Published online August 1, 2007
PEDIATRICS Vol. 120 No. 2 August 2007, pp. e283-e290 (doi:10.1542/peds.2006-2091)
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ARTICLE

Health-Related Quality of Life Among Preadolescent Children With Oral Clefts: The Mother's Perspective

Peter C. Damiano, DDS, MPHa,b, Margaret C. Tyler, MA, MSWa, Paul A. Romitti, MS, PhDc, Elizabeth T. Momany, PhDa, Michael P. Jones, PhDd, John W. Canady, MDe, Michael P. Karnell, PhDf,g and Jeffrey C. Murray, MDh,i,j

a Health Policy Research Program, Public Policy Center
b Departments of Preventive and Community Dentistry
c Epidemiology
d Biostatistics
e Plastic and Reconstructive Surgery
f Speech Pathology and Audiology
g Otolaryngology
h Pediatrics
i Pediatric Dentistry
j Biological Sciences, University of Iowa, Iowa City, Iowa


   ABSTRACT
TOP
 ABSTRACT
METHODS
 RESULTS
 DISCUSSION
 CONCLUSIONS
 REFERENCES
 
OBJECTIVE. The purpose of this study was to evaluate the factors that affect the health-related quality of life of preadolescent children with nonsyndromic oral clefts using the Pediatric Quality of Life Inventory instrument and to evaluate whether there were any differences related to the type of cleft or other factors.

METHODS. Data for this study were derived from telephone interviews with the parents of a statewide population of children who were in the Iowa Registry for Congenital and Inherited Disorders, were aged 2 to 12 years, had nonsyndromic oral clefts, and were born in Iowa between January 1, 1990, and December 31, 2000. Twenty-minute interviews were conducted with mothers of 104 children in the spring and summer of 2003; respondents then completed and mailed back Pediatric Quality of Life Inventory surveys 2 to 3 weeks after the interviews (69% participation rate).

RESULTS. After controlling for demographic characteristics, children with less severe speech problems had higher total Pediatric Quality of Life Inventory scores as well as higher physical and psychosocial health domain scores. Age and cleft type interacted, with younger children (aged 2–7 years) with a cleft lip or cleft lip and palate having higher health-related quality of life scores than children with an isolated cleft palate; however, this pattern was reversed for older children (aged 8–12 years).

CONCLUSIONS. Speech and aesthetic concerns seem to have been important factors affecting the health-related quality of life for children with oral clefts. These factors seem to be more important as children get closer to adolescence (ages 8–12 years) when acceptance by peers becomes more critical.

Key Words: health-related quality of life • oral clefts • cleft lip • cleft palate

Abbreviations: HRQoL—health-related quality of life • OC—oral cleft • PedsQL—Pediatric Quality of Life Inventory • CL—cleft lip • CLP—cleft lip and palate • CP—cleft palate • IRCID—Iowa Registry for Congenital and Inherited Disorders

The World Health Organization definition of health as not only the absence of disease but also the presence of factors that enhance physical, mental, and social well-being has led to a broader conceptualization of overall health that essentially translates into quality of life.1 Increasingly, successful outcomes of medical treatment for children are not defined solely in terms of cure, repair, or remission but also extend to the maintenance or improvement of patients' quality of life after treatment.2–4 This is especially true for children with chronic health conditions or conditions that require long-term treatment protocols, such as children with craniofacial abnormalities. The quality of the reconstructive and supportive procedures that a child with a chronic health condition receives can have an impact on their health-related quality of life (HRQoL).

Several studies have evaluated the HRQoL for adolescents and adults with oral clefts (OCs).5–8 Only a few, however, have attempted to measure the HRQoL of children with this condition, and none of these studies has used the Pediatric Quality of Life Inventory (PedsQL) to measure HRQoL.9,10 The PedsQL instrument was designed to measure HRQoL in children and to distinguish between healthy children and children with acute and chronic health conditions. The reliability and validity of the instrument has been demonstrated in the literature,11–15 and it has been used to evaluate the HRQoL for children with many different health conditions, including heart disease, cancer, asthma, diabetes, spinal muscular atrophy, psychiatric problems, arthritis, and cerebral palsy.16–26

This article reports findings regarding HRQoL among a registry-based sample of children who had nonsyndromic OCs and were aged 2 through 12. We compared total, physical, and psychosocial quality-of-life scores of children with OCs with published norms for healthy children. We then examined the effects of cleft type (lip only [CL], palate only [CP], lip and palate [CLP]) on quality of life, controlling for other related variables. The primary research questions in this study were as follows:

  1. What is the HRQoL reported for children with OCs using the PedsQL (mother's perspective), and how does it compare with published norms for other children?
  2. What factors are associated with total, physical, and psychosocial quality-of-life scores in children with OCs?
  3. Does the HRQoL vary for children with different types of clefts (CL, CP, and CLP)?
In addition, from the literature, it is hypothesized that a relationship exists between the type of cleft and age of the child in terms of the HRQoL, making age an important covariate in this study.


   METHODS
TOP
 ABSTRACT
 METHODS
RESULTS
 DISCUSSION
 CONCLUSIONS
 REFERENCES
 
Data for this study were derived from telephone interviews with the parents of a statewide population of children who were aged 2 to 12, had nonsyndromic OCs, and were born in Iowa between January 1, 1990, and December 31, 2000.

Study Population
Ascertainment of children was conducted by the Iowa Registry for Congenital and Inherited Disorders (IRCID). The IRCID conducts active, population-based surveillance of pregnancy outcomes (elective terminations, stillbirths, and live births) and of diagnoses of birth defects among Iowa residents. For this project, liveborn infants who received a diagnosis of an OC within the first year of life were identified, and medical charts of these patients were reviewed by trained IRCID staff to identify liveborn infants who received a diagnosis of a nonsyndromic OC. Nonsyndromic clefts included those with no evidence of additional noncleft structural birth defects, recognized etiology, or evidence of significant mental retardation. Cases were reviewed by a clinical geneticist, and in most cases, physical examinations and family histories were obtained by the same. Records of patients with nonsyndromic OCs were then matched to State of Iowa death certificate data to identify the current vital status of these patients and to exclude from the study patients who were deceased.

For each living patient who had a diagnosis of a nonsyndromic OC, an extensive search of available local, Internet, and commercial databases was conducted to find the current name, address, and telephone number of the patient's birth mother. Mothers who currently did not have custody of the patient (eg, child placed for adoption or foster care, mother was incarcerated) were excluded from the study. For the remaining eligible patients, IRCID staff contacted a local physician of each patient (generally a pediatrician or family practitioner) to inform the physician of our desire to contact the patient for study. After the physician notification, the IRCID mailed a letter to the birth mother of the patient to invite her to participate in the study; she was also asked to sign and return an informed consent form releasing her contact information, including telephone number, to the study interview team. Previous studies have indicated that the mother is overwhelmingly the person in the family with the most knowledge about a child's health. All correspondence, consent forms, and study instruments used were approved by the University of Iowa institutional review board.

Survey Instrument and Process
When the signed informed consent was received, a 20-minute telephone interview was conducted with the mother, which included questions about the type and severity of the child's cleft, location and type of cleft care received thus far, access to care, health status, clinical outcomes (eg, satisfaction with esthetics and speech), and social outcomes (eg, school performance, parenting stress). Items that addressed problems with speech and satisfaction with facial appearance were based on clinical measures and developed in collaboration with expert clinicians at the University of Iowa Hospitals and Clinics and the University of Pittsburgh. The speech problem scale consisted of 5 items that measured how often the child experienced difficulty with speech. These were coded as follows: 1 = never, 2 = sometimes, 3 = usually, and 4 = always. Higher scores would indicate more difficulty. To facilitate easier interpretation when using the scale with other measures, we reverse-coded each item and linearly transformed the scores to a 0 to 100 metric in which a higher score would indicate fewer problems: 100 = never, 66.7 = sometimes, 33.3 = usually, and 0 = always.

The 15-question Children and Adolescent Health Measurement Initiative Children with Special Health Care Needs screening instrument was also included as 1 indicator of the health of the child and to evaluate how frequently children with an oral cleft meet the established criteria of being a child with special health care needs.27 Interviews were conducted between March and August 2003.

PedsQL HRQoL Instrument
At the end of the telephone interview, biological mothers were asked to complete a written PedsQL survey that was mailed to them. The PedsQL version 4.0 is a 23-item instrument designed to measure age-specific HRQoL in children and adolescents aged 2 through 18. The PedsQL 4.0 provides an overall measure of quality of life in the domains of physical (8 items), emotional (5 items), social (5 items), and school (5 items) functioning. Each item asks the parent to indicate how often the child has experienced problems in specific areas (eg, walking, playing with other children, feeling sad or blue) in the previous month. Item responses are arranged in a 5-point Likert-type format, ranging from 0 (never a problem) to 4 (almost always a problem). A total psychosocial scale score is calculated from the individual emotional, social, and school domains.

Analysis
After the descriptive statistics were calculated for all variables, mean scores were computed for the PedsQL Total Quality of Life scale score, as well as for the Psychosocial Health scale score. Reliability analyses of these scale scores yielded very similar results to those published for the PedsQL, with alphas ranging from 0.81 to 0.91. Spearman rank-order tests were used to evaluate bivariate relationships between the PedsQL scores and the demographic characteristics of the children, as well as between the scores and the speech and aesthetic outcomes. One-way analysis of variance was used to evaluate differences in PedsQL scores by type of cleft and between the scores for all children with OCs and published data of a normative sample of "healthy" children.21,22 We examined potential differences in the HRQoL total scores and domain scores by type of OC using ordinary least squares regression to control for demographic differences, as well as for satisfaction with appearance and speech. Our initial models showed substantial violations of the regression assumptions that residuals be normally and symmetrically distributed. To address these problems, we logit-transformed our dependent variables (individual PedsQL scores) using the formula ln[(score – 1)/100 – (score – 1)]. This change improved normality and homoscedasticity, improving the reliability and interpretability of the results.

Iowa statewide norms were available for comparison for some of the survey items. These norms were derived from telephone interviews with the parents of 1041 Iowa children aged 2 through 12 obtained from the Iowa Child and Family Household Health Survey, a statewide study of the health and well-being of children in Iowa conducted in 2000. Specific information about the method used in that study can be found at http://ppc.uiowa.edu/health/ICHHS/iowachild2000/ichhs2000.htm.

Interview and PedsQL Participation Rates
A total of 937 children were identified by the IRCID as having been born in Iowa with an OC between 1990 and 2000. A total of 482 children were excluded from the study for (1) not having an OC (eg, submucous cleft, bifid uvula), (2) having received a diagnosis of syndromic, (3) having moved out of state, (4) being deceased, or (5) not living with the birth mother. Of the remaining 455 children, 129 could not be located; although some telephone or address information was found for another 81 of the children, the families could not be contacted by telephone. Of the 245 located families, 181 consented to participate in the study and 64 refused. A total of 151 mothers actually completed an interview for participation rates of 62% of the locatable families and 83% of the consenting families. Nonresponse bias tests indicated that nonresponders (ie, consented but did not complete an interview or PedsQL) and nonparticipants (ie, did not consent to participate) were similar to participants regarding the age of the mother and the child at the time of the interview and the gestational age of the child (ie, <37 weeks versus ≥37 weeks). Parents of male children with a CL were less likely to respond, and parents of male children with a CLP were less likely to participate. After the telephone interviews, 104 mothers returned the written PedsQL, for response rates of 69% of those participating, 57% of those consenting, and 42% of locatable eligible participants. Only results from the interviews with the 104 mothers who returned the PedsQL are presented in these analyses.


   RESULTS
TOP
 ABSTRACT
 METHODS
 RESULTS
DISCUSSION
 CONCLUSIONS
 REFERENCES
 
Demographic data for the 104 patients from whom PedsQL data were obtained are presented in Table 1 by type of cleft, with reference to state population figures for 2- to 12-year-old children in Iowa gathered as part of a different study of the health and well-being of children in the state. With the exception of a higher proportion of boys and a larger percentage with incomes greater than $75000 per year, sample demographics generally reflect those of the overall population of 2- to 12-year-old children in Iowa.


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  		TABLE 1 Demographic Characteristics of Children by Type of Cleft

 
Results of the bivariate relationships between the demographic characteristics of the children with OCs and the PedsQL total quality-of-life scores, as well as between the scores for physical and psychosocial components, are shown in Table 2. Higher household income was associated with higher total quality-of-life scores, as well as with high physical and psychosocial scores. A higher number in the household, a higher health status rating, and the mother's being married were similarly associated with higher scores. It should be noted that the mother's marital status was highly correlated with the number of people in the household.


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  		TABLE 2 Spearman Rank Correlations Between Demographic Characteristics and Quality-of-Life (PedsQL) Scale Scores

 
The total and component PedsQL scores were compared by type of cleft and compared with published normative PedsQL data for a "healthy" population (Table 3). The 3 cleft groups did not differ from each other on any of the PedsQL scales. The cleft lip and/or palate sample as a whole, however, scored significantly lower than norms for healthy children on the psychosocial health component scale.


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  		TABLE 3 Quality of Life in Children With Cleft Lip and/or Palate Compared With Norms for a Healthy Sample

 
Some aesthetic and speech concerns were found to have important bivariate associations with a higher quality-of-life rating (Table 4). The parent's perception of how happy the child was with his or her facial appearance was associated with higher total and psychosocial health PedsQL scores (but not with physical health). The mother's happiness with the child's facial appearance was not related to higher PedsQL scores, however. The parents' perception of the quality of the child's speech as measured by the 5-item scale indicated that the quality of the child's speech was highly correlated with the total, physical, and psychosocial health rating of the child.


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  		TABLE 4 Spearman's Rank Correlations Between Aesthetic and Speech Outcomes and Quality-of-Life (PedsQL) Scale Scores

 
The results of the final multivariate model are shown in Table 5. This model includes significant demographic variables as well as the type of cleft, age group of the child, and the hypothesized interaction between the 2. After controlling for demographic characteristics, children with less severe speech problems had higher total PedsQL scores as well as higher physical and psychosocial health domain scores. Children with CP had significantly higher PedsQL scores than children with CLP. Both categories of younger children (ages 2–4 and 5–7) had higher PedsQL scores than children who were 8 to 12 years of age. Note that in the final model, the child's gender and how happy the child was with his or her facial appearance were excluded because they were not significant in the preliminary models. Marital status was excluded from the final model because of its high correlation with household income; income was deemed to be the variable of greater explanatory value.


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  		TABLE 5 Regression Results for Relationships With PedsQL Scores: Parsimonious Models

 
To understand better the results in Table 5, we first calculated the predicted outcomes scores from the first model for all study participants. Second, we computed the average outcome scores for each combination of cleft type and age group. Third, we plotted these values, which appear in Figs 1, 2, and 3. Of particular interest is the interaction viewed as the crossed lines in the figures.


Figure 1
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  		FIGURE 1 Relationship of age and cleft type to total PedsQL HRQoL scores.

 

Figure 2
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  		FIGURE 2 Relationship of age and cleft type to PedsQL physical health score.

 

Figure 3
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  		FIGURE 3 Relationship of age and cleft type to PedsQL psychosocial health score.

 
Among children with CL and CLP, total HRQoL was lower for 2- to 4-year-olds than for 5- to 7-year-olds. It was also significantly lower among 8- to 12-year-olds (Fig 1). HRQoL scores for children with CP only, however, showed a markedly different relationship to child age. Scores were similar for children in the 2- to 4- and 5- to 7-year age groups but were substantially higher for the oldest group (ages 8–12). HRQoL scores for children with CP were also much lower than for children with the other 2 types of OCs except in the oldest age category, in which they were higher.

This interaction between child age and cleft type and its effect on the PedsQL total score held in the predictions of both the physical and psychosocial domain scores. The scores for children with CL and CLP increased from ages 2 to 4 years to 5 to 7 years but were lower for 8- to 12-year-olds, whereas scores for children with CP generally increased with age (Figs 2 and 3).


   DISCUSSION
TOP
 ABSTRACT
 METHODS
 RESULTS
 DISCUSSION
CONCLUSIONS
 REFERENCES
 
Better speech outcomes for preadolescent children with nonsyndromic OCs was significantly related to better total HRQoL scores, as well as to better physical and psychosocial domain scores using the PedsQL instrument, after controlling for other important factors. The relationship between cleft type and quality of life was more complicated, however, changing according to a child's age group. The presence of CL (the visually apparent component of OCs) may be of particular importance in this equation. The HRQoL scores increased for children with CL or CLP from ages 2 to 4 to ages 5 to 7. However, they declined for children who were aged 8 to 12, an age when physical appearance becomes more important with regard to social interaction and self-evaluation. The reverse pattern was true for children with an isolated CP, for whom older children had higher HRQoL scores. Increasing scores for children with CP could be related to improvements in speech that could occur after surgery and speech therapy.

Although this is the first study to evaluate the HRQoL from the mother's perspective in a population-based sample of preadolescent children with nonsyndromic OCs, the findings can be related to some published articles. In the only other published study to address specifically quality of life in children with craniofacial anomalies, including cleft lip and/or palate, Warschausky et al10 compared scores for 26 children with cleft lip and/or palate on the Child Health Questionnaire Parent Form with scores for 26 children with other craniofacial anomalies (eg, craniosynostosis). Results of this analysis showed that children with clefts scored within normal limits on both the physical and psychosocial scale scores of the Child Health Questionnaire.

Literature regarding the psychosocial domains of quality of life in this population is relatively extensive; psychological adjustment, self-esteem, and social interaction all have been evaluated and are relevant to the outcomes found in this study. For example, Millard and Richman28 found that speech and facial appearance were related to overall adjustment of children with OCs. Because their population of children and adolescents were aged 8 to 17, the changing patterns for younger children were not evident. However, their finding that children with CP had greater problems with behavioral and emotional difficulties related to speech problems may be interpreted to differ from our results. Although again an imperfect comparison, Broder and Strauss29 found that among 7-year-olds, children with CLP had lower self-concepts than children with other cleft types, a finding that could also be interpreted to differ from the findings here.

This study is the first to evaluate HRQoL for children with oral clefts using the PedsQL instrument. Results indicated that the scales were reliable and that the instrument can be useful in evaluating the HRQoL for children with OCs. The PedsQL can be helpful in that it is (1) 1 of the few HRQoL instruments that has repeatedly been shown to be reliable and valid for children who are as young as 2 years and have chronic health conditions, and (2) short enough to be included as 1 subcomponent in a broader study.

Children with nonsyndromic clefts have also been reported to have delays in cognitive development,30 brain structural asymmetries,31 higher lifetime mortality at all ages,32 and a wide range of genetic and environmental etiologies.33,34 All of these variables can now begin to be accounted for in future studies of HRQoL to determine whether subgroups can be defined on the basis of social, physical, or genetic characteristics.

The use of parent interviews for this data collection means, by definition, that these results reflect parental perception, which is 1 important yet specific outcome of cleft care. There is obviously important information that can be gained from other sources that would add a different dimension, such as clinical examinations to evaluate the physical and psychosocial health of a child with an OC. As with all studies, these results should be generalized and interpreted carefully. Although this study represents 1 of the few population-based studies of children with OCs, data were gathered from a single state in which a cleft team provides care for at least half of all children with OCs in the state. Because this study involved children only up to age 12, the HRQoL for these children as they reach adolescence and adulthood could change again. Additional study of this population as they age could provide valuable information about the HRQoL of adolescents and adults with different types of oral clefts. More studies about other views of the HRQoL for children with OCs, such as from the child's and the treating health professionals'35 perspective, should be explored in more detail as well. Conflicting perspectives among parents, children, and care providers could have important implications for the successful treatment of children.


   CONCLUSIONS
TOP
 ABSTRACT
 METHODS
 RESULTS
 DISCUSSION
 CONCLUSIONS
REFERENCES
 
The HRQoL varied for preadolescent children by type of cleft and by age. The HRQoL was better among children with CL and CLP than among those with CP until age 7, then declined for 8- to 12-year-olds. Children with CP demonstrated the opposite pattern, with better HRQoL observed in the oldest age group. One reason could be the difference in outward appearance related to lip involvement and the relative importance of facial appearance as the child gets closer to adolescence. The PedsQL proved beneficial as an instrument for evaluating the HRQoL for preadolescent children with an OC.


   ACKNOWLEDGMENTS
 
This research was supported by a grant from the National Institutes for Dental and Craniofacial Research (P60 DE-13076) and a grant from the Centers for Disease Control and Prevention (U50/CCU 7132380).


   FOOTNOTES
 
Accepted Jan 26, 2007.

Address correspondence to Peter C. Damiano, DDS, MPH, Health Policy Research Program, University of Iowa, Public Policy Center, 227 S Quadrangle, Iowa City, IA 52242. E-mail: peter-damiano{at}uiowa.edu

The authors have indicated they have no financial relationships relevant to this article to disclose.


   REFERENCES
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 METHODS
 RESULTS
 DISCUSSION
 CONCLUSIONS
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PEDIATRICS (ISSN 1098-4275). ©2007 by the American Academy of Pediatrics




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