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Pediatric hearing impairment caregiver experience: Impact of duration of hearing loss on parental stress
International Journal of Pediatric Otorhinolaryngology (2008) 72, 1693—1703
www.elsevier.com/locate/ijporl
Pediatric hearing impairment caregiver experience: Impact of duration of hearing loss on parental stress Jareen Meinzen-Derr a,b,1,*, Lynne H.Y. Lim c,1, Daniel I. Choo b, Samantha Buyniski b, Susan Wiley d a
Center for Epidemiology and Biostatistics, Cincinnati Children’s Hospital Medical Center, 3333 Burnet Avenue, MLC 5041, Cincinnati, OH 45229, United States b Division of Pediatric Otolaryngology, Cincinnati Children’s Hospital Medical Center, University of Cincinnati, Cincinnati, OH 45229, United States c Department of Otolaryngology-Head and Neck Surgery, National University Hospital and National University Singapore, Singapore, Singapore d Division of Developmental and Behavioral Pediatrics, Cincinnati Children’s Hospital Medical Center, University of Cincinnati, Cincinnati, OH 45229, United States Received 13 June 2008; received in revised form 11 August 2008; accepted 12 August 2008 Available online 25 September 2008
KEYWORDS Caregiver stress; Pediatric hearing loss; Deaf/hard of hearing children
Summary Objectives: Caregivers of children who are deaf/hard of hearing have been reported to have greater stress than caregivers of children with normal hearing. The time of diagnosis is a particularly stressful time and stress levels may change over time based on varying needs at different life events. Thus, we hypothesized that stress experienced by caregivers evolves over time and is impacted by the duration since the diagnosis of hearing loss. Methods: The 68-item pediatric hearing impairment caregiver experience (PHICE) is a validated questionnaire used to measure stress. The PHICE was administered to 152 caregivers of children with permanent hearing loss. Domain scores were converted into z-scores for analysis of trends of stress over time. Results: Parents of children whose hearing loss was identified more than 60 months ago reported higher stress levels regarding educational aspects of their child’s needs as compared to parents of children with less than 24 months or 24—60 months duration since diagnosis. Parents of children diagnosed with hearing loss within the preceding
* Corresponding author at: Center for Epidemiology and Biostatistics, Cincinnati Children’s Hospital Medical Center, 3333 Burnet Avenue, MLC 5041, Cincinnati, OH 45229, United States. Tel.: +1 513 636 7789; fax: +1 513 636 7509. E-mail address: [email protected] (J. Meinzen-Derr). 1 Co-first authors. 0165-5876/$ — see front matter # 2008 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2008.08.005
24 months reported higher stress levels in the area of healthcare than parents of children diagnosed greater than 24 months ago. Conclusions: Parental stressors change over time with respect to the time of diagnosis of hearing impairment. This phenomenon was observed irrespective of the age of diagnosis of hearing loss. As professionals serving families of children with hearing loss, we should be aware of changing stressors over time and identify the appropriate support services for families to meet those changing needs. By addressing those evolving stressors, the families’ ability to support and improve the outcomes for their children who are deaf or hard of hearing may be enhanced. # 2008 Elsevier Ireland Ltd. All rights reserved.
1. Background The diagnosis of a hearing loss is a critical life event with profound effects on parents and the family system. An extensive body of literature has been published in recent years focusing on stress in parents who have deaf or hard of hearing children [1— 10]. Findings from studies specific to stress levels reported by parents of children with hearing loss have been inconsistent. Some evidence suggests that hearing parents of children who are deaf/hard of hearing (hoh) feel more stress than parents of hearing children [3,8], while some studies report no difference in stress levels between the two groups of parents [4—6]. The types of stress reported by parents of children who are deaf/hoh may be different from that of parents of hearing children and specific to the child’s hearing impairment, such as stress about degree of hearing loss, age of identification [11], language ability [6], and mode of communication [10]. Until we understand the types of stress that burden parents and caregivers of children who are deaf/hoh, we cannot provide the appropriate support services and interventions necessary to strengthen the parent—child relationship critical to a child’s ultimate developmental success. Studies have not investigated the change of stress factors over time, which may vary over the course of a child’s development. Additionally, there are few measures of caregiver stress specific to hearing loss. The pediatric hearing impairment caregiver experience (PHICE) is a measure that was developed and validated specifically on families of children with hearing loss.
1.1. The pediatric hearing impairment caregiver experience Lim et al. developed and validated a questionnaire examining various stressors families may experience related to their child’s hearing loss. The development and validity of the PHICE is detailed in Appendix A. Briefly, the PHICE was created using
an ‘‘expert panel’’ consisting of professionals caring for children who were deaf/hoh and caregivers of children who were deaf/hoh. The 11-member team consisted of specialist doctors from the Departments of Developmental and Behavioral Pediatrics, Psychology, Pediatric Otolaryngology, a medical social worker, a speech rehabilitation therapist, an aural rehabilitation therapist, an audiologist, and program/education directors/principals of the state’s early intervention system and schools for the deaf (both oral and signing). Caregivers that were asked to be involved had children with bilateral hearing loss of any degree or etiology, regardless of caregiver’s hearing status. Semi-structured individual caregiver interviews were conducted and tape-recorded with consent. Detailed shorthand notes were taken simultaneously. Answers to questions were categorized and written summaries were made of categories. Questions were then created from the interviews and tested among additional caregivers of children with hearing loss. The PHICE is a 68-item questionnaire which covers 8 domains of stress. These domains include: communication (10 questions), education (7 questions), emotional well being (11 questions), equipment (3 questions), financial (4 questions), healthcare (14 questions), social (8 questions), and support (11 questions). Parents rate stress on an 8-point Likert scale ranging from no stress to extremely high stress (see Appendix B). Internal reliability, or internal consistency, was determined using Cronbach’s alpha for the 68 items on the PHICE. Cronbach’s alpha is a statistic with a range of 0—1 that is used to determine if a set of questions measure the same idea [12]. The PHICE was found to have an overall reliability of 0.96, which is considered an extremely high coefficient. Test—retest reliability, used to assess the consistency of the PHICE from one time to another is currently ongoing (no data yet available). As stress levels may change over time according to a variety of needs at different life events, we predicted that stress felt by caregivers evolved over time and would be impacted by the duration since the diagnosis of hearing loss.
Pediatric hearing impairment caregiver experience
2. Methods This study utilized a cross-sectional design with data collected from two time points. Between November 2003 and December 2004, 150 PHICE surveys were mailed to caregivers of children who had hearing loss and had been seen within our institution. Between June and August 2007, parents or caregivers of children with permanent hearing loss attending otolaryngology, audiology, or aural rehabilitation appointments at a pediatric tertiary care center were asked to complete the PHICE. Families were eligible if their child had any degree of permanent bilateral sensorineural hearing loss. This study was approved by the Institutional Review Board of our institution and written informed consent was obtained from all caregivers who participated in the study. Demographic information was collected as part of the survey. Child specific information included gender, race, age of identification of hearing loss, etiology of hearing loss, type and degree of hearing loss, type of educational environment the child is currently attending, and any developmental issues. Family specific information included race, marital status, income level, employment status, and educational level of the parents. The 8-point Likert scale on the PHICE was collapsed into a 3-point scale for the purpose of the analysis. Items that represented no stress (not applicable, no stress, very low stress) were given a score of 1, items that represented some stress (low stress, moderate stress) were given a score of 2, and items that represented high stress (high stress, very high stress, extremely high stress) were given a score of 3. Scores were summed in each domain and over the entire survey. Lower scores indicate lower stress relative to higher scores, with a possible range in total scores from 68 points (lowest possible stress) to 204 points (highest possible stress).
2.1. Statistical analysis Continuous variables (i.e., age) were reported as means with standard deviations or medians with ranges. Categorical variables (i.e., gender) were reported as proportions. Comparisons between continuous variables and duration since the diagnosis of hearing loss (as a categorical variable) were tested with ANOVA or Kruskal Wallis test (nonparametric test) as appropriate. Comparisons between categorical variables and duration since the diagnosis of hearing loss (as a categorical variable) were tested using chi-square analysis or Fisher’s Exact test as appropriate. Frequencies of each of the PHICE questions were computed. The rela-
1695 tionships between the individual questions and duration since diagnosis categories were evaluated with chi-square analysis. Domain scores (support, social, emotional, financial, healthcare, equipment, education, and communication) were converted to z-scores for analysis. A standard z-score allows for a better understanding of how many standard deviations above or below the mean an individual’s score was relative to the entire sample. Because each domain had a different minimum and maximum possible score, z-scores were also used to compare the PHICE scores across domains. Mean z-scores were adjusted (Least-Square means reported) for potential confounders and covariates that were found to be significantly associated with parental stress. All statistical analyses were conducted using SAS1 version 9.1 (Cary, NC).
3. Results 3.1. Subject demographics The return rate of the mailed surveys was 48% (72 returned completed surveys). Ninety-eight subjects were asked to complete the PHICE while waiting in clinic; no caregiver declined this study. Five surveys were excluded, as they were filled out twice by the same family and the first survey was kept in the analyses. Five surveys were excluded due to incomplete data on the PHICE. An additional eight subjects were excluded from the analyses due to missing information on the age of the diagnosis of the child’s hearing loss. A total of 152 surveys were included in the final analyses; 62 from the mailed survey group and 90 from the clinic group. Differences between the two study groups (mailed survey group and clinic group) were evaluated. Subjects in the mailed survey group were more likely to be Caucasian (88.7% vs. 45.6%, p = 0.04), to have family members with hearing loss (24.2% vs. 13.3%, p = 0.09), have a stressful event occur within the past 6 months (35.5% vs. 22.2%, p = 0.07), and have severe to profound hearing loss (72.9% vs. 52.3%, p = 0.006). Children in the mailed survey group had their hearing loss identified at significantly younger median [range] ages compared to the clinic group (10 months [0—64] vs. 15 months [0—132], respectively, p = 0.002). The finding may be due to the frequent clinical follow up evaluations of children with previously identified hearing losses resulting in a skewed population that is encountered in any given clinic sample. Of the 152 families who were included in the analyses, 81% were filled out by a mother; 12%
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Table 1 Subject characteristics Subject characteristics Male (%) Race, Caucasian Cause of hearing loss known Age of identification of hearing loss in months Age at time of study in months
Duration since diagnosis (in months) <24, N = 37
24—59.9, N = 45
17 28 20 9
26 38 25 12
(46) (76) (54) (0—120)
(58) (84) (56) (0—132)
60, N = 70 36 57 43 15
p-Value
(51) (81) (61) (0—96)
0.6 0.6 0.7 0.28
157.5 (65—257)
<0.0001
22 (6—130)
56 (24—187)
Degree of HL Severe to profound Moderate to severe Mild
19 (51) 14 (38) 4 (11)
29 (66) 11 (25) 4 (9)
41 (62) 23 (35) 2 (3)
0.95
Type of device used None Hearing aids Cochlear implant
3 (8) 24 (65) 10 (27)
2 (4) 21 (47) 22 (49)
12 (17) 33 (47) 25 (36)
0.07
Communication mode Speech Speech + sign Sign Other
21 9 8 8
(32) (24) (22) (22)
25 5 10 5
(56) (11) (22) (11)
31 23 14 2
(44) (33) (20) (3)
Education Fully mainstreamed Deaf education (oral or sign) Home or no school Daycare/preschool Other
4 4 17 9 3
(11) (11) (46) (24) (8)
12 14 5 4 10
(27) (31) (11) (9) (22)
28 31 9 0 5
(40) (44) (9)
a
Significant medical problems in the last 6 months
11 (30)
8 (18)
0.01
<0.0001
(7)
15 (21)
0.42
Caregiver characteristics Caregiver age in years Marital status–—married
31 (22—49) 27 (73)
35.5 (20—56) 35 (78)
41 (23—70) 47 (67)
Highest education Completed college/post-grad Some college HS grad Some HS
16 10 8 3
(43) (27) (22) (8)
24 10 10 1
(53) (22) (22) (2)
34 20 10 6
(49) (29) (14) (8)
7 7 8 4 9
(20) (20) (23) (11) (25)
9 6 5 6 16
(21) (14) (12) (14) (38)
12 12 12 8 19
(19) (19) (19) (13) (30)
0.93
29 (41) 17 (24) 22 (31)
0.99 0.10 0.55
Income level <$20,000 $20—40,000 $40—60,000 $60—80,000 >$80,000 Full time employment outside the home Other family members with hearing loss Stressful events in the past 6 months
15 (41) 6 (16) 8 (22)
19 (42) 4 (9) 12 (27)
Reported as N (%) for categorical variables and medians (ranges) for continuous variables. a Missing information on degree of hearing loss in 5 subjects.
<0.0001 0.46
0.68
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Table 2 Top 10 stressors reported among caregivers of children with hearing loss Item on PHICE
N (%) reporting high levels of stress
N (%) reporting low stress
Worries about my child’s safety Seeing my child frustrated because of his/her hearing loss Feeling uncertain about my child’s future Worries about others taking advantage of my child Costly medical equipment and expenses Worries about not doing enough for my child Having no time for myself Worries about my child losing or damaging his/her equipment My lack of signing skills Lack of educational opportunities for my child
39 36 35 31 31 31 30 24 23 21
77 68 74 70 57 78 51 80 58 49
were fathers, 5% were grandparents, 1% were foster parents, and in 1% both father and mother completed the survey. Ninety-four percent of caregivers were hearing. The median (range) age of child at time of study was 78.5 (6—257) months while the median (range) age of child at time of hearing loss diagnosis was 55.5 (3—227) months. The median (range) age of the caregiver was 37 (20—70) years. The majority of children (95%, n = 142) had a bilateral hearing loss. Regarding duration since the diagnosis of hearing loss, 24% (n = 37) had a child who had been diagnosed in the last 23 months, 30% (n = 45) of families had a child who was diagnosed 24—60 months ago, and 46% (n = 70) had a child diagnosed greater than 60 months prior to the survey. Table 1 shows differences across duration since diagnosis categories for both child and caregiver characteristics. No differences were seen with respect to gender or race among the three groups. The degree of hearing loss was severe to profound in over 50% in each group. Children who had been diagnosed with their hearing loss for 24 months were more likely to report speech as their primary mode of communication than children who had been diagnosed <24 months ago ( p = 0.01).
(26) (24) (23) (21) (21) (21) (20) (16) (15) (14)
(51) (45) (49) (46) (38) (52) (34) (53) (38) (32)
3.2. Caregiver stress Over 20% of all caregivers reported high levels of stress regarding worrying about child safety, seeing their child frustrated, uncertainty about their child’s future, worrying about others taking advantage of their child, and having no time for one’s self (Table 2). The duration since the hearing loss diagnosis was correlated with the domain scores using Pearson’s correlation coefficient. Duration since diagnosis was significantly positively correlated with the education (rho = 0.25, p = 0.002) and social domain (rho = 0.20, p = 0.02). Duration was significantly negatively correlated with the emotional domain (rho = 0.16, p = 0.046) indicating that the longer the time since the diagnosis of their child’s hearing loss, the stress related to emotional well being decreased. Differences in mean domain scores across the three groups were assessed using ANOVA. Significant increases in scores ( p 0.05) were seen with increasing duration since identification regarding the education domain (7 questions) and the social domain (8 questions) (Table 3). To better understand how stress evolves while families gain experience with their child’s hearing
Table 3 Unadjusted domain scores across groups Domain scores
<24, N = 37
24—59.9, N = 45
60, N = 70
Total possible range in scores
Communication Education * Emotional well being Equipment Financial Healthcare Social ** Support
14.9 9.3 17.3 4.9 6.4 20.6 12.6 14.7
15.1 10.9 16.5 1.8 5.9 19.9 13.6 15.2
14.5 11.6 16.1 4.8 6.5 19.7 14.4 15.2
10—30 7—21 11—33 3—9 4—12 14—42 8—24 11—33
* **
(3.6) (2.3) (4.2) (1.4) (1.7) (4.4) (3.3) (3.2)
p = 0.0008 for comparison among groups. p = 0.047 for comparison among groups.
(4) (3.1) (4.5) (1.3) (2) (4.9) (3.4) (4.1)
(3.8) (3.2) (4.3) (1.7) (2.2) (5.5) (3.8) (4.3)
1698 loss, domain z-scores were adjusted for confounders that may influence the relationship between stress and duration since diagnosis. Potential confounders included caregiver education, hearing loss severity, communication strategy, stressful events in past 6 months, additional medical problems in past 6 months, and current hearing aid or cochlear implant use. Certain models were also adjusted for race, age at diagnosis, and child’s current age if these were significant in the models. Adjusted mean scores that were significantly different across groups are illustrated in Fig. 1. Stress related to education, equipment, and support was at its lowest for caregivers of children diagnosed in the last 24 months. Stress related to healthcare and emotional well being was at its highest for this same group of caregivers. No significant differences were seen among groups regarding stress related to the social domain ( p = 0.35) and finances ( p = 0.26). Total stress showed a slight increase over time (from a z-score of 0.51 among <24 month group to 0.90 among the 60 month group), though this increase was not statistically significant ( p = 0.32). Although the main analyses focused on three duration categories, domain adjusted z-scores were plotted as spline curves over multiple age categories to illustrate a broader sense of change over a wider range of duration since diagnosis (Figs. 2 and 3). Consistent with earlier findings of increased stress over the three time since diagnosis groups, a significant increase in stress related to the areas of education ( p = 0.002) and support ( p = 0.008) was noted over time (Fig. 2). The association between duration since diagnosis and stress related to equipment, although statistically significant, did not appear to be linear. Duration 73—108 months and
J. Meinzen-Derr et al.
Fig. 2 Adjusted mean z-scores over time since the diagnosis of hearing loss was made for the domains of education, equipment, and support.
>156 months had the highest mean levels of stress, which were both significantly ( p < 0.05) different than the other duration groups. Stress related to healthcare and emotional well being appeared to decrease with time (Fig. 3) although these findings did not meet statistical significance ( p = 0.2 and p = 0.09, respectively). Stress related to communication remained relatively high across all duration categories, with only a slight decrease over time ( p = 0.08). Although these findings were only considered as marginally significant trends, they present a shift in the types of stress felt by caregivers as they gain experience with their child’s hearing loss.
4. Discussion The findings from this cross-sectional study provide an initial effort to address how stress may change for
Fig. 1 Mean z-scores, adjusted for confounders which included caregiver education, HL severity, communication strategy, and other stressful events. Numbers along bottom represent p-values for the comparison of the three groups.
Fig. 3 Adjusted mean z-scores over time since the diagnosis of hearing loss was made for the domains of healthcare, communication, and emotional well being.
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families of children with hearing loss depending on the time since identification of hearing loss. Families of children recently identified with hearing loss (within 24 months of the study) reported higher levels of stress in the area of healthcare (interacting with healthcare systems and/or physicians) and emotional well being. Families of children with a longer duration of experience adjusting to their child’s hearing loss had more concerns in the area of education and support systems. Although total stress was shown to slightly increase over duration groups, this finding did not represent the fluctuations in types of stress that was felt by the cohort of caregivers. Studies that have compared parental stress between parents with deaf/hoh children to normative samples of parents with hearing children have not been consistent with their findings. Pipp-Siegal et al., suggested that stress levels among mothers of children who are deaf or hard of hearing are not clinically higher according to the Parental Stress Index (PSI) than a normative sample of mothers of hearing children [6]. However, 13% of the study mothers had stress scores above the clinical cutoff level (>90th percentile). This finding is similar to that reported by Lederberg and Golbach, who reported that 9—13% of mothers of 3—4-year-old children who were deaf were above the clinical cutoff on the PSI [3]. They also reported differences in maternal stress between mothers of deaf/hoh children and mother of hearing children only among children who were 2 years of age, but not mothers of 3 or 4 year olds. Meadow-Orlans reported that 25% of mothers of children who were deaf/hoh had scores that were considered clinically high [4], though no differences were seen between the study group and the normative sample on the PSI. These types of observations and our clinical experience served as an impetus to develop, validate and utilize the PHICE in order to have an instrument that would be relevant to this specific patient population and potentially help guide family-centered care. Many studies on stress felt by parents of children who are deaf/hoh have a focused attention on hearing aid fitting or the decision regarding cochlear implants. Spahn et al. surveyed 57 mothers and 46 fathers of 57 children with cochlear implants [9]. Using the Symptom Checklist-90-R (SCL-90-R) to assess stress, investigators reported that 25% of mothers and 25% of fathers had high levels of psychic stress. Quittner et al. studied stress using the SCL90-R in children 24 months of age with cochlear implants [8]. Using the normative sample on the SCL-90-R for comparison, high stress was highly correlated with increased depression and anxiety in mothers of children with cochlear implants. Bur-
ger et al. compared the change in stress levels over time between parents of children with cochlear implants and those hearing aids [13]. Their findings suggested that parents of children with cochlear implants had a slight, though non-statistically significant reduction in stress over time (between preand post-implant) compared to parents of children with hearing aids. Investigators also noted that the time point immediately after diagnosis for both groups was perceived as the most stressful for families. Age at the onset of deafness has been shown to be related to maternal stress. In a study of 42 mothers of children who were deaf, stress was higher if the child’s deafness was identified prior to 18 months of age [11]. Interestingly, the child’s chronological age was not related to the overall stress score, though mothers of ‘‘younger’’ children had higher mean stress scores than mothers of ‘‘older’’ children. It was difficult, however, to determine if there were any modifying effects regarding the time of identification and the study age of the child. In other words, the study did not discuss how duration since the diagnosis affected maternal stress levels. Findings from the current study suggest that the duration since the diagnosis of the hearing loss played a significant role in caregiver stress, even after controlling for the age of the child at the time of the study. The initial grieving process of many caregivers upon the diagnosis may create or add to stress. Over time, caregivers gain both knowledge and experience with hearing loss, which could reduce certain aspects of stress. Results from the current study indicate that while certain types of stress diminished over time, others appeared to increase. Stress surrounding educational concerns naturally increased over the duration since diagnosis, as children began to enter the school system. Stress around equipment needs and support also increased with time following diagnosis of hearing loss. It is possible that as parents gain more understanding of the needs of their child who is deaf/hoh, certain specific equipment (e.g., vibrating alarms) become important. In contrast, stress around healthcare issues decreased with increasing duration. The initial high levels may have been due to the initial stress felt by caregivers regarding number of clinical visits, initiation into therapies, and also comfort level with clinicians. As families become familiar with aspects of clinical care, the stress in this domain decreases. This may indeed be a common trend that crosses any medical condition and the family interactions with the healthcare system. Interestingly, stress focused on communication remained high across groups (even upon controlling for primary communication
1700 mode), though it was slightly lower for the 60 month duration group. This finding speaks to a common theme felt by many caregivers regarding the impact of deafness on communication and the subsequent influence this has on family. A longitudinal study, following caregivers over time, will allow us to evaluate what components of stress are altered over time. It is interesting to note that a finding during the development of the PHICE was that caregivers who were deaf/hoh themselves and utilized American Sign Language as their primary language had the least stress of all caregivers. The majority of caregivers in the current study, however, were hearing. Studies on parental stress have not uniformly reported characteristics that are specific to a child who is deaf/hoh, such as primary mode of communication, age of hearing loss identification, type of school or intervention a child may be receiving. Yet, stress of caregivers of deaf/hoh children involves a complex model of demographic characteristics, characteristics of perceptions of the caregiver (e.g., education, social support), and factors related to child’s hearing loss (e.g., degree of hearing loss, age of identification, language ability) [2,6]. In addition, most studies do not address the different areas stress may be felt by caregivers, but rather classify people as either feeling high or low levels of stress. The current study was able to take into account such factors that are related to a child who is deaf/hoh. In addition, a major strength of the current study is the use of stress-specific domains: communication, education, emotional well being, equipment, financial, healthcare, social, and support. By categorizing stress reported by the caregiver, clinicians are able to identify potential areas of concern and address the needs of the family in terms of interventions and support mechanisms. This study examined the effect duration since the identification of the hearing loss has on caregiver stress. Longitudinal research should be employed to effectively assess how this stress changes over time within families. The current study utilized a fairly new tool for assessing caregiver stress. Although other widely accepted tools are available for measuring stress (i.e., Parental Stress Index), these tools do not have questions that are pertinent to how a caregiver may feel about the specific relevant needs of their deaf/hoh child. Investigators have taken the steps to ensure that the PHICE represented this group of parents and professionals were included in the development of the questionnaire, to create a valid tool (Appendix A). Test—retest reliability is currently ongoing and a longitudinal study would further validate changes in stress over time. Dr. L. Lim is
J. Meinzen-Derr et al. currently completing a similar study in a Singapore population of caregivers of children with hearing impairment. The goal of the study is to evaluate the validity of the questionnaire in a different cultural and ethnic context. Many factors play a role in parental stress and it is important to realize that stress is not necessarily static. By understanding how stressors change with experience with hearing loss, we can better anticipate areas of need and support for the families we serve, with the goal of improving child outcomes.
Appendix A. Development of the PHICE This study was approved by the Institutional Review Board, and informed consents were obtained. An ‘‘expert panel’’ consisting of professionals caring for children who were deaf/hoh and their caregivers was assembled. The 11-member team consisted of specialist doctors from the Divisions of Developmental and Behavioral Pediatrics, Psychology, Pediatric Otolaryngology, a medical social worker, a speech rehabilitation therapist, an aural rehabilitation therapist, an audiologist, and program/education directors/principals of the state’s early intervention system, an oral deaf school program, and a school for the deaf. The early intervention program cares for families of infants with newly diagnosed hearing loss. The majority of children at the school for the deaf utilize a total communication approach with signing, oral and lip-reading when necessary. The expert panel was interviewed in face-to-face personal interviews to help identify issues which were possible stressors for caregivers of children with hearing loss. Questions for a semi-structured interview were developed from this information along with review of the literature. Stress was defined as ‘‘specific external and/or internal demands that are appraised as taxing or exceeding the resources of the person’’ [1]. A stressor is ‘‘any stimulus that makes demands upon an organism requiring adaptation or adjustment’’ [2]. Primary caregiver was defined as the person who provided the most care to the child in terms of time, including a parent, relative, teacher or care-taker. Caregivers participating were of children from the state’s early intervention program, the school for the deaf, or the oral deaf school program. Primary caregivers were included in the study if they were (1) caregivers of children with bilateral hearing loss of any degree, etiology, and type (conductive, sensorineural or mixed); (2) any race or gender; (3) with
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hearing or with a hearing loss. Caregivers were excluded if their child had significant medical conditions causing significantly more stress than related to hearing loss, such that the caregiver cannot differentiate between the causes of the stress. The caregivers were a highly heterogeneous group with varied social income, race, child’s duration and degree of hearing loss severity, child’s intervention status (non-aided/HA/CI). As such, ‘‘information-rich’’ caregivers were identified to ensure that all possible issues causing stress were considered. As the interpretation developed, caregivers were selected to further enrich, confirm and disconfirm our understandings [3]. The number of caregivers was sampled to the point of saturation, with no new information identified [4]. Semi-structured individual caregiver interviews using the interview guide questions were then conducted in private areas at the schools or the hospital. Due to the heterogeneity of caregivers, individual interviews were conducted rather than focus groups so that information would be more accurate and forthcoming. The trained interviewers were the principal investigator (Lim) and two psychology graduate students. Interviews were tape-recorded with consent, and detailed shorthand notes were taken simultaneously. Interviewers sat into the meetings of other interviewers for internal monitoring of quality. Qualitative research methodology was used to interpret the data, emphasizing the iterative processes of describing, organizing, connecting, corroborating and legitimating [5]. The organizational style used was the ‘‘editing’’ style in the first rounds of readings where the plan was to explore, without prior preconceptions of the topic. Segments of the text were then organized into categories. The ‘‘template’’ organizing style was used later when transcripts were reread. Many revisions and re-categorizations were made as new understandings evolved during the review. The interpretive process was terminated when there was no new information
encountered. In the final presentation, the caregiver issues were categorized for presentation to a clinical audience. The three interviewers and two additional researchers familiar with qualitative questionnaire studies independently read all interview transcripts. Written summaries were made of categories and the issues involved in each. The researchers then met and developed preliminary interpretations and meanings. Following these initial discussions, the researchers individually developed categories of stress. Thereafter, a consensus of main issue and themes was reached. Questions were then developed so for development of a quantitative questionnaire. This was then pre-tested on caregivers to verify that the caregivers understood the questions. The majority of the PHICE development focused on content validity, which consists of a judgment by ‘‘experts’’ whether the tool appears appropriate for the measurement of stress among caregivers of children who are deaf/hoh [6,7]. Because no ‘‘gold standard’’ measure exists for quantifying stress specifically related to having a child who is deaf/hoh, criterion validity, the correlation of a scale with some other measure of a gold standard, is difficult to establish [7].
1. 2.
3. 4. 5. 6.
Appendix B. Parent/Caregiver Questionnaire The following is a list of possible issues face by parents or caregivers of children with a hearing impairment. Please think about the past 6 months and tell us what level of stress related to each issue, if any, you experienced as a result of caring for your child with a hearing impairment. If an issue does not pertain to you or your child, please circle ‘‘not applicable’’. If you have more than one child with hearing impairment, please think of your older child when answering this questionnaire
Please indicate the level of stress, if any, you have experienced with each issue during the past 6 months
Not applicable
No stress
Very low stress
Low stress
Moderate stress
High stress
Very high stress
Extremely high stress
Child has temper tantrums related to hearing loss. Child care provider/schools/teachers/ out-of-home caregiver’s lack of signing skills. Working together with education professionals. Worries about my child losing or damaging his/her equipment. Being unable to go to work. Having difficulties accepting that my child is hearing impaired.
1
2
3
4
5
6
7
8
1
2
3
4
5
6
7
8
1
2
3
4
5
6
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8
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7. 8. 9. 10. 11. 12. 13. 14. 15. 16. 17. 18. 19. 20. 21. 22. 23. 24. 25 26. 27. 28. 29. 30. 31. 32. 33. 34. 35. 36. 37. 38. 39. 40. 41. 42. 43. 44. 45. 46. 47. 48. 49.
Please indicate the level of stress, if any, you have experienced with each issue during the past 6 months
Not applicable
No stress
Very low stress
Low stress
Moderate stress
High stress
Very high stress
Extremely high stress
Hearing aids or cochlear implants not meeting expectations. Delayed cochlear implantation. Feeling that healthcare providers do not listen to me. Inadequate support or understanding from an employer. Child uses hearing loss as an excuse to behave badly. Feeling uncertain about my child’s future. Sound of child’s voice is irritating, loud or shrill. Distinguishing between behavior and communicating problems. Obtaining special learning materials for my child (e.g., books, captioned videos). Equipment interfering with child’s activities. Worries about not doing enough for my child. Not being able to attend to the needs of other family members. Worries that my child is getting good medical care. Working together with doctors. Worries about deaf community’s perceptions of my decisions for my child. Lack of interpreters for child. Worries about others taking advantage of my child. Worries about my child’s safety. My lack of signing skills. Worries about choosing the best form of communication for child (oral, sign, total) Worries about decisions I have made for my child’s education. Inadequate insurance coverage for treatment of hearing impairment. Feeling partly responsible for my child’s hearing impairment Feeling uncertain about disciplining my child. Traveling to medical appointments. Feeling confused about medical information. Not able to take time off from work to tend to child’s needs. Lack of childcare or babysitters for child. Worries about my child’s ability to make friends. My child’s lack of signing skills. Difficulty communicating with my child. Child’s behavior in school. Problems dealing with insurance company. Tired due to caring for my child with hearing impairment. Change in my relationship with my partner or spouse. Keeping large number of medical appointments. Working with the nursing or rehabilitation team. Understanding the laws related to hearing impairment. Difficulty getting information about hearing impairment or rehabilitation. Worries about how my child fits into the hearing community. Health care provider’s lack of signing skills. Child’s academic achievement in school. Lack of educational opportunities for my child.
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50. 51. 52. 53. 54. 55. 56. 57. 58. 59. 60. 61. 62. 63.
64. 65. 66. 67. 68.
Please indicate the level of stress, if any, you have experienced with each issue during the past 6 months
Not applicable
No stress
Very low stress
Low stress
Moderate stress
High stress
Very high stress
Extremely high stress
Costly medical equipment and expenses. Having no time for myself. Overwhelmed by information on caring for a child with hearing impairment. Difficulty getting medical and rehabilitation care. Worries about whether my child should have a hearing aid, cochlear implant, or no aid. Inadequate support or understanding from spouse or partner. Lack of support network of parents facing similar problems. Problems discussing sexual issues with my child. Immediate family and relative’s lack of signing skills. Child’s reading ability. Caring for the hearing aid or implant. Costly daily aids like vibrating alarms and TTY services. Relocation to be near resources that meet my child’s special needs. Worries about risks associated with medical treatment (e.g., surgery, hearing equipment). Knowing that my child was diagnosed with hearing impairment late. Worrying about success of my child’s therapy or rehabilitation. Inadequate support or understanding from friends and relatives. Lack of a large deaf community. Seeing my child frustrated because of his/her hearing loss.
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References [1] M. Hintermair, Sense of coherence: a relevant resource in the coping process of mothers of deaf and hard-of-hearing children? J. Deaf Stud. Deaf Educ. 9 (2004) 15—26. [2] M. Hintermair, Parental resources, parental stress, and socioemotional development of deaf and hard of hearing children, J. Deaf Stud. Deaf Educ. 11 (2006) 493—513. [3] A.R. Lederberg, T. Golbach, Parenting stress and social support in hearing mothers of deaf and hearing children: a longitudinal study, J. Deaf Stud. Deaf Educ. 7 (2002) 330— 345. [4] K. Meadow-Orlans, Stress, support, and deafness: perceptions of infants’ mothers and fathers, J. Early Intervent. 18 (1994) 91—102. [5] K.P. Meadow-Orlans, S. Dyssegaard, B. Smith-Gray, Infants who are deaf or hard of hearing, with and without physical/ cognitive disabilities, Am. Ann. Deaf 140 (1995) 279—286. [6] S. Pipp-Siegel, A.L. Yoshinaga-Itano, C. Sedey, Predictors of parental stress in mothers of young children with hearing loss, J. Deaf Stud. Deaf Educ. 7 (2002) 1—17.
[7] A.L. Quittner, R.L. Jackson, D.N. Glueckauf, Chronic parenting stress: moderating versus mediating effects of social support, J. Pers. Soc. Psychol. 59 (1990) 1266—1278. [8] A.L. Quittner, J.T. Rouiller, R.L. Steck, Cochlear implants in children: a study of parental stress and adjustment, Am. J. Otol. 12 (Suppl.) (1991) 95—104. [9] C. Spahn, B. Richter, I. Zschocke, E Wirsching, M. Lohle, The need for psychosocial support in parents with cochlear implanted children, Int. J. Pediatr. Otorhinolaryngol. 57 (2001) 45—53. [10] M.T. Greenberg, Family stress and child competence: the effects of early intervention for families with deaf infants, Am. Ann. Deaf 128 (1983) 407—417. [11] M.M. Konstantareas, V. Lampropoulou, Stress in Greek mothers with deaf children. Effects of child characteristics, family resources and cognitive set, Am. Ann. Deaf 140 (1995) 264—270. [12] L.J. Cronbach, Coefficient alpha and the internal structure of tests, Psychometrika 16 (1951) 297—334. [13] T. Burger, C. Spahn, B. Richter, S. Eissele, E. Lohle, J. Bengel, Parental distress: the initial phase of hearing aid and cochlear implant fitting, Am. Ann. Deaf 150 (2005) 5—10.
Available online at www.sciencedirect.com
www.elsevier.com/locate/ijporl
Pediatric hearing impairment caregiver experience: Impact of duration of hearing loss on parental stress Jareen Meinzen-Derr a,b,1,*, Lynne H.Y. Lim c,1, Daniel I. Choo b, Samantha Buyniski b, Susan Wiley d a
Center for Epidemiology and Biostatistics, Cincinnati Children’s Hospital Medical Center, 3333 Burnet Avenue, MLC 5041, Cincinnati, OH 45229, United States b Division of Pediatric Otolaryngology, Cincinnati Children’s Hospital Medical Center, University of Cincinnati, Cincinnati, OH 45229, United States c Department of Otolaryngology-Head and Neck Surgery, National University Hospital and National University Singapore, Singapore, Singapore d Division of Developmental and Behavioral Pediatrics, Cincinnati Children’s Hospital Medical Center, University of Cincinnati, Cincinnati, OH 45229, United States Received 13 June 2008; received in revised form 11 August 2008; accepted 12 August 2008 Available online 25 September 2008
KEYWORDS Caregiver stress; Pediatric hearing loss; Deaf/hard of hearing children
Summary Objectives: Caregivers of children who are deaf/hard of hearing have been reported to have greater stress than caregivers of children with normal hearing. The time of diagnosis is a particularly stressful time and stress levels may change over time based on varying needs at different life events. Thus, we hypothesized that stress experienced by caregivers evolves over time and is impacted by the duration since the diagnosis of hearing loss. Methods: The 68-item pediatric hearing impairment caregiver experience (PHICE) is a validated questionnaire used to measure stress. The PHICE was administered to 152 caregivers of children with permanent hearing loss. Domain scores were converted into z-scores for analysis of trends of stress over time. Results: Parents of children whose hearing loss was identified more than 60 months ago reported higher stress levels regarding educational aspects of their child’s needs as compared to parents of children with less than 24 months or 24—60 months duration since diagnosis. Parents of children diagnosed with hearing loss within the preceding
* Corresponding author at: Center for Epidemiology and Biostatistics, Cincinnati Children’s Hospital Medical Center, 3333 Burnet Avenue, MLC 5041, Cincinnati, OH 45229, United States. Tel.: +1 513 636 7789; fax: +1 513 636 7509. E-mail address: [email protected] (J. Meinzen-Derr). 1 Co-first authors. 0165-5876/$ — see front matter # 2008 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2008.08.005
24 months reported higher stress levels in the area of healthcare than parents of children diagnosed greater than 24 months ago. Conclusions: Parental stressors change over time with respect to the time of diagnosis of hearing impairment. This phenomenon was observed irrespective of the age of diagnosis of hearing loss. As professionals serving families of children with hearing loss, we should be aware of changing stressors over time and identify the appropriate support services for families to meet those changing needs. By addressing those evolving stressors, the families’ ability to support and improve the outcomes for their children who are deaf or hard of hearing may be enhanced. # 2008 Elsevier Ireland Ltd. All rights reserved.
1. Background The diagnosis of a hearing loss is a critical life event with profound effects on parents and the family system. An extensive body of literature has been published in recent years focusing on stress in parents who have deaf or hard of hearing children [1— 10]. Findings from studies specific to stress levels reported by parents of children with hearing loss have been inconsistent. Some evidence suggests that hearing parents of children who are deaf/hard of hearing (hoh) feel more stress than parents of hearing children [3,8], while some studies report no difference in stress levels between the two groups of parents [4—6]. The types of stress reported by parents of children who are deaf/hoh may be different from that of parents of hearing children and specific to the child’s hearing impairment, such as stress about degree of hearing loss, age of identification [11], language ability [6], and mode of communication [10]. Until we understand the types of stress that burden parents and caregivers of children who are deaf/hoh, we cannot provide the appropriate support services and interventions necessary to strengthen the parent—child relationship critical to a child’s ultimate developmental success. Studies have not investigated the change of stress factors over time, which may vary over the course of a child’s development. Additionally, there are few measures of caregiver stress specific to hearing loss. The pediatric hearing impairment caregiver experience (PHICE) is a measure that was developed and validated specifically on families of children with hearing loss.
1.1. The pediatric hearing impairment caregiver experience Lim et al. developed and validated a questionnaire examining various stressors families may experience related to their child’s hearing loss. The development and validity of the PHICE is detailed in Appendix A. Briefly, the PHICE was created using
an ‘‘expert panel’’ consisting of professionals caring for children who were deaf/hoh and caregivers of children who were deaf/hoh. The 11-member team consisted of specialist doctors from the Departments of Developmental and Behavioral Pediatrics, Psychology, Pediatric Otolaryngology, a medical social worker, a speech rehabilitation therapist, an aural rehabilitation therapist, an audiologist, and program/education directors/principals of the state’s early intervention system and schools for the deaf (both oral and signing). Caregivers that were asked to be involved had children with bilateral hearing loss of any degree or etiology, regardless of caregiver’s hearing status. Semi-structured individual caregiver interviews were conducted and tape-recorded with consent. Detailed shorthand notes were taken simultaneously. Answers to questions were categorized and written summaries were made of categories. Questions were then created from the interviews and tested among additional caregivers of children with hearing loss. The PHICE is a 68-item questionnaire which covers 8 domains of stress. These domains include: communication (10 questions), education (7 questions), emotional well being (11 questions), equipment (3 questions), financial (4 questions), healthcare (14 questions), social (8 questions), and support (11 questions). Parents rate stress on an 8-point Likert scale ranging from no stress to extremely high stress (see Appendix B). Internal reliability, or internal consistency, was determined using Cronbach’s alpha for the 68 items on the PHICE. Cronbach’s alpha is a statistic with a range of 0—1 that is used to determine if a set of questions measure the same idea [12]. The PHICE was found to have an overall reliability of 0.96, which is considered an extremely high coefficient. Test—retest reliability, used to assess the consistency of the PHICE from one time to another is currently ongoing (no data yet available). As stress levels may change over time according to a variety of needs at different life events, we predicted that stress felt by caregivers evolved over time and would be impacted by the duration since the diagnosis of hearing loss.
Pediatric hearing impairment caregiver experience
2. Methods This study utilized a cross-sectional design with data collected from two time points. Between November 2003 and December 2004, 150 PHICE surveys were mailed to caregivers of children who had hearing loss and had been seen within our institution. Between June and August 2007, parents or caregivers of children with permanent hearing loss attending otolaryngology, audiology, or aural rehabilitation appointments at a pediatric tertiary care center were asked to complete the PHICE. Families were eligible if their child had any degree of permanent bilateral sensorineural hearing loss. This study was approved by the Institutional Review Board of our institution and written informed consent was obtained from all caregivers who participated in the study. Demographic information was collected as part of the survey. Child specific information included gender, race, age of identification of hearing loss, etiology of hearing loss, type and degree of hearing loss, type of educational environment the child is currently attending, and any developmental issues. Family specific information included race, marital status, income level, employment status, and educational level of the parents. The 8-point Likert scale on the PHICE was collapsed into a 3-point scale for the purpose of the analysis. Items that represented no stress (not applicable, no stress, very low stress) were given a score of 1, items that represented some stress (low stress, moderate stress) were given a score of 2, and items that represented high stress (high stress, very high stress, extremely high stress) were given a score of 3. Scores were summed in each domain and over the entire survey. Lower scores indicate lower stress relative to higher scores, with a possible range in total scores from 68 points (lowest possible stress) to 204 points (highest possible stress).
2.1. Statistical analysis Continuous variables (i.e., age) were reported as means with standard deviations or medians with ranges. Categorical variables (i.e., gender) were reported as proportions. Comparisons between continuous variables and duration since the diagnosis of hearing loss (as a categorical variable) were tested with ANOVA or Kruskal Wallis test (nonparametric test) as appropriate. Comparisons between categorical variables and duration since the diagnosis of hearing loss (as a categorical variable) were tested using chi-square analysis or Fisher’s Exact test as appropriate. Frequencies of each of the PHICE questions were computed. The rela-
1695 tionships between the individual questions and duration since diagnosis categories were evaluated with chi-square analysis. Domain scores (support, social, emotional, financial, healthcare, equipment, education, and communication) were converted to z-scores for analysis. A standard z-score allows for a better understanding of how many standard deviations above or below the mean an individual’s score was relative to the entire sample. Because each domain had a different minimum and maximum possible score, z-scores were also used to compare the PHICE scores across domains. Mean z-scores were adjusted (Least-Square means reported) for potential confounders and covariates that were found to be significantly associated with parental stress. All statistical analyses were conducted using SAS1 version 9.1 (Cary, NC).
3. Results 3.1. Subject demographics The return rate of the mailed surveys was 48% (72 returned completed surveys). Ninety-eight subjects were asked to complete the PHICE while waiting in clinic; no caregiver declined this study. Five surveys were excluded, as they were filled out twice by the same family and the first survey was kept in the analyses. Five surveys were excluded due to incomplete data on the PHICE. An additional eight subjects were excluded from the analyses due to missing information on the age of the diagnosis of the child’s hearing loss. A total of 152 surveys were included in the final analyses; 62 from the mailed survey group and 90 from the clinic group. Differences between the two study groups (mailed survey group and clinic group) were evaluated. Subjects in the mailed survey group were more likely to be Caucasian (88.7% vs. 45.6%, p = 0.04), to have family members with hearing loss (24.2% vs. 13.3%, p = 0.09), have a stressful event occur within the past 6 months (35.5% vs. 22.2%, p = 0.07), and have severe to profound hearing loss (72.9% vs. 52.3%, p = 0.006). Children in the mailed survey group had their hearing loss identified at significantly younger median [range] ages compared to the clinic group (10 months [0—64] vs. 15 months [0—132], respectively, p = 0.002). The finding may be due to the frequent clinical follow up evaluations of children with previously identified hearing losses resulting in a skewed population that is encountered in any given clinic sample. Of the 152 families who were included in the analyses, 81% were filled out by a mother; 12%
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Table 1 Subject characteristics Subject characteristics Male (%) Race, Caucasian Cause of hearing loss known Age of identification of hearing loss in months Age at time of study in months
Duration since diagnosis (in months) <24, N = 37
24—59.9, N = 45
17 28 20 9
26 38 25 12
(46) (76) (54) (0—120)
(58) (84) (56) (0—132)
60, N = 70 36 57 43 15
p-Value
(51) (81) (61) (0—96)
0.6 0.6 0.7 0.28
157.5 (65—257)
<0.0001
22 (6—130)
56 (24—187)
Degree of HL Severe to profound Moderate to severe Mild
19 (51) 14 (38) 4 (11)
29 (66) 11 (25) 4 (9)
41 (62) 23 (35) 2 (3)
0.95
Type of device used None Hearing aids Cochlear implant
3 (8) 24 (65) 10 (27)
2 (4) 21 (47) 22 (49)
12 (17) 33 (47) 25 (36)
0.07
Communication mode Speech Speech + sign Sign Other
21 9 8 8
(32) (24) (22) (22)
25 5 10 5
(56) (11) (22) (11)
31 23 14 2
(44) (33) (20) (3)
Education Fully mainstreamed Deaf education (oral or sign) Home or no school Daycare/preschool Other
4 4 17 9 3
(11) (11) (46) (24) (8)
12 14 5 4 10
(27) (31) (11) (9) (22)
28 31 9 0 5
(40) (44) (9)
a
Significant medical problems in the last 6 months
11 (30)
8 (18)
0.01
<0.0001
(7)
15 (21)
0.42
Caregiver characteristics Caregiver age in years Marital status–—married
31 (22—49) 27 (73)
35.5 (20—56) 35 (78)
41 (23—70) 47 (67)
Highest education Completed college/post-grad Some college HS grad Some HS
16 10 8 3
(43) (27) (22) (8)
24 10 10 1
(53) (22) (22) (2)
34 20 10 6
(49) (29) (14) (8)
7 7 8 4 9
(20) (20) (23) (11) (25)
9 6 5 6 16
(21) (14) (12) (14) (38)
12 12 12 8 19
(19) (19) (19) (13) (30)
0.93
29 (41) 17 (24) 22 (31)
0.99 0.10 0.55
Income level <$20,000 $20—40,000 $40—60,000 $60—80,000 >$80,000 Full time employment outside the home Other family members with hearing loss Stressful events in the past 6 months
15 (41) 6 (16) 8 (22)
19 (42) 4 (9) 12 (27)
Reported as N (%) for categorical variables and medians (ranges) for continuous variables. a Missing information on degree of hearing loss in 5 subjects.
<0.0001 0.46
0.68
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Table 2 Top 10 stressors reported among caregivers of children with hearing loss Item on PHICE
N (%) reporting high levels of stress
N (%) reporting low stress
Worries about my child’s safety Seeing my child frustrated because of his/her hearing loss Feeling uncertain about my child’s future Worries about others taking advantage of my child Costly medical equipment and expenses Worries about not doing enough for my child Having no time for myself Worries about my child losing or damaging his/her equipment My lack of signing skills Lack of educational opportunities for my child
39 36 35 31 31 31 30 24 23 21
77 68 74 70 57 78 51 80 58 49
were fathers, 5% were grandparents, 1% were foster parents, and in 1% both father and mother completed the survey. Ninety-four percent of caregivers were hearing. The median (range) age of child at time of study was 78.5 (6—257) months while the median (range) age of child at time of hearing loss diagnosis was 55.5 (3—227) months. The median (range) age of the caregiver was 37 (20—70) years. The majority of children (95%, n = 142) had a bilateral hearing loss. Regarding duration since the diagnosis of hearing loss, 24% (n = 37) had a child who had been diagnosed in the last 23 months, 30% (n = 45) of families had a child who was diagnosed 24—60 months ago, and 46% (n = 70) had a child diagnosed greater than 60 months prior to the survey. Table 1 shows differences across duration since diagnosis categories for both child and caregiver characteristics. No differences were seen with respect to gender or race among the three groups. The degree of hearing loss was severe to profound in over 50% in each group. Children who had been diagnosed with their hearing loss for 24 months were more likely to report speech as their primary mode of communication than children who had been diagnosed <24 months ago ( p = 0.01).
(26) (24) (23) (21) (21) (21) (20) (16) (15) (14)
(51) (45) (49) (46) (38) (52) (34) (53) (38) (32)
3.2. Caregiver stress Over 20% of all caregivers reported high levels of stress regarding worrying about child safety, seeing their child frustrated, uncertainty about their child’s future, worrying about others taking advantage of their child, and having no time for one’s self (Table 2). The duration since the hearing loss diagnosis was correlated with the domain scores using Pearson’s correlation coefficient. Duration since diagnosis was significantly positively correlated with the education (rho = 0.25, p = 0.002) and social domain (rho = 0.20, p = 0.02). Duration was significantly negatively correlated with the emotional domain (rho = 0.16, p = 0.046) indicating that the longer the time since the diagnosis of their child’s hearing loss, the stress related to emotional well being decreased. Differences in mean domain scores across the three groups were assessed using ANOVA. Significant increases in scores ( p 0.05) were seen with increasing duration since identification regarding the education domain (7 questions) and the social domain (8 questions) (Table 3). To better understand how stress evolves while families gain experience with their child’s hearing
Table 3 Unadjusted domain scores across groups Domain scores
<24, N = 37
24—59.9, N = 45
60, N = 70
Total possible range in scores
Communication Education * Emotional well being Equipment Financial Healthcare Social ** Support
14.9 9.3 17.3 4.9 6.4 20.6 12.6 14.7
15.1 10.9 16.5 1.8 5.9 19.9 13.6 15.2
14.5 11.6 16.1 4.8 6.5 19.7 14.4 15.2
10—30 7—21 11—33 3—9 4—12 14—42 8—24 11—33
* **
(3.6) (2.3) (4.2) (1.4) (1.7) (4.4) (3.3) (3.2)
p = 0.0008 for comparison among groups. p = 0.047 for comparison among groups.
(4) (3.1) (4.5) (1.3) (2) (4.9) (3.4) (4.1)
(3.8) (3.2) (4.3) (1.7) (2.2) (5.5) (3.8) (4.3)
1698 loss, domain z-scores were adjusted for confounders that may influence the relationship between stress and duration since diagnosis. Potential confounders included caregiver education, hearing loss severity, communication strategy, stressful events in past 6 months, additional medical problems in past 6 months, and current hearing aid or cochlear implant use. Certain models were also adjusted for race, age at diagnosis, and child’s current age if these were significant in the models. Adjusted mean scores that were significantly different across groups are illustrated in Fig. 1. Stress related to education, equipment, and support was at its lowest for caregivers of children diagnosed in the last 24 months. Stress related to healthcare and emotional well being was at its highest for this same group of caregivers. No significant differences were seen among groups regarding stress related to the social domain ( p = 0.35) and finances ( p = 0.26). Total stress showed a slight increase over time (from a z-score of 0.51 among <24 month group to 0.90 among the 60 month group), though this increase was not statistically significant ( p = 0.32). Although the main analyses focused on three duration categories, domain adjusted z-scores were plotted as spline curves over multiple age categories to illustrate a broader sense of change over a wider range of duration since diagnosis (Figs. 2 and 3). Consistent with earlier findings of increased stress over the three time since diagnosis groups, a significant increase in stress related to the areas of education ( p = 0.002) and support ( p = 0.008) was noted over time (Fig. 2). The association between duration since diagnosis and stress related to equipment, although statistically significant, did not appear to be linear. Duration 73—108 months and
J. Meinzen-Derr et al.
Fig. 2 Adjusted mean z-scores over time since the diagnosis of hearing loss was made for the domains of education, equipment, and support.
>156 months had the highest mean levels of stress, which were both significantly ( p < 0.05) different than the other duration groups. Stress related to healthcare and emotional well being appeared to decrease with time (Fig. 3) although these findings did not meet statistical significance ( p = 0.2 and p = 0.09, respectively). Stress related to communication remained relatively high across all duration categories, with only a slight decrease over time ( p = 0.08). Although these findings were only considered as marginally significant trends, they present a shift in the types of stress felt by caregivers as they gain experience with their child’s hearing loss.
4. Discussion The findings from this cross-sectional study provide an initial effort to address how stress may change for
Fig. 1 Mean z-scores, adjusted for confounders which included caregiver education, HL severity, communication strategy, and other stressful events. Numbers along bottom represent p-values for the comparison of the three groups.
Fig. 3 Adjusted mean z-scores over time since the diagnosis of hearing loss was made for the domains of healthcare, communication, and emotional well being.
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families of children with hearing loss depending on the time since identification of hearing loss. Families of children recently identified with hearing loss (within 24 months of the study) reported higher levels of stress in the area of healthcare (interacting with healthcare systems and/or physicians) and emotional well being. Families of children with a longer duration of experience adjusting to their child’s hearing loss had more concerns in the area of education and support systems. Although total stress was shown to slightly increase over duration groups, this finding did not represent the fluctuations in types of stress that was felt by the cohort of caregivers. Studies that have compared parental stress between parents with deaf/hoh children to normative samples of parents with hearing children have not been consistent with their findings. Pipp-Siegal et al., suggested that stress levels among mothers of children who are deaf or hard of hearing are not clinically higher according to the Parental Stress Index (PSI) than a normative sample of mothers of hearing children [6]. However, 13% of the study mothers had stress scores above the clinical cutoff level (>90th percentile). This finding is similar to that reported by Lederberg and Golbach, who reported that 9—13% of mothers of 3—4-year-old children who were deaf were above the clinical cutoff on the PSI [3]. They also reported differences in maternal stress between mothers of deaf/hoh children and mother of hearing children only among children who were 2 years of age, but not mothers of 3 or 4 year olds. Meadow-Orlans reported that 25% of mothers of children who were deaf/hoh had scores that were considered clinically high [4], though no differences were seen between the study group and the normative sample on the PSI. These types of observations and our clinical experience served as an impetus to develop, validate and utilize the PHICE in order to have an instrument that would be relevant to this specific patient population and potentially help guide family-centered care. Many studies on stress felt by parents of children who are deaf/hoh have a focused attention on hearing aid fitting or the decision regarding cochlear implants. Spahn et al. surveyed 57 mothers and 46 fathers of 57 children with cochlear implants [9]. Using the Symptom Checklist-90-R (SCL-90-R) to assess stress, investigators reported that 25% of mothers and 25% of fathers had high levels of psychic stress. Quittner et al. studied stress using the SCL90-R in children 24 months of age with cochlear implants [8]. Using the normative sample on the SCL-90-R for comparison, high stress was highly correlated with increased depression and anxiety in mothers of children with cochlear implants. Bur-
ger et al. compared the change in stress levels over time between parents of children with cochlear implants and those hearing aids [13]. Their findings suggested that parents of children with cochlear implants had a slight, though non-statistically significant reduction in stress over time (between preand post-implant) compared to parents of children with hearing aids. Investigators also noted that the time point immediately after diagnosis for both groups was perceived as the most stressful for families. Age at the onset of deafness has been shown to be related to maternal stress. In a study of 42 mothers of children who were deaf, stress was higher if the child’s deafness was identified prior to 18 months of age [11]. Interestingly, the child’s chronological age was not related to the overall stress score, though mothers of ‘‘younger’’ children had higher mean stress scores than mothers of ‘‘older’’ children. It was difficult, however, to determine if there were any modifying effects regarding the time of identification and the study age of the child. In other words, the study did not discuss how duration since the diagnosis affected maternal stress levels. Findings from the current study suggest that the duration since the diagnosis of the hearing loss played a significant role in caregiver stress, even after controlling for the age of the child at the time of the study. The initial grieving process of many caregivers upon the diagnosis may create or add to stress. Over time, caregivers gain both knowledge and experience with hearing loss, which could reduce certain aspects of stress. Results from the current study indicate that while certain types of stress diminished over time, others appeared to increase. Stress surrounding educational concerns naturally increased over the duration since diagnosis, as children began to enter the school system. Stress around equipment needs and support also increased with time following diagnosis of hearing loss. It is possible that as parents gain more understanding of the needs of their child who is deaf/hoh, certain specific equipment (e.g., vibrating alarms) become important. In contrast, stress around healthcare issues decreased with increasing duration. The initial high levels may have been due to the initial stress felt by caregivers regarding number of clinical visits, initiation into therapies, and also comfort level with clinicians. As families become familiar with aspects of clinical care, the stress in this domain decreases. This may indeed be a common trend that crosses any medical condition and the family interactions with the healthcare system. Interestingly, stress focused on communication remained high across groups (even upon controlling for primary communication
1700 mode), though it was slightly lower for the 60 month duration group. This finding speaks to a common theme felt by many caregivers regarding the impact of deafness on communication and the subsequent influence this has on family. A longitudinal study, following caregivers over time, will allow us to evaluate what components of stress are altered over time. It is interesting to note that a finding during the development of the PHICE was that caregivers who were deaf/hoh themselves and utilized American Sign Language as their primary language had the least stress of all caregivers. The majority of caregivers in the current study, however, were hearing. Studies on parental stress have not uniformly reported characteristics that are specific to a child who is deaf/hoh, such as primary mode of communication, age of hearing loss identification, type of school or intervention a child may be receiving. Yet, stress of caregivers of deaf/hoh children involves a complex model of demographic characteristics, characteristics of perceptions of the caregiver (e.g., education, social support), and factors related to child’s hearing loss (e.g., degree of hearing loss, age of identification, language ability) [2,6]. In addition, most studies do not address the different areas stress may be felt by caregivers, but rather classify people as either feeling high or low levels of stress. The current study was able to take into account such factors that are related to a child who is deaf/hoh. In addition, a major strength of the current study is the use of stress-specific domains: communication, education, emotional well being, equipment, financial, healthcare, social, and support. By categorizing stress reported by the caregiver, clinicians are able to identify potential areas of concern and address the needs of the family in terms of interventions and support mechanisms. This study examined the effect duration since the identification of the hearing loss has on caregiver stress. Longitudinal research should be employed to effectively assess how this stress changes over time within families. The current study utilized a fairly new tool for assessing caregiver stress. Although other widely accepted tools are available for measuring stress (i.e., Parental Stress Index), these tools do not have questions that are pertinent to how a caregiver may feel about the specific relevant needs of their deaf/hoh child. Investigators have taken the steps to ensure that the PHICE represented this group of parents and professionals were included in the development of the questionnaire, to create a valid tool (Appendix A). Test—retest reliability is currently ongoing and a longitudinal study would further validate changes in stress over time. Dr. L. Lim is
J. Meinzen-Derr et al. currently completing a similar study in a Singapore population of caregivers of children with hearing impairment. The goal of the study is to evaluate the validity of the questionnaire in a different cultural and ethnic context. Many factors play a role in parental stress and it is important to realize that stress is not necessarily static. By understanding how stressors change with experience with hearing loss, we can better anticipate areas of need and support for the families we serve, with the goal of improving child outcomes.
Appendix A. Development of the PHICE This study was approved by the Institutional Review Board, and informed consents were obtained. An ‘‘expert panel’’ consisting of professionals caring for children who were deaf/hoh and their caregivers was assembled. The 11-member team consisted of specialist doctors from the Divisions of Developmental and Behavioral Pediatrics, Psychology, Pediatric Otolaryngology, a medical social worker, a speech rehabilitation therapist, an aural rehabilitation therapist, an audiologist, and program/education directors/principals of the state’s early intervention system, an oral deaf school program, and a school for the deaf. The early intervention program cares for families of infants with newly diagnosed hearing loss. The majority of children at the school for the deaf utilize a total communication approach with signing, oral and lip-reading when necessary. The expert panel was interviewed in face-to-face personal interviews to help identify issues which were possible stressors for caregivers of children with hearing loss. Questions for a semi-structured interview were developed from this information along with review of the literature. Stress was defined as ‘‘specific external and/or internal demands that are appraised as taxing or exceeding the resources of the person’’ [1]. A stressor is ‘‘any stimulus that makes demands upon an organism requiring adaptation or adjustment’’ [2]. Primary caregiver was defined as the person who provided the most care to the child in terms of time, including a parent, relative, teacher or care-taker. Caregivers participating were of children from the state’s early intervention program, the school for the deaf, or the oral deaf school program. Primary caregivers were included in the study if they were (1) caregivers of children with bilateral hearing loss of any degree, etiology, and type (conductive, sensorineural or mixed); (2) any race or gender; (3) with
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hearing or with a hearing loss. Caregivers were excluded if their child had significant medical conditions causing significantly more stress than related to hearing loss, such that the caregiver cannot differentiate between the causes of the stress. The caregivers were a highly heterogeneous group with varied social income, race, child’s duration and degree of hearing loss severity, child’s intervention status (non-aided/HA/CI). As such, ‘‘information-rich’’ caregivers were identified to ensure that all possible issues causing stress were considered. As the interpretation developed, caregivers were selected to further enrich, confirm and disconfirm our understandings [3]. The number of caregivers was sampled to the point of saturation, with no new information identified [4]. Semi-structured individual caregiver interviews using the interview guide questions were then conducted in private areas at the schools or the hospital. Due to the heterogeneity of caregivers, individual interviews were conducted rather than focus groups so that information would be more accurate and forthcoming. The trained interviewers were the principal investigator (Lim) and two psychology graduate students. Interviews were tape-recorded with consent, and detailed shorthand notes were taken simultaneously. Interviewers sat into the meetings of other interviewers for internal monitoring of quality. Qualitative research methodology was used to interpret the data, emphasizing the iterative processes of describing, organizing, connecting, corroborating and legitimating [5]. The organizational style used was the ‘‘editing’’ style in the first rounds of readings where the plan was to explore, without prior preconceptions of the topic. Segments of the text were then organized into categories. The ‘‘template’’ organizing style was used later when transcripts were reread. Many revisions and re-categorizations were made as new understandings evolved during the review. The interpretive process was terminated when there was no new information
encountered. In the final presentation, the caregiver issues were categorized for presentation to a clinical audience. The three interviewers and two additional researchers familiar with qualitative questionnaire studies independently read all interview transcripts. Written summaries were made of categories and the issues involved in each. The researchers then met and developed preliminary interpretations and meanings. Following these initial discussions, the researchers individually developed categories of stress. Thereafter, a consensus of main issue and themes was reached. Questions were then developed so for development of a quantitative questionnaire. This was then pre-tested on caregivers to verify that the caregivers understood the questions. The majority of the PHICE development focused on content validity, which consists of a judgment by ‘‘experts’’ whether the tool appears appropriate for the measurement of stress among caregivers of children who are deaf/hoh [6,7]. Because no ‘‘gold standard’’ measure exists for quantifying stress specifically related to having a child who is deaf/hoh, criterion validity, the correlation of a scale with some other measure of a gold standard, is difficult to establish [7].
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Appendix B. Parent/Caregiver Questionnaire The following is a list of possible issues face by parents or caregivers of children with a hearing impairment. Please think about the past 6 months and tell us what level of stress related to each issue, if any, you experienced as a result of caring for your child with a hearing impairment. If an issue does not pertain to you or your child, please circle ‘‘not applicable’’. If you have more than one child with hearing impairment, please think of your older child when answering this questionnaire
Please indicate the level of stress, if any, you have experienced with each issue during the past 6 months
Not applicable
No stress
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Child has temper tantrums related to hearing loss. Child care provider/schools/teachers/ out-of-home caregiver’s lack of signing skills. Working together with education professionals. Worries about my child losing or damaging his/her equipment. Being unable to go to work. Having difficulties accepting that my child is hearing impaired.
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7. 8. 9. 10. 11. 12. 13. 14. 15. 16. 17. 18. 19. 20. 21. 22. 23. 24. 25 26. 27. 28. 29. 30. 31. 32. 33. 34. 35. 36. 37. 38. 39. 40. 41. 42. 43. 44. 45. 46. 47. 48. 49.
Please indicate the level of stress, if any, you have experienced with each issue during the past 6 months
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Hearing aids or cochlear implants not meeting expectations. Delayed cochlear implantation. Feeling that healthcare providers do not listen to me. Inadequate support or understanding from an employer. Child uses hearing loss as an excuse to behave badly. Feeling uncertain about my child’s future. Sound of child’s voice is irritating, loud or shrill. Distinguishing between behavior and communicating problems. Obtaining special learning materials for my child (e.g., books, captioned videos). Equipment interfering with child’s activities. Worries about not doing enough for my child. Not being able to attend to the needs of other family members. Worries that my child is getting good medical care. Working together with doctors. Worries about deaf community’s perceptions of my decisions for my child. Lack of interpreters for child. Worries about others taking advantage of my child. Worries about my child’s safety. My lack of signing skills. Worries about choosing the best form of communication for child (oral, sign, total) Worries about decisions I have made for my child’s education. Inadequate insurance coverage for treatment of hearing impairment. Feeling partly responsible for my child’s hearing impairment Feeling uncertain about disciplining my child. Traveling to medical appointments. Feeling confused about medical information. Not able to take time off from work to tend to child’s needs. Lack of childcare or babysitters for child. Worries about my child’s ability to make friends. My child’s lack of signing skills. Difficulty communicating with my child. Child’s behavior in school. Problems dealing with insurance company. Tired due to caring for my child with hearing impairment. Change in my relationship with my partner or spouse. Keeping large number of medical appointments. Working with the nursing or rehabilitation team. Understanding the laws related to hearing impairment. Difficulty getting information about hearing impairment or rehabilitation. Worries about how my child fits into the hearing community. Health care provider’s lack of signing skills. Child’s academic achievement in school. Lack of educational opportunities for my child.
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50. 51. 52. 53. 54. 55. 56. 57. 58. 59. 60. 61. 62. 63.
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Please indicate the level of stress, if any, you have experienced with each issue during the past 6 months
Not applicable
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Very low stress
Low stress
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High stress
Very high stress
Extremely high stress
Costly medical equipment and expenses. Having no time for myself. Overwhelmed by information on caring for a child with hearing impairment. Difficulty getting medical and rehabilitation care. Worries about whether my child should have a hearing aid, cochlear implant, or no aid. Inadequate support or understanding from spouse or partner. Lack of support network of parents facing similar problems. Problems discussing sexual issues with my child. Immediate family and relative’s lack of signing skills. Child’s reading ability. Caring for the hearing aid or implant. Costly daily aids like vibrating alarms and TTY services. Relocation to be near resources that meet my child’s special needs. Worries about risks associated with medical treatment (e.g., surgery, hearing equipment). Knowing that my child was diagnosed with hearing impairment late. Worrying about success of my child’s therapy or rehabilitation. Inadequate support or understanding from friends and relatives. Lack of a large deaf community. Seeing my child frustrated because of his/her hearing loss.
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References [1] M. Hintermair, Sense of coherence: a relevant resource in the coping process of mothers of deaf and hard-of-hearing children? J. Deaf Stud. Deaf Educ. 9 (2004) 15—26. [2] M. Hintermair, Parental resources, parental stress, and socioemotional development of deaf and hard of hearing children, J. Deaf Stud. Deaf Educ. 11 (2006) 493—513. [3] A.R. Lederberg, T. Golbach, Parenting stress and social support in hearing mothers of deaf and hearing children: a longitudinal study, J. Deaf Stud. Deaf Educ. 7 (2002) 330— 345. [4] K. Meadow-Orlans, Stress, support, and deafness: perceptions of infants’ mothers and fathers, J. Early Intervent. 18 (1994) 91—102. [5] K.P. Meadow-Orlans, S. Dyssegaard, B. Smith-Gray, Infants who are deaf or hard of hearing, with and without physical/ cognitive disabilities, Am. Ann. Deaf 140 (1995) 279—286. [6] S. Pipp-Siegel, A.L. Yoshinaga-Itano, C. Sedey, Predictors of parental stress in mothers of young children with hearing loss, J. Deaf Stud. Deaf Educ. 7 (2002) 1—17.
[7] A.L. Quittner, R.L. Jackson, D.N. Glueckauf, Chronic parenting stress: moderating versus mediating effects of social support, J. Pers. Soc. Psychol. 59 (1990) 1266—1278. [8] A.L. Quittner, J.T. Rouiller, R.L. Steck, Cochlear implants in children: a study of parental stress and adjustment, Am. J. Otol. 12 (Suppl.) (1991) 95—104. [9] C. Spahn, B. Richter, I. Zschocke, E Wirsching, M. Lohle, The need for psychosocial support in parents with cochlear implanted children, Int. J. Pediatr. Otorhinolaryngol. 57 (2001) 45—53. [10] M.T. Greenberg, Family stress and child competence: the effects of early intervention for families with deaf infants, Am. Ann. Deaf 128 (1983) 407—417. [11] M.M. Konstantareas, V. Lampropoulou, Stress in Greek mothers with deaf children. Effects of child characteristics, family resources and cognitive set, Am. Ann. Deaf 140 (1995) 264—270. [12] L.J. Cronbach, Coefficient alpha and the internal structure of tests, Psychometrika 16 (1951) 297—334. [13] T. Burger, C. Spahn, B. Richter, S. Eissele, E. Lohle, J. Bengel, Parental distress: the initial phase of hearing aid and cochlear implant fitting, Am. Ann. Deaf 150 (2005) 5—10.
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