Family-Centered Early Intervention Deaf/Hard of Hearing (FCEI-DHH): Call to Action.

This Call to Action is the eighth and final article in this special issue on Family-Centered Early Intervention (FCEI) for children who are deaf or hard of hearing (DHH) and their families, or FCEI-DHH. Collectively, these articles highlight evidence-informed actions to enhance family well-being and to optimize developmental outcomes among children who are DHH. This Call to Action outlines actionable steps to advance FCEI-DHH supports provided to children who are DHH and their families. It also urges specific actions to strengthen FCEI-DHH programs/services and systems across the globe, whether newly emerging or long-established. Internationally, supports for children who are DHH are often siloed, provided within various independent sectors such as health/medicine, education, early childhood, and social and disability services. With this Call to Action, we urge invested parties from across relevant sectors to join together to implement and improve FCEI-DHH programs/services and systems, build the capacity of early intervention (EI) Providers and other professionals, extend research regarding FCEI-DHH, and fund EI supports, systems, and research, all with the aim of advancing outcomes for families and their children who are DHH.

Family-Centered Early Intervention Deaf/Hard of Hearing (FCEI-DHH): Methods.

This is the fourth article in a series of eight that comprise a special issue on family-centered early intervention (FCEI) for children who are deaf or hard of hearing (DHH) and their families, FCEI-DHH. This article describes the co-production team and the consensus review method used to direct the creation of the 10 Principles described in this special issue. Co-production is increasingly being used to produce evidence that is useful, usable, and used. A draft set of 10 Principles for FCEI-DHH and associated Tables of recommended behaviors were developed using the knowledge creation process. Principles were refined through two rounds of eDelphi review. Results for each round were analyzed using measures of overall group agreement and measures that indicated the extent to which the group members agreed with each other. After Round 2, with strong agreement and low to moderate variation in extent of agreement, consensus was obtained for the 10 Principles for FCEI-DHH presented in this special issue. This work can be used to enhance evolution of FCEI-DHH program/services and systems world-wide and adds to knowledge in improvement science.

Family-Centered Early Intervention Deaf/Hard of Hearing (FCEI-DHH): Foundation Principles.

This article is the fifth in a series of eight articles that comprise a special issue on Family-Centered Early Intervention (FCEI) for children who are deaf or hard of hearing (DHH) and their families, or FCEI-DHH. The 10 FCEI-DHH Principles are organized conceptually into three sections (a) Foundation Principles, (b) Support Principles, and (c) Structure Principles. Collectively, they describe the essential Principles that guide FCEI for children who are DHH and their families. This article describes the Foundation Principles (Principles 1 and Principle 2). The Foundation Principles emphasize the essential elements of ensuring that families with children who are DHH can access early intervention (EI) and other appropriate supports, as well as highlight the need for provision of EI that is family-centered. Implementation of these FCEI-DHH Principles is intended to improve the lives and the outcomes of children who are DHH and their families around the globe.

Family-Centered Early Intervention Deaf/Hard of Hearing (FCEI-DHH): Cultural & Global Implications.

This article is the third in a series of eight articles that comprise this special issue on family-centered early intervention for children who are deaf or hard of hearing and their families (FCEI-DHH). It highlights the origins of FCEI-DHH in Western contexts and well-resourced locations and emphasizes the role of culture(s) in shaping FCEI-DHH. This article also cautions against the direct application of the 10 FCEI-DHH Principles presented in this issue across the globe without consideration of cultural implications. Cultural perceptions of decision-making processes and persons who can be decision-makers in FCEI-DHH are explored. Deaf culture(s) and the benefits of exposure to DHH adults with diverse backgrounds are introduced. Structural inequities that impact families' access to FCEI-DHH programs/services and systems, within and among nations and regions, are noted. The need to consider the cultural influences on families is emphasized; this applies to all levels of FCEI, including the development of systems through implementation of supports.

Family-Centered Early Intervention Deaf/Hard of Hearing (FCEI-DHH): Support Principles.

This article is the sixth in a series of eight articles that comprise a special issue on Family-Centered Early Intervention (FCEI) for children who are deaf or hard of hearing (DHH) and their families, or FCEI-DHH. The Support Principles article is the second of three articles that describe the 10 Principles of FCEI-DHH, preceded by the Foundation Principles, and followed by the Structure Principles, all in this special issue. The Support Principles are composed of four Principles (Principles 3, 4, 5, and 6) that highlight (a) the importance of a variety of supports for families raising children who are DHH; (b) the need to attend to and ensure the well-being of all children who are DHH; (c) the necessity of building the language and communication abilities of children who are DHH and their family members; and (d) the importance of considering the family's strengths, needs, and values in decision-making.

Family-Centered Early Intervention Deaf/Hard of Hearing (FCEI-DHH): Guiding Values.

This article is the second of eight articles in this special issue on Family-Centered Early Intervention (FCEI) for children who are deaf or hard of hearing (DHH) and their families, or FCEI-DHH. Five foundational values that guide FCEI-DHH are described, providing an evidence-informed, conceptual context for the 10 FCEI-DHH Principles and other articles presented in this issue. These values are applicable for Early Intervention (EI) Providers and other professionals on FCEI teams, as well as for FCEI-DHH programs/services and systems. The five key values include (1) being family-centered, (2) responding to diversity, (3) involving invested parties, especially families and individuals who are DHH, (4) supporting holistic child development, and (5) ensuring fundamental human rights. These evidence-informed values are considered essential to the effective provision of FCEI-DHH supports.

Family-Centered Early Intervention Deaf/Hard of Hearing (FCEI-DHH): Introduction.

This article is the first of eight articles in this special issue on Family-Centered Early Intervention (FCEI) for children who are deaf or hard of hearing (DHH), or FCEI-DHH. In 2013, a diverse panel of experts published an international consensus statement on evidence-based Principles guiding FCEI-DHH. Those original Principles have been revised through a coproduction process involving multidisciplinary collaborators and an international consensus panel, utilizing the best available evidence and current understanding of how to optimally support children who are DHH and their families. This revision (referred to as expanded Principles) was motivated by the need to incorporate (a) input from family leaders and DHH leaders, (b) broader international and cultural perspectives, (c) new empirical evidence, and (d) research in human development. This Introduction provides an overview of the rationale, purposes, and main content areas to be addressed throughout the special issue.

Family-Centered Early Intervention Deaf/Hard of Hearing (FCEI-DHH): Structure Principles.

This article is the seventh in a series of eight articles that comprise a special issue on family-centered early intervention for children who are deaf or hard of hearing and their families, or FCEI-DHH. This article, Structure Principles, is the third of three articles (preceded by Foundation Principles and Support Principles) that describe the 10 FCEI-DHH Principles. The Structure Principles include 4 Principles (Principle 7, Principle 8, Principle 9, and Principle 10) that highlight (a) the importance of trained and effective Early Intervention (EI) Providers, (b) the need for FCEI-DHH teams to work collaboratively to support families, (c) the considerations for tracking children's progress through developmental assessment, and (d) the essential role of progress monitoring to continuously improve systems.

Aided Hearing Moderates the Academic Outcomes of Children With Mild to Severe Hearing Loss.

OBJECTIVES: There are very limited data regarding the spoken language and academic outcomes of children with mild to severe hearing loss (HL) during the elementary school years, and the findings of these studies are inconsistent. None of these studies have examined the possible role of aided hearing in these outcomes. This study used a large cohort of children to examine these outcomes and in particular to examine whether aided hearing moderates the effect of HL with regard to these outcomes. DESIGN: The spoken language, reading, writing, and calculation abilities were measured after second and fourth grades in children with mild to severe HL (children who are hard of hearing; CHH, n = 183) and a group of children with normal hearing (CNH, n = 91) after the completion of second and fourth grades. Also, among the CHH who wore hearing aids, aided better-ear speech intelligibility index values at the age of school entry were obtained. RESULTS: Oral language abilities of the CHH with mild and moderate HL were similar to the CNH at each grade. Children with moderately-severe HL (better-ear pure tone threshold >59 but <76 dB HL) had significantly poorer oral language and reading skills than the CNH at each grade. The children with mild and moderate HL did not differ from the CNH in oral language or reading. No differences were found between the CHH regardless of severity and CNH with regard to spelling, passage writing, or calculation. The degree to which hearing aids provided audible speech information played a moderating role in the oral language outcomes of CHH and this moderation of language mediated the relationship between the unaided hearing ability of the CHH and their academic outcomes. CONCLUSIONS: As a group, children with mild and moderate HL have good outcomes with regard to language and academic performance. Children with moderately-severe losses were less skilled in language and reading than the CNH and CHH children with mild and moderate losses. Audibility provided by hearing aids was found to moderate the effects of HL with respect to these outcomes. These findings emphasize the importance of including the effects of clinical interventions such as aided hearing when examining outcomes of CHH.

Medical Referral Patterns and Etiologies for Children With Mild-to-Severe Hearing Loss.

OBJECTIVES: To (1) identify the etiologies and risk factors of the patient cohort and determine the degree to which they reflected the incidence for children with hearing loss and (2) quantify practice management patterns in three catchment areas of the United States with available centers of excellence in pediatric hearing loss. DESIGN: Medical information for 307 children with bilateral, mild-to-severe hearing loss was examined retrospectively. Children were participants in the Outcomes of Children with Hearing Loss (OCHL) study, a 5-year longitudinal study that recruited subjects at three different sites. Children aged 6 months to 7 years at time of OCHL enrollment were participants in this study. Children with cochlear implants, children with severe or profound hearing loss, and children with significant cognitive or motor delays were excluded from the OCHL study and, by extension, from this analysis. Medical information was gathered using medical records and participant intake forms, the latter reflecting a caregiver's report. A comparison group included 134 children with normal hearing. A Chi-square test on two-way tables was used to assess for differences in referral patterns by site for the children who are hard of hearing (CHH). Linear regression was performed on gestational age and birth weight as continuous variables. Risk factors were assessed using t tests. The alpha value was set at p < 0.05. RESULTS: Neonatal intensive care unit stay, mechanical ventilation, oxygen requirement, aminoglycoside exposure, and family history were correlated with hearing loss. For this study cohort, congenital cytomegalovirus, strep positivity, bacterial meningitis, extracorporeal membrane oxygenation, and loop diuretic exposure were not associated with hearing loss. Less than 50% of children underwent imaging, although 34.2% of those scanned had abnormalities identified. No single imaging modality was preferred. Differences in referral rates were apparent for neurology, radiology, genetics, and ophthalmology. CONCLUSIONS: The OCHL cohort reflects known etiologies of CHH. Despite available guidelines, centers of excellence, and high-yield rates for imaging, the medical workup for children with hearing loss remains inconsistently implemented and widely variable. There remains limited awareness as to what constitutes appropriate medical assessment for CHH.

Effect of Context and Hearing Loss on Time-Gated Word Recognition in Children.

OBJECTIVES: The purpose of this study was to examine word recognition in children who are hard of hearing (CHH) and children with normal hearing (CNH) in response to time-gated words presented in high- versus low-predictability sentences (HP, LP), where semantic cues were manipulated. Findings inform our understanding of how CHH combine cognitive-linguistic and acoustic-phonetic cues to support spoken word recognition. It was hypothesized that both groups of children would be able to make use of linguistic cues provided by HP sentences to support word recognition. CHH were expected to require greater acoustic information (more gates) than CNH to correctly identify words in the LP condition. In addition, it was hypothesized that error patterns would differ across groups. DESIGN: Sixteen CHH with mild to moderate hearing loss and 16 age-matched CNH participated (5 to 12 years). Test stimuli included 15 LP and 15 HP age-appropriate sentences. The final word of each sentence was divided into segments and recombined with the sentence frame to create series of sentences in which the final word was progressively longer by the gated increments. Stimuli were presented monaurally through headphones and children were asked to identify the target word at each successive gate. They also were asked to rate their confidence in their word choice using a five- or three-point scale. For CHH, the signals were processed through a hearing aid simulator. Standardized language measures were used to assess the contribution of linguistic skills. RESULTS: Analysis of language measures revealed that the CNH and CHH performed within the average range on language abilities. Both groups correctly recognized a significantly higher percentage of words in the HP condition than in the LP condition. Although CHH performed comparably with CNH in terms of successfully recognizing the majority of words, differences were observed in the amount of acoustic-phonetic information needed to achieve accurate word recognition. CHH needed more gates than CNH to identify words in the LP condition. CNH were significantly lower in rating their confidence in the LP condition than in the HP condition. CHH, however, were not significantly different in confidence between the conditions. Error patterns for incorrect word responses across gates and predictability varied depending on hearing status. CONCLUSIONS: The results of this study suggest that CHH with age-appropriate language abilities took advantage of context cues in the HP sentences to guide word recognition in a manner similar to CNH. However, in the LP condition, they required more acoustic information (more gates) than CNH for word recognition. Differences in the structure of incorrect word responses and their nomination patterns across gates for CHH compared with their peers with NH suggest variations in how these groups use limited acoustic information to select word candidates.

Assessing Vocal Development in Infants and Toddlers Who Are Hard of Hearing: A Parent-Report Tool.

The main purpose of the current investigation was to determine whether the Vocal Development Landmarks Interview-Experimental Version (VDLI-E) was sensitive to variation in the vocal development of infants and toddlers who are hard of hearing. The VDLI-E is an interactive parent interview that uses audio samples of authentic infant vocalizations to make targeted vocal behaviors clear and understandable to parents without the need for technical terms, verbal descriptions, or adult modeling of infant productions. The VDLI-E was found to be sensitive to age and hearing and was related to performance on concurrent measures of early auditory skills, expressive vocabulary, and overall expressive language abilities. These findings provide preliminary support for the utility of this measure in monitoring the impact of early auditory experiences on vocal development for 6- to 18-month-old children who are hard of hearing.

An Introduction to the Outcomes of Children with Hearing Loss Study.

The landscape of service provision for young children with hearing loss has shifted in recent years as a result of newborn hearing screening and the early provision of interventions, including hearing technologies. It is expected that early service provision will minimize or prevent linguistic delays that typically accompany untreated permanent childhood hearing loss. The post-newborn hearing screening era has seen a resurgence of interest in empirically examining the outcomes of children with hearing loss to determine if service innovations have resulted in expected improvements in children's functioning. The Outcomes of Children with Hearing Loss (OCHL) project was among these recent research efforts, and this introductory article provides background in the form of literature review and theoretical discussion to support the goals of the study. The Outcomes of Children with Hearing Loss project was designed to examine the language and auditory outcomes of infants and preschool-age children with permanent, bilateral, mild-to-severe hearing loss, and to identify factors that moderate the relationship between hearing loss and longitudinal outcomes. The authors propose that children who are hard of hearing experience limitations in access to linguistic input, which lead to a decrease in uptake of language exposure and an overall reduction in linguistic experience. The authors explore this hypothesis in relation to three primary factors that are proposed to influence children's access to linguistic input: aided audibility, duration and consistency of hearing aid use, and characteristics of caregiver input.

Epilogue: Conclusions and Implications for Research and Practice.

The primary purpose of the epilogue article is to synthesize the key findings from the Outcomes of Children with Hearing Loss (OCHL) study by presenting a set of 10 major conclusions. The conclusion statements provide a concise summary of the main results related to children's auditory and language outcomes and factors identified as moderators of these outcomes. The second section of this article summarizes the primary clinical implications that follow from the OCHL study in relation to three questions: (1) Can we afford to be complacent about the current outcomes of children who are hard of hearing? (2) Which malleable factors can be addressed to promote success through implementation of best practices? and (3) Which nonmalleable factors are consequential and what are their implications for practice? The authors end with some future research directions for the OCHL project.

Afterword: Lessons Learned About Multicenter Research Collaboration.

In this brief afterword, we discuss the challenges and lessons learned in the process of implementing a multisite, longitudinal study. Some of the lessons learned by the research team are shared regarding research design and analysis, strategies implemented to reduce threats to validity, and techniques used to promote teamwork and collaboration across sites.

Outcomes of Children with Hearing Loss: Data Collection and Methods.

OBJECTIVES: The primary objective of this article was to describe recruitment, data collection, and methods for a longitudinal, multicenter study involving children with bilateral mild to severe hearing loss. The goals of this research program were to characterize the developmental outcomes of children with mild to severe bilateral hearing loss during infancy and the preschool years. Furthermore, the researchers examined how these outcomes were associated with the child's hearing loss and how home background and clinical interventions mediated and moderated these outcomes. DESIGN: The participants in this study were children who are hard of hearing (CHH) and children with normal hearing (CNH) who provided comparison data. CHH were eligible for participation if (1) their chronological age was between 6 months and 7 years of age at the time of recruitment, (2) they had a better-ear pure-tone average of 25 to 75 dB HL, (3) they had not received a cochlear implant, (4) they were from homes where English was the primary language, and (5) they did not demonstrate significant cognitive or motor delays. Across the time span of recruitment, 430 parents of potential children with hearing loss made contact with the research group. This resulted in 317 CHH who qualified for enrollment. In addition, 117 CNH qualified for enrollment. An accelerated longitudinal design was used, in which multiple age cohorts were followed long enough to provide overlap. Specifically, children were recruited and enrolled continuously across an age span of 6.5 years and were followed for at least 3 years. This design allowed for tests of time (period) versus cohort age effects that could arise by changes in services and technology over time, yet still allowed for examination of important developmental relationships. RESULTS: The distribution of degree of hearing loss for the CHH showed that the majority of CHH had moderate or moderate-to-severe hearing losses, indicating that the sample undersampled children with mild HL. For mothers of both CHH and CNH, the distribution of maternal education level showed that few mothers lacked at least a high school education and a slight majority had completed a bachelor's degree, suggesting that this sample of research volunteers was more advantaged than the United States population. The test battery consisted of a variety of measures concerning participants' hearing and behavioral development. These data were gathered in sessions during which the child was examined by an audiologist and a speech-language examiner. In addition, questionnaires concerning the child's behavior and development were completed by the parents. CONCLUSION: The Outcomes of Children with Hearing Loss study was intended to examine the relationship between variation in hearing ability across children with normal and mild to severe hearing loss and variation in their outcomes across several domains of development. In addition, the research team sought to document important mediators and moderators that act between the hearing loss and the outcomes. Because the study design provided for the examination of outcomes throughout infancy and early childhood, it was necessary to employ a number of different measures of the same construct to accommodate changes in developmental performance across age. This resulted in a large matrix of measures across variable types and developmental levels, as described in this manuscript.

Language Outcomes in Young Children with Mild to Severe Hearing Loss.

OBJECTIVES: This study examined the language outcomes of children with mild to severe hearing loss during the preschool years. The longitudinal design was leveraged to test whether language growth trajectories were associated with degree of hearing loss and whether aided hearing influenced language growth in a systematic manner. The study also explored the influence of the timing of hearing aid fitting and extent of use on children's language growth. Finally, the study tested the hypothesis that morphosyntax may be at particular risk due to the demands it places on the processing of fine details in the linguistic input. DESIGN: The full cohort of children in this study comprised 290 children who were hard of hearing (CHH) and 112 children with normal hearing who participated in the Outcomes of Children with Hearing Loss (OCHL) study between the ages of 2 and 6 years. CHH had a mean better-ear pure-tone average of 47.66 dB HL (SD = 13.35). All children received a comprehensive battery of language measures at annual intervals, including standardized tests, parent-report measures, and spontaneous and elicited language samples. Principal components analysis supported the use of a single composite language score for each of the age levels (2, 3, 4, 5, and 6 years). Measures of unaided (better-ear pure-tone average, speech intelligibility index) and aided (residualized speech intelligibility index) hearing were collected, along with parent-report measures of daily hearing aid use time. Mixed modeling procedures were applied to examine the rate of change (227 CHH; 94 children with normal hearing) in language ability over time in relation to (1) degree of hearing loss, (2) aided hearing, (3) age of hearing aid fit and duration of use, and (4) daily hearing aid use. Principal components analysis was also employed to examine factor loadings from spontaneous language samples and to test their correspondence with standardized measures. Multiple regression analysis was used to test for differential effects of hearing loss on morphosyntax and lexical development. RESULTS: Children with mild to severe hearing loss, on average, showed depressed language levels compared with peers with normal hearing who were matched on age and socioeconomic status. The degree to which CHH fell behind increased with greater severity of hearing loss. The amount of improved audibility with hearing aids was associated with differential rates of language growth; better audibility was associated with faster rates of language growth in the preschool years. Children fit early with hearing aids had better early language achievement than children fit later. However, children who were fit after 18 months of age improved in their language abilities as a function of the duration of hearing aid use. These results suggest that the language learning system remains open to experience provided by improved access to linguistic input. Performance in the domain of morphosyntax was found to be more delayed in CHH than their semantic abilities. CONCLUSION: The data obtained in this study largely support the predictions, suggesting that mild to severe hearing loss places children at risk for delays in language development. Risks are moderated by the provision of early and consistent access to well-fit hearing aids that provide optimized audibility.

Quantity and Quality of Caregivers' Linguistic Input to 18-Month and 3-Year-Old Children Who Are Hard of Hearing.

OBJECTIVES: The primary objective of this study was to examine the quantity and quality of caregiver talk directed to children who are hard of hearing (CHH) compared with children with normal hearing (CNH). For the CHH only, the study explored how caregiver input changed as a function of child age (18 months versus 3 years), which child and family factors contributed to variance in caregiver linguistic input at 18 months and 3 years, and how caregiver talk at 18 months related to child language outcomes at 3 years. DESIGN: Participants were 59 CNH and 156 children with bilateral, mild-to-severe hearing loss. When children were approximately 18 months and/or 3 years of age, caregivers and children participated in a 5-min semistructured, conversational interaction. Interactions were transcribed and coded for two features of caregiver input representing quantity (number of total utterances and number of total words) and four features representing quality (number of different words, mean length of utterance in morphemes, proportion of utterances that were high level, and proportion of utterances that were directing). In addition, at the 18-month visit, parents completed a standardized questionnaire regarding their child's communication development. At the 3-year visit, a clinician administered a standardized language measure. RESULTS: At the 18-month visit, the CHH were exposed to a greater proportion of directing utterances than the CNH. At the 3-year visit, there were significant differences between the CNH and CHH for number of total words and all four of the quality variables, with the CHH being exposed to fewer words and lower quality input. Caregivers generally provided higher quality input to CHH at the 3-year visit compared with the 18-month visit. At the 18-month visit, quantity variables, but not quality variables, were related to several child and family factors. At the 3-year visit, the variable most strongly related to caregiver input was child language. Longitudinal analyses indicated that quality, but not quantity, of caregiver linguistic input at 18 months was related to child language abilities at 3 years, with directing utterances accounting for significant unique variance in child language outcomes. CONCLUSIONS: Although caregivers of CHH increased their use of quality features of linguistic input over time, the differences when compared with CNH suggest that some caregivers may need additional support to provide their children with optimal language learning environments. This is particularly important given the relationships that were identified between quality features of caregivers' linguistic input and children's language abilities. Family supports should include a focus on developing a style that is conversational eliciting as opposed to directive.

Trends and Predictors of Longitudinal Hearing Aid Use for Children Who Are Hard of Hearing.

OBJECTIVES: Children who are hard of hearing (CHH) have restricted access to acoustic and linguistic information. Increased audibility provided by hearing aids (HAs) influences language outcomes, but the advantages of appropriately fit HAs can only be realized if children wear their devices on a consistent basis. The purpose of this article was to characterize long-term HA use in CHH, based on parent-report measures, and identify factors that influence longitudinal trends in HA use. DESIGN: Participants were parents of 290 children with mild to severe hearing loss. At every visit, parents estimated the average amount of time the child used HAs per day during the week and on the weekends. Parent reports of daily HA use were analyzed to determine if different patterns of HA use were observed longitudinally during the study. Independent predictor variables were then related to longitudinal trends in HA use within three age groups (infant, preschool, school age). RESULTS: On average across multiple visits, parents reported that their children wore their HAs for 10.63 hr per day (SD = 3.29). Data logging values were lower than parent-report measures (M = 8.44, SD = 4.06), suggesting that parents overestimated daily HA use. The majority of children in each age group wore HAs at least 8 hr per day from their first research testing interval to their last, based on parent-report measures. Maternal education level predicted longitudinal trends in HA use for infants and school-age CHH. Degree of hearing loss was related to trends in school-age children only. CONCLUSIONS: These results indicated that the majority of CHH increased HA use over time, but a sizable minority demonstrated a low level of use or decreased use in the time period studied. Maternal education level influenced longitudinal trends in daily HA use. Degree of hearing loss influenced trends in school-age children only. Audiologists and early intervention service providers might aid in improving HA use by providing regular hands-on training with the HAs and individualized problem-based strategies to address the challenges families experience with attaining a high level of use. Families may also benefit from practical demonstrations of the benefits of consistent HA use, such as hearing loss simulations, examples of listening in noise with and without HAs, or listening to malfunctioning HAs.