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. 2020 Aug 21;15(8):e0237815. doi: 10.1371/journal.pone.0237815

Mothers’ perceptions of universal newborn hearing screening in transitional Albania

Birkena Qirjazi 1,2,#, Ervin Toçi 1,2,*,#, Eduard Tushe 3,, Genc Burazeri 1,, Enver Roshi 1,
Editor: Bolajoko O Olusanya4
PMCID: PMC7444524  PMID: 32822384

Abstract

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.

Introduction

Screening newborns for hearing impairment or hearing loss is important as this is one of the most frequent congenital conditions, affecting about 1–6 newborns per 1000 live births [1,2] with much higher rates among high-risk infants admitted to neonatal intensive care unit (NICU) [3]. In addition, about half of hearing impaired newborns have no known risk factors [1] highlighting the necessity to screen all newborns. Furthermore, early detection and treatment of hearing loss is important for ensuring the normal development of speech and language and avoiding negative effects on cognitive, academic and social-emotional skills of affected children [4].

Even though universal newborn hearing screening (UNHS) is mainly implemented in developed countries [5], the practice is expanding in developing countries as well [6]. The main challenge with UNHS in developed countries is the low rate of screening follow-up. For example, in USA in 2012 about one third of newborns that did not pass the first hearing screening test failed to receive timely follow-up [7]. On the other hand, implementation of UNHS in developing countries faces challenges of different natures, including limited coverage, high unaffordable cost, lack of health personnel, poor infrastructure, poor access to rural areas, socio-economic differences, etc. [8].

Besides these factors, people’s perceptions of health services constitutes an important determinant of utilization of available health services and a key element for ensuring that health interventions and services are successful [9]. Various factors contribute to shape one’s perceptions about health services being offered in a certain area including culture, traditions and different inherent characteristics of the health system/facility, the quality of interactions with the health personnel, etc. [9] with health staff behavior potentially moderating the effect of health services on patient satisfaction level [10].

There is a direct link between perception of a health service and users’ satisfaction. Research shows that the perception about the quality of a health service is strongly and directly linked with the satisfaction users get from that service and the future use of such services [11].

In this context, parents’ perception about newborn hearing screening could play an important role in improving screening program participation rates, decrease of the refusal rates and ensure that more newborns will be tested as well [12].

In Albania, a small country in South East Europe, the EUSCREEN project, universal newborn hearing screening component, started to be implemented on January 1st 2018 on three country sites and namely in Tirana, the capital city of Albania, Kukës and Pogradec municipalities [13]. Parents' opinion on certain aspects of hearing screening is important and the "acceptability" of the screening instrument by the target population is one of the criteria to be taken into consideration when applying a particular screening procedure to a community [14]. Therefore, in the context of the EUSCREEN project, a survey was carried out in order to shed light on mothers’ perceptions of various aspects of universal newborn hearing screening in Albania.

Methodology

Type of study

This is a cross-sectional study conducted during May-June 2019.

Population study and sampling

The target population of the study comprised the mothers of newborn babies in the maternity hospitals where newborn hearing screening is being implemented in Albania, and more specifically in Tirana, Kukës and Pogradec.

Since there are approximately a total of 11500 births each year in these 3 districts (about 80% in Tirana) and due to the inability to include the entire birth cohort in the study over a year, we decided to draw a sample of mothers who gave birth to infants in these districts’ maternity hospitals. WINPEPI statistical program was used to calculate the sample size based on the following parameters: alpha error = 5%, study power = 80%, assumed proportion of mothers in favor of hearing screening = 50% (this level enables maximization of sample size); acceptable difference = 4.3%; source population = 11500. The application of these parameters generated a sample size of 497 individuals.

In total, 512 mothers were interviewed (200 at Koco Gliozheni Maternity Hospital in Tirana, 200 at Queen Geraldine Maternity Hospital in Tirana as well, 57 at Kukës Maternity Hospital and 55 at Pogradec Maternity Hospital). It should be noted that among the 400 births included in the two maternity hospitals in Tirana, in 76 of cases the mothers did not reside in Tirana but in other districts, as many future mothers choose to give birth in one of the Tirana maternities. Mothers who gave birth to babies in Tirana but did not reside in Tirana lived in a large variety of cities and other areas of Albania, from south to north.

Data collection and data collection instrument

In each of the maternities included in the study, the survey instrument (questionnaire) was applied consecutively to the mothers who gave birth to one or more living children in these maternity hospitals during May-June 2019, until the preset number of questionnaires (at least) was reached.

Information on mothers’ opinions on the acceptability of newborn hearing screening was collected through a face-to-face administered questionnaire.

The questionnaire contained key socio-demographic data of mothers, such as: residence (urban vs. rural); age, marital status, education level; employment status, social class, economic situation, and overall health status.

The next section of the questionnaire contained specific questions regarding the acceptability of newborn hearing screening. Questions covered issues such as: being aware that the baby is being tested for hearing; being informed on the fact that screening is provided at this maternity hospital and the sources of this information; mothers’ impression of hearing screening process (on a 1–5 scale where 1 means "very satisfactory" and 5 "not at all satisfactory"); the level of stress or anxiety they experienced while waiting for the hearing screening response (on a 1–5 scale where 1 means "extremely" and 5 means "not at all"); mothers’ preference to be informed as soon as their child has hearing problems (yes vs. no); importance of newborn hearing screening to parents (yes vs. no); willingness to pay themselves for screening their newborn baby (yes vs. no).

The questionnaire used for this survey has been built from the local experts (the authors of this paper), based on browsing of the appropriate literature listed in the references and picking up those items considered as relevant for the study objectives. Therefore, the questionnaire has face validity, construct validity and content validity.

Ethical issues

All mothers included in this study were approached by EUSCREEN staff screening newborns’ hearing at respective maternity hospitals.

In order to be part of newborn hearing screening, each mother was required to give a written informed consent. If such consent was not retrieved, then the newborn was not screened, the mother’s interviewing was not done and this particular baby was not part of UNHS anymore.

All mothers accepting to screen their baby were required to give a formal oral consent for participating in the actual study.

All mothers agreed to participate after being thoroughly informed about the survey.

EUSCREEN project in Albania has been approved by the project counterpart in Albania, i.e. the University of Medicine, and the Ministry of Health and Social Protection of Albania.

Data analysis

Absolute numbers and the corresponding percentages were reported, in order to describe the data. Chi-square test was used to detect any statistically significant difference when comparing categorical variables.

All data analyzes were performed using the Statistical Package for Social Sciences (SPSS) statistical software, version 19.

Results

General information

A total of 512 mothers who had a live birth in one of the four selected maternity hospitals participated in the survey. The maternal mean age was 28.6 years ± 5.5 years, ranging from a minimum of 16 years (1 case) to a maximum of 47 years (1 case). Table 1 shows the general characteristics of participating mothers by birth survey site. More than one fourth (27.3%) of mothers were between 16 and 25 years old, about one quarter (24.4%) had secondary education or less, 56% resided in urban areas, 98% were married at the time of the survey, about half (50.2%) were employed, 91.2% declared to belong to middle social class, 80.9% had average economic status and 92.4% declared to have very good or good general health. Significantly higher proportions of mothers in Tirana had university education, resided in urban areas and had very good or good economic status, compared to mothers from other survey sites (Table 1). On the other hand, the proportion of employment was significantly higher among Pogradec mothers whereas the proportion of mothers with very good or good health status was higher among Kukës and Pogradec mothers compared to Tirana (Table 1). There were no significant age, marital status and social class differences by survey site (Table 1).

Table 1. General characteristics of participating mothers.

Variable Total N = 512 Survey site P-value
Tirana n = 400 Kukës n = 57 Pogradec n = 55
Age-group
16–25 years 140 (27.3)* 100 (25.0) 21 (36.8) 19 (34.5) 0.088
26–30 years 193 (37.7) 149 (37.3) 21 (36.8) 21 (38.2)
>30 year 179 (35.0) 151 (37.8) 13 (22.8) 15 (27.3)
Education level
Secondary or les 125 (24.4) 105 (26.3) 11 (19.3) 9 (16.4) <0.001
High school 203 (39.6) 131 (32.8) 36 (63.2) 36 (65.5)
University 184 (35.9) 164 (41.0) 10 (17.5) 10 (18.2)
Residence
Urban 288 (56.3) 256 (64.0) 15 (26.3) 17 (30.9) <0.001
Rural 224 (43.8) 144 (36.0) 42 (73.7) 38 (69.1)
Marital status
Single †† 10 (2.0 6 (1.5) 2 (3.6) 2 (3.6) 0.375
Married 502 (98.0) 394 (98.5) 55 (96.4) 53 (96.4)
Employment status
Employed 257 (50.2) 202 (50.5) 20 (35.1) 35 (63.6) 0.013
Not employed 245 (47.9) 190 (47.5) 37 (64.9) 18 (32.7)
Student 10 (2.0) 8 (2.0) 0 (0.0) 2 (3.6)
Social class
Low 20 (3.9) 18 (4.5) 1 (1.8) 1 (1.8) 0.131
Middle 467 (91.2) 358 (89.5) 55 (96.5) 54 (98.2)
High 25 (4.9) 24 (6.0) 1 (1.8) 0 (0.0)
Economic status
Very good, good 82 (16.0) 81 (20.3) 1 (1.8) 0 (0.0) <0.001
Average 414 (80.9) 307 (76.8) 53 (93.0) 54 (98.2)
Poor, very poor 16 (3.1) 12 (3.0) 3 (5.3) 1 (1.8)
General health status
Very good, goo 473 (92.4) 361 (90.3) 57 (100.0) 55 (100.0) 0.019
Average 37 (7.2) 37 (9.3) 0 (0.0) 0 (0.0)
Poor, very poor 2 (0.4) 2 (0.5) 0 (0.0) 0 (0.0)

* Absolute number and column percentage.

† P-value according to chi-square test.

†† Including divorced and widowed.

Mother’s perceptions about various aspects of newborn hearing screening

Table 2 shows mothers’ perceptions about different aspects of newborn hearing screening. More than 9 in 10 mothers (93%) knew what their baby was being tested for, only one third (33%) were informed beforehand that newborn hearing screening was being offered at the respective maternity hospital where they gave birth, 94% were very satisfied or satisfied with newborn hearing screening and about 62% thought that waiting for the hearing screening result was very stressful or stressful (Table 2).

Table 2. Mothers’ perception about various aspects of newborn hearing screening.

Knowing what the newborn is being tested for, n (%) Informed that hearing screening is offered in this maternity, n (%) Very satisfied or satisfied with newborn hearing screening, n (%) Waiting for screening result is very stressful or stressful, n (%)
Total 476 (93.0) 169 (33.0) 482 (94.1) 315 (61.5)
Survey site NS *** * ***
Tirana 369 (92.3) 81 (20.3) 371 (92.8) 235 (58.8)
Kukës 57 (100.0) 40 (70.2) 56 (98.2) 48 (84.2)
Pogradec 50 (90.9) 48 (87.3) 55 (100.0) 32 (58.2)
Age-group NS NS NS NS
16–25 years 125 (89.3) 47 (33.6) 127 (90.7) 95 (67.9)
26–30 years 182 (94.3) 72 (37.3) 185 (95.9) 118 (61.1)
>30 year 169 (94.4) 50 (27.9) 170 (95.0) 102 (57.0)
Education level *** ** NS NS
Secondary or les 101 (80.8) 31 (24.8) 114 (91.2) 74 (59.2)
High school 192 (94.6) 86 (42.4) 194 (95.6) 134 (66.0)
University 183 (99.5) 52 (28.3) 174 (94.6) 107 (58.2)
Residence NS * NS NS
Urban 269 (93.4) 79 (27.4) 267 (92.7) 172 (59.7)
Rural 207 (92.4) 90 (40.2) 215 (96.0) 143 (63.8)
Marital status NS NS NS NS
Single †† 8 (80.0) 5 (50.0) 9 (90.0) 6 (60.0)
Married 468 (93.2) 164 (32.7) 473 (94.2) 309 (61.6)
Employment status * NS NS NS
Employed 246 (95.7) 88 (34.2) 241 (93.8) 159 (61.9)
Not employed 220 (89.8) 78 (31.8) 231 (94.3) 152 (62.0)
Student 10 (100.0) 3 (30.0) 10 (100.0) 4 (40.0)
Social class *** * ** **
Low 11 (55.5) 7 (35.0) 15 (75.0) 11 (55.0)
Middle 440 (94.2) 160 (34.3) 442 (94.6) 280 (60.0)
High 25 (100.0) 2 (8.0) 25 (100.0) 24 (96.0)
Economic status *** NS *** NS
Very good, good 79 (96.3) 19 (23.2) 77 (93.9) 45 (54.9)
Average 390 (94.2) 146 (35.3) 395 (95.4) 259 (62.6)
Poor, very poor 7 (43.8) 4 (25.0) 10 (62.5) 11 (68.8)
General health status * ** NS **
Very good, goo 439 (92.8) 166 (35.1) 444 (93.9) 282 (59.6)
Average 36 (97.3) 3 (8.1) 36 (97.3) 31 (83.8)
Poor, very poor 1 (50.0) 0 (0.0) 2 (100.0) 2 (100.0)

NS–not significant.

* P<0.05

** P<0.01

*** P<0.001 (P-value according to chi-square test).

†† Including divorced and widowed.

Significantly higher proportions of highly educated, employed, high social class, very good or good economic or health status mothers knew what their newborn baby was being tested for compared to their respective counterparts (Table 2).

Significantly higher proportions of mothers in Kukës (70.2%) and Pogradec (87.3%) were informed that hearing screening was offered in the maternity hospital where they gave birth compared to Tirana mothers (20.3%) and such proportion was higher among rural, high school and university, low and middle social class mothers and those with very good/good self-reported health status compared to their respective counterparts.

Significantly higher proportions of mothers in Pogradec and Kukës, and those belonging to higher social class and economic status were very satisfied/satisfied with the newborn hearing screening.

Lastly, waiting for the results of newborn hearing screening was very stressful/stressful for significantly higher proportions of Kukës mothers, those belonging to high social class and those experiencing poor/very poor health status (Table 2).

Source of information about newborn hearing screening

Mothers reporting to have been informed that newborn hearing screening is offered in the maternity where they gave birth (n = 169 or 33% of participating mothers, see Table 2) were asked to mention the source of such information (Table 3). The majority of informed mothers (67.3%) were informed that newborn hearing screening was offered at this maternity by the medical staff at the time they were admitted to the maternity hospital, followed by information from family members, relatives or friends (in 8.6% of cases), local television programs talking about newborn hearing screening (4.9%), Internet (3.7%), posters in maternity hospital premises (3.7%) whereas other sources of information were less frequent.

Table 3. Source of information among mothers who know that newborn hearing screening is provided at the maternity hospital where they gave birth.

Information source Absolute number Percentage
Various relatives or friends 14 8.6
Internet 6 3.7
Work colleagues who had previously giving birth in this maternity hospital 1 0.6
From previous birth 2 1.2
At the women’s consultory 5 3.1
In the hospital when I was hospitalized (from staff working here) 109 67.3
Poster placed in maternity hospital premises 6 3.7
Relatives who work at the maternity hospital 3 1.9
Relatives undergoing hearing test in maternity 2 1.2
Relatives previously giving birth 8 4.9
Television (in general) 3 1.9
TV at the maternity waiting hall 3 1.9
Total 162* 100.0

* Any discrepancy with the total number (in this case n = 169) is due to missing information.

Other aspects related to the newborn hearing screening

Participating mothers were also asked the following questions: "If your child has hearing problems, would you prefer to know this early?"; "Do you think it's important for all babies to be tested for hearing in the early days of life?" and "Would you be willing to pay for a hearing test?" Analysis of their responses revealed that all mothers (100%) included in the study answered “Yes” to each of these questions (Table 4).

Table 4. Opinions related to other aspects of newborn hearing screening.

Question Absolute number Percentage
If your child has hearing problems, would you prefer to know this early?—Yes 512 100.0
Do you think it's important for all babies to be tested for hearing in the early days of life?—Yes 512 100.0
Would you be willing to pay for a hearing screening?—Yes 512 100.0

Discussion

This study generated novel and interesting information that sheds light on various aspects related to the acceptability of universal newborn hearing screening program, an activity being piloted in three regions of Albania, in the framework of the EUSCREEN project.

The overwhelming majority (93%) of mothers who have given birth to children in the maternities included in the EUSCREEN project in Albania were aware of what their baby was being tested for. This suggests that the staff involved in hearing screening in all 4 participating maternity hospitals has adequately and appropriately explained to mothers the details of hearing test.

Knowledge of what the baby was being screened for was significantly and positively related to educational level, social class, economic status and overall health status. This finding has important implications for the future, suggesting that hearing screening explanation and education may need to be adapted to various educational, social, economic, and health groups for greater effectiveness.

Two-thirds of mothers who gave birth to babies in the maternities included in the EUSCREEN project in Albania were not informed that hearing screening was provided at these maternities. This finding is similar to previous research in developing countries. For example a study from Nigeria, where there is no national universal newborn hearing screening but neonatal hearing screening is offered in the frame of research projects, suggested that about 63% of mothers were not aware of neonatal hearing screening as well [15]. This finding suggests that greater work needs to be done so that prospective mothers and the general public are informed about the screening of newborn hearing and the places where this testing is offered. Research shows that better informed mothers have more positive attitudes toward universal newborn hearing screening programs [16].

A significantly higher percentage of babies born to mothers residing in Kukës and Pogradec, in rural areas, mothers with secondary or tertiary education, mothers with low or middle social status and mothers with very good/good health status were informed that newborn hearing screening was provided in the maternity hospital compared to their respective counterparts. These findings suggest that screening information efforts should target less informed groups.

On the other hand, about 80% of mothers in Tirana were not informed that hearing screening was provided in the two Tirana maternity hospitals. This result seems to be partly influenced by the fact that about 20% of births here occurred among mothers residing in other cities not being specifically covered by awareness campaigns for newborn hearing screening. Another explanation can be found in the much more dynamic, active and very heterogeneous population in Tirana compared to Kukës and Pogradec, where the latter are relatively small communities and the information is disseminated faster and more effectively than in Tirana. However, the finding regarding Tirana suggests that information campaigns in big cities should be different from those employed in small cities in terms of the aggressiveness of the coverage and the ways in which information is communicated.

About 67% of mothers informed about newborn hearing screening had received this information at the maternity ward after giving birth, by the relevant medical staff there, just as it occurs in most countries [16]. In Nigeria mothers learned about the availability of newborn hearing screening from antenatal clinics (28%), media (11%), family members or friends (17%), Internet (44%), etc. [15]. In Hong Kong about 69% of mothers learned about neonatal hearing screening at the postnatal ward premises and other information sources included previous screening of older children (16.9%), friends or relatives (5.9%) etc. [16]. It seems that in Albania, for the time being, the information sources other than the maternity hospitals where UNHS is taking place play a rather small role in informing the population about this screening program. On the other hand, international research suggests that most mothers would like to be informed about newborn hearing screening well in advance and probably not in maternity hospital premises [17]. The implication for the future is that there is need to improve and expand UNHS awareness campaigns in Albania to cover also mass media, social media and the Internet.

The overwhelming majority (94.1%) of mothers who gave birth to children in the maternities included in the EUSCREEN project in Albania were either very satisfied or satisfied with the hearing screening provided to their babies. This data suggests that the staff involved in hearing screening in all 4 maternities in Albania has performed the procedure properly, gaining the confidence of mothers. Parents’ positive attitudes towards UNHS has also been reported in literature [16, 18]. Also, there was a positive relationship between satisfaction with newborn hearing screening with social class and economic status, in accordance with previous research [15].

On the other hand, a smaller percentage of mothers in Tirana are more satisfied with hearing screening compared to mothers who gave birth in Kukës or Pogradec. The implication is that measures should also be taken in Tirana to increase the percentage of mothers who are very satisfied/satisfied with the screening of their newborn baby. In this regard, efforts should be focused on mothers with low social status and disadvantaged economic status.

In our study about two-thirds of mothers (61.5%) were very stressful or stressful waiting for the hearing screening result. Being anxious about the screening results is also reported by previous research and it seems to be mainly associated with the inconclusive message (the result of the screening test is not definitive and there is need for further testing) at the end of each screening stage [19]. In addition, the role of screeners seems to be fundamental for reassuring parents about the need to be screened again [19]. The implications are that a better job should be done by the relevant hearing screening staff for supporting mothers and informing them about the screening procedure, stages and interpretation of results and intervention opportunities.

All mothers included in the current study (100%) stated that they would prefer to know early if their child has hearing problems. This finding best supports the value and importance of hearing screening in newborns as this procedure provides the fastest possible responses (meeting future mothers' expectations) and ensures early detection of the problem, greatly increasing the chances of early treatment, and potential full rehabilitation of infants affected by these conditions. The willingness to know early if own child has a hearing problem is supported by international research [16, 20]. Knowing early does not take away the grief that comes with the unfavorable diagnosis but it could give parents the needed time to somehow getting used to this situation and it can give them a sense of being able to take action quickly [20].

The importance of early detection and early treatment is also supported by the opinions of the mothers included in the study, where all mothers (100%) stated that it is very important for all babies to be tested for hearing in the early days of life, similar to international research results showing a very high level of parental agreement with regard to implementation and importance of newborn hearing screening [15, 18]. This is an additional argument in favor of universal hearing screening of all newborns, as this procedure is not only of medical and general population health benefit but it also enables the achievement of an aspiration, expectation or ideal of future mothers as an integral part of the universal right to good health.

As literature review shows, there are evidence-based arguments to support the implementation of UNHS [5]. However, early identification needs to be followed by timely fitting, amplification and family support for the affected babies; this requires effective follow-up of babies with hearing loss being detected by UNHS and often this represents the real challenge of the UNHS [7]. Research shows that on the one hand parents do support early identification of hearing loss but on the other hand they are rightly concerned about the effectiveness of follow-up and appropriate treatment for the affected babies [21]. Indeed, early identification [within the first 6 months of life] of hearing loss or impairment is accompanied by many different challenges for the parents, including timing, early amplification issues, information and support, daily management issues, etc. [21], issues that call for the development of services that appropriately address early identification of hearing impairment [21]. In addition, it should be kept in mind that most mothers have rather a limited understanding of their baby’s hearing development milestones [16], which means that passing the newborn screening test is not a full assurance that future hearing problems will not occur and therefore mothers have to be able to continually monitor the baby’s growth and detect any warning signs that something is not right with baby’s hearing [16]. The relevant implication is that prospective mothers need to be better educated about child hearing development milestones as well.

All mothers included in the study (100%) stated that they would be willing to pay for their child's hearing medical control. This finding assures us once again about the public perception that universal newborn hearing screening is important, and that is why all possible efforts should be taken to achieve full coverage of all babies born in Albania. The finding specifically suggests that such activity is so positively perceived and highly appreciated by mothers as there may be room for discussions about potential parental involvement in universal hearing screening funding schemes (in the event of an absolute inability to be 100% covered by public funds), economic contribution which undoubtedly needs to be tailored to the socio-economic level and other factors and based on the results of extensive consultations with all stakeholders.

Study limitations

Our study has several limitations. The interviewing of consecutive mothers showing up to give birth in the selected maternity hospitals does not exclude the selection bias. However, we covered all the UNHS pilot sites in Albania and also selected the mothers proportional to size of respective maternities, in order to reduce the selection bias. On the other hand, the study sample is rather small and does not allow the unconditional generalization of the results. Finally, the cross-sectional nature of the study does not allow us to draw any causal conclusions.

The strong point of the present study is that it offers for the first time a glimpse on mothers’ perception about newborn hearing screening, a largely under researched topic in Albania.

Conclusions

In Albania mothers in general had positive attitudes toward universal newborn hearing screening program. The overwhelming majority of mothers giving birth to maternity hospitals where UNHS is being implemented were aware what their newborn is tested for, meaning the screening staff has done its job quite well. On the other hand, about two-thirds of mothers didn’t know the hearing screening was offered in these maternity hospitals with considerable differences between the capital and two other regions, implying further targeted information efforts. About two-thirds of mothers were stressed or anxious waiting for the testing results. All mothers were in favor of early detection of hearing problems among newborn babies, they wished to know early if their baby has a hearing problem and they are willing to contribute financially for testing the hearing of their newborn babies. The various significant socio-demographic differences noticed regarding mother’s awareness, information and satisfaction rates about various UNHS elements and perceived stress of screening results should guide information and education campaigns. The public willingness to financially contribute in order to screen the hearing of newborns might be an opportunity for scaling-up the UNHS in the country.

Supporting information

S1 Database

(SAV)

Data Availability

All relevant data are within the manuscript and its Supporting Information file.

Funding Statement

This project has received funding from the European Union’s Horizon 2020 research and innovation program under grant agreement No 733352. This study sponsor had no role in the study design, or the collection, analysis and interpretation of the data or in the writing of the report or in the decision to submit the article for publication.

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Decision Letter 0

Bolajoko O Olusanya

7 Jul 2020

PONE-D-20-07930

Mothers’ perceptions of universal newborn hearing screening in transitional Albania

PLOS ONE

Dear Dr. Toçi,

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Reviewers' comments:

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Comments to the Author

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Reviewer #1: No

Reviewer #2: Yes

**********

2. Has the statistical analysis been performed appropriately and rigorously?

Reviewer #1: No

Reviewer #2: No

**********

3. Have the authors made all data underlying the findings in their manuscript fully available?

The PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception (please refer to the Data Availability Statement in the manuscript PDF file). The data should be provided as part of the manuscript or its supporting information, or deposited to a public repository. For example, in addition to summary statistics, the data points behind means, medians and variance measures should be available. If there are restrictions on publicly sharing data—e.g. participant privacy or use of data from a third party—those must be specified.

Reviewer #1: Yes

Reviewer #2: Yes

**********

4. Is the manuscript presented in an intelligible fashion and written in standard English?

PLOS ONE does not copyedit accepted manuscripts, so the language in submitted articles must be clear, correct, and unambiguous. Any typographical or grammatical errors should be corrected at revision, so please note any specific errors here.

Reviewer #1: No

Reviewer #2: Yes

**********

5. Review Comments to the Author

Please use the space provided to explain your answers to the questions above. You may also include additional comments for the author, including concerns about dual publication, research ethics, or publication ethics. (Please upload your review as an attachment if it exceeds 20,000 characters)

Reviewer #1: A cross-sectional study was carried out to explore parents’ perceptions about various elements three sites of UNHS in Albania and included 512 mothers. While the study is a needed study as there is paucity of research from this region, there are several concerns.

1. The words 'mothers' and 'parents' are used interchangeably throughout the manuscript, making it difficult for a reader to derive conclusions.

2. The questionnaire development does not follow standard procedures for questionnaire development and validation. There is no mention who were these 'local experts'?Also no mention of the what was this 'appropriate literature' challenges the overall validity of the questionnaire and thereby the methodology. The usage of terms like 'thought to have' should be avoided in scientific writing.

3. The study was approved by Ministry of Health and Social Protection and University of Medicine, however there is no mention of Institutional committee approval. Also why was oral consent considered valid? Shouldnt a written informed consent be taken from all the participants.

4. The analysis was restricted to Chi-square test, and no detailed statistical exploration was carried out.

5. Were any standard checklists used to determine the general health status as well as economic status?

6. Clinical implications, future directions and limitations could have been added to enrich the discussion further.

7 Overall, the manuscript would benefit if read and corrected by a native English language expert in scientific writing.

Reviewer #2: 1.The manuscript is technically sound, and the data support the conclusions . However the conclusion should be precise and reflect the study objective.

2.Statistical analysis were performed but the author should use logistic regression to aid determination of factors that were most likely responsible for perception of mothers.

3. The authors made all data underlying the findings in their manuscript fully available.

4.The manuscript was presented in an intelligible fashion and written in standard English however the word stress/stressed used need to be clarified to avoid ambiguity.

**********

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Reviewer #1: No

Reviewer #2: Yes: Abayomi Oladapo Somefun

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PLoS One. 2020 Aug 21;15(8):e0237815. doi: 10.1371/journal.pone.0237815.r002

Author response to Decision Letter 0


8 Jul 2020

Response to Reviewers

Manuscript PONE-D-20-07930: “Mothers’ perceptions of universal newborn hearing screening in transitional Albania”

We thank the reviewers for their constructive comments which have helped us to improve the content and format of the manuscript, which we hope is now acceptable for publication in the PLOS ONE Journal.

Below, we address point-by-point all the reviewers’ comments:

Reviewer #1:

1. “The words 'mothers' and 'parents' are used interchangeably throughout the manuscript, making it difficult for a reader to derive conclusions.”

Response: We thank the reviewer for this valuable comment and suggestion. In line with reviewer’s suggestion, we decided to keep only the word “mother’s” and therefore in the revised manuscript we have now only used the terms “mother’s”, where appropriate.

2. “The questionnaire development does not follow standard procedures for questionnaire development and validation. There is no mention who were these 'local experts'? Also no mention of the what was this 'appropriate literature' challenges the overall validity of the questionnaire and thereby the methodology. The usage of terms like 'thought to have' should be avoided in scientific writing”

Response: Point well-taken. In the revised manuscript, we have now reformulated the relevant section in order to reflect the concerns and suggestions of the reviewer, so that now this section reads like this: “The questionnaire used for this survey has been built from the local experts (the authors of this paper), based on browsing of the appropriate literature listed in the references and picking up those items considered as relevant for the study objectives. Therefore, the questionnaire has face validity, construct validity and content validity.”

3a. “The study was approved by Ministry of Health and Social Protection and University of Medicine, however there is no mention of Institutional committee approval.

Response: We thank the reviewer for bringing this point into our attention. We are trying to explain again the situation regarding this point: The EUSCREEN in Albania has been approved by the Ministry of Health and Social Protection (MoHSP) and the implementing partner, the University of Medicine. We (the project coordinator in Albania) have the document stating such an agreement and approval and, if the reviewer thinks it necessary, we can make this document available to him/her or to any other interested party. This approval is signed by the Minister of Health and Social Protection and the Rector of the University of Medicine. The Bio-ethical Committee that is functioning in Albania is dependent on the MoHSP and therefore, the agreement made by the Minister of Health has covered the issue of the Bio-ethical Committee as well. This approval includes all data collection process in the framework of EUSCREEN study in Albania, including hearing screening tests and other questionnaires for the mothers and or staff. For example, in the framework of EUSCREEN data collection in Albania, we ask the mothers also about various risk factors for hearing impairment (such as family history, genetic disorders, use of antibiotics during pregnancy etc.), and many other socio-demographic factors (age, education, residence, financial situation, etc.). Other investigators from the EUSCREEN consortium have also come to Albania and interviewed the staff for their purposes, and they have even published papers based on that information and have stated exactly the approval given by the MoHSP and the agreement between MoHSP and the University of Medicine as a proof of ethical approval of the study (reference: Bussé AM, Qirjazi B, Goedegebure A, et al. Implementation of a neonatal hearing screening programme in three provinces in Albania. Int J Pediatr Otorhinolaryngol. 2020;134:110039. doi:10.1016/j.ijporl.2020.110039). In this context, the data collection made for the purposes of this study is in total agreement with the approval given by the MoHSP and following the data collection in the framework of EUSCREEN in Albania.

In the manuscript we have explained this situation with the following paragraph: “The UNHS project in Albania was approved by Ministry of Health and Social Protection, in collaboration with the implementing partner, the University of Medicine. This approval comprised all elements of screening procedure, including various data collection activities from the mothers (i.e. demographic and socioeconomic questionnaire, parents’ opinion on newborn hearing screening, reasons for refusing screening, etc.).”

3b. “Also why was oral consent considered valid? Shouldnt a written informed consent be taken from all the participants.”

Response: Point well-taken. In order to be part of the newborn screening, each mother has to give a written informed consent, which is administered by the hospital staff. If such written consent was not given, the mother and the baby could not be part of the UNHS. Among mothes giving their written consent to be part of the screening process, for every other survey we formally asked also for the oral consent of the mothers.

In order for this to be clear, we have now reformulated the respective section in the manuscript, so as now it reads like this: “In order to be part of hearing screening, each mother was required to give a written informed consent. If such consent was not retrieved, then the newborn was not screened, the mother’s interviewing was not done and this particular baby was not part of UNHS anymore. All mothers accepting to screen their baby were required to give a formal oral consent for participating in the actual study.”

4. “The analysis was restricted to Chi-square test, and no detailed statistical exploration was carried out”

Response: We thank the reviewer for this comment. Actually, the data analysis was guided by the nature of the variables in our disposal. We kindly remind the reviewer that this is a small mainly descriptive study that does not aim to engage into complex statistical models or analysis but it rather tries to give an overall picture of mother’s perceptions about UNHS in Albania. On the other hand, the other papers from international area also have reported simple analytical tools in order to give such a picture. At the end, we did not aim to detect the predictors or factors related to various mothers’ perceptions and therefore the chi square test was regarded as an optimal test to check for the present differences in our case.

5. “Were any standard checklists used to determine the general health status as well as economic status?”

Response: We thank the reviewer for this comment. Actually, the general health status and the economic status of the mothers was self-reported, based on the well-recognized questions: “How would you rate your general health status? (ranging from 1 (Very good) to 5 (very poor)” and “On a scale from1 (very poor) to 5 (not poor), how do you consider yourself?”

6. “Clinical implications, future directions and limitations could have been added to enrich the discussion further”

Response: We thank the reviewer for this suggestion. In the revised manuscript we have now made some additional efforts to address the points raised by the reviewer. The added paragraphs are listed below:

“On the other hand, international research suggests that most mothers would like to be informed about newborn hearing screening well in advance and probably not in maternity hospital premises [17]. The implication is that there is need to improve and expand UNHS awareness campaigns to cover also mass media, social media and the Internet”

“In addition, it should be kept in mind that most mothers have rather a limited understanding of their baby’s hearing development milestones [16], which means that passing the newborn screening test is not a full assurance that future hearing problems will not occur and therefore mothers have to be able to continually monitor the baby’s growth and detect any warning signs that something is not right with baby’s hearing [16]. The relevant implication is that prospective mothers need to be better educated about child hearing development milestones as well.”

In line with reviewer’s suggestion, we have now added a “Study limitations” section, which in the revised manuscript reads like this:

“Our study has several limitations. The interviewing of consecutive mothers showing up to give birth in the selected maternity hospitals does not exclude the selection bias. However, we covered all the UNHS pilot sites in Albania and also selected the mother’s proportional to size, in order to reduce the selection bias. On the other hand, the study sample is rather small and does not allow the unconditional generalization of the results. Finally, the cross-sectional nature of the study does not allow us to draw any causal conclusions.

The strong point of the present study is that it offers for the first time a glimpse on mothers’ perception about newborn hearing screening, an under researched topic in Albania.”

7. “Overall, the manuscript would benefit if read and corrected by a native English language expert in scientific writing”

Response: We thank the reviewer for this suggestion. We have now revised the English language of the manuscript and have done all the necessary changes in order to comply with this suggestion. The changes are marked throughout the manuscript, where necessary.

Reviewer #2:

1. “The manuscript is technically sound, and the data support the conclusions . However the conclusion should be precise and reflect the study objective.”

Response: We thank the reviewer for this valuable comment and suggestion. We have gone through our results and discussion and feel that in general they are in concordance with each-other; we have tried to structure the discussion following our results. In addition, we have now expanded the discussion as to include some other relevant topics and have also added the “Study limitation” section. We do hope that these changes will satisfy the reviewer’s concern about this point.

2. “Statistical analysis were performed but the author should use logistic regression to aid determination of factors that were most likely responsible for perception of mothers.”

Response: We thank the reviewer for this comment. Actually, the data analysis was guided by the nature of the variables in our disposal. We kindly remind the reviewer that this is a small mainly descriptive study that does not aim to engage into complex statistical models or analysis but it rather tries to give an overall picture of mother’s perceptions about UNHS in Albania. On the other hand, the other papers from international area also have reported simple analytical tools in order to give such a picture. At the end, we did not aim to detect the predictors or factors related to various mothers’ perceptions and therefore the chi square test was regarded as an optimal test to check for the present differences in our case.

3. “The authors made all data underlying the findings in their manuscript fully available.”

Response: We agree.

3. “The manuscript was presented in an intelligible fashion and written in standard English however the word stress/stressed used need to be clarified to avoid ambiguity.”

Response: We thank the reviewer for this suggestion. We have now added the word “anxious” next to the word “stressed” in order to not confuse the reader. In the revised manuscript, the only sentence where the word “stressed” is used, now reads like this “About two-thirds of mothers were stressed or anxious waiting for the testing results.”

In the other circumstances where the word “stress” is used it is followed by an explanation that this refers to the anxiety waiting for the hearing screening results. For example, in the manuscript, lines 294-298 state that “In our study about two-thirds of mothers (61.5%) were very stressful or stressful waiting for the hearing screening result. Being anxious about the screening results is also reported by previous research and it seems to be mainly associated with the inconclusive message (the result of the screening test is not definitive and there is need for further testing) at the end of each screening stage [18]”. To our opinion, the reader is clear enough that the stress here refers to the anxiety related to waiting for the hearing screening results.

Attachment

Submitted filename: Response to reviewers.doc

Decision Letter 1

Bolajoko O Olusanya

4 Aug 2020

Mothers’ perceptions of universal newborn hearing screening in transitional Albania

PONE-D-20-07930R1

Dear Dr. Toçi,

We’re pleased to inform you that your manuscript has been judged scientifically suitable for publication and will be formally accepted for publication once it meets all outstanding technical requirements.

Within one week, you’ll receive an e-mail detailing the required amendments. When these have been addressed, you’ll receive a formal acceptance letter and your manuscript will be scheduled for publication.

An invoice for payment will follow shortly after the formal acceptance. To ensure an efficient process, please log into Editorial Manager at http://www.editorialmanager.com/pone/, click the 'Update My Information' link at the top of the page, and double check that your user information is up-to-date. If you have any billing related questions, please contact our Author Billing department directly at authorbilling@plos.org.

If your institution or institutions have a press office, please notify them about your upcoming paper to help maximize its impact. If they’ll be preparing press materials, please inform our press team as soon as possible -- no later than 48 hours after receiving the formal acceptance. Your manuscript will remain under strict press embargo until 2 pm Eastern Time on the date of publication. For more information, please contact onepress@plos.org.

Kind regards,

Bolajoko O. Olusanya, MBBS, FMCPaed, FRCPCH, PhD

Academic Editor

PLOS ONE

Additional Editor Comments (optional):

Reviewers' comments:

Reviewer's Responses to Questions

Comments to the Author

1. If the authors have adequately addressed your comments raised in a previous round of review and you feel that this manuscript is now acceptable for publication, you may indicate that here to bypass the “Comments to the Author” section, enter your conflict of interest statement in the “Confidential to Editor” section, and submit your "Accept" recommendation.

Reviewer #1: All comments have been addressed

**********

2. Is the manuscript technically sound, and do the data support the conclusions?

The manuscript must describe a technically sound piece of scientific research with data that supports the conclusions. Experiments must have been conducted rigorously, with appropriate controls, replication, and sample sizes. The conclusions must be drawn appropriately based on the data presented.

Reviewer #1: Partly

**********

3. Has the statistical analysis been performed appropriately and rigorously?

Reviewer #1: Yes

**********

4. Have the authors made all data underlying the findings in their manuscript fully available?

The PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception (please refer to the Data Availability Statement in the manuscript PDF file). The data should be provided as part of the manuscript or its supporting information, or deposited to a public repository. For example, in addition to summary statistics, the data points behind means, medians and variance measures should be available. If there are restrictions on publicly sharing data—e.g. participant privacy or use of data from a third party—those must be specified.

Reviewer #1: Yes

**********

5. Is the manuscript presented in an intelligible fashion and written in standard English?

PLOS ONE does not copyedit accepted manuscripts, so the language in submitted articles must be clear, correct, and unambiguous. Any typographical or grammatical errors should be corrected at revision, so please note any specific errors here.

Reviewer #1: Yes

**********

6. Review Comments to the Author

Please use the space provided to explain your answers to the questions above. You may also include additional comments for the author, including concerns about dual publication, research ethics, or publication ethics. (Please upload your review as an attachment if it exceeds 20,000 characters)

Reviewer #1: Satisfactory changes and justification done. However, i suggestion to separate the questionnaire into the demographic details part and the questionnaire aspect

**********

7. PLOS authors have the option to publish the peer review history of their article (what does this mean?). If published, this will include your full peer review and any attached files.

If you choose “no”, your identity will remain anonymous but your review may still be made public.

Do you want your identity to be public for this peer review? For information about this choice, including consent withdrawal, please see our Privacy Policy.

Reviewer #1: No

Acceptance letter

Bolajoko O Olusanya

11 Aug 2020

PONE-D-20-07930R1

Mothers’ perceptions of universal newborn hearing screening in transitional Albania

Dear Dr. Toçi:

I'm pleased to inform you that your manuscript has been deemed suitable for publication in PLOS ONE. Congratulations! Your manuscript is now with our production department.

If your institution or institutions have a press office, please let them know about your upcoming paper now to help maximize its impact. If they'll be preparing press materials, please inform our press team within the next 48 hours. Your manuscript will remain under strict press embargo until 2 pm Eastern Time on the date of publication. For more information please contact onepress@plos.org.

If we can help with anything else, please email us at plosone@plos.org.

Thank you for submitting your work to PLOS ONE and supporting open access.

Kind regards,

PLOS ONE Editorial Office Staff

on behalf of

Dr. Bolajoko O. Olusanya

Academic Editor

PLOS ONE

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