Eligibility criteria

The population, concepts, and context approach were referred to when developing the inclusion criteria to address the research questions [41, 42]. First, for the type of participants (population), although the studies focused on parents of DHH children, in terms of early intervention, the inclusion criteria would be for children falling within the chronological age range of newborn to preschool. Specifically, parents whose children’s chronological ages ranged from 0 to 6 years would be included. Additionally, parents whose children underwent CI surgery before 6 years old, regardless of their current chronological age, would be included. Studies including children with prelingual or late-onset deafness, hearing aid users, CI users, or CI candidates were included. Second, the concept may comprise details pertaining to instruments that evaluate “parental emotional status,” and/or “parental ecological support systems,” and/or “parental quality of life.” Lastly, the context must be English-language-only peer-reviewed publications with no date restrictions, and the articles discussed the PES and/or parental support systems and/or parental quality of life among parents of DHH children using validated instruments.

Types of evidence sources

This study included only peer-reviewed articles that were written in English with no date restrictions. Furthermore, pre-prints, pilot studies, case studies, protocol articles, proceeding articles, literature review articles, systematic reviews, or scoping reviews were not included in this study. Qualitative studies that do not use validated instruments were excluded from the present study. Therefore, only studies that used validated instruments and had parents of DHH children, whose chronological age ranged from 0 to 6 years or had undergone CI surgery between 0 and 6 years old were included. More specifically, articles that relate to parents of preschoolers/toddlers/infants who have hearing loss (either congenital or late-onset hearing loss) were included in this study.

Information sources

To discover articles pertinent to the research question, this study used Medline (via OVID and EMBSCO), Scopus, and Web of Science (WOS). Before entering the article title and abstract screens, articles that discussed adults and/or did not include hearing loss were excluded. The bibliographic databases were searched in March 2023.

Search

Medical subject headings and keywords for the search were covered in the following three key domains:

1. Population: parents of DHH children.
2. Measurement: questionnaire, scale, survey, inventory, or instrument.
3. Concept: quality of life, emotional state of parents, or ESS.

The full electronic search strategy comprised (parent* OR father* OR mother* OR caregiver* OR famil*) AND (children with hearing loss OR children with hearing impairment OR deaf children OR children with deaf or hard of hearing) AND (scale* OR test* OR questionnaire* OR assessment* OR measure* OR inventor* OR instrument*) AND (parent emotion* OR parental emotional readiness OR parental support OR parental support system* OR parental ecological support system* OR parental ecological support OR quality of life). Although the input of Boolean and search terms varies from database to database, it will be adjusted according to the system’s interface and input method; however, the relevant Boolean and keyword searches are clearly linked to the three key domains.

Selection of sources of evidence

The first author performed a literature search for potentially relevant publications measuring PES and ESS among children with hearing loss. Before the first and second authors separately assessed the titles and abstracts of the original articles (proportionate agreement = 0.82), the first author screened and excluded non-hearing loss-related articles after deleting duplicate publications. Extracted studies that used validated instruments for evaluating parental outcomes (either PES or ESS) among DHH children who had their hearing devices at the age of 0–6 years regardless of whether the hearing device was either bilateral hearing aids, bilateral CI, or bimodal were included, and subsequent full-text data extraction finalized the application of the inclusion criteria. During the full-text review phase, the fourth author crosschecked approximately 20% of the articles (proportionate agreement = 0.84). Disagreements over eligibility were addressed by obtaining a third opinion from the third author in both the title and abstract screening and full-text review phases (Fig 1). The review process was performed using the Covidence software during the title and abstract screening and full-text reviews, whereas Microsoft Excel was used during the data extraction phase. Covidence provides the ideal user experience for researchers conducting a systematic/scoping review during the title and abstract screening phase [43].

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Fig 1. Flowchart of the article review process (PRISMA-ScR).

https://doi.org/10.1371/journal.pone.0305748.g001

Data charting

The present database inquiry was conducted between February and March 2023. The first author developed the data-charting table (in Microsoft Excel) for use in the full-text review section and sent it to the second and third authors for additional review and correction while checking the title and abstract. Once the data extraction table’s initial iteration was completed, the second and third authors revised it as necessary.

Data items

To characterize the data from the included articles, a set of variables was defined prior to their extraction for this study. After the second and third authors reviewed the data-charting table, the extracted information was inserted into the table according to the variables listed in the Microsoft Excel sheet. Author(s) and publication year, research design, participant characteristics number of participants, gender of the parents and children, and age-related variables (including child’s age of deafness diagnosis, chronological age, and age at cochlear implantation), child’s device setting, objective(s) of the study, instrument(s) used, and outcomes were listed as variables. This information would summarize existing instruments for evaluating PES and ESS among parents of DHH children.

Synthesis of results

The data and findings from the included studies are integrated and presented in Table 1.

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Table 1. Characteristics of the included studies on parental emotional status and ecological support systems among parents of DHH children.

https://doi.org/10.1371/journal.pone.0305748.t001

Selection of sources of evidence

Of the 3,060 articles retrieved from the databases (Medline OVID [685] and EMBSCO [1,631], Scopus [59], and WOS [685]), 2,680 were excluded owing to duplication of articles and articles’ subjects, abstracts, and/or title headings. Next, after reviewing 139 full-text articles, 117 articles were excluded for the following reasons: the age of the DHH children, either chronological or age at implantation, was not within 0–6 years, regardless of whether the hearing device was either bilateral hearing aids, bilateral CI, unilateral CI or bimodal (n = 51; 43.59%); did not contain validated instrument (n = 23; 19.66%); not related to the present study’s aims (e.g., outcomes were focused on children rather than parents, and outcomes were not related to PES or ESS; n = 30; 25.64%); the types of research designs did not conform to the study’s inclusion criteria (mentioned in the methodology; n = 7; 5.98%); the articles were not written in English (n = 4; 3.42%); did not provide child’s age-related information (n = 1; 0.85%); and the full-text cannot be accessed (n = 1; 0.85%). Overall, 22 articles met the inclusion criteria (Fig 1).

Characteristics of the sources of evidence

The included articles were published from 2007 to 2022, and approximately half (10 articles) of them were published within five years (since 2019). In addition, most studies had cross-sectional designs (n = 14; 63.63%), whereas case-control study designs (n = 5; 22.73%) and cohort study designs (n = 3; 13.63%) comprised the minority. Of the included studies, four (18.18%) included parents of hearing children as the control group [45, 49, 59, 64]. Furthermore, one study that administered a qualitative instrument was included in the present study because it used a validated instrument and simultaneously interpreted the findings from both qualitative and quantitative approaches, which met the present study’s inclusion criteria [56]. Moreover, nine (40.9%) of the included studies focused on parents of children with at least a CI only, whereas only one study focused on parents of children who are CI candidates only. Moreover, six (27.3%) and two (9.1%) of the included studies comprised parents of children with different types of hearing devices and HA users only, respectively.

Of the parents of DHH children in the included studies, mothers represented approximately 54.4% (n = 947), whereas fathers represented only 25.4% (n = 443). Some studies referred to the responders as parents (n = 347; 19.9%) rather than specifying them as either fathers or mothers; therefore, the number did not count into either mothers or fathers [47, 49, 55, 59]. Moreover, among the study population, a portion of the children who experienced hearing loss were born to mothers (n = 3), fathers (n = 14), or parents (n = 5) with a history of hearing loss [49–51, 53]. Notably, a small percentage of grandparents participated in the study (n = 3; 0.2%), refer to Table 1 for details.

Results of individual sources of evidence

Of the included studies, approximately 22 and 10 evaluated PES and ESS among parents of DHH children, respectively. Among them, different questionnaires were identified and targeted various related concepts (Table 2), including PES (number of instruments = 23) and ESS (number of instruments = 12). Except for the Clarke Modification of the Holroyd Questionnaire on Resources and Stress (Clarke QRS; 0.68), the Parental Involvement on Child Care Index (range 0.67–0.68), and the social relationships domain (0.66) of the World Health Organisation Quality of Life Questionnaire–Short Form (WHOQOL-BREF), the majority of the instruments demonstrated acceptable (> 0.70) to good (> 0.80) internal consistency reliability (Table 2). Moreover, only a few condition-specific instruments were developed and used for evaluating either PES or ESS among parents of children with CI, that is, the Parental Attitudes of Various Aspects of Cochlear Implantation (PAVACI) questionnaire [64] and Parenting Stress-CI modules-early childhood (EC) version [47] for PES, and Children with Cochlear Implants: Parental Perspectives Questionnaire (CCIPP; [65–69]) for both PES and ESS.

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Table 2. Outcomes and instruments used related to parental emotional status and ecological support systems among the included studies.

https://doi.org/10.1371/journal.pone.0305748.t002

Summary of evidence

This scoping review ascertained suitable instruments for evaluating PES and ESS within the clinical context while also consolidating pivotal factors impacting PES and ESS in the context of early intervention for children with DHH children, especially for parents of children undergoing the CI journey. The current shortage of instruments adapted for parents of DHH children is evidenced by the limited number of studies included in this scoping review. Although numerous assessments are accessible to parents of children with CI, acknowledging that these instruments frequently incorporate challenging components that are mainly intended for post-CI only is needed.

Why does evaluating parental emotional status and ecological support systems matter?

Seven (31.8%) of the articles included in the present analyses exclusively recruited individuals of a single gender, who were fathers only (n = 2; [53, 57]) and mothers only (n = 5; [24, 44, 56, 61, 63]). The representative participants’ distribution was unsurprising as mothers were frequently observed to be the child’s caregivers, and the fathers were frequently observed as breadwinners. Fathers showed low involvement with their children, considering that they were mostly invisible in the intervention sessions [29]. A study by Hintermair et al. [53] demonstrated that almost 76% of fathers were rarely involved in their child’s early intervention sessions with 15% of whom were not involved at all. Although studies have shown no significant correlation between fathers’ working hours and frequency of involvement in a child’s intervention [50, 53], most of the fathers mentioned that the main reason for being absent in their child’s intervention was the conflicting time schedule between the early intervention and their working hours [53]. However, fathers are no longer regarded as outsiders but as participants in child rearing, and their presence significantly impacts their child’s development [57, 73].

Hintermair et al. [53] reported that although fathers’ involvement may have been absent during the early intervention session, they remained a visible and integral part of the family-centered intervention framework. One of the key components of early intervention remains to be the engaged participation of family members [25]. Family member involvement plays a key role in family-centered interventions, whether it applies to selecting educational interventions for the child or making decisions regarding hearing interventions. This involvement includes choices such as total communication, sign language, or auditory–verbal interventions for educational interventions, and the selection of hearing aids or CIs for hearing interventions. The decision about CI surgery for children, particularly those who may benefit from it, frequently relies upon a thorough assessment conducted by professionals and adherence to certain implantation criteria. Nevertheless, the final decision about the pursuit of implantation often depends on the parents.

Further, several included studies indicated that PES significantly correlated with parental involvement in intervention sessions among parents of DHH children [28, 47, 52] and positively associated with their marital quality [29, 57]. In other words, parents with emotional stability, including low depression or stress levels, tend to engage more in their child’s intervention sessions, and providing emotional and behavioral support from the spouse would enhance their satisfaction with the marital relationship. A supportive spouse who not only participated in intervention sessions but also offered emotional support would establish an ideal home setting for children, thereby promoting their overall growth and progress. These study results may help researchers identify the indicators that may affect parents’ PES and ESS, which can be used as variables in future research to explore the causal relationship among parental involvement, marital relationship, and PES and ESS.

Emotionally stable parents receiving sufficient ESS during their child’s intervention journey can positively affect their child’s speech and hearing development [62]. However, this intervention journey is not a short-term effort but requires a long-term commitment from the parents. Committing to interventions that require constant involvement may be challenging for parents who do not have adequate ESS and stable PES [102, 103]. In addition, parents juggle multiple roles during the intervention journey, including being the child’s mother/father, teacher, case monitor, speech therapist, and driver, and must attend various interventional-related appointments that may cause exhaustion [103]. Negative emotions such as being overwhelmed, depressed, regretful, or helpless may emerge when a child fails to meet expectations or when the parents are blamed for missed appointments because of work schedules [30, 103]. These negative emotions can make it challenging for healthcare professionals to recommend appropriate interventions. Therefore, parents’ emotional preparedness and the adequacy of their ESS for their children’s intervention journey, particularly the CI journey, must be understood. The following section examines the relevant instruments for PES and ESS assessment in a clinical setting.

Instruments for evaluating parental emotional status and ecological support systems evaluation among the included studies

The present review includes 22 studies, with 23 and 12 validated instruments used for evaluating PES and ESS, respectively, among parents of DHH children. Although several established validating instruments exist for evaluating PES and ESS in parents of DHH children, the available instruments mostly comprise generic measures that have been validated using evidence-based approaches, including the PSI (either short form [76] or full version [89]), WHOQOL-BREF [96], DASS [83], MSPSS [78], and others (Table 2; [26, 72, 73, 75, 80–83, 85–88, 90–95, 97–101]). These instruments can be employed for assessing either PES or ESS. However, generic methods possess the inherent capacity to exhibit reduced sensitivity or yield inconsistent outcomes when employed to assess certain populations or circumstances, particularly in PES [44, 49, 60]. As evidenced by the findings of this scoping review, no discernible disparity in parental stress levels was noted between parents of DHH children and parents of hearing children [49]. However, parents of children with CI experienced higher stress levels than parents of hearing population [59, 62, 64]. Nevertheless, upon closer examination of parental stress levels, the stress indices of these parents (both pre- and post-CI) remained within the fair range [44, 56, 60]. Notably, among the abovementioned studies, only two studies used condition-specific instruments for evaluating parental stress levels [59, 60]. Therefore, the observed disparities that appear substantial lack substantive significance.

Conversely, an interesting finding was observed regarding ESS. Parents of children with moderate hearing loss and CI experienced lower social support levels than parents of hearing children [49, 57]. Meanwhile, the findings indicated that parents who exhibit more emotional stability, characterized by lower depression, anxiety, and stress levels, or who are engaged in their child’s daily activities or intervention sessions, tend to receive greater assistance from their family, friends, or spouse [49, 53, 57, 62, 63]. However, acknowledging that some of the population and item design logic behind the instruments were not originally intended for children with disabilities, for instance, MSPSS [49, 63], which was devised by university undergraduate students [78], is significant. Consequently, critically examining and assessing their psychometric features before their use with the population with disabilities particularly those who are DHH are necessary.

Condition-specific instruments for parents of children with CI

Owing to the irreversible nature of CI surgery, parents must have a positive emotional status and adequate ESS throughout their long-term CI journey (including pre- and post-CI surgery) to not only increase their involvement intention but also to facilitate their child’s spoken language development [30]. For example, although hearing aids did not benefit their children and the children kept removing them because of discomfort, parents still needed to ensure that their children consistently wore the hearing aids to meet the requirements for undergoing CI surgery. Dealing with their children’s uncooperativeness during this process was challenging and caused negative experiences for the parents. If the children have undergone CI surgery, parental involvement will affect the child’s speech and hearing development effectiveness [62, 104, 105]. Thus, parents should exert more effort to cope with mapping sessions and speech therapies, that is, they must attend all the appointments without affecting their daily routine. This can be a challenging task because they have to balance their work schedule, attend therapy sessions, and handle the financial cost of CI devices, which can be emotionally burdensome [106]. Therefore, for healthcare professionals who are working with families of children with CI, the condition-specific instrument design is important to acquire a better understanding of the families’ circumstances. Of note, although the present scoping review identified mostly generic measures among the instruments examined, few instruments were observed to be condition-specific measures, specifically designed for assessing parents of children with CI. Examples of such instruments include the CCIPP [67–70], PAVACI [65], and Parenting Stress-CI modules–EC [48].

The CCIPP scale contains several versions, each indicating slight variation in terms of domains and items [68–70]. The number of domains ranged from 8 to 11, whereas the number of items ranged from 48 to 74. In the study of Peker et al. [59] employed the instrument developed by Incesulu [70], and when compared with Archbold’s version [67, 68], it became apparent that Archbold’s version encompassed a larger quantity of items. In Archbold’s version, additional items were included in several domains to gain insight into the present condition of parents and children with CI, especially the parental-related domains (“decision making” and “process of implantation”), such as the parents’ potential hesitations or concerns regarding decision-making, the expected outcomes for their child following CI surgery, and the support they received from others. Nevertheless, of note, both versions primarily prioritize the assessment of the child’s quality of life rather than that of the parents, and the generalizability of these findings may be limited. For example, based on the findings of the “process of implantation” domain, whether parents expressed concerns regarding their child’s outcome or lacked sufficient support from others following CI surgery is difficult to ascertain. Besides, another limitation of using the CCIPP is that it was designed for parents whose children had CI for at least 3 years. This scale cannot be used to measure parental concerns and support systems among parents of children with CI candidates and newly implantees (within 1 year). Consequently, if healthcare professionals use the CCIPP to measure parental quality of life, its practical limitations and interpretive power may be questioned.

Similarly, to assess parental attitudes toward their children’s CI, the PAVACI instrument was developed [65]. This instrument comprises 70 items distributed across six different domains. A subtle distinction exists between the CCIPP and PAVACI scales in terms of the composition of the samples used during their respective development processes. The development of the PAVACI scale was specifically informed by data collected from a cohort of parents and children 1 year following CI surgery. However, although this scale demonstrates suitability in evaluating the outcomes of parents with newly implanted children (1 year of CI experience), its applicability may be limited when assessing parents whose children have not yet undergone CI surgery. Furthermore, half of the items, specifically 35 items, were related to parents’ support systems, encompassing several aspects, including services provided by the CI center, rehabilitation programs, and decision-making processes. Half of the items remained targeted at the child’s outcomes regarding the child’s communication, academics, social skills, and social abilities. Thus, the PAVICI scale’s content may not be appropriate for evaluating PES in relation to their child’s CI and their ESS during the CI journey. Therefore, in the context of clinical practice, individuals who lack proficiency in counseling, such as audiologists who frequently interact with families, encounter difficulties in understanding the parental emotions and the support they need.

Currently, the Parenting Stress-CI modules–EC (15 items) is considered the most suitable condition-specific instrument for evaluating the emotional status of parents who have children with CI [48]. The Parenting Stress-CI modules–EC demonstrates a greater connection and closeness to the familial circumstances of children with CI than the PSS or PSI scales. Specifically, the scale now includes items addressing concerns related to fear of losing the CI, managing the CI, or dealing with individuals staring at a child’s CI. These items emphasize the stress experienced by parents because of the appearance of the CI and the potential financial and managerial risks it encompasses. The published research demonstrated that the instrument shows favorable reliability and validity levels [48]. Also, the scale underwent a convergent validity test using the PSI-SF, and the findings were satisfactory, that is, the correlation between the two measures approached a significant level (r = .66, p < .001). Despite the inclusion of inquiries regarding potential CI-related risks, including financial costs and CI appearance, and the examination of the impact of family dynamics on parental stress levels, considering that the instrument still has its limitations is essential. Notably, the population recruitment during the instrument’s development phase may present certain limitations, as most participants were Caucasians. Therefore, the generalizability of the Parenting Stress-CI modules–EC to other ethnic groups remains uncertain. Furthermore, the scale employed in the Cejas et al. [48] study lacked different domains. Consequently, the interpretation of parental stress levels solely relied on the overall score, making it difficult to recognize specific stressors for families or parents. Specifically, without a detailed analysis, ascertaining the specific aspects (CI appearance, financial costs, family support, healthcare professionals’ support, and child’s expectation outcomes) that contribute to the experience of stress is challenging. Moreover, as the scale must be accompanied by social support-related instruments to understand the parent’s current emotional status and support systems, healthcare professionals should be aware of the limitations of the scale. Simultaneously, employing additional instruments pertaining to social support to comprehensively comprehend the prevailing emotional condition and support systems of parents of children with CI is imperative.

Limitations

This scoping review skips over gray literature, such as unpublished works or theses, as well as peer-reviewed publications written in languages other than English, such as Mandarin and Malay. Considering the multi-lingual nature of Malaysia, the exclusion of Mandarin and Malay literature may result in a missed opportunity to include valuable information that can be used in academic and clinical contexts within the country. This exclusionary approach may result in the omission of studies relevant to the present study’s objectives. Furthermore, note that this scoping review did not evaluate the quality of the studies included in the analyses. However, all included articles were extracted from bibliographic databases and had undergone the rigorous process of peer review before publication. Lastly, based on the findings of previous studies, the present study identified various factors that could influence PES and ESS. Notably, key indicators such as ethnicity, economic status, area of living, and culture have not been thoroughly examined [107]. This suggests that the results of the included studies do not necessarily apply to Asian cultures or even multicultural Malaysians. Future studies should investigate these significant indicators to better understand how PES and ESS are affected across different populations.