Modelling the cost-effectiveness of a newborn hearing screening programme; usability and pitfalls.

OBJECTIVE: The EUSCREEN project concerns the study of European vision and hearing screening programmes. Part of the project was the development of a cost-effectiveness model to analyse such programmes. We describe the development and usability of an online tool to enable stakeholders to design, analyse or modify a newborn hearing screening (NHS) programme. DESIGN: Data from literature, from existing NHS programmes, and observations by users were used to develop and refine the tool. Required inputs include prevalence of the hearing impairment, test sequence and its timing, attendance, sensitivity, and specificity of each screening step. Outputs include the number of cases detected and the costs of screening and diagnostics. STUDY SAMPLE: Eleven NHS programmes with reliable data. RESULTS: Three analyses are presented, exploring the effect of low attendance, number of screening steps, testing in the maternity ward, or screening at a later age, on the benefits and costs of the programme. Knowledge of the epidemiology of a staged screening programme is crucial when using the tool. CONCLUSIONS: This study presents a tool intended to aid stakeholders to design a new or analyse an existing hearing screening programme in terms of benefits and costs.

An Albanian translation of a questionnaire for self-reported tinnitus assessment.

OBJECTIVE: To our knowledge, there is no published study investigating the characteristics of people experiencing tinnitus in Albania. Such a study would be important, providing the basis for further research in this region and contributing to a wider understanding of tinnitus heterogeneity across different geographic locations. The main objective of this study was to develop an Albanian translation of a standardised questionnaire for tinnitus research, namely the European School for Interdisciplinary Tinnitus Research-Screening Questionnaire (ESIT-SQ). A secondary objective was to assess its applicability and usefulness by conducting an exploratory survey on a small sample of the Albanian tinnitus population. DESIGN AND STUDY SAMPLE: Three translators were recruited to create the Albanian ESIT-SQ translation following good practice guidelines. Using this questionnaire, data from 107 patients attending otolaryngology clinics in Albania were collected. RESULTS: Participants reporting various degrees of tinnitus symptom severity had distinct phenotypic characteristics. Application of a random forest approach on this preliminary dataset showed that self-reported hearing difficulty, and tinnitus duration, pitch and temporal manifestation were important variables for predicting tinnitus symptom severity. CONCLUSIONS: Our study provided an Albanian translation of the ESIT-SQ and demonstrated that it is a useful tool for tinnitus profiling and subgrouping.

Cost-Effectiveness of Neonatal Hearing Screening Programs: A Micro-Simulation Modeling Analysis.

OBJECTIVES: Early detection of neonatal hearing impairment moderates the negative effects on speech and language development. Universal neonatal hearing screening protocols vary in tests used, timing of testing and the number of stages of screening. This study estimated the cost-effectiveness of various protocols in the preparation of implementation of neonatal hearing screening in Albania. DESIGN: A micro-simulation model was developed using input on demography, natural history of neonatal hearing impairment, screening characteristics and treatment. Parameter values were derived from a review of the literature and expert opinion. We simulated multiple protocols using otoacoustic emissions (OAE) and automated auditory brainstem response (aABR), varying the test type, timing and number of stages. Cost-effectiveness was analyzed over a life-time horizon. RESULTS: The two best protocols for well infants were OAE followed by aABR (i.e., two-stage OAE-aABR) testing in the maternity ward and single-aABR testing. Incremental cost-effectiveness ratios were €4181 and €78,077 per quality-adjusted life-year gained, respectively. Single-aABR screening led to more cases being detected compared to a two-stage screening program. However, it also resulted in higher referral rates, which increased the total costs of diagnostics. Multi-staged screening decreased referral rates but may increase the number of missed cases due to false-negative test results and nonattendance. CONCLUSIONS: Only the 2-stage OAE-aABR (maternity ward) protocol was below the willingness-to-pay threshold of €10,413 for Albania, as suggested by the World Health Organization, and was found to be cost-effective. This study is among the few to assess neonatal hearing screening programs over a life-time horizon and the first to predict the cost-effectiveness of multiple screening scenarios.

Implementation of Newborn Hearing Screening in Albania.

Newborn hearing screening (NHS) was implemented in Albania in four maternity hospitals in 2018 and 2019. Implementation outcome, screening outcome, and screening quality measures were evaluated. Infants were first screened by midwives and nurses before discharge from the maternity hospital and returned for follow-up screening. Acceptability, appropriateness, feasibility, adoption, fidelity, coverage, attendance, and stepwise and final-referral rates were assessed by onsite observations, interviews, questionnaires, and a screening database. A post hoc analysis was performed to identify reasons for loss to follow up (LTFU) in a multivariate logistic regression. In total, 22,818 infants were born, of which 96.6% were screened. For the second screening step, 33.6% of infants were LTFU, 40.4% for the third, and 35.8% for diagnostic assessment. Twenty-two (0.1%) were diagnosed with hearing loss of ≥40 dB, six unilateral. NHS was appropriate and feasible: most infants are born in maternity hospitals, hence nurses and midwives could perform screening, and screening rooms and logistic support were supplied. Adoption among screeners was good. Referral rates decreased steadily, reflecting increasing skill. Occasionally, screening was repeated during a screening step, contrary to the protocol. NHS in Albania was implemented successfully, though LTFU was high. It is important to have effective data tracking and supervision throughout the screening.

Mothers' perceptions of universal newborn hearing screening in transitional Albania.

The universal newborn hearing screening (UNHS) component of the multi-center EUSCREEN project is being piloted in Albania since January 1st 2018. The aim of this study was to explore mothers' perceptions about various elements of UNHS in Albania. A cross-sectional study was carried out in the three sites of UNHS in Albania, namely in Tirana, Kukës and Pogradec during May-June 2019. During this period 512 consecutively approached mothers giving birth to included maternity hospitals were interviewed face-to-face about different aspects of UNHS. Basic socio-demographic and socioeconomic information was also collected. Mean age of participating mothers was 28.6 years ± 5.5 years. The overwhelming majority (93%) of mothers knew what their baby was being tested for, 33% were aware that hearing screening was offered in maternity hospital, 94% were very satisfied/satisfied with UNHS and about 62% were very stressed/stressed waiting for screening results, with significant sociodemographic and socioeconomic differences. The main information source about UNHS was screening staff in the maternity hospitals where mothers gave birth, reported in 67% of cases. All mothers (100%) agreed on the importance of early detection of newborn hearing problems, all mothers were willing to be informed early if their newborn baby had a hearing problem and all mothers were willing to contribute financially for testing the hearing of their newborn baby. These findings should guide information and education campaigns about UNHS in Albania. The public willingness to financially support neonatal hearing testing should be considered as an opportunity to achieve universal newborn hearing screening in the country.

Implementation of a neonatal hearing screening programme in three provinces in Albania.

OBJECTIVES: The EUSCREEN study compares the cost-effectiveness of paediatric hearing screening programmes and aims to develop a cost-effectiveness model for this purpose. Alongside and informed by the development of the model, neonatal hearing screening (NHS) is implemented in Albania. We report on the first year. METHODS: An implementation plan was made addressing objectives, target population, screening protocol, screener training, screening devices, care pathways and follow up. NHS started January 1st, 2018 in four maternity hospitals: two in Tirana, one in Pogradec and one in Kukës, representing both urban and rural areas. OAE-OAE-aABR was used to screen well infants in maternity hospitals, whereas aABR-aABR was used in neonatal intensive care units and in mountainous Kukës for all infants. Screeners' uptake and attitudes towards screening and quality of screening were assessed by distributing questionnaires and visiting the maternity hospitals. The result of screening, diagnostics, follow up and entry into early intervention were registered in a database and monitored. RESULTS: Screeners were keen to improve their skills in screening and considered NHS valuable for Albanian health care. The number of "fail" outcomes after the first screen was high initially but decreased to less than 10% after eight months. In 2018, 11,507 infants were born in the four participating maternity hospitals, 10,925 (94.9%) of whom were screened in the first step. For 486 infants the result of screening was not registered. For the first screen, ten parents declined, eight infants died and one infant was discharged before screening could be performed. In 1115 (10.2%) infants the test either could not be performed or the threshold was not reached; 361 (32,4%) of these did not attend the second screen. For the third screen 31 (34.4%) out of 90 did not attend. Reasons given were: parents declined (124), lived too far from screening location (95), their infant died (11), had other health issues (7), or was screened in private clinic (17), no reason given (138). CONCLUSIONS: Implementation of NHS in Albania is feasible despite continuing challenges. Acceptance was high for the first screen. However, 32.4% of 1115 infants did not attend the second screen, after a "fail" outcome for the first test.

Neonatal hearing screening in Albania: results from an ongoing universal screening program.

The paper describes the outcomes of an ongoing universal hearing screening program in Tirana, Albania. The main objectives of the project were the evaluation of the feasibility of a neonatal hearing screening program in Albania, and an evaluation of the prevalence of risk factors in the NICU environment. One thousand five hundred and sixty-one (1561) infants from both the WB and NICU were screened with transient evoked otoacoustic emissions (TEOAE). A detailed history of risk factors was collected in each case, thus it was possible to evaluate the main factors influencing the output of the screening program. It was concluded that the program had the capacity to identify infants with congenital hearing loss provided that an informative component is well-structured and delivered. Also, although the prevalence of risk factors appeared high, the reduction of 'case leakage' would allow the precise estimation of the incidence of hearing loss in the Albanian population.