Mobile Health School Screening and Telemedicine Referral to Improve Access to Specialty Care in Rural Alaska: Integrating Mixed Methods Data to Contextualize Trial Outcomes.

OBJECTIVES: To understand factors associated with outcomes in a cluster-randomized controlled trial that evaluated a telemedicine specialty referral intervention for school hearing screenings in 15 rural Alaskan communities. DESIGN: Hearing Norton Sound was a mixed methods cluster-randomized controlled trial that compared a telemedicine specialty referral pathway (intervention) to a standard primary care referral pathway (control) for school hearing screenings. As a mixed methods trial, both quantitative and qualitative data were collected, analyzed, and integrated. Main trial results are published elsewhere, but integration of community-specific quantitative outcomes and qualitative results have not yet been reported. The constant comparative method was used to analyze qualitative data from semistructured interviews with six stakeholder groups across all 15 communities. Descriptive statistics were used to describe community-specific proportions of follow-up in both trial years. Qualitative and quantitative results were integrated to reveal relationships between contextual factors and follow-up outcomes across communities. RESULTS: The Hearing Norton Sound trial enrolled 1481 children from October 2017 to March 2019, with a total of 790 children requiring referral. Of the children who referred in the telemedicine specialty referral pathway communities (intervention), 68.5% received follow-up (268/391), compared to 32.1% (128/399) in primary care referral communities (control)(previously reported). When broken down by community, the mean proportion receiving follow-up was 75.26% (SD 22.5) and 37.9% (SD 11.4) for the telemedicine specialty referral communities and primary care referral communities, respectively. For qualitative data collection, semistructured interviews were conducted with 101 individuals between December 2018 and August 2019. Six stakeholder groups participated: elders (n = 14), parents (n = 25), children (n = 11), teachers/school staff (n = 18), principals (n = 6), and healthcare providers/clinic staff (n = 27). Six overall factors related to the outcomes of the telemedicine specialty referral pathway emerged during analysis: clinic capacity, personnel ownership and engagement, scheduling, telemedicine equipment/processes, communication, and awareness of the need for follow-up. We integrated these factors with the community-specific follow-up percentages and found associations for four of the six qualitative factors: clinic capacity, personnel ownership and engagement, communication, and awareness. An association was not seen for scheduling and telemedicine equipment/processes, which had variable relationships with the follow-up outcome. CONCLUSIONS: The Hearing Norton Sound trial demonstrated that a telemedicine specialty referral pathway can close the gap on children lost to follow up after school hearing screening. As a whole, the intervention profoundly increased the proportion of children receiving follow-up, but there was variability in outcomes within and between communities. To understand this variability, we analyzed community-specific intervention outcomes alongside community member feedback on factors related to the intervention. We identified four key factors that contributed to the success of the intervention. Attention to these factors will be essential to successful adaptation and implementation of this telemedicine specialty referral intervention and other similar interventions in future work in rural Alaska and beyond.

Prevalence of Childhood Hearing Loss in Rural Alaska.

OBJECTIVES: Childhood hearing loss has well-known lifelong consequences. Certain rural populations are at higher risk for infection-related hearing loss. For Alaska Native children, historical data on hearing loss prevalence suggest a higher burden of infection-related hearing loss, but updated prevalence data are urgently needed in this high-risk population. DESIGN: Hearing data were collected as part of two school-based cluster-randomized trials in 15 communities in rural northwest Alaska over two academic years (2017-2019). All enrolled children from preschool to 12th grade were eligible. Pure-tone thresholds were obtained using standard audiometry and conditioned play when indicated. The analysis included the first available audiometric assessment for each child (n = 1634 participants, 3 to 21 years), except for the high-frequency analysis, which was limited to year 2 when higher frequencies were collected. Multiple imputation was used to quantify the prevalence of hearing loss in younger children, where missing data were more frequent due to the need for behavioral responses. Hearing loss in either ear was evaluated using both the former World Health Organization (WHO) definition (pure-tone average [PTA] > 25 dB) and the new WHO definition (PTA ≥ 20 dB), which was published after the study. Analyses with the new definition were limited to children 7 years and older due to incomplete data obtained on younger children at lower thresholds. RESULTS: The overall prevalence of hearing loss (PTA > 25 dB; 0.5, 1, 2, 4 kHz) was 10.5% (95% confidence interval [CI], 8.9 to 12.1). Hearing loss was predominately mild (PTA >25 to 40 dB; 8.9%, 95% CI, 7.4 to 10.5). The prevalence of unilateral hearing loss was 7.7% (95% CI, 6.3 to 9.0). Conductive hearing loss (air-bone gap of ≥ 10 dB) was the most common hearing loss type (9.1%, 95% CI, 7.6 to 10.7). Stratified by age, hearing loss (PTA >25 dB) was more common in children 3 to 6 years (14.9%, 95% CI, 11.4 to 18.5) compared to children 7 years and older (8.7%, 95% CI, 7.1 to 10.4). In children 7 years and older, the new WHO definition increased the prevalence of hearing loss to 23.4% (95% CI, 21.0 to 25.8) compared to the former definition (8.7%, 95% CI, 7.1 to 10.4). Middle ear disease prevalence was 17.6% (95% CI, 15.7 to 19.4) and was higher in younger children (23.6%, 95% CI, 19.7 to 27.6) compared to older children (15.2%, 95% CI, 13.2 to 17.3). High-frequency hearing loss (4, 6, 8kHz) was present in 20.5% (95% CI, 18.4 to 22.7 [PTA >25 dB]) of all children and 22.8% (95% CI, 20.3 to 25.3 [PTA >25 dB]) and 29.7% (95% CI, 27.0 to 32.4 [PTA ≥ 20 dB]) of children 7 years and older (limited to year 2). CONCLUSIONS: This analysis represents the first prevalence study on childhood hearing loss in Alaska in over 60 years and is the largest cohort with hearing data ever collected in rural Alaska. Our results highlight that hearing loss continues to be common in rural Alaska Native children, with middle ear disease more prevalent in younger children and high-frequency hearing loss more prevalent with increasing age. Prevention efforts may benefit from managing hearing loss type by age. Lastly, continued research is needed on the impact of the new WHO definition of hearing loss on field studies.

Telemedicine Referral to Improve Access to Specialty Care for Preschool Children in Rural Alaska: A Cluster-Randomized Controlled Trial.

OBJECTIVES: Preschool programs provide essential preventive services, such as hearing screening, but in rural regions, limited access to specialists and loss to follow-up compound rural health disparities. We conducted a parallel-arm cluster-randomized controlled trial to evaluate telemedicine specialty referral for preschool hearing screening. The goal of this trial was to improve timely identification and treatment of early childhood infection-related hearing loss, a preventable condition with lifelong implications. We hypothesized that telemedicine specialty referral would improve time to follow-up and the number of children receiving follow-up compared with the standard primary care referral. DESIGN: We conducted a cluster-randomized controlled trial in K-12 schools in 15 communities over two academic years. Community randomization occurred within four strata using location and school size. In the second academic year (2018-2019), an ancillary trial was performed in the 14 communities that had preschools to compare telemedicine specialty referral (intervention) to standard primary care referral (comparison) for preschool hearing screening. Randomization of communities from the main trial was used for this ancillary trial. All children enrolled in preschool were eligible. Masking was not possible because of timing in the second year of the main trial, but referral assignment was not openly disclosed. Study team members and school staff were masked throughout data collection, and statisticians were blinded to allocation during analysis. Preschool screening occurred once, and children who were referred for possible hearing loss or ear disease were monitored for follow-up for 9 months from the screening date. The primary outcome was time to ear/hearing-related follow-up from the date of screening. The secondary outcome was any ear/hearing follow-up from screening to 9 months. Analyses were conducted using an intention-to-treat approach. RESULTS: A total of 153 children were screened between September 2018 and March 2019. Of the 14 communities, 8 were assigned to the telemedicine specialty referral pathway (90 children), and 6 to the standard primary care referral pathway (63 children). Seventy-one children (46.4%) were referred for follow-up: 39 (43.3%) in the telemedicine specialty referral communities and 32 (50.8%) in the standard primary care referral communities. Of children referred, 30 (76.9%) children in telemedicine specialty referral communities and 16 (50.0%) children in standard primary care referral communities received follow-up within 9 months (Risk Ratio = 1.57; 95% confidence interval [CI], 1.22 to 2.01). Among children who received follow-up, median time to follow-up was 28 days (interquartile range [IQR]: 15 to 71) in telemedicine specialty referral communities compared with 85 days (IQR: 26 to 129) in standard primary care referral communities. Mean time to follow-up for all referred children was 4.5 (event time ratio = 4.5; 95% CI, 1.8 to 11.4; p = 0.045) times faster in telemedicine specialty referral communities compared with standard primary care referral communities in the 9-month follow-up time frame. CONCLUSIONS: Telemedicine specialty referral significantly improved follow-up and reduced time to follow-up after preschool hearing screening in rural Alaska. Telemedicine referrals could extend to other preventive school-based services to improve access to specialty care for rural preschool children.

Community Perspectives on Hearing Loss in Rural Alaska.

OBJECTIVES: The aim of this study is to present an explanatory model of hearing loss in the Bering Strait region of Alaska in order to contextualize the results of a cluster randomized trial and propose implications for regional hearing-related health care. DESIGN: To promote ecological validity, or the generalizability of trial findings to real world experiences, qualitative methods (focus groups and interviews) were used within a mixed methods cluster randomized trial evaluating school hearing screening and follow-up processes in 15 communities in the Bering Strait region of Alaska. Focus groups were held between April and August 2017, and semistructured interviews were conducted between December 2018 and August 2019. Convenience sampling was used for six of the 11 focus groups to capture broad community feedback. Purposive sampling was used for the remaining five focus groups and for all interviews to capture a variety of experiences with hearing loss. Audio recordings of focus groups and interviews were transcribed, and both notes and transcripts were deidentified. All notes and transcripts were included in the analysis. The constant comparative method was used to develop a codebook by iteratively moving between transcripts and preliminary themes. Researchers then used this codebook to code data from all focus groups and interviews using qualitative analysis software (NVIVO 12, QSR International) and conducted thematic analyses to distill the findings presented in this article. RESULTS: Participants in focus groups (n = 116) and interviews (n = 101) shared perspectives in three domains: etiology, impact, and treatment of hearing loss. Regarding etiology, participants emphasized noise-induced hearing loss but also discussed infection-related hearing loss and various causes of ear infections. Participants described the impact of hearing loss on subsistence activities, while also detailing social, academic, and economic consequences. Participants described burdensome treatment pathways that are repetitive and often travel and time intensive. Communication breakdowns within these pathways were also described. Some participants spoke positively of increased access via onsite hearing health care services in "field clinics" as well as via telemedicine services. Others described weaknesses in these processes (infrequent field clinics and communication delays in telemedicine care pathways). Participants also described home remedies and stigma surrounding the treatment for hearing loss. CONCLUSIONS: Patient-centered health care requires an understanding of context. Explanatory models of illness are context-specific ways in which patients and their networks perceive and describe the experience of an illness or disability. In this study, we documented explanatory models of hearing loss to foster ecological validity and better understand the relevance of research findings to real-life hearing-related experiences. These findings suggest several areas that should be addressed in future implementation of hearing health care interventions elsewhere in rural Alaska, including management of repetitious treatments, awareness of infection-mediated hearing loss, mistrust, and communication breakdowns. For hearing-related health care in this region, these findings suggest localized recommendations for approaches for prevention and treatment. For community-based hearing research, this study offers an example of how qualitative methods can be used to generate ecologically valid (i.e., contextually grounded) findings.