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.

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.

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.

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.

Changing the Paradigm for School Hearing Screening Globally: Evaluation of Screening Protocols From Two Randomized Trials in Rural Alaska.

OBJECTIVES: Diagnostic accuracy was evaluated for various screening tools, including mobile health (mHealth) pure-tone screening, tympanometry, distortion product otoacoustic emissions (DPOAE), and inclusion of high frequencies to determine the most accurate screening protocol for identifying children with hearing loss in rural Alaska where the prevalence of middle ear disease is high. DESIGN: Hearing screening data were collected as part of two cluster randomized trials conducted in 15 communities in rural northwest Alaska. All children enrolled in school from preschool to 12th grade were eligible. Analysis was limited to data collected 2018 to 2019 (n = 1449), when both trials were running and measurement of high frequencies were included in the protocols. Analyses included estimates of diagnostic accuracy for each screening tool, as well as exploring performance by age and grade. Multiple imputation was used to assess diagnostic accuracy in younger children, where missing data were more prevalent due to requirements for conditioned responses. The audiometric reference standard included otoscopy, tympanometry, and high frequencies to ensure detection of infection-related and noise-induced hearing loss. RESULTS: Both the mHealth pure-tone screen and DPOAE screen performed better when tympanometry was added to the protocol (increase in sensitivity of 19.9%, 95% Confidence Interval (CI): 15.9 to 24.1 for mHealth screen, 17.9%, 95% CI: 14.0 to 21.8 for high-frequency mHealth screen, and 10.4%, 95% CI: 7.5 to 13.9 for DPOAE). The addition of 6 kHz to the mHealth pure-tone screen provided an 8.7 percentage point improvement in sensitivity (95% CI: 6.5 to 11.3). Completeness of data for both the reference standard and the mHealth screening tool differed substantially by age, due to difficulty with behavioral testing in young children. By age 7, children were able to complete behavioral testing, and data indicated that high-frequency mHealth pure-tone screen with tympanometry was the superior tool for children 7 years and older. For children 3 to 6 years of age, DPOAE plus tympanometry performed the best, both for complete data and multiply imputed data, which better approximates accuracy for children with missing data. CONCLUSIONS: This study directly evaluated pure-tone, DPOAE, and tympanometry tools as part of school hearing screening in rural Alaskan children (3 to 18+ years). Results from this study indicate that tympanometry is a key component in the hearing screening protocol, particularly in environments with higher prevalence of infection-related hearing loss. DPOAE is the preferred hearing screening tool when evaluating children younger than 7 years of age (below 2nd grade in the United States) due to the frequency of missing data with behavioral testing in this age group. For children 7 years and older, the addition of high frequencies to pure-tone screening increased the accuracy of screening, likely due to improved identification of hearing loss from noise exposure. The lack of a consistent reference standard in the literature makes comparing across studies challenging. In our study with a reference standard inclusive of otoscopy, tympanometry, and high frequencies, less than ideal sensitivities were found even for the most sensitive screening protocols, suggesting more investigation is necessary to ensure screening programs are appropriately identifying noise- and infection-related hearing loss in rural, low-resource settings.

The impact of telehealth on wait time for ENT specialty care.

Audiology in rural Alaska has changed dramatically in the past 6 years by integrating store and forward telemedicine into routine practice. The Audiology Department at the Norton Sound Health Corporation in rural Nome Alaska has used store-and-forward telemedicine since 2002. Between 2002 and 2007, over 3,000 direct audiology consultations with the Ear, Nose, and Throat (ENT) Department at the Alaska Native Medical Center in Anchorage were completed. This study is a 16-year retrospective analysis of ENT specialty clinic wait times on all new patient referrals made by the Norton Sound Health Corporation providers before (1992-2001) and after the initiation of telemedicine (2002-2007). Prior to use of telemedicine by audiology and ENT, 47% of new patient referrals would wait 5 months or longer to obtain an in-person ENT appointment; this dropped to 8% of all patients in the first 3 years with telemedicine and then less than 3% of all patients in next 3 years using telemedicine. The average wait time during the first 3 years using telemedicine was 2.9 months, a 31% drop compared with the average wait time of 4.2 months for the preceding years without telemedicine. The wait time then dropped to an average of 2.1 months during the next 3 years of telemedicine, a further drop of 28% compared with the first 3 years of telemedicine usage.

Hearing Norton Sound: a community randomised trial protocol to address childhood hearing loss in rural Alaska.

INTRODUCTION: The population in rural Alaska experiences a disproprionately high burden of infection-mediated hearing loss. While the state mandates school hearing screening, many children with hearing loss are not identified or are lost to follow-up before ever receiving treatment. A robust, tribally owned healthcare system exists in Alaska, but children with hearing loss must first be identified and referred for existing infrastructure to be used. This trial will evaluate a new school hearing screening and referral process in rural Alaska, with the goal of improving timely identification and treatment of childhood hearing loss. METHODS AND ANALYSIS: Comparative effectiveness community randomised trial testing digital innovations to improve school hearing screening and referral in 15 communities in the Norton Sound region of northwest Alaska, with data collection from October 2017 to February 2020. All children (K-12) attending school in Bering Strait School District with parental informed consent and child assent will be eligible (target recruitment n=1500). Participating children will undergo both the current school hearing screen and new mobile health (mHealth) screen, with screening test validity evaluated against an audiometric assessment. Communities will be cluster randomised to continue the current primary care referral process or receive telemedicine referral for follow-up diagnosis and treatment. The primary outcome will be time to International Statistical Classification of Diseases, 10th Revision, ear/hearing diagnosis from screening date, measured in days. Secondary outcomes will include: sensitivity and specificity of current school and mHealth screening protocols measured against a benchmark audiometric assessment (air and bone conduction audiometry, tympanometry and digital otoscopy); hearing loss prevalence; hearing-related quality of life; and school performance (AIMSweb). Intention-to-treat analysis will be used. ETHICS AND DISSEMINATION: This study has been approved by the Institutional Review Boards of Alaska Area, Norton Sound and Duke University and is registered on clinicaltrials.gov. Results will be distributed with equal emphasis on scientific and community dissemination. TRIAL REGISTRATION NUMBER: NCT03309553; Pre-results.

Hearing Norton Sound: mixed methods protocol of a community randomised trial to address childhood hearing loss in rural Alaska.

INTRODUCTION: Childhood hearing loss has implications for school achievement, economic outcomes and quality of life. This study will engage rural Alaska communities in research to improve the school hearing screening and referral process, partnering with stakeholders to develop a locally derived, evidence-based solution to improve timely identification and treatment of childhood hearing loss. METHODS AND ANALYSIS: Mixed methods community randomised trial in 15 communities in the Norton Sound region of northwest Alaska. Data collection will span from April 2017 until February 2020. Qualitative and mixed methods components are described in this protocol and the community randomised trial in the companion protocol. Focus groups and community events will be held leading up to the randomised trial to obtain community perspectives on childhood hearing loss in Alaska and elicit community input during trial protocol refinement (exploratory sequential stage). Stakeholder groups, including parents, children, teachers, school administrators and community health aides, will participate, along with community leaders, tribal leaders and community members. The randomised trial will be combined with qualitative, semi-structured interviews to elicit stakeholder perspectives on the intervention (explanatory sequential stage). The five stakeholder groups described above will participate in interviews. The study will conclude with additional focus groups and community events to discuss results and provide community insight for future implementation. Concluding focus groups will include policymakers, healthcare administrators, and tribal and community leaders in addition to the stakeholder groups. Informed consent and child assent will be required. Recordings will be transcribed and deidentified, with only stakeholder group recorded. Analyses will include categorical coding as well as narrative and thematic analysis. ETHICS AND DISSEMINATION: The Hearing Norton Sound study has been approved by the Institutional Review Boards of Alaska Area, Norton Sound, and Duke University, with trial registration on clinicaltrials.gov. Study results will be distributed with equal emphasis on scientific and community dissemination. TRIAL REGISTRATION NUMBER: NCT03309553; Results.