Abstract
Objective:
To describe the association between children’s orofacial cleft (OFC) and families’ quality of life (QoL), using the short-form Family Impact Scale questionnaire (FIS-SF). Also assessed were the psychometric properties of the FIS-SF, as well as whether certain demographic and clinical variables impacted the family.
Design:
Observational cross-sectional study.
Setting:
Tertiary care public children’s hospital in New South Wales, Australia.
Participants:
Parents/caregivers of children with OFC.
Main outcome measure(s):
The short-form Family Impact Scale questionnaire.
Results:
Two hundred and fourteen parents completed the FIS-SF. Excellent convergent validity was evident, but discriminant validity was weaker. Those children with velopharyngeal insufficiency/submucous cleft reported lower scores on the family/parental activity, and lower overall family impact scores than those with cleft lip (CL) or cleft palate (CP). Those with cleft lip and palate (CL/P) had poorer family/parental activity scores than those with CP. There were no systematic differences in FIS-SF scores by the child’s age, gender, private health insurance, living location, the number of cleft-related operations, or whether a child had commenced orthodontic treatment.
Conclusions:
The FIS-SF is a valid and reliable measure for determining the impact that OFC has on family QoL. The impact of OFC on children’s families differs according to cleft type.
Introduction
Orofacial clefts (OFC) are among one of the most common visible birth defects (Kadir et al., 2017). In Australia, the incidence of cleft lip and palate (CL/P) is 1.2 per 1000 births and for cleft palate (CP) is 1.4 per 1000 births (Abeywardana and Sullivan, 2008). These children experience compromised health, psychosocial well-being, and quality of life (QoL) (Wehby and Cassell, 2010), and many undergo prolonged and extensive treatment and rehabilitation from birth until adulthood. The prevailing aim of cleft rehabilitation is to enhance psychosocial well-being and QoL and to facilitate integration into the community (Klassen et al., 2012).
In recent years, there has been a move from the purely surgical treatment of patients with OFC to a more holistic, socio-environmental approach (Borrell-Carrió et al., 2004). In keeping with this, the last decade has increasingly seen traditional measures of health supplemented with QoL measures, and these are recognized as valid parameters for patient assessment in all aspects of health care, including oral health (Eckstein et al., 2011; Genderson et al., 2013; Queiroz et al., 2015). Oral-health-related quality of life (OHRQoL) is a report given by patients to assess how oral disease affects daily functioning and psychosocial well-being (Malden et al., 2008; Sischo and Broder, 2011; Ward et al., 2013). It is a multidimensional concept, increasingly recognized as an integral part of health and well-being (Sischo and Broder, 2011).
Contemporary concepts of child health encompass both the child and the family, due to the dependence of the child on the caregiver and the vital role played by the family in child health (Rothman et al., 1991; Krepner, 2000). Assessing the family’s perspective of the impact of a child’s condition is important, particularly because chronic childhood illness inevitably impacts the family and also because health care interventions usually address both parental/family needs and concerns as well as the child’s (Rothman et al., 1991). Accordingly, when assessing OHRQoL, it is imperative to consider not just the effect that OFC may have on the affected individual but also its impact on the parent, the family, and its day-to-day functioning.
Having a child with an orofacial condition has proven to affect the OHRQoL and daily functioning of the family. Locker and colleagues utilized the Family Impact Scale (FIS) to study the family impact in families with children who had pediatric dental, orthodontic, or oral-facial issues. Approximately three-quarters of caregivers conveyed a family impact “sometimes” or “often/everyday” over the previous 3 months. The highest impacts were on parent and family activities, parent emotions, and family conflict (Locker et al., 2002). Parents also reported increased stress and worry about their child and the need to take time off work for appointments.
The development and evaluation of the 14-item FIS for oral health research were carried out by Locker and colleagues in 2002 (Locker et al., 2002). The FIS was developed to determine the impact of a child’s oral-facial condition on family life and well-being, specifically the influence on parent and family activities, parental emotions, family conflict, and family finances (Antunes et al., 2014). Its psychometric properties have been assessed in a number of groups, settings, and conditions, including craniofacial conditions, and it has been found to have acceptable validity and reliability (Malden et al., 2008). Although the FIS is rapidly being considered an essential component of an OHRQoL assessment for children, a limitation is its length. Shorter forms have been found to reduce respondent burden and to allow for easier, more efficient application in clinical practice and research (Broder et al., 2012). The short-form Family Impact Scale questionnaire (FIS-SF) was developed in a New Zealand cohort of children, where it was shortened from 14 to 8 items and found to have good psychometric properties including acceptable reliability, validity, and responsiveness (Thomson et al., 2013).
Additional research using the FIS-SF tool is required in more clinical settings and in various populations. The aim of this study was to describe the impact (and associations) of OFC on the families of an Australian clinical pediatric sample. Also assessed were the psychometric properties of the FIS-SF, as well as the association of family impact with demographic and clinical characteristics such as age, gender, living location, cleft type, private health insurance, number of past cleft-related surgical operations, and commencement of orthodontic treatment.
Methods
Design
This was an observational cross-sectional study carried out with a consecutive clinical case series of children presenting for assessment at the Cleft Clinic, held at a major pediatric tertiary referral hospital in Sydney, Australia. The study was conducted from August 2015 to July 2016 and was approved by the Sydney Children’s Hospital Network Human Research Ethics Committee.
Procedure
All parents/caregivers presenting to their child’s appointment at the Cleft Clinic were invited to participate in the study and were asked to complete an FIS-SF questionnaire on the same day. Consent was obtained from both the parent and the child before proceeding.
Study sample
Included in the study were parents who were able to speak and read English, with a child aged 7 to 18 years affected by OFC. Excluded from the sample were parents who had a child with a known syndrome. The proposed minimum sample size of 80 to 100 was chosen because it was the number of patients it was thought feasible to recruit during a 12-month period.
Measures
Demographic and clinical information was collected and confirmed via an online hospital database. Demographic data collected included the child’s sex, age, postcode, and whether the family had private health insurance. Clinical data included the cleft classification, number of cleft-related operations, and whether orthodontic treatment had commenced. Postcodes were used to group patients into “Sydney metropolitan” or “rural” categories.
The 8-item FIS-SF comprises 3 subscales that measure parent/family activity (4 items), parental emotions (2 items), and family conflict (2 items). Parents completed the questionnaire and rated how much each impact has occurred over the previous 3 months. The item response options were: “never” (scoring 0); “once or twice” (1); “sometimes” (2); “often” (3); “every day or almost every day” (4). Scale scores were computed by summing the scores for all 8 items; subscale scores (for the parental emotions, parental/family activity, and family conflict subscales) were computed by summing scores for their constituent items. Scores for all scales were standardized by dividing the raw scale score by the number of items. Scale sores are interpreted so that the higher the overall score, the greater the family impact.
Parents also responded to the global family impact question “How much is the daily life of your family affected by the condition of his/her teeth, mouth or face?,” with response options of “not at all”’ (scoring 1), “very little” (2), “some” (3), “a lot” (4) and “very much” (5). A global health rating was included to assess the overall impact on the family of OFC, and the condition of the child’s teeth, mouth, and face, and to enable examination of the criterion validity of the multi-item FIS-SF (see below). Test–retest reliability was not examined.
Statistical Analysis
All statistical analyses were undertaken using IBM SPSS Statistics (version 23). Categorical variables were described using frequencies and percentages, and continuous variables were described using means and standard deviations (SDs) or medians and ranges/interquartile ranges (with data on cleft type presented by patient characteristics in Table 1). We then examined the responses to the FIS-SF items (data reported in Table 2). For the small number of questionnaires with missing data for a particular FIS-SF item, we assigned the mean item score (none had more than 10% of item responses missing). After the computation of scale scores and their associated descriptive statistics, cross-sectional criterion validity was evaluated by examining the gradient in mean scores on the FIS-SF across the ordinal response categories of the global family impact item (data reported in Table 3). An ascending gradient in mean scores would indicate acceptable validity; analysis of variance was used to determine the statistical significance of the observed gradient in mean scores. This was repeated for the parental emotions, parental/family activity, and family conflict subscales. We then determined impact prevalence by counting the number of FIS-SF items for which the response was “often” or “every day or almost every day.” That simple count was then recoded such that anyone with one or more of those responses was categorized as having an impact. We then reported “impact prevalence” as the percentage of individuals with an impact, with cross-tabulations and χ2 tests (and standardized residuals) used to determine the statistical significance of the observed differences in prevalence. After confirmation of the validity of the FIS-SF, we examined scale scores by patient characteristics (data reported in Table 4).
Cleft Type, by Demographic Characteristics and Treatment History (Brackets Contain Row Percentages Unless Otherwise Indicated).
Abbreviations: CL, cleft lip; CL/P, cleft lip and palate; CP, cleft palate; VPI/SMC, velopharyngeal insufficiency/submucous cleft.
a Column percentages.
b Data missing for 6 cases.
c P < .05; cross-tabulations and χ2 tests.
Items and Responses.
Mean Standardized FIS-S Scores and Prevalence of Impacts by Responses to the Global Family Impact Question.
Abbreviation: FIS, Family Impact Scale.
a P < .05; analysis of variance; all four groups differ significantly from one another.
b P < .05; analysis of variance; all groups differ significantly from one another, with the exception of the “Very much/A lot” and “Some” groups.
c P < .05; Cross-tabulations; all 4 groups differ significantly from one another.
d P < .05; Cross-tabulations; all four groups differ significantly from one another, with the exception of the “Very little” and “Not at all” groups.
Mean Standardized FIS-SF Scores and Subscale Scores by Sociodemographic and Clinical Characteristics (Brackets Contain Standard Deviation Unless Otherwise Indicated).
Abbreviations: CL, cleft lip; CL/P, cleft lip and palate; CP, cleft palate; FIS-SF, short-form Family Impact Scale questionnaire; VPI/SMC, velopharyngeal insufficiency/submucous cleft.
a P < .05; analysis of variance.
Results
A total of 214 parents completed the FIS-SF questionnaire, with only 1 parent declining to participate in the study. The mean age of the children was 11.1 years (SD = 3.6, range 6-19), and nearly two-thirds were male (Table 1). Cleft lip and palate cases comprised just over half of the sample, with CP cases the next most common, at one-fifth. Approximately two-thirds did not have private health insurance and just under half lived in the Sydney metropolitan area. Just under half of the children had undergone 2 to 3 cleft-related operations and half had commenced orthodontic treatment.
Most questionnaires (95.0%) were completed by the child’s mother. A total of 5 questionnaires had missing data (2 participants failed to answer 2 questions, and 3 failed to answer 1). The most common item unanswered was “during the last 3 months, because of your child’s teeth, lips, mouth or jaws, how often have you or another family member taken time off work?” Responses to the scale items are presented in Table 2. Cronbach α values for the total score and the parental emotions, parental/family activity, and family conflict subscales were 0.88, 0.75, 0.81, and 0.70, respectively.
The standardized overall FIS-SF scores ranged from 0 to 3.5, with 35 (16.4%) having a score of 0, and none having the maximum score. Acceptable criterion validity was demonstrated by the consistent gradients observed in FIS-SF mean scores and impact prevalence across the ordinal categories of the global family impact item (Table 3). Although numbers were small in some categories, the scale scores and impact prevalence were highest in those for whom the greatest family impact was reported.
There were no significant differences by cleft severity type in responses to the global oral health rating item. Families with a child with a velopharyngeal insufficiency/submucous cleft (VPI/SMC) reported significantly greater impacts on their daily life than did families with children affected by other cleft types (Table 4). They also had significantly higher family/parental activity scores, as well as greater overall family impact than those with CL and CP. Families with a child affected by CL/P reported worse scores on the family/parental activity subscale than those with a child affected by CP. Although there were no statistically significant age-group differences in FIS scores, there were consistent gradients in FIS scores across the age groups, with worse family impact (higher mean scores) and parental emotions subscale scores observed with older children. There were no systematic differences in FIS-SF scores by sex, private health insurance, living location, the number of cleft-related operations, or whether a child had commenced orthodontic treatment.
Discussion
This study set out to describe the association between OFC and families’ QoL and to more closely examine the psychometric properties of the FIS-SF. It found that the associations between the global health rating and FIS-SF scores were strong and in the hypothesized direction, thus demonstrating the measure’s excellent criterion validity. Family impact was highest in families who had a child with VPI/SMC. Although there were no differences in overall FIS scores by age group, family impact was greater on the parental emotions subscale for families with an older child with cleft.
This study focused on an important clinical group for which little is known of the various orofacial conditions’ family impact. It used established measures of family impact and validated those for these patients. There was an excellent response rate, with only 1 parent who did not consent to participate in the study. The actual number of participants was greater than had been anticipated because there were more patients attending the cleft clinic during the period of study data collection than there had been in the past; this gave us more statistical power than we needed. Using the data in Table 3, we conducted a post hoc power analysis which showed that, for 95% power to detect the observed difference in overall mean FIS score between the “Very much/A lot” and the “Very little” group, we would have needed 28 individuals in each of those categories (or an overall total of about 112 participants, far fewer than the 214 for whom we obtained data). Standardized FIS-SF scores ranged from 0 to 3.5 (out of a possible maximum of 4.0), indicating that the measure had the range of scores to be sensitive to variations in family impact among the sample included in the study (Scientific Advisory Committee of the Medical Outcomes Trust, 2002). A limitation of this study was that test–retest reliability was not assessed because of the structure of the cleft clinic and concern about participant burden. Moreover, the study was cross-sectional, and so the measure’s responsiveness in this group could not be assessed over time. We suggest that the question “during the last 3 months, because of your child’s teeth, lips, mouth or jaws, how often have you or another family member taken time off work?” might be reworded, in order to include people who do not work, but who may still find that their child’s OFC impedes their ability to carry out their daily tasks. Thus, “…time off work” could be modified to read “…time off work or your daily tasks.”
Different cleft types represent distinct clinical entities and require different treatment and rehabilitation protocols. A bilateral CL/P carries a burden of significant facial deformity and poor speech; it is traditionally considered to be the most severe cleft phenotype. Accordingly, our finding that the impact was greater on those families with a child affected by VPI/SMC was unexpected. Few other studies have investigated VPI/SMC as a distinct cleft type and so there are no other data to with which to compare this finding. Further investigation in other settings would be useful in this respect.
Families with a child with a CL/P reported worse family/parental activity than those with CP. CL/P repair involves an intensive treatment and rehabilitation protocol, requiring multiple appointments, surgeries and therefore more parental time off work, and less time for other family members. This could partly explain the finding that those with a child with a CL/P reported worse family/parental activity. To date, the only other study reporting the differential impact of cleft type on families is a Brazilian study (Antunes et al., 2014). They also reported a worse impact on families with a child who had a CL or CL/P than in those with CP. Locker and colleagues reported that families with children affected by OFC were affected more than those with a child with dental caries or malocclusion, demonstrating the condition’s pervasive effects on the functioning of parents and the family as a whole (Locker et al., 2002).
In the current study, family impact (particularly on the parental emotions subscale) was greater among families of children of older age. Although no studies have investigated the impact that a child’s age has on family impact, findings from several OHRQoL studies have reported the negative impact that OFC may have on a child’s social and emotional well-being, especially in older age groups (Broder et al., 2012; Ward et al., 2013; Broder et al., 2014). This may be partly explained by the fact that these children do not fit societal norms in terms of facial appearance and communication skills (Pope & Ward, 1997), and older children have to cope with facial differences in addition to typical adolescent concerns about appearance (Topolski et al., 2005). More studies are required to assess how family impact changes as a child gets older, so that researchers and health care professionals may gain further insight to facilitate changes and to institute better care.
There were no systematic differences in family impact by the number of surgical operations. Secondary surgical treatment for individuals with OFC is indicated to improve aesthetics and function. One possible explanation for this finding is that the child’s OHRQoL and family impact improved as a result of each surgery and therefore the child and the family were not affected by having undergone multiple surgeries. Indeed, a recent study by Broder and colleagues reported that those who underwent cleft-related surgery had significant improvements in self-rated OHRQoL (Broder et al., 2016). This is likely to have led to lower impact on the family too.
In Australia, those who live rurally have poorer access to health care, and travelling to multiple appointments involves arduous time and travel commitments. Our comparison of FIS-SF scores for families from metropolitan regions with those from rural regions revealed no differences, perhaps because our cleft clinic is organized to minimize difficulties for rural families. Appointment times are arranged to best suit their travel arrangements, and anyone in pain is offered an immediate appointment. This may, in part, explain why we found no difference; however, it may simply be that those families in remote regions are used to travelling long distances and accept this as part of their care.
To our knowledge, the association between family impact and whether orthodontic treatment had commenced has not been previously examined in children with OFC. We thought that the family impact may be worse during orthodontic treatment, due to greater travel to appointments, financial burden, and concerns about the effects that orthodontic treatment may have on the child’s speech, mastication, and social interaction, but no such association was observed. There are no studies in the literature which have assessed how orthodontic treatment may impact the family; however, as the implementation of the FIS measure in clinical research gains momentum, we may be able to better determine whether this affects families.
Future research may consider the application of the FIS-SF measure in longitudinal studies, with data from different patient populations to verify the measure and to confirm and extend the findings that have been reported here (and to determine the responsiveness of the measure). Families may be substantially impacted when there is a child in the family who has a cleft (Locker et al., 2002), and so assessing the family impact of child OFC may be useful in identifying the need and improving care for individuals with OFC (and their families). Further research in this area should aim to reduce the burden of OFCs on individuals and their families.
Implementation of the FIS measure in clinical practice represents a shift from the biomedical model—comprised of objective dental and medical assessments—to the socio-environmental model of health care, which incorporates reports of functional and psychosocial issues and encompasses how patients, and their families view and are influenced by their oral health (Sischo and Broder, 2011). Family Impact Scale data provide unique information to supplement clinical measures, enabling improved understanding of treatment needs and the identification of at-risk groups. Incorporating FIS data into clinical practice and research has essential benefits for patients and their families, as well as for developing interventions that care for the entire family (Sischo and Broder, 2011).
Conclusion
This article confirms the impact of OFC on family QoL and provides further verification of the favorable psychometric properties of the FIS-SF. Routinely monitoring the family impact of child OFC may be useful in identifying the need and improving care for individuals with OFC (and their families).
Footnotes
Appendix
Spearman Correlations Between Standardized FIS-SF Scores and Sociodemographic and Clinical Characteristics.
| FIS | Parental emotions | Parental/family activity | Family conflict | |
|---|---|---|---|---|
| Sex | 0.02 | 0.01 | 0.02 | 0.02 |
| Age | 0.14 | 0.14 | 0.11 | 0.12 |
| Cleft type | 0.29 | 0.01 | −0.11 | 0.01 |
| Private health insurance | 0.05 | 0.12 | −0.02 | 0.03 |
| Postcode | −0.10 | −0.12 | −0.07 | −0.07 |
| Cleft-related operations | 0.12 | 0.07 | 0.14 | 0.04 |
| Orthodontic treatment | 0.11 | 0.11 | 0.14 | 0.03 |
Abbreviation: FIS-SF, short-form Family Impact Scale questionnaire.
Acknowledgments
The authors are grateful to all the parents for their cooperation and participation in this study.
Authors' Note
Sally Hibberts is now affiliated with Department of Dentistry, The Children's Hospital at Westmead, Westmead, Australia.
Declaration of Conflicting Interests
The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding
The author(s) received no financial support for the research, authorship, and/or publication of this article.
References
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