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The Health Related Quality of Life of children with

CLP and their parents

Masterscriptie Orthopedagogiek Naam: Anouck Splinter Studentnummer: 5655684 Begeleidster AMC: Dr. Lotte Haverman Begeleider UvA: Drs. Ed de Bruin Tweede beoordelaar: Dr. Roos Rodenburg Amsterdam, augustus 2014

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Index: Abstract 3 Introduction 4 Method 7 Procedure 7 Participants 8 Measures 8 Socio-demographics 8

Health-related quality of life (HRQOL) 8

Satisfaction with appearance 9

Parental distress 9

Statistical analysis 11

Results 13

Socio-demographics 14

HRQOL 15

Satisfaction with appearance 18

Parental distress 19

Predictors of HRQOL in children with CLP 20

Predictors of clinical distress in parents 21

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Abstract:

Objectives: Having cleft lip and/or palate (CLP) and the therefore needed surgeries could have an impact on the health-related quality of life (HRQOL) of children. The aim of this study was to examine the HRQOL of children with CLP, the satisfaction of appearance of these children, the distress in parents of children with CLP, and the determinants of both the HRQOL of children and the distress in parents.

Methods: Participants were all children (0-18 years old) who registered at the website (www.hetklikt.nu) between December 2012 and February 2014 and filled in various questionnaires. Independent and one sample t-tests, univariate, multivariate and logistic regression analysis were used to analyze baseline data.

Results: Younger children with CLP (0-5 years old) scored lower on some HRQOL domains. Adolescents with CLP were more at risk for problems with psychosocial health, emotional and social functioning compared to the norm population. Satisfaction with appearance appeared to be a predictor for HRQOL. More than one third of the parents showed clinical levels of distress.

Conclusion: It seemed that children with CLP differ in their HRQOL from the norm population, but further research is needed. Also parents seemed affected by having a child with CLP. The study findings underline the relevance for systematically monitoring these children in daily clinical practice.

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Introduction:

One to two children per 1000 births will be born with a cleft lip or/and palate (CLP) and CLP is therefore one of the most common malformations in the Netherlands (Hoek, Kraaimaat, Admiraal, Kuijpers-Jagtman, & Verhaak, 2009). CLP occurs during the first weeks of pregnancy in which tissues of the lip and/or palate do not completely join together (Mossey, Little, Munger, Dixon, & Shaw, 2009). Different types of CLP are known due to the separate development of lip and palate. Children with only a cleft lip have a lip with a small notch or could have a complete split from their lip to their nose. Both could be unilateral or bilateral. Children with cleft lip and palate have besides a split in their lip, an opening in the roof of their mouth in which the palate was not completely closed either. Another type of CLP is an isolated cleft palate where no split in the lip is visible and only the palate is affected (Mossey et al., 2009). Having CLP can be seen as a chronic disease, because it occurs at birth, lasts longer than three months, and requires lifestyle changes, is incurable but medically manageable (Mokkink, van der Lee, Grootenhuis, Offringa, & Heymans, 2008).

Children with CLP have to undergo surgeries to normalize the face and to have less difficulties with speech and eating. Goals of the surgeries are to construct a symmetrical nose with open airways, a cleft area that has alveolar integrity and an intact palate which allows normal speech development (Marcusson, Paulin & Ostrup, 2002). Despite the surgical procedures, children with CLP could still have facial disfigurement, dental problems and speech/hear difficulties (Baker, Owens, Stern, & Willmot, 2009; Feragen, Borge & Rumsey, 2009), and consequently multidisciplinary care from birth to adulthood is required (Mossey et al., 2009).

Having CLP and the needed reconstruction surgeries could have an impact on the health-related quality of life (HRQOL) (Damiano et al., 2007). According to the World Health Organization (WHO), quality of life refers to the perception of individuals about their life related to their goals, expectations, concerns and standards in the culture and value system in which they live. When the physical symptoms, functional status as well as disease impact on psychological and social functioning are also incorporated in this concept, it could be defined as HRQOL. Measuring the HRQOL is crucial to assess the influence of children’s disease on their life (Haverman et al., 2012).

Children with chronic conditions have a lower HRQOL in comparison with healthy children (Engelen et al., 2009). Only few studies have assessed the HRQOL of children with CLP and conflicting findings have been found on the HRQOL in children with CLP. Damiano et al. (2007) and Sagheri et al. (2009) reported that children between 2 and 12 years old with

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CLP had lower HRQOL scores compared to healthy children. Other research showed that the HRQOL of children with CLP was relatively high and children with CLP had equivalent or better scores than healthy children (Bos & Prahl, 2011; Kramer et al., 2009).

Different studies did examine determinants for HRQOL, such as age, gender, kind of cleft, speech and satisfaction with appearance. Age is one of the most investigated predictors where children between 12 and 15 years old had significantly lower HRQOL scores than children below 12 years old (Bos & Prahl, 2011). This study suggested that older children are possibly more aware of their cleft lip and have more treatment experience. In contrast,

Damiano et al. (2007) showed higher HRQOL scores of younger children aged 2-7 than older children aged 8-12. A study regarding gender showed that girls had higher HRQOL scores in comparison with boys (Kramer et al., 2009). Boys seemed as well to have more difficulties with adjustment (Berger & Dalton, 2011). Also the type of cleft seemed to be a predictive factor where children with only a cleft palate had significantly higher HRQOL scores than children with cleft lip and palate (Damiano et al., 2007) and children with only a cleft lip scored higher on HRQOL compared to both children with only a cleft palate and children with cleft lip and palate (Sagheri et al., 2009). Another determinant of the HRQOL is the speech of the child where children with less severe speech problems had higher total HRQOL scores (Damiano et al., 2007). Furthermore, Richman et al. (2012) stated that satisfaction with appearance may be a better predictor for poor outcomes than objective variables such as age, gender or cleft type. Higher levels of satisfaction with appearance was associated with a higher HRQOL score of children aged 2-12 years (Damiano et al., 2007). It is important to identify determinants of HRQOL to provide targeted interventions for children who are most at risk with the aim to increase the HRQOL of children with CLP (Barlow & Ellard, 2006; Klassen et al., 2012; Kramer et al., 2007).

Physical attractiveness is of high value in our society (Lockhart, 2003). While Slifer et al. (2003) did not find differences between children with CLP and healthy children in their satisfaction with appearance, Hunt et al. (2006) showed that participants between 8 and 21 years old with CLP were less happy with their appearance in comparison with participants without CLP and were most unhappy with their nose, lips and teeth. However, another study found that children with CLP were significantly more satisfied about some parts of their face such as teeth, eyes, ears, hair and chin in comparison with their healthy peers, but were only more dissatisfied about their lips (Berger & Dalton, 2009). Age (Feragen & Borge, 2010), having scars (Hunt et al., 2006) and the visibility of the cleft (Berger & Dalton, 2009) are all associated with the satisfaction of appearance. Children with a visible cleft are almost 2 times

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more likely to be teased than children with a non-visible cleft (Feragen & Borge, 2010). Having been teased in general is found as a determinant of how happy someone is with their appearance where being teased can lead to unhappiness with facial appearance (Hunt et al., 2006).

Having a child with CLP impacts the whole family, especially the parents (Baker, Owens, Stern, & Willmot, 2009). A mixture between grief about the impairment and delight about the new born could arise when parents are confronted with having a child with CLP (Nelson, Kirk, Caress, & Glenny, 2012) which could also be traumatic for these parents (Despars et al., 2011). As mentioned earlier, these children undergo a lot of surgical procedures and this could be challenging for parents as their role as protector of their child (Nelson et al., 2012). Hatzmann et al. (2008) found that parents of children with chronic conditions have a lower HRQOL in comparison with parents of healthy children. Parents of chronically ill children reported more difficulties with social and daily functioning, vitality, sleep, and having more negative feelings and less positive emotions compared to parents of healthy children. Furthermore, they spent less time doing leisure activities (Hatzmann, Peek, Heymans, Maurice-Stam, & Grootenhuis, 2013). However, parents of children of CLP were not included in that sample. Studies on distress in parents of children with CLP showed that some parents have elevated levels of clinical distress as a result of their child’s CLP

(Hasanzadeh et al., 2014; Kramer et al., 2007), but the general distress of parents of children with CLP was relatively low (Baker et al., 2009; Kramer, Baethge, Sinikovic, & Schliephake, 2007). Despars et al. (2011) found significantly more posttraumatic stress symptoms in mothers of children with CLP than mothers of children without CLP. In addition, lower levels of perceived social support predicted more parental distress (Baker et al., 2009). Clinical-demographic variables of the child didn't seem to be a predictor for parental distress (Baker et al., 2009).

All eligible research on parental distress did not make a distinction between parents of biological children with CLP or parents of adopted children with CLP. Nowadays, adopting children with special needs as CLP is increasingly more common (Hansson, Ostman, & Becker, 2013; Hansson, Svensson, & Becker, 2012). The existing literature about adopting children with special needs didn’t examine the HRQOL of these children and the impact on parents was not measured. Previous studies stated that adopted children lagged behind in communication compared to non-adopted children (Dalen & Theie, 2014) and adopted children with special needs showed more problems on communication than adopted children without special needs (Welsh & Viana, 2012).

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Across the aforementioned studies, there is no consensus about the HRQOL of

children with CLP. The use of different questionnaires, different age groups along with either parent report or self-report among these studies leads to a lack of uniformity. It is therefore difficult to compare the various studies and research is needed (Bos & Prahl, 2011; Barlow & Ellard, 2006). Also, no previous studies have assessed the HRQOL of Dutch children with CLP. Besides the HRQOL of children with CLP, literature about distress in parents of

children with CLP is scarce (Barlow & Ellard, 2006). Therefore, the main research question in this study is: what is the impact of having CLP on the HRQOL of Dutch children and how will parents be affected by having a child with CLP? To answer this main question, the following questions will be examined; (1) what is the HRQOL of children with CLP compared to a healthy norm population and to children with other chronic conditions? (2) what is the satisfaction with appearance of children with CLP? (3) what is the parental distress in parents of children with CLP? (4) what are the determinants of the HRQOL of children with CLP? and (5) what are the determinants of the distress of their parents? It is

hypothesized that children with CLP have a lower HRQOL than healthy children, but no difference will be found compared to chronically ill children. Satisfaction with appearance, especially with the lip, nose and mouth among children with CLP will be lower than healthy children and satisfaction with appearance will be a predictor for the HRQOL of these

children. A generally low parental stress level is hypothesized, but a minority of the parents will show elevated levels of distress. Furthermore, perceived level of social support is associated with the distress of parents.

Method: Procedure:

The data for this study was collected online via the KLIK website (www.hetklikt.nu). KLIK (in Dutch: Kwaliteit Leven In Kaart, in English: quality of life in clinical practice) is a web-based application developed by the research team of the Emma Children’s hospital AMC for the use of patient reported outcomes and parent reported outcomes in daily clinical practice (Haverman, Engelen, van Rossum, Heymans, & Grootenhuis, 2011). It is mainly designed to improve the communication about psychosocial problems between doctor and patient

(Haverman et al., 2012). Since December 2012, all children with CLP who are in care at the multidisciplinary team of the Emma’s children hospital were invited to use KLIK. This multidisciplinary team consists of surgeons, a pediatrician, a clinical geneticist, a dentist, a speech therapist and a pediatric worker. All patients have received a letter in which KLIK is

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introduced as a new part of the existing care. The letter included psychoeducation about the use of questionnaires during a doctor’s consultation and an explanation of the procedure of registration was described. Parents and the children were asked to register at the KLIK website. They actively had to confirm that they read the general terms also including how their privacy will be managed by using KLIK. Immediately after registration, patients received an email containing an unchangeable password that enables them to login the website to get access to the questionnaires. Prior to the doctor’s consultation, an automatic email was sent to remind parents and patients to fill in the questionnaires on the KLIK website. The completed questionnaires were discussed systematically during the consultation so that psychosocial problems can be identified and if necessary, a psychosocial referral could be made. Parents and children were asked to complete the questionnaires approximately every 3 months depending on the date of their consultation. KLIK is designed for clinical practice, but the data can also be used for scientific research. Patients and/or their parents had to give actively permission during registration for using their answers for research. The current study was approved by the medical ethics committee of the AMC.

Participants:

Eligible participants included all registered patients (0-18 years) with CLP and their parents at the KLIK website. No distinction was made between the different types of CLP. For this study, all patients registered between December 2012 and February 2014 were included. Only the data of the patients (or their parents when patients were younger than 12 years) who gave permission for using their data for research were used for analyzing.

Measures:

Socio-demographics:

Information on socio-demographic characteristics (child’s age, sex, type of education,

doubled classes and sports activities) were obtained through a questionnaire. Also the parents were asked about their age, country of birth, sex, marital status, highest level of education and current work situation.

HRQOL:

In general, there is little agreement about which questionnaires should be used to measure the HRQOL, but generic patient reported outcomes (PRO) instruments are advantageous because of the possibility to compare different chronic disease groups and to compare patients with

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healthy children (Klassen et al., 2012). Therefore, generic HRQOL questionnaires were used to assess the HRQOL and either proxy report or self-report was obtained depending on the child’s age. Children below 8 years old are not cognitively able to report their own HRQOL (Fekkes et al., 2000) and thus parents were asked to report their perception of the child’s HRQOL. Children between 8 and 18 years old completed the questionnaires themselves. The applied instruments were in Dutch language, but some were also available in English.

TAPQOL:

The TNO-AZL Preschool Children Quality of Life (TAPQOL) is a 43-item generic, multi-dimensional questionnaire which is developed to assess the HRQOL of very young children from 0 to 5 years of age (Fekkes et al., 2000). Only proxy-report was used. The TAPQOL contains 20 items of physical functioning, 9 items of emotional functioning, 10 items of social functioning and 4 items of communication. For completing the questionnaire, parents were asked how often a requested problem has occurred in the past three months on a 3-point response scale. For example, parents were asked if their child cried at night. Parents had the possibility to choose between never, occasionally and often. When parents reported a problem as occasionally or often, they had to assess next how well the child reacts in relation to this problem (fine, not so good, quite bad, bad). A higher score (0-100) represents a better HRQOL of the child. Also a higher score on the scales ‘problem behavior’ and ‘anxiety’ indicates less problem behavior and less anxiety. Fekkes et al. (2000) has shown a good validity and reliability (Cronbach’s α ranged between .66 and .89) for this measurement and the reliability in the present study ranged from .47-.97. At the KLIK website, this

questionnaire was also available in English.

PedsQL:

The Pediatric Quality of Life Inventory 4.0 (PedsQL) is a 23-item generic questionnaire to assess the HRQOL of children and adolescents (6-18 years) (Varni, Burwinkle, Seid, & Skarr, 2003). Parents as well as children can complete this questionnaire. In the current study, the parent proxy report (6 or 7 years old) and self-reports (8-18 years old) of the PedsQL was used. This questionnaire consists of 8 items of physical functioning, 5 items of emotional functioning and social functioning and 5 items of school functioning. Children and parents were asked to register the experienced problems of the different domains during the last week on a 5-point response scale. For example, parents were asked if their child had problems with running (proxy-report) and the children were asked if it was hard for them to run (self-report).

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They could choose to rate a problem as (0) never a problem, (1) almost never a problem, (2) sometimes a problem, (3) often a problem or (4) almost always a problem. A higher score (0-100) on the PedsQL indicates a better HRQOL. Children who scored one standard deviation or more below the mean score of the norm population were considered as an at-risk group for a low HRQOL (Varni et al., 2003). Calculating the proportion of children with a low HRQOL is a more clinical way of looking at the data (Haverman et al., 2012). It has been shown that the PedsQL is a measurement with a good feasibility, reliability (α= 0.89 child; α= 0.92 parent) and validity (Varni et al., 2003). It is also an adequate assessment to measure the HRQOL for pediatric research in the Netherlands (Engelen et al., 2009) and an useful instrument for examining the HRQOL of children with CLP (Damiano et al., 2007). In the present study-sample, Cronbach’s alpha ranged from .78-.97. At the KLIK website, this questionnaire was also available in English.

Satisfaction with Appearance:

SWA:

The Satisfaction With Appearance (SWA) questionnaire is developed for adolescents (8-18 years) with facial abnormalities to measure their perceived satisfaction with their appearance (Cleft Psychology special interest group, as cited in Berger & Dalton, 2009). Parent proxy report and self-reports are available. Only the self-report (8-18 years) was used in the current study. This questionnaire contains 20 general and specific questions about facial appearance and adolescents can answer on a 10-point Likert scale from 0 (very unhappy) to 10 (very happy). For example, children were asked how happy they were with their lips. A higher score (0-10) indicates a higher facial satisfaction. The reliability of the current study sample was good (Cronbach’s alpha = .93). No normdata of the SWA is published. For the current sample, unpublished normdata including mean scores of the different SWA items of both girls and boys aged between 9 and 16 years old (n=761) from the UK was used as a comparison. This normdata only included the mean scores of 13 different items of the SWA for every age in the range of both girls and boys.

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Parental distress:

DT-P:

Distress Thermometer for Parents (DT-P) is a short screening measurement to identify distress of parents of children aged 0-18 years old with a chronic health condition developed by the research team of the Emma's children hospital (Haverman et al., 2013). This

measurement consists of a distress thermometer and a problem list with additional questions. Parents could rate their overall distress on a thermometer from 0 (no distress) to 10 (extreme distress) with a score ≥ 4 indicating clinical levels of distress. Besides the thermometer, a problem list is included in the DT-P to obtain information about the sources of distress and can identify what support is needed for an individual parent. This list includes 7 items of practical functioning, 4 items of family/social functioning, 9 items of emotional functioning, 7 items of physical functioning, 2 items of cognitive functioning and 5 or 7 items of parenting functioning (the items depend on whether a child is younger than 2 years old or older,

respectively) and some additional questions. For example, parents were asked if they received enough social support and if they wanted to talk to a professional. Parents could answer these items with either 'yes' or 'no'. It has shown good reliability (α= ≥ 0.90) and validity

(Haverman et al., 2013). In the current study-sample, Cronbach’s alphas ranged from .54-.84.

Statistical analyses:

Statistical Package for Social Sciences (SPSS) version 21 was used to analyze the data in this study. First, all socio-demographic characteristics of both children and parents were assessed by descriptive statistics. Second, baseline differences between (1) participants and non-participants, (2) the study group and the Dutch normgroup, (3) the study group and children with other chronic illnesses were measured. Differences on gender were examined by Chi-square tests and differences on age were examined using independent sample T-tests. The HRQOL of children with CLP aged 0-18 years old was examined by the use of 2 different questionnaires which led to different statistical analyses. HRQOL scores of both

questionnaires were computed according to the manual. Based on plots on face value and a Kolmogorov-Smirnov test, the distribution of the HRQOL of children (0-5 years old) was not considered normally distributed and therefore a Mann-Whitney U test was used to analyze HRQOL scores compared to the norm group. To measure the extent of these differences, effect size r was calculated by dividing the Z-score of the different TAPQOL scales by the squared root of the sample. An effect size of .10 was considered as small, .30 as medium and

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.50 as large (Cohen, 1988). Normal distribution was considered for the PedsQL scores and independent sample T-tests were used to compare children with CLP with the Dutch norm group, as well as with the group with children with other chronic illnesses for every different age group. To measure the strength of the PedsQL results, Cohen’s D effect sizes were

calculated by dividing the difference in mean scores between children with CLP and the norm population by the pooled SD by both groups. Effect sizes from .20 were considered as small, .50 as medium and .80 and higher as large (Cohen, 1988). To adjust for multiple testing, the significance level was calculated by using the Bonferroni correction. A significance level of .004 was applied to the HRQOL of children aged 0-5 years old; that is .05 divided by the number of 13 subscale levels of the TAPQOL. For the HRQOL of children aged 6-18 years applied a significance level of .008. This level was calculated per age group and was formed by .05 divided by the number of 6 subscales of the PedsQL.

Further analyses were performed to examine the percentage of both children with CLP and healthy children with a low HRQOL. Children with a low HRQOL had a score ≥ 1 SD below the mean of the norm group (PedsQL) which was calculated by scores. These Z-scores were reached by subtracting the mean of the normgroup from the mean of the child’s HRQOL domain score according to their age group divided by the standard deviation of the norm group. Children with a Z-score lower than a value of – 1.00 were considered as having low HRQOL. Subsequently, Chi square tests were used to compare the percentage of children with CLP with low HRQOL with the percentage of children in the norm group with low HRQOL.

Descriptive analyses were used for the satisfaction with appearance among the study sample. The unpublished normdata was used to compare the satisfaction of appearance of children with CLP and healthy children. The unpublished data only included the mean scores of the different SWA items for every age (9-16) of both girls and boys. Therefore, the mean age of both girls and boys with CLP was calculated and the corresponding SWA score of the norm group was used to perform One sample T-tests. Bonferroni correction was used to adjust for multiple testing. A significance level of .004 was formed by dividing the number of 13 SWA items by .05.

The distress of parents of children with CLP was measured by descriptive statistics. A thermometer level equally or above 4 indicated clinical levels of distress. Also the problem list and additional questions were analyzed by descriptive statistics.

Linear regression analyses were performed to predict HRQOL of children with CLP (PedsQL). The selected predictors age, gender, SWA and adoption were all included in an

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univariate analysis separately. Variables with P. <.20 were included in the final linear multivariate regression analyses. Checking for multicolinearity (high correlation between predictors, correlation > .70), no variables correlated with each other at .70 level which led to no exclusion of variables. All four variables had P < .20 on some HRQOL scores, but only two variables were selected for the final regression. Because SWA and adoption had more P-values lower than .20 on different HRQOL scores than age and gender, only SWA and adoption were included in the final linear multivariate regression analysis. No further analyses were performed for the TAPQOL scores because of the excessively skewed distribution of the scale scores (strong ceiling effect).

To assess predictors for the clinical distress of parents (a score ≥ 4), logistic regression analyses were conducted. The most reported problems (≥20%) of the problem list by parents were included in an univariate regression separately; leisure activities/relaxing, keeping emotions under control, depression, feeling tense or nervous, sleep, fatigue, memory and social support. Also socio-demographic variables as the age of the child and the parent, gender of the child and parent, marital status, current work situation, country of birth,

education level and adoption were included in an univariate analysis separately. Because only 7 variables could be included in the final logistic regression analysis, variables with P < 0.10 were selected for this analysis. Checking for multicolinearity (high correlation between predictors, correlation > .70), keeping emotions under control and depression correlated at .72 level and therefore one needed to be excluded. Because keeping emotions under control had a lower P-value (P = .000) in comparison with depression (P = 0.003), depression was excluded from the final regression analysis. Leisure acitivities/relaxing, keeping emotions under

control, social support, fatigue, memory, current work situation and feeling tense or nervous were included in the final logistic regression analysis.

Results:

Between December 2012 and February 2014, 276 children with CLP and their parents had received a letter including information about the use of KLIK. Figure 1 describes the participants of this study. A distinction was made between non-registered patients and

registered patients which was based on registration at the KLIK website. Of the 276 children, 113 children registered at the KLIK website (40.9%). These registered patients were further distinguished in non-participants and participants. Non-participants (N=38, 33.6%) were patients who didn’t give permission for using their data for research and/or patients who only

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completed the socio-demographic questionnaires. A total of 75 registered patients and their parents participated (66.4%). This study only used the baseline data of the participants.

Figure 1. Flow diagram of the participants

Socio-demographic:

The socio-demographics of children with CLP and their parents are shown in table 1. The mean age of the children was 4.98 years (SD= 4.29) and 39 of them was female (42.9%). The mean age of the parents was 39.13 years (SD= 7.52) and 84.7% of the questionnaires were filled in by mothers (N=60). Of all parents, 95.9% (N=71) were born in the Netherlands and 70 parents (94.6%) were married or living together. Both registered patient en non-participants were pooled (N=201) to make a comparison with the non-participants on age and gender. Participants (Mean= 5.40, SD= 4.40) were significantly younger than the

non-All approached patients N=276 Non-registered patients N=163 Registered patients N=113 Non-participants N=38 Participants N=75 TAPQOL N=43 PedsQL 6-7 N=8 PedsQL 8-12 N=13 PedsQL 13-18 N=5 SWA N=17 DT-P N=68 Children 0 - 1.5 years N= 19 Children 1.5 - 5 years N= 24

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participants (Mean= 9.58, SD= 4.20, P <.05) and the gender of both participants and non-participants was comparable.

Table 1. Characteristics of participants (N=75) and non-participants (N=201)

Child characteristics N Mean SD N Mean SD

Age (years) 75 5.38 4.43 201 9.23 4.34*

N % N %

Gender (female) 31 41.3 89 44.3

Different age groups

0-5 years 43 57.3 44 21.9

6-7 years 8 10.7 26 12.9

8-12 years 18 24.0 91 45.3

13-18 years 6 8.0 40 19.9

Adoption (yes) 12 16.0

Parental characteristics N Mean SD

Age (years)¹ 72 39.13 7.52

N %

Gender (mother)³ 61 84.7

Country of birth (Netherlands)² 71 95.9

Marital status² Married/living together 70 94.6 Divorced 1 1.4 Single 3 4.1 Education² Low 5 6.8 Middle 28 37.8 High 41 55.4

Paid work (yes)² 53 71.6

2) ² 1 missing; 1 parent didn't complete the socio-demographic questionnaire

4) * P < .05, p-value was measured by independent sample t-test

3) ³ 3 missing; 1 parent didn't complete the socio-demographic questionnaire, 1 foster mother and 1 filled in 'both father and mother'

Participants Non-participants

1) ¹ 3 missing; 1 parent didn't complete the socio-demographic questionnaire, 1 parent filled in the child's birth date, 1 parent filled in the date of registration

HRQOL:

TAPQOL:

The results are shown in table 2. In comparison with the norm group (Mean= 2.55, SD= 0.92), children with CLP (Mean= 2.01, SD= 1.37, P< .05) were significantly younger. Children with CLP between 0 and 5 years old scored significantly higher on appetite (P< .004) and problem behavior (P< .004), which indicates that children with CLP showed less problem behavior

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than the norm group. Children with CLP scored significantly lower on communication (P<.004) and stomach (P< .004) than the norm group. Specifically, children with CLP had more difficulties with understanding what other people said, talking clearly, saying and making it clear what he/she meant and had more abdominal pain and were more nauseas than healthy children. Effect sizes were considered as small to medium, ranging from -.19 to -. 24. No differences were found between girls and boys.

A comparison was also made within the group of children with CLP, between adopted (N=4) and biological children (N=39). Except for one scale, no differences were found. Adopted children (Mean= 52.08, SD= 5.51) scored significantly lower than biological children (Mean= 82.74, SD= 2.89, P< .05) on communication (data not shown).

Table 2. The HRQOL of children with CLP (0-5 years old) compared to the norm population

Effect size

Subscale N Median Range N Median Range P r

Sleeping 339 87.50 6.25 - 100.00 43 81.25 31.25 - 100.00 .088 -.09 Appetite 339 83.33 25.00 - 100.00 43 100.00 16.67 - 100.00 .000* -.20 Lungs 339 100.00 0.00 - 100.00 43 100.00 8.33 - 100.00 .065 -.09 Stomach 338 100.00 33.33 - 100.00 43 83.33 0-100.00 .000* -.19 Skin 339 91.67 41.67 - 100.00 43 100.00 50.00 - 100.00 .996 -.00 Motor functioning 289 100.00 75.00 - 100.00 24 100.00 50.00 - 100.00 .287 -.06 Social functioning 292 100.00 16.67 - 100.00 24 100.00 50.00 - 100.00 0.81 -.01 Problem behavior 340 71.43 7.14 - 100.00 43 85.71 7.14 - 100.00 .000* -.24 Communication 285 93.75 50.00 - 100.00 24 81.25 43.75 - 100.00 .000* -.24 Anxiety 340 83.33 33.33 - 100.00 43 83.33 16.67 - 100.00 .104 -.08 Positive mood 340 100.00 50.00 - 100.00 43 100.00 50.00 - 100.00 .172 -.07 Liveliness 339 100.00 50.00 - 100.00 43 100.00 50.00 - 100.00 .279 -.06 1) Higher scores indicate better HRQOL (0-100)

2) * P ≤ .004 (with Bonferroni correction), p-values were measured by Mann-Whitney U tests

Norm population Children with CLP

PedsQL:

The results are presented in table 3. The HRQOL scores of children with CLP aged 6-7 and 8-12 years old were not different from either healthy children and chronically ill children. Only children with CLP aged 13-18 years old had a significant lower school functioning (P < .008) than healthy children and the effect size was large. No difference was found in comparison with chronically ill children. Boys didn’t differ from girls on the different HRQOL scales.

When adopted children with CLP (N= 8) were compared with biological children with CLP (N= 18), a significant result was found on social functioning. Adopted children (mean=

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98.75, SD= 2.50) scored significantly higher on this scale (mean= 76.74, SD= 19.34, P< .05). No differences were found on the other scales (data not shown).

Figure 2 shows the prevalence of both children with CLP and healthy children with low HRQOL scores and the differences between both groups. There were no differences found between children with CLP and healthy children aged 6 and 7 years old. Children with CLP between 8 and 12 years old were more at risk for problems with psychosocial health (P< .008), emotional functioning (P< .008) and social functioning (P< .008) compared to healthy children. A significant higher risk for problems with social functioning (P< .008) and school functioning (P <.008) was found among children with CLP aged 13-18 years than for healthy children.

Table 3. HRQOL of children with CLP (6-18 years old) compared to the norm population and chronically ill children

Effe ct siz e Effe ct siz e

Subscale: N Mean SD N Mean SD P d N Mean SD P d

6-7 ye ar old T otal score 8 88.32 4.92 61 86.07 8.29 .458 -.27 11 80.34 9.39 .043 -.85 Physical health 8 94.53 4.94 61 88.83 9.43 .099 -.60 11 82.39 11.38 .012 -1.07 Psychosocial health 8 85.00 6.67 61 84.59 9.23 .904 -.04 11 79.24 10.99 .207 -.52 Emotional functioning 8 77.50 7.07 61 78.44 12.76 .839 .07 11 74.09 12.61 .465 -.27 Social functioning 8 93.75 7.91 61 89.02 11.21 .253 -.42 11 84.09 10.68 .046 -.90 School functioning 8 83.75 15.53 61 86.31 10.80 .552 .24 11 79.55 16.65 .584 -.25 8-12 ye ars old T otal score 13 74.58 20.69 192 82.31 8.83 .205 .88 26 80.64 9.32 .331 .65 Physical health 13 84.38 26.15 192 85.25 8.85 .906 .10 26 82.21 12.14 .724 -.18 Psychosocial health 13 69.36 20.58 192 80.75 10.34 .070 1.10 26 79.81 10.43 .105 1.00 Emotional functioning 13 66.15 28.22 192 76.85 13.76 .199 .78 26 78.85 13.21 .145 .96 Social functioning 13 71.54 21.05 192 86.51 12.24 .025 1.22 26 83.27 12.80 .083 .92 School functioning 13 70.38 20.15 192 78.88 11.90 .158 .71 26 77.31 13.13 .204 .53 13-18 ye ars old T otal score 5 75.65 20.20 148 83.14 8.99 .455 .83 25 77.09 9.40 .802 .15 Physical health 5 88.75 15.40 148 86.76 9.21 .643 -.22 25 81.00 12.00 .218 -.65 Psychosocial health 5 68.67 23.64 148 81.21 10.22 .302 1.23 25 75.00 9.56 .585 .66 Emotional functioning 5 67.00 29.28 148 77.53 15.01 .467 .70 25 71.40 16.62 .759 .26 Social functioning 5 79.00 20.43 148 90.14 11.37 .291 .98 25 83.40 12.97 .534 .34 School functioning 5 60.00 22.36 148 75.95 12.68 .008* 1.26 25 70.20 15.17 .214 .67

1) higher scores indicate higher HRQOL (scores 0-100)

2)** P < .008 (with bonferroni correction), p-values at independent sample t-tests: CLP versus norm, CLP versus chronically ill children

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Figure 2. Proportion (%) of both children with CLP and the norm population with low HRQOL scores (6-18 years old) and the differences between these groups

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Satisfaction with Appearance:

The mean scores of the different satisfaction with appearance items are shown in table 4.The mean score of the total satisfaction with appearance was 7.53 (SD=1.80) where a score of 4.19 was the lowest and 9.81 the highest. Boys with CLP were most dissatisfied about their teeth (6.40), mouth (6.60) and nose (7.20) whereas girls with CLP reported nose (5.29), mouth (5.43) and speech (5.86) as most dissatisfied items. There were no differences found between girls and boys with CLP. Only a trend was shown at the overall appearance where girls (Mean=6.14, SD= 2.41) showed less satisfaction with their overall appearance in comparison with boys (Mean= 8.80, SD= 1.69, P = .03). Satisfaction with appearance scores were equal between adopted children with CLP and biological children with CLP.

In comparison with healthy boys, boys with CLP were significantly more satisfied with their chin (P< .004). The effect size was considered as large. Girls with CLP were more satisfied about their eyes (P< .004) than healthy girls with a medium effect size.

Table 4. Satisfaction with appearance between children with CLP (N=17, 8-18 years old) and English students ( N=761, 9-16 years old)

P d P d

Items Mean SD Mean SD Mean SD Mean SD

Face 8.10 2.18 6.52 3.21 .048 -.49 6.14 3.34 6.29 2.74 .911 .05 Overall appearance 8.80 1.69 7.10 2.61 .011 -.65 6.14 2.41 6.24 2.39 .919 .04 Profile 8.40 1.9 6.46 3.08 .010 -.63 7.57 2.23 5.85 2.71 .087 -.63 Good looking 7.80 2.04 6.28 1.45 .043 -1.05 6.57 3.69 3.60 2.43 .077 -1.22 Nose 7.20 3.05 7.00 3.08 .840 -.06 5.29 3.95 6.39 2.65 .487 .42 Mouth 6.60 3.47 7.24 2.55 .574 .25 5.43 3.69 7.34 2.35 .220 .81 Chin 9.00 1.41 7.02 2.72 .002* -.73 8.86 1.68 6.41 2.41 .008 -1.02 Teeth 6.40 2.99 6.26 3.22 .886 -.04 6.57 3.26 5.61 3.38 .465 -.28 Hair 8.40 1.40 8.30 2.56 .072 -.04 8.71 1.98 7.43 2.79 .136 -.46 Eyes 9.40 1.35 8.45 2.37 .053 -.40 9.57 0.79 8.15 2.23 .003* -.64 Ears 8.70 1.77 7.08 2.85 .018 -.57 8.29 1.98 7.38 2.45 .271 -.37 Speech 7.60 2.88 7.39 2.69 .823 -.08 5.86 3.13 7.32 2.41 .263 .61 Hearing 8.60 1.9 8.10 2.59 .426 -.19 7.00 3.27 7.71 2.4 .586 .30

1) Range from 0-10; higher scores indicate higher satisfaction with appearance

2)*P ≤ .004 (with bonferroni correction), p-values at one-sample t-tests: CLP versus the scores of the norm group items

Children with CLP Norm population

Boys Girls

Children with CLP Norm population

Parental distress:

The results are presented in table 5. Clinical levels of distress were measured in 24 parents of children with CLP (35.3%). Parents reported most problems (higher than 20%) with fatigue (46%), sleep (32%) and feeling tense or nervous (29%). Furthermore, 97% of all parents (N=66) were satisfied with the amount of social support and 17 parents (25%) wanted or maybe wanted to speak to a professional.

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Between parents of adopted children and parents of biological children with CLP were not found any differences on the levels of distress (data not shown). There were also no

differences found on distress level between fathers and mothers of the study group (data not shown).

Table 5. Problem list of the distress thermometer for parents (N=68)

N: % N: % Practical problems: Housing 64 94.1 4 5.9 Work/study 61 89.7 7 10.3 Finances/insurance 62 91.2 6 8.8 Housekeeping 56 82.4 12 17.6 Transport 64 94.1 4 5.9

Child care/child supervision 59 86.8 9 13.2

Leisure activities/relaxing 54 79.4 14 20.6

Family/social problems:

Dealing with (ex) partner 63 92.6 5 7.4

Dealing with family 62 91.2 6 8.8

Dealing with friends 66 97.1 2 2.9

Interacting with your child(ren) 61 89.7 7 10.3

Emotional problems:

Keeping emotions under control 52 76.5 16 23.5

Self-confidence 59 86.8 9 13.2

Fears 59 86.8 9 13.2

Depression 51 75.0 17 25.0

Feeling tense or nervous 48 70.6 20 29.4

Loneliness 65 95.6 3 4.4 Feelings of guilt 59 86.8 9 13.2 Use of substances 65 95.6 3 4.4 Intrusive thoughts 58 85.3 10 14.7 Physical problems: Eating 65 95.6 3 4.4 weight 60 88.2 8 11.8 Sleep 46 67.6 22 32.4 Fatigue 37 54.4 31 45.6 Out of shape 56 82.4 12 17.6 Pain 58 85.3 10 14.7 Sexuality 64 94.1 4 5.9 Cognitive problems: Concentration 55 80.9 13 19.1 Memory 51 75.0 17 25.0 Additional questions

Enough social support 2 2.9 66 97.1

Lack of understanding 62 91.2 6 8.8

Having a chronic disease 61 89.7 7 10.3

Getting along medical staff 3 4.4 65 95.6

Talk to a professional 51 75.0 17 25.0

1) Percentages in bold were the most reported problems by parents (> 20%)

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Predictors of HRQOL in children with CLP:

The results of the univariate analysis and multivariate regression analysis are shown in table 6 and 7. The variance of the different scales ranged from 14-46%. Adoption was significantly associated with psychosocial health and emotional functioning where adopted children with CLP had fewer problems with psychosocial health and emotional functioning than biological children with CLP. No significant association was found between adoption and social

functioning. Further, satisfaction with appearance was significantly related to different

HRQOL scales. Children with CLP with higher scores of their satisfaction of appearance had higher total HRQOL scores. Also children with a higher satisfaction of appearance scored higher on psychosocial health and emotional functioning. Satisfaction of appearance was not associated with social and school functioning.

Table 6. Univariate regression analysis of PedsQL scores of children with CLP

Age -.18 -.06 -.23 -.10 -.19 -.33^

Gender .22 .14 .24 .37^ .15 .08

Adoption .30^ .17 .34^ .39^ .41^ .08

Satisfaction with Appearance .48^ .31 .50^ .53^ .38^ .43

1) correlation coefficients were shown between the variables and the different domains of the HRQOL 2) ^ variables with P < .20 were selected for the multivariate regression analysis

Total score Physical health Psychosocial health Emotional functioning Social functioning School functioning

Table 7. Adoption and Satisfaction with appearance predicting HRQOL (PedsQL scores) in children with CLP

B SE β B SE β B SE β B SE β B SE β B SE β

Adoption 20.16 10.22 .41 8.05 9.45 .21 26.62 11.75 .45* 36.62 16.76 .43* 25.52 12.19 .45 17.73 11.78 .34

Satisfaction with appearance 4.49 1.89 .50* 2.23 1.75 .32 5.70 2.17 .52* 8.62 3.10 .55* 4.16 2.25 .40 4.32 2.18 .44

R² .40 .14 .45 .46 .35 .30

F 4.57 1.13 5.75 6.00 3.73 2.97

1) *P< .05

Social functioning School functioning Total score Physical health Psychosocial health Emotional

functioning

Predictors of clinical distress in parents:

The results of the univariate analysis are presented in table 8, and the logistic regression analysis in table 9. Leisure activities/relaxing, keeping emotions under control, enough social support, fatigue, memory, feeling tense or nervous and current work situation were all not significantly related to clinical levels of distress in parents of children with CLP.

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Table 8. Univariate regression analysis of clinical distress levels in parents of children with CLP

Leisure activities/relaxing .46^

Keeping emotions under control .46^

Depression .36^

Enough social support -.24^

Sleep .08

Fatigue .44^

Memory .50^

Feeling tense or nervous .54^

Adoption -.17 Age child -.06 Gender child .18 Age parent -.10 Gender parent -.02 Education -.10 Country of birth -.01 Marital status .05

Current work situation -.21^

2) ^ variables with P < .10 were selected for the logistic regression analysis

Clinical distress*

1) * distress scores of 4-10, correlation coefficients were shown between clinical distress and the different variables

Table 9. Logistic regression analysis of predictors for clinical levels of distress in parents of children with CLP

B SE Wald

Leisure activities/relaxing 1.11 1.00 1.23

Keeping emotions under control 1.17 .87 1.80

Enough social support -.22 .99 0.00

Fatigue .83 .75 1.22

Memory 1.26 .93 1.86

Feeling tense or nervous .44 .99 0.20

Current work situation -.49 .77 0.40

1) * P < .05

Discussion:

The current study examined the HRQOL of children with CLP aged 0-18 years old, the prevalence of children with a low HRQOL, the satisfaction of appearance of these children, the parental distress and the determinants of both the HRQOL of the children and parental distress.

The hypothesis about the HRQOL of children with a cleft lip can be partially supported. HRQOL scores of children with CLP (6-18 years old) were equal compared to

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chronically ill children. Children with CLP between 0 and 5 years old had less appetite problems, less problem behavior, but these children had more difficulties with understanding what other people said, talking clearly, saying and making it clear what he/she meant and had more abdominal pain and were more nauseas than healthy children. Compared to the norm population, it seemed that children with CLP between 6 and 18 years old as a group do not differ in their HRQOL, except for school functioning in the 13-18 years old group. Children with CLP in this age group scored significantly lower on school functioning in comparison with healthy children. However, the determinants were not measured and it is not clear why these children showed lower school functioning. For example, Haverman et al. (2012) showed that children with juvenile idiopathic arthritis with more school absence had a lower school functioning in comparison with children with less school absence. Also, children with hearing difficulties seemed to have more problems with following teaching in class which directly led to poorer academic functioning (Teasdale & Sorensen, 2007) and poorer school achievement was found in children with CLP in comparison with their healthy classmates (Wehby et al., 2014). School absence and having speech/hear difficulties could be a possible explanation for the lower scores on school functioning and should be further investigated in future research.

Although it seemed that children with CLP as a group function as well as healthy children, differences in prevalence of low HRQOL between the study group and healthy children showed that children with CLP were more at risk for problems on several HRQOL domains. Children with CLP (8-12 years old) were more likely to have problems with psychosocial health, emotional and social functioning than healthy children. Besides school functioning, adolescents with CLP between 13 and 18 years old were just as 8-12 years old children more at risk for problems with social functioning. These results indicate that social functioning is an important issue when patients with CLP reach adolescence. Though, it remains unclear in this study what factors may place these children more at risk for problems with social functioning. It is known that children with CLP have been more teased in

comparison with healthy children (Berger & Dalton, 2009; Hunt et al., 2006). Social functioning and teasing seemed correlated in a way that children who were teased had less social confidence, had less comfort in trusting and depending on others and worried more about being abandoned in relationships in comparison with children who were not teased (Ledley et al., 2006). Having been teased might be a possible explanation for the experienced problems with social functioning and should be included as a possible predictor of lower social functioning in future research.

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In measuring the HRQOL of children with CLP, a distinction was also made between adopted children (N=12) and biological children. Whereas adopted children (N=4) between 0 and 5 years old scored lower on communication than biological children, adopted children aged 6-18 years (N=8) old scored higher on social functioning in comparison with biological children. Furthermore, adoption was associated with HRQOL scores. Adopted children had less problems with psychosocial health and emotional functioning than biological children with CLP. However, adopted children with CLP were not separately compared with the norm population but these results suggest that adopted children with CLP experience less problems than biological children with CLP. This study was the first in examining the HRQOL of adopted children and based on the study results, it remains unclear why these adopted children with CLP scored better on psychosocial health and emotional functioning. Therefore, further research on the HRQOL of adopted children with CLP is needed.

Another aim of this study was to investigate the satisfaction with appearance of children with CLP ( 8-18 years old). Both girls and boys with CLP mentioned most

dissatisfaction about their nose and mouth but boys were also dissatisfied about their teeth and girls about their speech. Special attention should be addressed to girls as a trend was found towards more dissatisfaction in girls about their appearance than in boys. This difference between girls and boys in appearance was also found in previous literature (Feragen & Borge, 2010) and girls in general were more likely to be dissatisfied with their physical appearance than boys (Delfabbro et al., 2011). It seems that both girls and boys do not differ from healthy children in their satisfaction with appearance and even reported to be more satisfied with some parts of their face than healthy children, which was not in agreement with the

hypothesis. Boys with CLP were more satisfied with their chin and girls were more satisfied with their eyes than the comparison group. Similarly, Berger and Dalton (2009) showed that 11-16 years old children with CLP were more satisfied with some parts of their appearance in comparison with healthy children, but children with CLP were more dissatisfied about their lip. With the current cross-sectional data, it is difficult to determine why someone is satisfied or dissatisfied with their appearance. It is possible that adolescents may have had corrective surgeries and orthodontic procedures which could lead to more satisfaction compared to the situation before these reconstructions. Another explanation could be that the answer is social desirable where children might think they hurt the multidisciplinary team by telling them their dissatisfaction about their face. It is also possible that some adolescents have accepted their different appearance.

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One of the strengths of this study is the examination of determinants of HRQOL of children with CLP. Satisfaction of appearance was found to be an important predictor of HRQOL scores. Children with CLP who were more satisfied with their appearance had higher total HRQOL, better psychosocial health and better emotional functioning. The

multidisciplinary team should not only address the physical aspects of having a cleft clip during consultation, but it seems crucial to ask children about their satisfaction with appearance and possible improvements could be discussed. Because children with CLP seems in this study more satisfied with their appearance than healthy children, which is in contrast to the expectation, and while satisfaction with appearance seems to be an important factor, it is questionable if the used norm data is a good reflection of the Dutch norm

population. Cultural differences between English students and Dutch students could also cause bias. Therefore, future research should focus on obtaining normdata of the satisfaction of appearance in the Dutch norm population.

The hypothesis about the clinical distress in parents of children with CLP can be supported in which more than one third of all parents showed elevated levels of distress. Problems with fatigue, sleep and feeling tense or nervous were the most reported by parents. This is in accordance with an earlier study in which the author stated that parents of children with chronically ill children experienced some problems (Hatzmann et al., 2013). There were no predictors found for the clinical levels of distress in parents of children with CLP. This could be expectable because five out of seven possible predictors (except for perceived social support and current work situation) in the final analysis were gained from the problem list. Moreover, the separate problem domains of this list seemed to be significantly correlated with the overall distress of parents (Haverman et al., 2013). Even social support, which was

hypothesized, didn’t appear to be a predictive factor. Almost all parents in the study sample, except for two, reported to have no problems with perceived social support. A quarter of all parents (maybe) wanted to talk to a professional, which was not associated with clinical levels of distress in parents. Though, it is important for the multidisciplinary team to know which parents and why parents wanted to talk to a professional. The answers of the problem list can be used to evaluate the cause of the parents’ distress and what support is needed for every parent (Haverman et al., 2013).

A few limitations of this study must be mentioned. One of the main limitations is the small sample of participants, especially in the 8-18 years old age group. This age group showed most HRQOL problems and this suggests that having CLP affected children the most in adolescence. Also, the oldest child in this sample was sixteen, which didn’t allow to state

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anything about seventeen and eighteen year old adolescents. Comparing participants with non-participants, adolescents (8-18 years old) were less willing to participate in this study compared to younger children. This is not surprising as parents of younger children decide whether or not to take part in a study. Further research on the HRQOL of these children, mainly with a larger sample of adolescents, is needed.

While the current study showed distress and various problems among parents of children with CLP, a comparison with a norm population of parents was not made. Currently, norm data of the used questionnaire (DT-P) did not exist, but future research should focus on obtaining data about the distress and problems of parents in the Dutch norm population.

Another limitation of this study was the lack of making a distinction between the different cleft types in examining the HRQOL of these children. Studies have shown the impact of different cleft types on HRQOL. Damiano et al. (2007) found that children with only an isolated cleft palate in comparison with children with cleft lip and palate had significantly higher PedsQL scores. Sagheri et al. (2009) showed that children with only an isolated cleft palate and children with cleft lip and palate, in comparison with children with only a cleft lip, have a lower HRQOL. This can be due to more hear and speech difficulties in children with only an isolated cleft palate and cleft lip and palate. A distinction in cleft lips was not made in this study because every patient who were in care of the multidisciplinary team were invited to register at the website regardless their cleft type and the type of cleft was not asked during registration. Based on the mentioned literature, it is advisable to include the different cleft types in future studies.

Although the PedsQL measures different aspect of the HRQOL of these children, not all specific issues of having CLP as type of cleft, hear and speech difficulties are incorporated in this instrument. There is still not a PRO instrument designed especially for children with CLP, which addresses the unique issues of this group (Eckstein et al., 2011; Klassen et al., 2012). In addition, Gosain and Chim (2011) mentioned the importance of a well-validated outcome measure to evaluate the HRQOL of children with CLP at different ages. How the various aspects of HRQOL of children with CLP might change over time, from birth to adulthood, is still unknown. This study analyzed baseline cross-sectional data which do not permit drawing causal inferences. In the near future, HRQOL research in children with CLP should be extended so as to get a better understanding of the impact of having CLP during children’s development. A strength of the current study was the collection of data by using a website where children and their parents could fill in questionnaires. This is reliable, valid, less time consuming and more efficient in comparison with paper and pencil (Engelen et al.,

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2011). This website could also be used for longitudinal research in which children and their parents can complete several questionnaires during the child’s development.

In conclusion, this study included participants with a wide age range and generated more insight in the HRQOL of Dutch children with CLP and the impact of having a child with CLP on their parents. A significant amount of children with CLP, mainly during adolescence, had lower HRQOL scores than healthy children. Satisfaction with appearance seems to be an important factor and some parents show elevated levels of distress. These findings underline the relevance of systematically monitoring these children and their parents in daily clinical practice to identify problems in an early stage, get a better understanding of the impact of having CLP and provide them the care they need the most.

Acknowledgements

I would like to thank Professor Martha Grootenhuis for giving me the opportunity to write my Masterthesis at the psychosocial department of the Emma’s children hospital AMC and to be a part of the KLIK team. I’d also like to thank Ed de Bruin for being my supervisor at the University of Amsterdam and providing me with feedback. At last, a special thanks to my supervisor at the Emma’s children hospital, Dr. Lotte Haverman, from whom I have learned a lot about decent research. It has been a ‘process’, but thank you for all the time you have spent on me! I worked with great pleasure at this project and I am very glad you and Martha gave me the possibility to continue my work as a member of the KLIK team!

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