mean age: 7.74 ± 0.7 years; 16 girls) operated for CLP and 30 healthy controls (mean age: 8.03 ± 0.7 years; 15 girls). Self and proxy-rated OHRQoL was assessed by the child oral health impact profile, a reliable and valid questionnaire designed for use with children and teenagers. Results: Patients had lower quality of life scores than controls for functional well-being (22.1 ± 1.2 vs. 27.9 ± 0.74;P< 0.001), emotional well-being (32.3 ± 1.4 vs. 37.3 ± 0.6; P = 0.002), and self-esteem (24.4 ± 0.9 vs. 29.2 ± 0.4;P< 0.001). Redo surgeries (n = 10) and a visible facial difference (n = 17) were the main depreciative determinants of the OHRQoL, particularly for emotional well-being (30.2 ± 1.7 vs. 35.2 ± 1.4;P= 0.003 and 29.6 ± 1.8 vs. 36.8 ± 1.3; P = 0.003; respectively). Conclusion: Children with repaired CLP have a lower OHRQoL than their healthy peers. Psychosocial care, orthodontics, and speech therapy are the main needs for further care aimed at improving their quality of life.
Keywords: Child oral health impact profile, cleft lip, cleft palate, oral health-related quality of life
|How to cite this article:|
Christophe AL, Nicole NM, Gérard EB, Alexis N. Oral health-related quality of life of children with repaired cleft lip and palate in Yaounde, Cameroon: A cross-sectional study. Int J Adv Med Health Res 2020;7:61-6
|How to cite this URL:|
Christophe AL, Nicole NM, Gérard EB, Alexis N. Oral health-related quality of life of children with repaired cleft lip and palate in Yaounde, Cameroon: A cross-sectional study. Int J Adv Med Health Res [serial online] 2020 [cited 2021 Apr 10];7:61-6. Available from: https://www.ijamhrjournal.org/text.asp?2020/7/2/61/305448
| Introduction|| |
In Africa, usually qualified by the zoological term “hare's beak”, cleft lip and palate (CLP) constitute a real family drama due to the various interpretations to which they are subjected to. Even in cleft palate, the morphological concerns take precedence over the functional manifestations, obscuring orthodontics, speech, and maxillofacial growth. Primary CLP repair, therefore, appears to be a specific request from parents.
Once this demand is satisfied, willingly during free surgery campaigns, families are mostly lost to follow-up. Hence, little is known about the apprehensions of families concerning the outcomes of the surgery and the impact of the latter on their daily lives. Moreover, the needs for additional care within the framework of comprehensive CLP care are also unknown.
The oral health-related quality of life (OHRQoL), which “reflects people's comfort when eating, sleeping and engaging in social interaction; their self-esteem; and their satisfaction with respect to their oral health”, appear therefore as a fundamental tool to consider to deal with these issues.
Available literature shows some discrepancies concerning the outcomes of CLP care as far as the quality of life is concerned. While many studies find children and adolescents with CLP appear to have good psychosocial functioning and resilience,, evidence also suggests that individuals with CLP have significantly poor overall oral health-related quality of life and in all dimensions compared with schoolchildren without CLP.
Thus, in this study, we sought to assess in a low-income setting (Cameroon), the OHRQoL of children who underwent CLP surgery and compare it with that of their healthy peers.
| Methods|| |
We carried out a single-center, cross-sectional, descriptive, and analytical study at the Yaounde Central Hospital, Cameroon. Files of all patients who underwent CLP surgery whether it was primary or a redo, between January 2014 and December 2018 were reviewed.
Children aged 4–18 years old at the time of the study were selected regardless of their gender (”patients group”). The parents were summoned with their children to the hospital by phone call on June, July, and August 2019, corresponding to school holidays. After obtaining the informed consent from parents, we filled out for each of the children a form including items capturing age, gender, type of cleft at birth, associated malformation or chronic condition (such patients were excluded), and prior history of surgical intervention. Participants were then classified as having either a visible facial difference (lip or nose or orthognathic involvement in the cleft issue) or a nonvisible facial difference (only palate involvement in the cleft issue).
After selecting patients, controls were consecutively and non-randomly selected according to their age (4–18 years) and as many girls as boys. These controls were selected among children and teenagers, free from any chronic condition or malformation, attending usual ear nose and throat consultation for acute diseases not related to the oral sphere and for which an informed consent from their parents had been obtained.
The OHRQoL of patients and controls was measured using the Child Oral Health Impact Profile (COHIP) questionnaire. This instrument has demonstrated excellent psychometric properties. The COHIP assesses self-reported (patients and control) and proxy-reported (parents) OHRQoL with five discrete domains and an overall score range of 5–170 [Table 1]: Oral Health (specific oral symptoms; ten items; range of 10–50); Functional Well-being (ability to carry out specific everyday tasks; six items; range of 6–30); Emotional Well-being (peer interactions and mood states; eight items; range of 8–40); School (tasks associated with the school environment; four items; range of 4–20); Self-esteem (positive feelings about self; six items; range of 6–136). Both patients and parents answered the questionnaire, but parents intervened only when the patient did not give a precise answer or had difficulty answering.
The COHIP uses a 5-point Likert scale (”never” = 1, “almost never” = 2, “sometimes” = 3, “fairly often” = 4, and “almost all the time” = 5); higher scores indicate better quality of life.
Statistical analysis was undertaken using the SPSS® software, version 21.0 (IBM, Chicago, Illinois). Data were presented in proportion for categorical variables and mean and standard error of mean or standard deviation where indicated for continuous variables. Subscale scores were computed as the average of the responses on that subscale. The overall COHIP score was computed by summing the subscale scores.
Unpaired t-test or analysis of variance was used to compare means between 2 or more groups. Proportions were analyzed using a contingency table and Chi-square analyses. P < 0.05 was considered statistically significant.
The study was approved by the ethics committee of the Faculty of Medicine and Biomedical Sciences of the University of Yaounde I. The COHIP questionnaire was administered in French on an individual and confidential basis. Families were not paid for their participation.
| Results|| |
Presentation of the study population
The study population consisted of 27 patients and 30 controls [Figure 1].
The mean age of patients was 7.74 ± 0.7 years and 16 were aged <7-years-old. The Male:Female sex ratio was 0.7 (11 boys and 16 girls). [Table 2] illustrates the demographic data of patients and controls.
According to Kernahan and Stark classification of CLP, at birth, there were 11 cases (41%) of the cleft of the primary palate (lip n = 7 ; lip and alveolus n = 4), 10 cases (37%) of cleft of the secondary palate (soft palate n = 4, hard and soft palate n = 6) and both were found in 6 cases (22%). Therefore, 17 patients (63%) had a visible facial difference after surgery. In unilateral CLP (n = 11), the left side was involved in 6 cases and the right in 5 cases (P = 0.9).
Primary surgery, performed in 17 cases (63%) had been at an average age of 2.98 ± 1.07 years and redo carried out in 10 cases (37%) at an average age of 5.3 ± 0.87 years
The average hospital stay was 3.4 ± 1.6 days (2–5 days) postoperative.
Oral health-related quality of life assessment
The COHIP questionnaire response rate was 85.2% among patients and 83.8% among controls
The overall COHIP score was 141 ± 4.3 for patients and 157.6 ± 1.2 for controls (P < 0.001).
More specifically, the functional well-being (27.9 ± 0.4 vs. 22.1 ± 1.2, P < 0.001), emotional well-being (37.3 ± 0.6 vs. 32.3 ± 1.4, P = 0.002) and self-esteem (29.2 ± 0.4 vs. 24.4 ± 0.9, P < 0.001) subscale scores were better in controls [Figure 2].
|Figure 2: Subscales child oral health impact profile scores of patients versus controls|
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Oral health related quality of life determinants
The overall COHIP score for boys was higher than that of girls, particularly for functional well-being and self-esteem subscales. However, the differences were not significant, as shown in [Table 3].
|Table 3: Overall and subscales COHIP scores of patients by gender, age and number of surgery done|
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Likewise, the overall and subscales COHIP scores of patients aged ≥7 years were higher than those aged >7 years, although the difference was not significant as set out in [Table 3].
The overall COHIP score (133.7 ± 5.5 vs. 153.5 ± 5.0; P = 0.014), emotional well-being subscale score (29.6 ± 1.8 vs. 36.8 ± 1.3; P = 0.003) and oral health subscale score (42.5 ± 2.0 vs. 48.2 ± 0.9; P = 0.16) were lower for patients with a visible facial difference compared to those with a nonvisible facial difference as shown in [Figure 3].
|Figure 3: Subscales child oral health impact profile scores of patients according to the visibility of their facial difference|
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The overall and subscale COHIP scores of patients who underwent redo surgery were lower than that of those who underwent primary surgery but this was significant only for emotional well being subscale score (30.2 ± 1.7 vs. 35.2 ± 1.4; P = 0.03) [Table 3].
| Discussion|| |
Quality of life is a complex, multidimensional, and subjective concept. This subjectivity explains the proliferation of quality of life assessment tools. If none has been developed specifically for CLP, over the past 15 years, many instruments were set up, assessing the quality of life related to the orofacial health of children with craniofacial conditions. So, with this multitude of instruments, it became difficult to compare results between studies, even if they mostly dealt with the same domains of quality of life such as physical health, self-esteem, behavior, psychological distress, and relationships with peers.
In this study, we used the COHIP questionnaire, one of the most used tool in this purpose and judged to be of quality. It was developed from a pool of 54 items chosen after a review of the literature and experts to reach the 34 current items. It has excellent reliability and concurrent validity in different countries and communities in Asia, Europe, and America and has been tested in children and teenagers with various craniofacial conditions. Futhermore, unlike the others, this questionnaire includes a specific dimension, the school environment, where the child faces his speaking skills and his peers.
The COHIP response rate in this study was high similar to 85.7% obtained by François-Fiquet et al. This result can be explained by our desire to administer the questionnaire ourselves, the parents, and their children having been specially summoned for the study. It would have been possible to administer the questionnaire by phone interviews. This would have allowed us to increase the number of inclusions, but the rate of responses would probably have been lower. It is unclear whether receiving patients in the consultation could influence their responses. However, it is undeniable that it has influenced the response rate through better participant awareness.
The overall OHRQoL of patients with repaired CLP, chiefly for functional well-being, emotional well-being, and self-steem subscales, was lower than that of their healthy peers. Broder and Wilson-Genderson in 2016 using the same questionnaire to analyze the effects of surgery for cleft lip and/or palate on self-and caregiver rated OHRQoL among youth, made the same observation. However, for Warschausky et al. in 2002, Jokovic et al. in 2004, Locker and al in 2005 or Wogelius et al. in 2009, presence of CLP had a little impact on the quality of life of patients compared to controls. These differences could be because the children in these studies are following a cleft center protocol treatment with advanced treatment, including not only surgery but also the orthodontic treatment and speech therapy. Therefore, a more comprehensive CLP care with orthodontic and speech-therapy should be included in our setting as it has shown a good effect on OHRQoL in other studies.
Regarding functional well-being, Ward et al. determining the impact of orofacial clefts on the OHRQOL of affected children reached the same conclusions as the present study. Patients with repaired CLP are more likely to have chewing and certain words pronunciation difficulties as well as being well understood when they speak. These chewing difficulties are related to malocclusions due to either anomaly in position, number, and shape of teeth or to a contraction of the maxillary arch. Likewise, after surgery, persistent oro-nasal fistulas or velopharyngeal insufficiency may cause nasalization of the voice. Improving this functional well-being would therefore require adequate orthodontic care but, above all a good primary surgery technique, raising the need for perpetual surgeon's training in our milieu.
Like in this study, Ward et al. and Kortelainen et al. found that the presence of facial cleft, even repaired, impaired emotional well-being. CLP patients are readily anxious and upset. They are subject to intimidation and teasing and they worried about their fellow's perception. In the study of Masnari et al. children described fellow's looks of pity or fear, a phenomenon that reduces self-esteem. However, they can have a negative appreciation of their facial appearance (nose, lip, mouth, profile) without having a poor self-esteem. Personalized psychosocial care, coupled with a compassionate family environment, prove to be essential for these patients. Media or public health interventions may be warranted in this context to reduce discrimination and prejudice against children and teenagers with facial conditions.
In this study, the OHRQoL was lower in patients aged >7 years, even though the difference observed was not significant. This result suggests that teenagers with repaired CLP would have a lower quality of life than younger children. These results match with that of Ward et al. For Damiano et al., the difference in external appearance related to lip or nose involvement in the cleft process would have a greater impact on the quality of life as the child is approaching adolescence.
In the present study, as in that of Feragen and Borge, patients with a visible facial difference had lower overall, oral health, and emotional well-being scores than those with a nonvisible facial difference. This trend is widely described in literature, more often concerning self-esteem and interpersonnal relationships with peers. With the current cultural emphasis on appearence, finding means to foster patient's resilience is essential. Here too, as demonstrated by Topolski et al, an affectionate and protective family unit would effectively compensate this discomfort.
All these factors overlap and will vary depending on the patient's gender. In the study of Broder and Strauss, female teenagers had a lower emotional well-being and overall quality of life score compared to male teenagers. These findings are consistent with those of many authors related to the gender difference in appearance standards and attractiveness. Girls internalize these standards and are therefore at a high risk of worry of appearance-related self-esteem associated with their facial difference. Families and caregivers should be vigilant about this aspect in young girls. In the present study, such an influence of gender was not found, probably due to the lower proportion of teenagers in the study population.
The emotional well-being score of patients subject to redo surgeries was lower compared to that of those with primary surgery. Broder et al. made the same observation. This may indicate that these multi-operated patients would have a more severe defect related to their cleft, or that their families have unrealistic expectations or that the surgical technique used was not approriate, raising again the need for perpetual surgeon's training.
The main limitation of our study was the small sample size due to the single-center recruitment considering the low incidence of CLP (0.8–2.7 for 1000 births). However, notwithstanding this limitation, the results of this study provide an interesting overview of the thoughts, apprehensions, and experiences of families living with CLP children in Cameroon as well as the need for a comprehensive CLP care program in our practice and not just the surgery.
| Conclusion|| |
OHRQoL of children with repaired CLP at the Yaounde Central Hospital seems to be lower than that of their healthy peers. This quality of life is mainly negatively influenced by the visible nature of the CLP and redo surgeries. Specifically, functional well-being, emotional well-being, and self-esteem are the most affected domains of the OHRQoL. Psychosocial care and functional rehabilitation (orthodontics and speech therapy) in a framework of a multidisciplinary team-based comprehensive cleft clinic, therefore, appear as the further priority interventions to consider in these patients aimed at improving their quality of life.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Bob'Oyono JM, Bengondo C, Andze G, Ndjolo A, Bengono G. Ten years of chelioplasties for congenital cleft lip in Yaounde Central Hospital. Rev afr Otorhinolaryngol Chir Cervicofac 2005;3:18-22.
Gift HC, Atchison KA. Oral health, health, and health-related quality of life. Med Care 1995;33:NS57-77.
Gilchrist F, Rodd H, Deery C, Marshman Z. Assessment of the quality of measures of child oral health-related quality of life. BMC Oral Health 2014;14:40.
Baker SR, Owens J, Stern M, Willmot D. Coping strategies and social support in the family impact of cleft lip and palate and parents' adjustment and psychological distress. Cleft Palate Craniofac J 2009;46:229-36.
Feragen KB, Borge AI, Rumsey N. Social experience in 10-year-old children born with a cleft: Exploring psychosocial resilience. Cleft Palate Craniofac J 2009;46:65-74.
Queiroz Herkrath AP, Herkrath FJ, Rebelo MA, Vettore MV. Measurement of health-related and oral health-related quality of life among individuals with nonsyndromic orofacial clefts: A systematic review and meta-analysis. Cleft Palate Craniofac J 2015;52:157-72.
Broder HL, Wilson-Genderson M. Reliability and convergent and discriminant validity of the child oral heath impact profile (COHIP Child's version). Commun Dentist Oral Epidemiol 2007;35:20-31.
Kernahan DA, Stark RB. A new classification for cleft lip and cleft palate. Plast Reconstr Surg Transplant Bull 1958;22:435-41.
Topolski TD, Edwards TC, Patrick DL. Quality of life: How do adolescents with facial differences compare with other adolescents? Cleft Palate Craniofac J 2005;42:25-32.
Broder HL, Wilson-Genderson M, Sischo L. Oral health-related quality of life in youth receiving cleft-related surgery: Self-report and proxy ratings. Qual Life Res 2017;26:859-67.
François-Fiquet C, Dupouy M, Daoud S, Poli-Merol ML. Fentes labio-palatines: Qualité de vie (VSP-A) patients et parents. À propos de 51 familles. Ann Chir Plast Esthet 2014;15:900.
Warschausky S, Kay JB, Buchman S, Halberg A, Berger M. Health-related quality of life in children with craniofacial anomalies. Plast Reconstr Surg 2002;110:409-14.
Jokovic A, Locker D, Tompson B, Guyatt G. Questionnaire for measuring oral health-related quality of life in eight- to ten-year-old children. Pediatr Dent 2004;26:512-8.
Locker D, Jokovic A, Tompson B. Health-related quality of life of children aged 11 to 14 years with orofacial conditions. Cleft Palate Craniofac J 2005;42:260-6.
Wogelius P, Gjørup H, Haubek D, Lopez R, Poulsen S. Development of Danish version of child oral-health-related quality of life questionnaires (CPQ8-10 and CPQ11-14). BMC Oral Health 2009;9:11.
Ward JA, Vig KW, Firestone AR, Mercado A, da Fonseca M, Johnston W. Oral health-related quality of life in children with orofacial clefts. Cleft Palate Craniofac J 2013;50:174-81.
Kortelainen T, Tolvanen M, Luoto A, Ylikontiola LP, Sa×ndor GK, Lahti S. Oral health-related quality of life among cleft lip and/or palate and school children. Cleft Pal Craniofac J 2016;53:172.
Hunt O, Burden D, Hepper P, Stevenson M, Johnston C. Self-reports of psychosocial functioning among children and young adults with cleft lip and palate. Cleft Palate Craniofac J 2006;43:598-605.
Masnari O, Landolt MA, Roessler J, Weingaertner SK, Neuhaus K, Meuli M, et al
. Self- and parent-perceived stigmatisation in children and adolescents with congenital or acquired facial differences. J Plast Reconstr Aesthet Surg 2012;65:1664-70.
Hunt O, Burden D, Hepper P, Johnston C. The psychosocial effects of cleft lip and palate: A systematic review. Eur J Orthod 2005;27:274-85.
Damiano PC, Tyler MC, Romitti PA, Momany ET, Jones MP, Canady JW, et al
. Health-related quality of life among preadolescent children with oral clefts: The mother's perspective. Pediatrics 2007;120:283-90.
Feragen KB, Borge AI. Peer harassment and satisfaction with appearance in children with and without a facial difference. Body Image 2010;7:97-105.
Feragen KB, Kvalem IL, Rumsey N, Borge AI. Adolescents with and without a facial difference: The role of friendships and social acceptance in perceptions of appearance and emotional resilience. Body Image 2010;7:271-9.
Broder H, Strauss RP. Self-concept of early primary school age children with visible or invisible defects. Cleft Palate J 1989;26:114-7.
Lawler M, Nixon E. Body dissatisfaction among adolescent boys and girls: The effects of body mass, peer appearance culture and internalization of appearance ideals. J Youth Adolesc 2011;40:59-71.
Vanderas AP. Incidence of cleft lip, cleft palate, and cleft lip and palate among races: A review. Cleft Palate J 1987;24:216-25.
[Figure 1], [Figure 2], [Figure 3]
[Table 1], [Table 2], [Table 3]