Quality of life of parents and siblings of children with cochlear implants.

OBJECTIVES: This study examined the quality of life (QoL) of the parents and siblings of hearing-impaired children with cochlear implants (CIs).
DESIGN: This is a cross-sectional, questionnaire-based study. The questionnaire consists of three sub-domains - interaction, emotional well-being and support for the hearing-impaired child and the overall QoL -- and two open-ended questions for participants to provide comments and suggestions to enhance their family's QoL. A total of 63 questionnaires were e-mailed or mailed to families who met the inclusion criteria.
SETTING: The study was conducted under the Center for Rehabilitation & Special Needs, Faculty of Health Sciences, Universiti Kebangsaan Malaysia, Kuala Lumpur. PARTICIPANTS: A total of 79 parents and 23 siblings from 44 families of children with CI participated in this study. MAIN OUTCOME MEASURES: The mean score for each of the sub-domain and the overall QoL for both subject groups were computed. The answers for the open-ended questions were listed and organized into themes.
RESULTS: There were significant correlations between the overall QoL score and each of the test domains for the parents' group (p < 0.01). For the siblings' group, only the interaction and support domains were significant. Interaction was the main sub-domain affecting the parents' QoL but for siblings, this could not be determined. A total of 60% of the pooled comments were classified as 'concerns'. As for the suggestions, 38.7% requested some form of support, mainly financial (41.7%).
CONCLUSIONS: Families with hearing impaired children with CI have numerous concerns that need to be addressed, even though, on average, they were satisfied with their overall QoL.

Introduction

Hearing loss impacts the quality of life (QoL) of the affected individuals (Looi et al., 2016; Umansky et al., 2011; Wake et al., 2004), even if the loss was unilateral (Borton et al., 2010). In the literature, numerous QoL studies following cochlear implantation have reported improvement of the cochlear implant (CI) users' QoL (Cohen et al., 2004; Isarin et al., 2015; Liu et al., 2016; Orabi et al., 2006; Vermeire et al., 2005). However, cochlear implantation has not only directly improved the QoL of the implant users but also affected the QoL of their immediate family members. Spahn et al. (2004), for example, studied the effect on parents who have hearing-impaired children with CIs. Such studies fall into the category of ‘third-party disability’ studies, (Grawburg et al., 2013; Scarinci et al., 2012, 2009), in which the disability of a family member impacts others in the family. The current study will report how parents and siblings view their family's QoL, when a member of the family is a child or sibling with severe to profound hearing loss who is a CI user.

Within the World Health Organization's (WHO) International Classification of Functioning, Disability and Health (ICF), even when family members do not have a health condition, they may still experience impairment, limitations and restrictions on participation in activities, if a child or sibling in the family has a disability. Consequently, the family's QoL may be affected. Scarinci et al. (2009), for example, studied 10 spouses of older people with hearing impairment and found that their activities and participation were restricted because of the hearing impairment of their significant others.

As a unit, the immediate family members of a child consist of parents and siblings. When a child in a family has a disability, the psychological impact is felt not only by the parents (Spahn et al., 2004), but also by the siblings of the disabled child (Cuzzocrea et al., 2014; Emerson and Giallo, 2014). Moyson and Roeyers (2012) who investigated the QoL of siblings of children with intellectual disability, concluded that siblings can define their own QoL and that they do have a different ‘concept’ of the family's QoL. The study by Cuzzocrea et al. (2014) found associations between family characteristics and the emotional and relationship difficulties of siblings of children with disabilities. These findings suggest it is worthwhile to include siblings in family QoL studies.

The present study aimed to investigate the QoL of the parents and siblings of children with CIs. Specifically, the objectives of the study were to: (i) quantify the parents' and siblings' perspectives on the impact of cochlear implantation on their lives in three domains: interaction, emotional well-being and support for the hearing-impaired child/sibling and the overall QoL; (ii) correlate the investigated family sub-domains (interaction, emotional well-being and support) and the QoL scores for each study group; (iii) determine the main domain affecting the QoL of parents and siblings; (iv) explore the associations of duration of implant experience, socioeconomic status and parents' education levels on the overall QoL and each of the investigated domains; and (v) document the comments and suggestions from the participants to further improve their family's QoL.

Method

Ethical considerations

The study protocol was approved by the UKM Research Ethical Committee (approval number UKM 1.5.3.5/244/NN-118-2011).

Participants

A purposive sampling method was utilized. Sample size calculation (Krejcie and Morgan, 1970) showed that a total of 67 subjects were needed for each group. Subjects were recruited from the family members of children who had undergone cochlear implantation under the Universiti Kebangsaan Malaysia (UKM) Cochlear Implant Program. The inclusion criteria for parent(s) were having a hearing-impaired child below 12 years of age who had been fitted at least unilaterally with a CI and for a duration of at least a year. For siblings, the inclusion criteria were as follows: must be between 13 and 18 years old; parents must be participating in the study and must be living in the same house as the sibling with the CI and be able to understand the instructions on the questionnaire. If more than one sibling met the inclusion criteria, the eldest child was selected. Families with more than one disabled child and more than one hearing-impaired child using either hearing aids or CIs were excluded from the study.

Instruments

The questionnaire used was made available in Malay and English language versions. It was adapted from The Beach Centre Family Quality of Life Questionnaire (Hoffman et al., 2006) and The Parent Questionnaire, Children with Cochlear Implants: Parental Perspectives (O'Neill et al., 2004). It included two sections. Section I is about demographic details. Section II has three sub-domains in IIA (interaction, emotional well-being and support for the hearing-impaired child) and, in IIB, there are four questions, on the family's overall QoL and two open-ended questions requesting participants to provide comments and suggestions to further improve their family's QoL.

The Beach Center Family Quality of Life Questionnaire (Hoffman et al., 2006) was originally developed to assess the QoL of families with children with disabilities. It has five domains (family interaction, parenting, emotional well-being, physical/material well-being and disability-related support) and 25 questions. On the other hand, The Parent Questionnaire, Children with Cochlear Implants: Parental Perspectives (O'Neill et al., 2004) is a closed-format questionnaire with 10 sub-domains and 74 questions. The sub-domains are communication, general functioning, self-reliance, well-being and happiness, social relationships, education, process of implantation, effects of implantation, decision to implant and supporting the child. Preceding the first five sub-domains is a sentence, ‘Thinking about your child now compared to before implantation…,’ suggesting the sub-domains are about changes experienced by parents, post-cochlear implantation.

Sub-domains ‘Interaction’ and ‘Emotional well-being,’ in Section IIA of the adapted questionnaire, consist of 20 questions extracted from The Parent Questionnaire, Children with Cochlear Implants: Parental Perspectives (O'Neill et al., 2004). Meanwhile, the questions in the ‘Support’ domain are from the Beach Center Family Quality of Life Questionnaire (Hoffman et al., 2006). Questions in Section IIA have been selected based on the purpose of this study. In each of these three sub-domains, there are 10 questions/domain for parents. For siblings, there are only seven out of the 10 questions/domain. The ratings in Section IIA are based on the 5-Likert scale, with positive items ranging from 1 (strongly disagree) to 5 (strongly agree) and negative items scoring the reverse. In Section IIB, scoring for the family's QoL ranges from 1 (strongly dissatisfied) to 5 (strongly satisfied).

Test procedure

Questionnaire preparation and validation

Relevant items from the original versions of the questionnaires were first identified, according to the objectives of the study. A back-to-back translation technique (Chen and Boore, 2010; Maneesriwongul and Dixon, 2004) was used to develop the Malay and English versions of the questionnaire. A pilot study was then conducted on four subject groups from families with children who used hearing aids: parents who spoke Malay and parents who spoke English as their primary language and siblings whose main language was Malay and those whose main language was English. Amendments to the questionnaire were made according to the responses received. The coefficient of reliability, Cronbach's Alpha, α, was computed to assess the internal consistencies of the instrument in which a cut-off point of 0.6 would be acceptable (Nunnally, 1978).

Questionnaire distribution

The finalized questionnaire was emailed or mailed to the identified participants. The participants (parents and siblings) were reminded to answer the questions independently to get their true views on the domains being studied. In addition, they were asked to return the questionnaires within two weeks after receipt.

Results

Instruments' internal consistencies

The α values for the parents' questionnaire ranged between 0.67 and 0.83. For the siblings' questionnaire, the α values were between 0.67 and 0.85. Therefore, the questionnaires were reliable and valid for the purpose of this study (Nunnally, 1978).

Participants' demographics

Of the 63 questionnaires distributed to families who met the inclusion criteria, 44 families returned the questionnaires, giving a response rate of 70%. The participants were families with cochlear implanted children aged between 3.9 and 11.8 years old (mean age = 9.3 ± 2.5 years) with the mean implant experience of 5.6 ± 2.3 years (ranging from 1.9 to 10 years). A total of 79 parents (41 mothers and 38 fathers) and 23 siblings (11 sisters and 12 brothers) participated. In some of these cases, both parents evaluated their child.

The age range of the siblings was 12.4–17.8 years old (mean = 15.3 ± 1.7 years). The highest education levels attained by the parents ranged from primary school to postgraduate degrees, with the majority having secondary education (48%), followed by Bachelor's degrees (32%). Most of the respondents were of Malay ethnicity (60%), while respondents of Chinese ethnicity comprised 30% and respondents of other ethnicities 10% of the participants. Almost 40% of the families had total monthly income between Malaysian Ringgit (RM) 7501.00 to 10,000.00, which could be classified as upper middle class in the Malaysian context.

QoL domains

Section IIA

Overall, the mean scores for the ‘interaction’ sub-domain for parents and siblings were 4.06 ± 0.55 and 3.79 ± 0.52, respectively. Parents rated highest (4.53 ± 0.60) for the statement ’My child's relationship with me has improved,’ while siblings rated highest (4.39 ± 0.50) for the statement ‘My sibling knows when I want his/her attention because he/she can hear me calling’.

For the ‘emotional well-being’ domain, both parents and siblings rated highest for these two statements ‘It is important to me that my child/sibling could hear sounds from the traffic for safety reason’ and ‘My child/sibling continues to be a happy person and good/fun to be with’. Interestingly, for the statement ‘The whole process of cochlear implantation is stressful’, parents had an average score of 3.06 ± 1.16, which was associated with ‘unsure,’ while siblings had a mean of 2.30 ± 1.1, which was associated with ‘agreed’. The overall mean scores for this sub-domain were 3.96 ± 0.47 for parents and 3.77 ± 0.60 for siblings.

In the third sub-domain, which was the ‘support’ domain, both groups rated almost the same. The parents had a mean overall score of 4.38 ± 0.42 and the siblings scored 4.24 ± 0.42. Both rated highest for the statement ‘Regular tuning and checking of the implant system are essential’.

Section IIB

Asked whether they were satisfied with the interaction among family members, parents gave a mean score of 4.38 ± 0.54, while siblings had a mean score of 4.09 ± 0.90. For the question of overall satisfaction as to their emotional wellbeing, the mean scores obtained were 4.30 ± 0.63 and 4.26 ± 0.49 for parents and siblings, respectively. For the question on overall satisfaction as to the support for the CI child, parents had a mean score of 4.05 ± 1.07, while siblings had a mean score of 4.17 ± 0.72. The mean scores for the overall family's QoL were not different. Parents had a mean score of 4.28 ± 0.57 and siblings had a mean score of 4.20 ± 0.50, indicating they were generally satisfied with the family's QoL.

The responses to the open-ended questions have been combined as most of the siblings did not answer the questions. Table 1 lists the comments given by the participants. Table 2 shows the three themes which occurred most frequently from an analysis of the qualitative data of the participants' comments and suggestions. The themes were identified by grouping answers with similar meanings. The groupings were cross-checked by two of the authors to ensure similarity of analysis.

Table 1

Comments given by the participants (parents and siblings) classified as ‘advantages’ and ‘concerns’.

Advantages		Concerns
Comments	%	Comments	%
Improved listening skill	16	Worry if the implant fail	10
Clearer speech	14	Financial problem	8
Improved localization skill	4	Not satisfied with the speech language therapy services	8
Good family support	4	High cost of device maintenance	6
Improved learning skill due to better hearing	2	Not satisfied with the current mainstream education	6
		Unconductive school environment	6
		Teachers are not aware and lack of knowledge about CI	4
		Worry about the child's future education	4
		No school visit by the speech language pathologist	4
		Lack of improvement in speech	4
TOTAL	40	TOTAL	60

Table 2

Breakdowns for suggestions from participants for the top three major themes emerged from the qualitative data of the open-ended questions.

Support		Educational issues		Consistent appointments
Suggestions	%	Suggestions	%	Suggestions	%
Financial support	41.7	Teach teachers (about hearing loss and CIs)	50.0	Speech language pathologists	40.0
Orientation to the CI	33.3	Placement of special teacher in the class	28.6	Audiologists (for programming)	30.0
Emotional support	8.3	Teachers to use FM system	7.1	Child psychologist	10.0
Family support	8.3	Teachers to motivate the CI child	7.1	Family counselor	10.0
Counseling service	4.2	Vocational training	7.1	Physiotherapist	10.0
General health	4.2

Hypotheses testing

Pearson correlation analyses showed significant correlations between each of the sub-domain in section IIA and the overall QoL of parents in Section IIB (for interaction domain r = 0.513, p = 0.001; emotional well-being r = 0.351, p = 0.002; support r = 0.435, p = 0.001). For siblings, however, the emotional well-being domain was not significantly correlated with the QoL (for interaction domain r = 0.656, p = 0.001; emotional well-being r = 0.383, p = 0.071; support r = 0.683, p = 0.001). Thus, the first hypothesis was accepted for parents but partially accepted for siblings.

Multiple linear regressions were performed in order to determine which sub-domain was the main one that interacted with the overall family QoL of both groups when considered together. For parents, F (3,75) = 13.307, p < 0.05 and r2 = 0.347 with the prediction model obtained was as follow:*p < 0.05.

Interaction was the main sub-domain contributing to the satisfactory QoL for parents. The emotional well-being domain was no longer significant when the three factors were considered together.

Similar analysis done for siblings also revealed significant F values in which F(2,20) = 10.895, p < 0.05 and r2 = 0.521 but none of the factors remained significant. Hence, the main domain contributing to the siblings' QoL could not be determined. The prediction model was as below:

The 3 × 3 Multivariate Analysis of Variance (MANOVA) was conducted to examine the association between the following demographic factors (duration of implant experience, parents' education levels and socioeconomic status) and each of the three test domains. In this analysis, duration of implant experience was treated as numerical data, while the other two variables were categorical. Table 3 summarizes the results.

Table 3

Findings from the Multivariate Analysis of Variance (MANOVA) for both study groups to examine the effects of duration of CI experience, socioeconomic status and parents' education levels on the family QoL sub-domains.

Groups	Parents			Siblings
	Duration of CI# experience	Socioeconomic status	Parents' education levels	Duration of CI# experience	Socioeconomic status	Parents' education levels
Pillai's Trace (p)	*0.01	0.23	0.58	0.15	0.96	0.55
Partial eta (ȵ2)	0.56	0.06	0.05	0.89	0.05	0.15
Test of between-subjects effects for duration of CI experience
Sub-domains	Interaction	Emotional well-being	Support
Significance value, p	*0.004	*0.039	*0.031
Partial eta (ȵ2)	0.594	0.524	0.533

#CI = Cochlear implant.

∗p < 0.05.

Only the duration of implant experience was a significant factor for parents [F (90,144) = 2. 064, p = 0.001] but not for siblings. Further ‘test of between-subjects’ effects conducted revealed significant associations between the duration of implant experience and each of the test domains. Data for siblings were non-significant.

Discussion

This study examined QoL in families with parents and siblings of children with CI, following the concept of third party disability (Scarinci et al., 2012, 2009). In general, both parents and siblings agreed they were satisfied with the overall family QoL. In our study, the interaction sub-domain was significantly correlated with the QoL of parents and siblings of the CI children. In fact, when all three sub-domains were considered, interaction was the main sub-domain affecting the QoL of parents (regression model 1). The longer the duration of the CI experience, the better (Table 3). Our findings on family QoL were partly consistent with an earlier study which reported that parents were most satisfied with increased communication, improved social relations and self-reliance of the child (Huttunen et al., 2009). Unfortunately, we could not determine the main sub-domain affecting the siblings' QoL even though the prediction model (regression model 2) was significant. In contrast to consistent findings that the use of CI improved communication skills among CI users and their family members and social relations (Almeida et al., 2015; Looi et al., 2016), Liu et al. (2016) reported that improvement in speech recognition scores among pediatric CI recipients in South West China did not correlate with improvement in the QoL of these children, suggesting that speech perception ability and, therefore, oral interaction skills achieved by these children following cochlear implantation might not always contribute to their QoL. However, in that study, they measured only the QoL of the implantees, not that of their families.

In the ‘interaction’ sub-domain, parents rated highest for the item ‘My child relationship with me has improved,’ while siblings rated highest for ‘My sibling knows when I want his/her attention because he/she can hear me calling’. According to Moyson and Roeyers (2012), joint activities is one of the domains of siblings' QoL. Having a severely and profoundly hearing-impaired sibling who could now hear through his/her CI device might have brought great satisfaction to the siblings.

The emotional well-being sub-domain was significantly associated with the parents' QoL but not with the siblings'. This result was interesting, even though it could have been affected by the relatively small sample sibling participants. As the other two domains achieved statistically significant correlations with the QoL of the siblings' group, this finding warrants some discussion.

The age of the siblings who participated in this study ranged from 12 years and 4 months (thereafter, denoted as 12;4) to 17;8 years old, with a mean age of 15.3 ± 1.7 years. During these teenage years, emotional well-being might not have been the siblings' main focus. However, Moyson & Roeyers (2012) listed several sibling QoL domains that could be described as ‘emotional well-being,’ such as acceptance, mutual understanding, exchanging experience and private time. This study found that siblings rated highest (4.70 ± 0.47) for ‘My sibling continues to be a happy sibling and good/fun to be with’. This rating of nearly 5 suggests that the sibling participants strongly agreed with this statement. The parents rated almost equally the statements ‘It is important to me that my child could hear sounds from the traffic for safety reasons’ (4.63 ± 0.59) and ‘My child continues to be a happy child and good/fun to be with’ (4.62 ± 0.56). These results partly supported the earlier finding by Huttunen et al. (2009) who reported that parents had high expectations that auditory information obtained via a CI would improve their child's safety in traffic and their child would be able to join socially in the hearing world. In this domain, both participant groups rated lowest for the item ‘The whole process of implantation was stressful’. The siblings rated 2.30 ± 1.11, suggesting they agreed with this statement, while parents rated 3.06 ± 1.16 suggesting they were generally ‘unsure’. In a study by Spahn et al. (2004), who measured psychological distress and QoL of 52 mothers and 42 fathers of 55 CI children, 21% of the parents showed enhanced psychological distress when going through the implant process and the parents' condition was not correlated with the CI children speech status. Huttunen et al. (2009) found that parents reported that CI affected their children and, therefore, their lives in a variety of ways that could not be summarized by a single scale. Another noteworthy statement in this sub-domain that was rated relatively low by the siblings was ‘I/other children in the family resented the time and attention taken up by my implanted sibling’ with a mean score of 2.70 ± 1.22, which ranged between ‘agreed’ (2) to ‘unsure’ (3). This finding was partially consistent with that reported by Cuzzocrea et al. (2014) who found that siblings of children with disabilities had higher stress levels than do their peers. This finding needs to be explored further in future studies.

For the support domain, both groups rated highest for the statement ‘Regular tuning and checking of the implant system are essential’. This was further emphasized in Table 2, where 30% of the participants wanted more appointments with the audiologist for mapping. Both groups also rated second highest for the statement ‘Important for the CI professionals to visit home/school at least 1x/year,’ which was also highlighted in Table 1, where some participants raised the ‘concern’ of not being visited by speech language pathologists (SLPs) at home/school and being not satisfied with speech therapy services. A total of 40% of the participants indicated a need for more appointments with the SLPs. A study by Davis and Gavidia-Payne (2009) showed that professional support was one of the strongest predictors of family QoL, when faced with the challenges of having children with disabilities. Thus, this issue needs to be addressed by the CI team members.

The responses from the participants for open-ended questions in Section IIB provided a slightly different view. Even though parents and siblings generally agreed they were satisfied with their families' overall QoL, there were significantly more ‘concerns’ listed by the participants than ‘advantages’. ‘Worry if the implant breakdown, device maintenance, financial problems’ (see Table 1) are concerns related to financial issues faced by the families. This is consistent with a study reported by Moroe and Kathrada (2016), in which families voiced their concerns which included financial difficulties, schooling and vocational prospects for their children with CIs. In our study, 50% of the participants who raised education-related issues suggested that teachers should have better knowledge of CI and supported children with CI in the classrooms.

Strengths and limitations of the study

This is the first study to report on the QoL of both parents and siblings (within a family) of hearing-impaired children with CIs. The relatively small number of sibling participants, however, is a limitation of this study. The results, however, suggest that siblings' well-being can be affected differently whenever there is a hearing-impaired child with a CI in the family.

The current study focused on only three sub-domains while there can be additional domains affecting the families' QoL. Our current findings from the multiple linear regression analyses showed that only 34.7% of the variances in the parents' QoL could be explained by these three domains, while for the siblings, the interaction and support domains explained 52.1% of the variances. Thus, it is suggested that, in future, approaches such as those used by Moroe and Kathrada (2016) or Moyson and Roeyers (2012) can be incorporated in the design of studies to capture even more data on family domains.

Conclusion

Overall, our results suggest that families do have a lot of concerns and need various forms of support to improve their families' QoL, even though they agree that cochlear implantation improved the overall family QoL. These concerns need to be addressed by CI team members to ensure a more holistic management approach for the families of children with CI.

Declaration of interest statement

The authors report no declarations of interest.