The Cleft Palate–Craniofacial Journal 00(00) pp. 000–000 Month 2015 Ó Copyright 2015 American Cleft Palate–Craniofacial Association
ORIGINAL ARTICLE Oral Health–Related Quality of Life in Children in Chile Treated for Cleft Lip and Palate: A Case-Control Approach ´ ´ D.D.S, Cesar Pedro C. Aravena, D.D.S., Ph.D., Tania Gonzalez, D.D.S, Tamara Oyarzun, Coronado, D.D.S, M.Sc. Objective: To compare the oral health–related quality of life of patients treated for cleft lip and/or cleft palate (CL/P) versus unaffected children between 8 and 15 years of age using a Spanish-language version of the Child Oral Health Impact Profile (COHIP-Sp) administered to a Chilean population. Design: A cross-sectional study with a matched case-control design was used. Methods: Participants were 48 children (mean age 11.3 years) with a history of CL/P from three cities in Chile and one group of 96 children (mean age 11.2 years) unaffected by CL/P. The COHIP-Sp was applied to both groups. Quality of life was compared according to the overall score and the average score of items and domains on the COHIP-Sp scale between the two groups (Mann-Whitney U test; P , .05). Results: The COHIP-Sp score was 94.1 6 19.3 in children with CL/P and 97.1 6 15.6 for the control group (P ¼ .31). A significantly lower score was observed in the group with CL/P in the domains ‘‘functional well-being’’ (P ¼ .001) and ‘‘school environment’’ (P ¼ .001); the only average in favor of the quality of life in children with CL/P was in ‘‘self-image’’ (P ¼ .0002). Conclusion: The oral health–related quality of life of children with a history of CL/P was similar to that of the control group. Nevertheless, a lower quality of life was observed concerning items associated with speech and being understood by other people. Further study into the risk factors associated with surgery and rehabilitative treatment is recommended. KEY WORDS:
child; Chile; cleft lip; cleft palate; oral health; quality of life
Region in the south of Chile show this prevalence increasing to 2.03 per 1000 live births (Rosa, 2010). The treatment of patients with CL/P seeks to achieve normal growth and development of the face, complete closure of the cleft, comprehensible speech, normal hearing, a good esthetic result, and consequently correct socialization (Eberlinc and Kozelj, ˇ 2012). There are, however, certain sequelae that will influence quality of life, such as difficulties in eating, speaking, oral hygiene, esthetics, growth, and other physical problems such as ear infections, alterations in hearing, and a psychosocial burden (Ward et al., 2013). Oral health complications are numerous, including dental agenesis and supernumerary and poorly positioned teeth (Chapados, 2000), and cause speech disorders such as hypernasality and facial alterations such as nose and mouth asymmetry, which also affect the individual’s self-image, social affiliation, and adaptation (Broder et al., 1994; Berk et al., 2001). In response to this situation, the measurement of oral health–related quality of life has become an important health indicator and reveals the functional and psychosocial results of oral diseases and conditions (Cohen, 1997, quoted by Do and Spencer, 2008). Broder and Wilson-Genderson (2007) adapted and validated the Child Oral Health Impact Profile (COHIP) in English to measure quality of life in terms of oral health, functional well-being, social-emotional well-being, school
Cleft lip and/or cleft palate (CL/P) is one of the most frequent congenital malformations, with a worldwide prevalence estimated at 0.8/1000 live births (World Health Organization, Human Genetics Programme, 2002), and there is a significant variation in global incidence, depending on geography, gender, race, and ethnic group, among others (Rozendaal et al., 2012). South America has one of the highest frequencies in the world: in Bolivia, it is 2.5 per 1000 live births and in Argentina, 1.5 per 1000 live births (Nazer et al., 2010). Colombia and Brazil have reported a prevalence of 1 per 1000 live births (Hurtado et al., 2008; Raposo-do-Amaral et al., 2011). In Chile, the reported frequency is 1.8 per 1000 live births, with 452 new cases reported annually (Chilean Ministry of Health [MINSAL], 2009); however, local reports in the Los R ´ıos
Dr. Aravena is Professor, Institute of Anatomy, Histology and Pathology, Faculty of Medicine, Universidad Austral de Chile, Valdivia, Chile. Ms. Gonza´lez and Ms. Oyarzun ´ are Dentists, location. Mr. Coronado is Professor, Department of Morphofunction, Faculty of Medicine, Universidad Diego Portales, Santiago, Chile. Financial support: This study had private financial support from the researchers and the School of Dentistry, Universidad Austral de Chile. Submitted March 2015; Revised May 2015; Accepted May 2015. Address correspondence to: Dr. Pedro C. Aravena, School of Dentistry, Universidad Austral de Chile, 1640 Rudloff Street, Valdivia, Chile. E-mail [email protected]. DOI: 10.1597/15-095 0
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environment, and self-image in children 8 to 15 years old, recruiting participants with various oral health conditions such as craniofacial, orthodontic, and general pediatric dental anomalies. This scale presented a high internal consistency and an excellent test-retest reliability, contributing a valuable instrument for the assessment of the oral health–related quality of life in children with CL/P (Broder et al., 2014). The use of the COHIP in children with CL/P could show the impact of this condition and sequelae after treatment such as esthetic and speech problems, nervousness when speaking in public or at school (Chetpakdeechit et al., 2009) or obvious self-image problems (Berk et al., 2001); therefore, the use of the COHIP at the local level could reveal similar and relevant issues to ascertain the oral health of this population. In light of the bibliographic references presented, we propose the following hypothesis: children between 8 and 15 with a history of surgically treated CL/P exhibit an oral health–related quality of life similar to children unaffected by this condition in a Chilean population. The aim of this study was to determine oral health– related quality of life by means of an adapted Spanishlanguage version of the COHIP in children and adolescents between 8 and 15 years old treated surgically for nonsyndromic CL/P and to compare it with children and adolescents unaffected by CL/P in a Chilean population. METHODS Design and Patients A cross-sectional study with a matched case-control design was conducted and approved for the Ethical and Research Committee of the Regional of Health Department of Chile (Register No. 152/2014). Forty-eight children and adolescents with a history of CL/P participated (average age 11.3 6 2.3 years; 62.5% male) and were treated surgically in primary care hospitals in Chile: Hospital Base in Valdivia, San Borja Arriara´n Hospital in Santiago, and the Assistance Network for Children With Cleft Lip/Palate in Puerto Montt City. In addition, there was a control group of 96 children and adolescents unaffected by CL/P (average age 11.2 6 2.3 years; 46.8% male) from public schools in Valdivia, Chile. For matching the two study groups, a number of children were selected at a 2:1 ratio between unaffected children and those with a history of CL/P and of similar age, gender, and the same DMFT index (Lucas et al., 2000), as this index was statistically similar between the two groups (CL/P group DMFT index ¼ 2.2 6 2.8; control group DMFT index ¼ 2.7 6 2.6; P . .05). The group of children with a history of CL/P was selected by convenience sampling. Those children between 8 and 15 years of age were included who could read and write in Spanish, who agreed voluntarily to
participate through a written informed consent, and whose parents signed an informed consent. Children or adolescents who presented a disabling medical condition, syndromic CL/P, or mental disorders were excluded. The children in the control group were selected by means of simple random sampling from five public schools in Valdivia. Regarding the external validity of our results, the sample size was based on the incidence rate of children with CL/P in Chile calculated on the basis of a power of 80%, 95% confidence level according to the overall population of Valdivia consistent with its overall birth rate and rate of live births with CL/P in Chile ¼ 1.8/1000 (MINSAL, 2009). This calculation detected a difference of 13.3 points on the overall COHIP score between children with CL/P and the control group (Broder and Wilson-Genderson, 2007; Ward et al., 2013) and matching with double the number of subjects in the ´ control group (Argimon and Jimenez, 2004). This calculation provided a minimum number of 31 subjects between 8 and 15 years (EPIDAT 3.0, Statistical Analysis Software) Adaptation and Validity of the COHIP to Spanish The first stage was to adapt the COHIP to Spanish. This instrument is composed of five domains (well-being in oral health, functional well-being, social-emotional well-being, school environment, and self-image), with a total of 34 items. Each item is valued on a 5-item Likerttype scale ranging from 0 to 4 (4 ¼ never, 3 ¼ almost never, 2 ¼ sometimes, 1 ¼ fairly often, and 0 ¼ always), with the exception of the domain ‘‘self-image,’’ which presents a score assigned with an ordinal and increasing number (Broder, 2007). The questions with no response are scored as zero. The total score of the scale varies between 0 and 136, where a higher score corresponds to a better oral health–related quality of life (Broder and Wilson-Genderson, 2007). In its adaptation, the COHIP was translated from English to Spanish simultaneously by two investigators (T.G. and T.O.), who then agreed with a third Englishspeaking investigator on a definitive version called COHIP-Sp. This was taken to a qualitative review process in terms of face and construct validity by five experts in the treatment of children with CL/P and a focus group with 15 children from 8 to 15 years old from a public school in Valdivia. In this analysis, question 22 was modified to use the word withdrawn instead of insecure. The rough draft of the scale was applied on two occasions, 2 weeks apart, to a pilot group of 30 randomly selected children 8 to 15 years of age from a public school in Valdivia. The total score obtained in both applications was analyzed by means of the intraclass correlation coefficient R. This test had a high test-retest reliability (intraclass correlation coefficient
Aravena et al., QUALITY OF LIFE IN CHILEAN CHILDREN WITH CLEFT LIP/PALATE
TABLE 1 Sociodemographic Characteristics and Treatment History of Patient Group With Cleft Lip and/or Palate in Cities of Chile Valdivia, n ¼ 29 Age, average (SD) Gender Male, n (%) Cleft diagnosis Cleft lip, n (%) Cleft palate, n (%) Cleft lip-palate, n (%)
11.5 (2.5)
Santiago, Puerto Montt, n ¼ 12 n¼7 10.5 (2.1)
11.7 (1.7)
Total, N ¼ 48 11.3 (2.3)
19 (65.5)
6 (50.0)
5 (71.4)
30 (62.5)
3 (10.3)
2 (16.6)
0 (0.0)
5 (10.4)
8 (27.5)
5 (41.6)
0 (0.0)
13 (27.0)
18 (62.0)
5 (41.6)
7 (100.0)
30 (62.5)
Cleft side Left, n (%) Right, n (%) Bilateral, n (%) Median cleft palate, n (%)
7 (24.1) 7 (24.1) 7 (24.1)
2 (16.6) 3 (25.0) 2 (16.6)
6 (85.7) 0 (0.0) 1 (14.2)
15 (31.2) 10 (20.8) 10 (20.8)
8 (27.5)
5 (41.6)
0 (0.0)
13 (27.0)
Presurgical orthopedics Yes, n (%)
15 (51.7)
9 (75.0)
4 (57.1)
28 (58.3)
Number of surgeries Average no. (SD)
3.2 (2.0)
3.6 (2.9)
4.5 (1.6)
3.5 (2.2)
Age at first surgery Average months (SD)
9.5 (10.0)
6.4 (2.1)
5.8 (4.5)
8.2 (8.1)
Age at last surgery Average years (SD)
7.0 (4.5)
6.0 (4.0)
9.1 (3.8)
7.1 (4.3)
0
intraoral exploration mirror without magnification, applying the DMFT index. In the group of children with CL/P, independent variables related to the sociodemographic data and history of treatment for CL/P were recorded by interviewing the parents (Table 1): age (in years), gender (male/female), diagnosis of the cleft according to the classification of Clinical Guide for Cleft Lip-Palate (cleft lip, prepalatal cleft, or primary palate cleft; cleft palate or cleft hard and/or soft palate; cleft lip-palate; MINSAL, 2009), side affected by the cleft (left, right, bilateral, medial cleft palate), use of presurgical orthopedic apparatus (yes/no), number of surgeries under general anesthesia (No. of surgeries), age at first surgery (months), and age at last surgery (years). Only the child’s age (years) and gender (male/female) were recorded for the control group. The oral health–related quality of life score from the COHIP-Sp was recorded as a dependent variable, obtaining the average score of the participants from each group by items, domain, and sum of the total points from the scale. Data Analysis
[ICC] . .8) for questions 2, 9, 10, 11, 14, 17, 20, 23, 26, 28, and 30, and the total ICC of the instrument was .89 (Norman and Streiner, 2008). Application of the COHIP-Sp The second stage was to apply the COHIP-Sp. For this, participants were contacted by telephone between August and October 2014. In the group of children with CL/P, two investigators (T.G. and T.O.) applied the COHIP-Sp according to the geographical location and willingness of the family to participate. Sociodemographic data and history of treatment for CL/P were recorded based on an interview with the parents. For the control group, the investigators visited the children and parents at their schools after coordinating with the establishment’s administration. In both groups, the objective of the scale was explained orally. Once the children agreed to participate, they were given the scale in writing. Each child was left alone in a physical place free of visual or auditory interference. After administering the questionnaire, a basic oral examination was performed to match the training groups. For this, each child sat in a chair with full neck extension and maximum oral opening. An investigator (T.G.) used a flashlight with a frontal headpiece and a flat No. 5
The variables were analyzed with descriptive statistics, presenting the results with dispersion measurements and central tendency (average 6 standard deviation). To compare the quality of life between the study groups, the parametric/nonparametric distribution of the total average score of the scale in both was analyzed using the Shapiro-Wilk test (P , .05). According to the result, the scores were compared to total average, average of domains, and the items on the scale with the corresponding test (t test/Mann-Whitney U test). An index for a better quality of life was determined when the average of the score from the scale was significantly higher between the two groups (P , .05; 95% confidence interval). The analyses were done with the statistical R Core Team program (2013). RESULTS Table 1 presents the sociodemographic information, diagnostic history, and treatment for the group of children with CL/P. The distribution analysis of the averages of the score showed a nonparametric distribution in the group of children with CL/P (P¼ .47) and parametric for the control group (P¼.03). The average overall score of the COHIP-Sp was 94.1 6 19.3 for the group of children with CL/P and 97.1 6 15.6 for the control group, there being no statistically significant difference (P ¼ .31). However, the analysis of the average score in the domains on the scale revealed significantly unfavorable differences for the
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Table 2
Average Scale Score of the COHIP-Sp and Domains (Score Range 0–4 Points) Cleft Lip and/or Palate Group, n ¼ 48 Average (SD)
Control Group, n ¼ 96
CI 95%
Average (SD)
CI 95%
P Value‡
Oral health well-being (10)
24.5 (6)
(22.7–26.2)
25.1 (5.6)
(23.9–26.2)
.5490
Functional well-being (6) Had trouble biting off or chewing foods such as apple, carrot, or firm meat Had difficulty eating foods you would like to eat because of your teeth, mouth, or face Had trouble sleeping because of your teeth, mouth, or face Had difficultly saying certain words because of your teeth, mouth, or face Had people have difficulty understanding what you were saying because of your teeth, mouth, or face Had difficulty keeping your teeth clean because of your teeth, mouth, or face Social-emotional well-being (8)
16.8 (4.2)
(15.5–18.0)
19.2 (4.1)
(18.4–20.1)
.0013*
2.4 (1.1) 22.5 (7.4)
(2.1–2.7) (20.4–24.7)
School environment (4) Missed school for any reason because of your teeth, mouth, or face Had difficulty paying attention in school because of your teeth, mouth, or face Not wanted to speak/read aloud in class because of your teeth, mouth, or face Not wanted to go to school because of your teeth, mouth, or face
12.9 (2.6) 2.7 (1)
(12.1–13.7) (2.4–3)
Self-image (6) Been confident because of your teeth, mouth, or face Felt that you were attractive (good looking) because of your teeth, mouth, or face I have good teeth I feel good about myself When I am older, I believe (think) that I will have good teeth When I am older, I believe (think) that I will have good health COHIP-Sp (34)
17.2 (5.3) 2.5 (1.3) 2 2.3 3.4 3.4 3.4 94.1
Child Oral Health Impact Profile Domains and Items (n of Items)†
2.8 (1.2)
(2.5–3.2)
3.1 (1.2)
(2.8–3.3)
.3393
3.4 (0.9) 3.3 (1) 2.1 (1.2)
(3.1–3.7) (3.1–3.6) (1.7–2.4)
3.3 (0.9) 3.5 (0.8) 3.3 (0.9)
(3.1–3.5) (3.3–3.6) (3.1–3.5)
.4399 .5251 .0001*
2.5 (1.3)
(2.1–2.9)
3.1 (1.1)
(2.9–3.4)
.0033*
2.7 (0.1) 24.4 (5.6)
(2.5–3) (23.2–25.5)
.1054 .0981
14.3 (2.2) 3.3 (0.9)
(13.8–14.7) (3.1–3.4)
.0013* .0040*
3.1 (1.2)
(2.8–3.5)
3.6 (0.6)
(3.5–3.7)
.0045*
3.3 (1.1) 3.6 (0.8)
(3–3.6) (3.3–3.8)
3.6 (0.8) 3.6 (0.7)
(3.5–3.8) (3.5–3.8)
.0318* .6042
(15.8–18.8) (2.1–2.9)
13.9 (4.6) 1.9 (1.4)
(13–14.9) (1.6–2.2)
.0002* .0152*
(1.6–2.5) (1.9–2.7) (3–3.7) (3.1–3.7) (3.1–3.7) (88.5–99.7)
1.1 1.7 2.8 3.1 3.2 97.1
(0.8–1.3) (1.5–2.1) (2.5–3.1) (2.9–3.3) (3–3.4) (93.9–100.3)
.0001* .0246* .0117* .1267 .1749 .3190
(1.4) (1.3) (1.1) (1) (0.9) (19.3)
(1.1) (1.3) (1.3) (1.1) (1) (15.6)
† Items are shown in detail in those domains with statistically significant differences. Mann-Whitney U test. * P , .05.
children with CL/P in the areas of ‘‘functional well-being’’ (P ¼ .0013) and ‘‘school environment’’ (P ¼ .0013), whereas in the domain ‘‘self-image,’’ the difference was favorable to the group of children with CL/P (P ¼ .0002). The comparative analysis of the scores from the domains and items of the COHIP-Sp between the two groups appears in Table 2. DISCUSSION Oral health–related quality of life based on the COHIPSp in children with history of surgically treated CL/P showed indices similar to children unaffected in a Chilean population. Nevertheless, the influence of the condition of CL/P was observed in relation to a lower quality-of-life score in some aspects such as functional well-being and school environment. The present study found a greater number of male children with CL/P and the left side more affected with a diagnosis of cleft lip-palate, with demographic data similar to that reported in Dutch and Colombian populations (Lucas et al., 2000; Hurtado et al., 2008). The average of the oral health–related quality of life in the group of children
with CL/P was lower than the group control by only three points. This result is consistent with that reported by Broder and Wilson-Genderson (2007) and Ward et al. (2013) in a population of American children with CL/P. In terms of functional well-being, the lower score reported by these patients occurred in the questions related to speaking in public and speaking or reading aloud at school (Table 2). These results are consistent with those described by Chetpakdeechit et al. (2009) in a Swedish population. This can be explained because secondary cleft palates are often associated with speech problems such as hypernasality and more concretely velar insufficiency (Hudon, 1997, cited by Chapados, 2000) characterized by hypernasal resonance and reduced intraoral pressure when pronouncing consonants, which causes the audible nasal sound during speech (Smith & Guyette, 2004, cited by Sullivan et al., 2011), directly affecting their social integration and cultural adaptation in adulthood (Berk et al., 2001). In addition, it should be pointed out that children miss school, and their emotional control or social interaction is hindered for reasons associated with the teeth, mouth, and face (Chetpakdeechit et al., 2009) or the presence of otitis media with effusion (Tierney et al., 2015).
Aravena et al., QUALITY OF LIFE IN CHILEAN CHILDREN WITH CLEFT LIP/PALATE
The only domain that showed a positive statistically significant difference for the group of patients with CL/P was ‘‘self-image’’ (P ¼ .0002;Table 2). The literature is not conclusive on this topic. Reports from these patients show lower self-esteem in the population with a history of CL/P (Berk et al., 2001). Nevertheless, the studies by Hunt et al. (2005) and Vinaccia et al. (2008) present conclusions similar to this study, arguing that the patients’ acceptance of their CL/P tends to lead to a more favorable self-image. The limitations of this investigation relate to the validation of the scale in terms of the reduced number of participants in the study group and a validity analysis limited to the test-retest reliability and face validity. In terms of the selected sample, there may be influence on the results given the age differences, geographic condition, and socioeconomic and cultural status of the participants in the study groups and their comparability. Furthermore, the small number of participants with CL/P may cause those items on the scale to show a false statistical association with low quality-of-life scores observed in the group with CL/P. With respect to the application of the scale, the results could be affected by the high number of questions on the instrument, which could affect the child’s concentration, in addition the wide age range of the participants (8 to 15 years), which could influence level of reading comprehension of the questionnaire. Despite these limitations, the use of the COHIP scale demonstrated good reliability, similar to the coefficient level previously reported for an American population (Broder and Wilson-Genderson, 2007). In addition, to enhance the power of the study and the external validity of the results, the study groups were matched by considering double observations in the control group to increase the statistical power given the reduced ´ number of subjects with CL/P (Argimon and Jimenez, 2004), and the DMFT index avoided bias in differences in the quality-of-life results in relation to this variable (Lucas et al., 2000). In conclusion, oral health–related quality of life in children treated for CL/P was similar to the children unaffected by CL/P, being positive in terms of their selfimage. However, a lower quality of life was observed in areas such as functional well-being and school environment. This investigation has been a first step in the research line of oral health–related quality of life of patients with cleft lip-palate in the Latin American region. Nevertheless, it is suggested that studies in this vein should continue, and researchers should conduct risk factor analyses associated with the levels of quality of life of children with CL/P detectable in the surgical or rehabilitative treatment, as well as analyze the quality of life in children or adult populations in Hispano-America. Acknowledgments. The authors thank the surgeons and research team members at their three study sites: Dr. Fernando Salinas (Hospital Base in Valdivia), Dr. Roberto Pantoja (San Borja Arriara´n Hospital in Santiago), and Ms. Claudia Aguilera Susuki (School of Speech Therapy and
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Assistance Network for Children with Cleft Lip/Palate in Puerto Montt and Gantz Fundation of Child Cleft Palate in Santiago). We are grateful for all parents and children who participated voluntarily in this research. For them, our thanks and respect.
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Sullivan SR, Vasudavan S, Marrinan EM, Mulliken JB. Submucous cleft palate and velopharyngeal insufficiency: comparison of speech outcomes using three operative techniques by one surgeon. Cleft Palate Craniofac J. 2011;48:561–570. Tierney S, O’Brien K, Harman NL, Sharma RK, Madden C, Callery P. Otitis media with effusion: experiences of children with cleft palate and their parents. Cleft Palate Craniofac J. 2015;52:23–30. Vinaccia S, Quiceno JM, Ferna´ndez H, Calle LA, Naranjo M, Osorio J. Autoesquemas y habilidades sociales en adolescentes con ´ ´ diagnostico de labio y paladar hendido. Pensamiento Psicologico. 2008;4:123–135.
Ward JA, Vig KWL, 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;50:174–181. World Health Organization, Human Genetics Programme. Global Strategies to Reduce the Health-Care Burden of Craniofacial Anomalies: Report of WHO Meetings on International Collaborative Research on Craniofacial Anomalies. Geneva, Switzerland, 5–8 November 2000; Park City, Utah, USA 24–26 May 2001. Geneva, Switzerland: Human Genetics Programme, Management of Noncommunicable Diseases, World Health Organization; 2002.
ORIGINAL ARTICLE Oral Health–Related Quality of Life in Children in Chile Treated for Cleft Lip and Palate: A Case-Control Approach ´ ´ D.D.S, Cesar Pedro C. Aravena, D.D.S., Ph.D., Tania Gonzalez, D.D.S, Tamara Oyarzun, Coronado, D.D.S, M.Sc. Objective: To compare the oral health–related quality of life of patients treated for cleft lip and/or cleft palate (CL/P) versus unaffected children between 8 and 15 years of age using a Spanish-language version of the Child Oral Health Impact Profile (COHIP-Sp) administered to a Chilean population. Design: A cross-sectional study with a matched case-control design was used. Methods: Participants were 48 children (mean age 11.3 years) with a history of CL/P from three cities in Chile and one group of 96 children (mean age 11.2 years) unaffected by CL/P. The COHIP-Sp was applied to both groups. Quality of life was compared according to the overall score and the average score of items and domains on the COHIP-Sp scale between the two groups (Mann-Whitney U test; P , .05). Results: The COHIP-Sp score was 94.1 6 19.3 in children with CL/P and 97.1 6 15.6 for the control group (P ¼ .31). A significantly lower score was observed in the group with CL/P in the domains ‘‘functional well-being’’ (P ¼ .001) and ‘‘school environment’’ (P ¼ .001); the only average in favor of the quality of life in children with CL/P was in ‘‘self-image’’ (P ¼ .0002). Conclusion: The oral health–related quality of life of children with a history of CL/P was similar to that of the control group. Nevertheless, a lower quality of life was observed concerning items associated with speech and being understood by other people. Further study into the risk factors associated with surgery and rehabilitative treatment is recommended. KEY WORDS:
child; Chile; cleft lip; cleft palate; oral health; quality of life
Region in the south of Chile show this prevalence increasing to 2.03 per 1000 live births (Rosa, 2010). The treatment of patients with CL/P seeks to achieve normal growth and development of the face, complete closure of the cleft, comprehensible speech, normal hearing, a good esthetic result, and consequently correct socialization (Eberlinc and Kozelj, ˇ 2012). There are, however, certain sequelae that will influence quality of life, such as difficulties in eating, speaking, oral hygiene, esthetics, growth, and other physical problems such as ear infections, alterations in hearing, and a psychosocial burden (Ward et al., 2013). Oral health complications are numerous, including dental agenesis and supernumerary and poorly positioned teeth (Chapados, 2000), and cause speech disorders such as hypernasality and facial alterations such as nose and mouth asymmetry, which also affect the individual’s self-image, social affiliation, and adaptation (Broder et al., 1994; Berk et al., 2001). In response to this situation, the measurement of oral health–related quality of life has become an important health indicator and reveals the functional and psychosocial results of oral diseases and conditions (Cohen, 1997, quoted by Do and Spencer, 2008). Broder and Wilson-Genderson (2007) adapted and validated the Child Oral Health Impact Profile (COHIP) in English to measure quality of life in terms of oral health, functional well-being, social-emotional well-being, school
Cleft lip and/or cleft palate (CL/P) is one of the most frequent congenital malformations, with a worldwide prevalence estimated at 0.8/1000 live births (World Health Organization, Human Genetics Programme, 2002), and there is a significant variation in global incidence, depending on geography, gender, race, and ethnic group, among others (Rozendaal et al., 2012). South America has one of the highest frequencies in the world: in Bolivia, it is 2.5 per 1000 live births and in Argentina, 1.5 per 1000 live births (Nazer et al., 2010). Colombia and Brazil have reported a prevalence of 1 per 1000 live births (Hurtado et al., 2008; Raposo-do-Amaral et al., 2011). In Chile, the reported frequency is 1.8 per 1000 live births, with 452 new cases reported annually (Chilean Ministry of Health [MINSAL], 2009); however, local reports in the Los R ´ıos
Dr. Aravena is Professor, Institute of Anatomy, Histology and Pathology, Faculty of Medicine, Universidad Austral de Chile, Valdivia, Chile. Ms. Gonza´lez and Ms. Oyarzun ´ are Dentists, location. Mr. Coronado is Professor, Department of Morphofunction, Faculty of Medicine, Universidad Diego Portales, Santiago, Chile. Financial support: This study had private financial support from the researchers and the School of Dentistry, Universidad Austral de Chile. Submitted March 2015; Revised May 2015; Accepted May 2015. Address correspondence to: Dr. Pedro C. Aravena, School of Dentistry, Universidad Austral de Chile, 1640 Rudloff Street, Valdivia, Chile. E-mail [email protected]. DOI: 10.1597/15-095 0
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environment, and self-image in children 8 to 15 years old, recruiting participants with various oral health conditions such as craniofacial, orthodontic, and general pediatric dental anomalies. This scale presented a high internal consistency and an excellent test-retest reliability, contributing a valuable instrument for the assessment of the oral health–related quality of life in children with CL/P (Broder et al., 2014). The use of the COHIP in children with CL/P could show the impact of this condition and sequelae after treatment such as esthetic and speech problems, nervousness when speaking in public or at school (Chetpakdeechit et al., 2009) or obvious self-image problems (Berk et al., 2001); therefore, the use of the COHIP at the local level could reveal similar and relevant issues to ascertain the oral health of this population. In light of the bibliographic references presented, we propose the following hypothesis: children between 8 and 15 with a history of surgically treated CL/P exhibit an oral health–related quality of life similar to children unaffected by this condition in a Chilean population. The aim of this study was to determine oral health– related quality of life by means of an adapted Spanishlanguage version of the COHIP in children and adolescents between 8 and 15 years old treated surgically for nonsyndromic CL/P and to compare it with children and adolescents unaffected by CL/P in a Chilean population. METHODS Design and Patients A cross-sectional study with a matched case-control design was conducted and approved for the Ethical and Research Committee of the Regional of Health Department of Chile (Register No. 152/2014). Forty-eight children and adolescents with a history of CL/P participated (average age 11.3 6 2.3 years; 62.5% male) and were treated surgically in primary care hospitals in Chile: Hospital Base in Valdivia, San Borja Arriara´n Hospital in Santiago, and the Assistance Network for Children With Cleft Lip/Palate in Puerto Montt City. In addition, there was a control group of 96 children and adolescents unaffected by CL/P (average age 11.2 6 2.3 years; 46.8% male) from public schools in Valdivia, Chile. For matching the two study groups, a number of children were selected at a 2:1 ratio between unaffected children and those with a history of CL/P and of similar age, gender, and the same DMFT index (Lucas et al., 2000), as this index was statistically similar between the two groups (CL/P group DMFT index ¼ 2.2 6 2.8; control group DMFT index ¼ 2.7 6 2.6; P . .05). The group of children with a history of CL/P was selected by convenience sampling. Those children between 8 and 15 years of age were included who could read and write in Spanish, who agreed voluntarily to
participate through a written informed consent, and whose parents signed an informed consent. Children or adolescents who presented a disabling medical condition, syndromic CL/P, or mental disorders were excluded. The children in the control group were selected by means of simple random sampling from five public schools in Valdivia. Regarding the external validity of our results, the sample size was based on the incidence rate of children with CL/P in Chile calculated on the basis of a power of 80%, 95% confidence level according to the overall population of Valdivia consistent with its overall birth rate and rate of live births with CL/P in Chile ¼ 1.8/1000 (MINSAL, 2009). This calculation detected a difference of 13.3 points on the overall COHIP score between children with CL/P and the control group (Broder and Wilson-Genderson, 2007; Ward et al., 2013) and matching with double the number of subjects in the ´ control group (Argimon and Jimenez, 2004). This calculation provided a minimum number of 31 subjects between 8 and 15 years (EPIDAT 3.0, Statistical Analysis Software) Adaptation and Validity of the COHIP to Spanish The first stage was to adapt the COHIP to Spanish. This instrument is composed of five domains (well-being in oral health, functional well-being, social-emotional well-being, school environment, and self-image), with a total of 34 items. Each item is valued on a 5-item Likerttype scale ranging from 0 to 4 (4 ¼ never, 3 ¼ almost never, 2 ¼ sometimes, 1 ¼ fairly often, and 0 ¼ always), with the exception of the domain ‘‘self-image,’’ which presents a score assigned with an ordinal and increasing number (Broder, 2007). The questions with no response are scored as zero. The total score of the scale varies between 0 and 136, where a higher score corresponds to a better oral health–related quality of life (Broder and Wilson-Genderson, 2007). In its adaptation, the COHIP was translated from English to Spanish simultaneously by two investigators (T.G. and T.O.), who then agreed with a third Englishspeaking investigator on a definitive version called COHIP-Sp. This was taken to a qualitative review process in terms of face and construct validity by five experts in the treatment of children with CL/P and a focus group with 15 children from 8 to 15 years old from a public school in Valdivia. In this analysis, question 22 was modified to use the word withdrawn instead of insecure. The rough draft of the scale was applied on two occasions, 2 weeks apart, to a pilot group of 30 randomly selected children 8 to 15 years of age from a public school in Valdivia. The total score obtained in both applications was analyzed by means of the intraclass correlation coefficient R. This test had a high test-retest reliability (intraclass correlation coefficient
Aravena et al., QUALITY OF LIFE IN CHILEAN CHILDREN WITH CLEFT LIP/PALATE
TABLE 1 Sociodemographic Characteristics and Treatment History of Patient Group With Cleft Lip and/or Palate in Cities of Chile Valdivia, n ¼ 29 Age, average (SD) Gender Male, n (%) Cleft diagnosis Cleft lip, n (%) Cleft palate, n (%) Cleft lip-palate, n (%)
11.5 (2.5)
Santiago, Puerto Montt, n ¼ 12 n¼7 10.5 (2.1)
11.7 (1.7)
Total, N ¼ 48 11.3 (2.3)
19 (65.5)
6 (50.0)
5 (71.4)
30 (62.5)
3 (10.3)
2 (16.6)
0 (0.0)
5 (10.4)
8 (27.5)
5 (41.6)
0 (0.0)
13 (27.0)
18 (62.0)
5 (41.6)
7 (100.0)
30 (62.5)
Cleft side Left, n (%) Right, n (%) Bilateral, n (%) Median cleft palate, n (%)
7 (24.1) 7 (24.1) 7 (24.1)
2 (16.6) 3 (25.0) 2 (16.6)
6 (85.7) 0 (0.0) 1 (14.2)
15 (31.2) 10 (20.8) 10 (20.8)
8 (27.5)
5 (41.6)
0 (0.0)
13 (27.0)
Presurgical orthopedics Yes, n (%)
15 (51.7)
9 (75.0)
4 (57.1)
28 (58.3)
Number of surgeries Average no. (SD)
3.2 (2.0)
3.6 (2.9)
4.5 (1.6)
3.5 (2.2)
Age at first surgery Average months (SD)
9.5 (10.0)
6.4 (2.1)
5.8 (4.5)
8.2 (8.1)
Age at last surgery Average years (SD)
7.0 (4.5)
6.0 (4.0)
9.1 (3.8)
7.1 (4.3)
0
intraoral exploration mirror without magnification, applying the DMFT index. In the group of children with CL/P, independent variables related to the sociodemographic data and history of treatment for CL/P were recorded by interviewing the parents (Table 1): age (in years), gender (male/female), diagnosis of the cleft according to the classification of Clinical Guide for Cleft Lip-Palate (cleft lip, prepalatal cleft, or primary palate cleft; cleft palate or cleft hard and/or soft palate; cleft lip-palate; MINSAL, 2009), side affected by the cleft (left, right, bilateral, medial cleft palate), use of presurgical orthopedic apparatus (yes/no), number of surgeries under general anesthesia (No. of surgeries), age at first surgery (months), and age at last surgery (years). Only the child’s age (years) and gender (male/female) were recorded for the control group. The oral health–related quality of life score from the COHIP-Sp was recorded as a dependent variable, obtaining the average score of the participants from each group by items, domain, and sum of the total points from the scale. Data Analysis
[ICC] . .8) for questions 2, 9, 10, 11, 14, 17, 20, 23, 26, 28, and 30, and the total ICC of the instrument was .89 (Norman and Streiner, 2008). Application of the COHIP-Sp The second stage was to apply the COHIP-Sp. For this, participants were contacted by telephone between August and October 2014. In the group of children with CL/P, two investigators (T.G. and T.O.) applied the COHIP-Sp according to the geographical location and willingness of the family to participate. Sociodemographic data and history of treatment for CL/P were recorded based on an interview with the parents. For the control group, the investigators visited the children and parents at their schools after coordinating with the establishment’s administration. In both groups, the objective of the scale was explained orally. Once the children agreed to participate, they were given the scale in writing. Each child was left alone in a physical place free of visual or auditory interference. After administering the questionnaire, a basic oral examination was performed to match the training groups. For this, each child sat in a chair with full neck extension and maximum oral opening. An investigator (T.G.) used a flashlight with a frontal headpiece and a flat No. 5
The variables were analyzed with descriptive statistics, presenting the results with dispersion measurements and central tendency (average 6 standard deviation). To compare the quality of life between the study groups, the parametric/nonparametric distribution of the total average score of the scale in both was analyzed using the Shapiro-Wilk test (P , .05). According to the result, the scores were compared to total average, average of domains, and the items on the scale with the corresponding test (t test/Mann-Whitney U test). An index for a better quality of life was determined when the average of the score from the scale was significantly higher between the two groups (P , .05; 95% confidence interval). The analyses were done with the statistical R Core Team program (2013). RESULTS Table 1 presents the sociodemographic information, diagnostic history, and treatment for the group of children with CL/P. The distribution analysis of the averages of the score showed a nonparametric distribution in the group of children with CL/P (P¼ .47) and parametric for the control group (P¼.03). The average overall score of the COHIP-Sp was 94.1 6 19.3 for the group of children with CL/P and 97.1 6 15.6 for the control group, there being no statistically significant difference (P ¼ .31). However, the analysis of the average score in the domains on the scale revealed significantly unfavorable differences for the
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Table 2
Average Scale Score of the COHIP-Sp and Domains (Score Range 0–4 Points) Cleft Lip and/or Palate Group, n ¼ 48 Average (SD)
Control Group, n ¼ 96
CI 95%
Average (SD)
CI 95%
P Value‡
Oral health well-being (10)
24.5 (6)
(22.7–26.2)
25.1 (5.6)
(23.9–26.2)
.5490
Functional well-being (6) Had trouble biting off or chewing foods such as apple, carrot, or firm meat Had difficulty eating foods you would like to eat because of your teeth, mouth, or face Had trouble sleeping because of your teeth, mouth, or face Had difficultly saying certain words because of your teeth, mouth, or face Had people have difficulty understanding what you were saying because of your teeth, mouth, or face Had difficulty keeping your teeth clean because of your teeth, mouth, or face Social-emotional well-being (8)
16.8 (4.2)
(15.5–18.0)
19.2 (4.1)
(18.4–20.1)
.0013*
2.4 (1.1) 22.5 (7.4)
(2.1–2.7) (20.4–24.7)
School environment (4) Missed school for any reason because of your teeth, mouth, or face Had difficulty paying attention in school because of your teeth, mouth, or face Not wanted to speak/read aloud in class because of your teeth, mouth, or face Not wanted to go to school because of your teeth, mouth, or face
12.9 (2.6) 2.7 (1)
(12.1–13.7) (2.4–3)
Self-image (6) Been confident because of your teeth, mouth, or face Felt that you were attractive (good looking) because of your teeth, mouth, or face I have good teeth I feel good about myself When I am older, I believe (think) that I will have good teeth When I am older, I believe (think) that I will have good health COHIP-Sp (34)
17.2 (5.3) 2.5 (1.3) 2 2.3 3.4 3.4 3.4 94.1
Child Oral Health Impact Profile Domains and Items (n of Items)†
2.8 (1.2)
(2.5–3.2)
3.1 (1.2)
(2.8–3.3)
.3393
3.4 (0.9) 3.3 (1) 2.1 (1.2)
(3.1–3.7) (3.1–3.6) (1.7–2.4)
3.3 (0.9) 3.5 (0.8) 3.3 (0.9)
(3.1–3.5) (3.3–3.6) (3.1–3.5)
.4399 .5251 .0001*
2.5 (1.3)
(2.1–2.9)
3.1 (1.1)
(2.9–3.4)
.0033*
2.7 (0.1) 24.4 (5.6)
(2.5–3) (23.2–25.5)
.1054 .0981
14.3 (2.2) 3.3 (0.9)
(13.8–14.7) (3.1–3.4)
.0013* .0040*
3.1 (1.2)
(2.8–3.5)
3.6 (0.6)
(3.5–3.7)
.0045*
3.3 (1.1) 3.6 (0.8)
(3–3.6) (3.3–3.8)
3.6 (0.8) 3.6 (0.7)
(3.5–3.8) (3.5–3.8)
.0318* .6042
(15.8–18.8) (2.1–2.9)
13.9 (4.6) 1.9 (1.4)
(13–14.9) (1.6–2.2)
.0002* .0152*
(1.6–2.5) (1.9–2.7) (3–3.7) (3.1–3.7) (3.1–3.7) (88.5–99.7)
1.1 1.7 2.8 3.1 3.2 97.1
(0.8–1.3) (1.5–2.1) (2.5–3.1) (2.9–3.3) (3–3.4) (93.9–100.3)
.0001* .0246* .0117* .1267 .1749 .3190
(1.4) (1.3) (1.1) (1) (0.9) (19.3)
(1.1) (1.3) (1.3) (1.1) (1) (15.6)
† Items are shown in detail in those domains with statistically significant differences. Mann-Whitney U test. * P , .05.
children with CL/P in the areas of ‘‘functional well-being’’ (P ¼ .0013) and ‘‘school environment’’ (P ¼ .0013), whereas in the domain ‘‘self-image,’’ the difference was favorable to the group of children with CL/P (P ¼ .0002). The comparative analysis of the scores from the domains and items of the COHIP-Sp between the two groups appears in Table 2. DISCUSSION Oral health–related quality of life based on the COHIPSp in children with history of surgically treated CL/P showed indices similar to children unaffected in a Chilean population. Nevertheless, the influence of the condition of CL/P was observed in relation to a lower quality-of-life score in some aspects such as functional well-being and school environment. The present study found a greater number of male children with CL/P and the left side more affected with a diagnosis of cleft lip-palate, with demographic data similar to that reported in Dutch and Colombian populations (Lucas et al., 2000; Hurtado et al., 2008). The average of the oral health–related quality of life in the group of children
with CL/P was lower than the group control by only three points. This result is consistent with that reported by Broder and Wilson-Genderson (2007) and Ward et al. (2013) in a population of American children with CL/P. In terms of functional well-being, the lower score reported by these patients occurred in the questions related to speaking in public and speaking or reading aloud at school (Table 2). These results are consistent with those described by Chetpakdeechit et al. (2009) in a Swedish population. This can be explained because secondary cleft palates are often associated with speech problems such as hypernasality and more concretely velar insufficiency (Hudon, 1997, cited by Chapados, 2000) characterized by hypernasal resonance and reduced intraoral pressure when pronouncing consonants, which causes the audible nasal sound during speech (Smith & Guyette, 2004, cited by Sullivan et al., 2011), directly affecting their social integration and cultural adaptation in adulthood (Berk et al., 2001). In addition, it should be pointed out that children miss school, and their emotional control or social interaction is hindered for reasons associated with the teeth, mouth, and face (Chetpakdeechit et al., 2009) or the presence of otitis media with effusion (Tierney et al., 2015).
Aravena et al., QUALITY OF LIFE IN CHILEAN CHILDREN WITH CLEFT LIP/PALATE
The only domain that showed a positive statistically significant difference for the group of patients with CL/P was ‘‘self-image’’ (P ¼ .0002;Table 2). The literature is not conclusive on this topic. Reports from these patients show lower self-esteem in the population with a history of CL/P (Berk et al., 2001). Nevertheless, the studies by Hunt et al. (2005) and Vinaccia et al. (2008) present conclusions similar to this study, arguing that the patients’ acceptance of their CL/P tends to lead to a more favorable self-image. The limitations of this investigation relate to the validation of the scale in terms of the reduced number of participants in the study group and a validity analysis limited to the test-retest reliability and face validity. In terms of the selected sample, there may be influence on the results given the age differences, geographic condition, and socioeconomic and cultural status of the participants in the study groups and their comparability. Furthermore, the small number of participants with CL/P may cause those items on the scale to show a false statistical association with low quality-of-life scores observed in the group with CL/P. With respect to the application of the scale, the results could be affected by the high number of questions on the instrument, which could affect the child’s concentration, in addition the wide age range of the participants (8 to 15 years), which could influence level of reading comprehension of the questionnaire. Despite these limitations, the use of the COHIP scale demonstrated good reliability, similar to the coefficient level previously reported for an American population (Broder and Wilson-Genderson, 2007). In addition, to enhance the power of the study and the external validity of the results, the study groups were matched by considering double observations in the control group to increase the statistical power given the reduced ´ number of subjects with CL/P (Argimon and Jimenez, 2004), and the DMFT index avoided bias in differences in the quality-of-life results in relation to this variable (Lucas et al., 2000). In conclusion, oral health–related quality of life in children treated for CL/P was similar to the children unaffected by CL/P, being positive in terms of their selfimage. However, a lower quality of life was observed in areas such as functional well-being and school environment. This investigation has been a first step in the research line of oral health–related quality of life of patients with cleft lip-palate in the Latin American region. Nevertheless, it is suggested that studies in this vein should continue, and researchers should conduct risk factor analyses associated with the levels of quality of life of children with CL/P detectable in the surgical or rehabilitative treatment, as well as analyze the quality of life in children or adult populations in Hispano-America. Acknowledgments. The authors thank the surgeons and research team members at their three study sites: Dr. Fernando Salinas (Hospital Base in Valdivia), Dr. Roberto Pantoja (San Borja Arriara´n Hospital in Santiago), and Ms. Claudia Aguilera Susuki (School of Speech Therapy and
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Assistance Network for Children with Cleft Lip/Palate in Puerto Montt and Gantz Fundation of Child Cleft Palate in Santiago). We are grateful for all parents and children who participated voluntarily in this research. For them, our thanks and respect.
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Cleft Palate–Craniofacial Journal, March 2016, Vol. 53 No. 2
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