Assessing hearing loss in older adults with a single question and person characteristics; Comparison with pure tone audiometry in the Rotterdam Study.

INTRODUCTION: Hearing loss (HL) is a frequent problem among the elderly and has been studied in many cohort studies. However, pure tone audiometry-the gold standard-is rather time-consuming and costly for large population-based studies. We have investigated if self-reported hearing loss, using a multiple choice question, can be used to assess HL in absence of pure tone audiometry.
METHODS: This study was performed within 4,906 participants of the Rotterdam Study. The question (in Dutch) that was investigated was: 'Do you have any difficulty with your hearing (without hearing aids)?'. The answer options were: 'never', 'sometimes', 'often' and 'daily'. Mild hearing loss or worse was defined as PTA0.5-4(Pure Tone Average 0.5, 1, 2 & 4 kHz) ≥20dBHL and moderate HL or worse as ≥35dBHL. A univariable linear regression model was fitted with the PTA0.5-4 and the answer to the question. Subsequently, sex, age and education were added in a multivariable linear regression model. The ability of the question to classify HL, accounting for sex, age and education, was explored through logistic regression models creating prediction estimates, which were plotted in ROC curves.
RESULTS: The variance explained (R2) by the univariable regression was 0.37, which increased substantially after adding age (R2 = 0.60). The addition of sex and educational level, however, did not alter the R2 (0.61). The ability of the question to classify hearing loss, reflected in the area under the curve (AUC), was 0.70 (95% CI 0.68, 0.71) for mild hearing loss or worse and 0.86 (95% CI 0.85, 0.87) for moderate hearing loss or worse. The AUC increased substantially when sex, education and age were taken into account (AUC mild HL: 0.73 (95%CI 0.71, 0.75); moderate HL 0.90 (95%CI 0.89, 0.91)).
CONCLUSION: Self-reported hearing loss using a single question has a good ability to detect hearing loss in older adults, especially when age is accounted for. A single question cannot substitute audiometry, but it can assess hearing loss on a population level with reasonable accuracy.

Introduction

Age-related hearing loss (ARHL) is considered to be one of the most common chronic disorders among the elderly [1-3]. Generally, world-wide life expectancy increases, resulting in an upsurge of age-related health problems, including hearing loss. The prevalence of hearing loss in people over 65 years old ranges from 29% to 47%, based on the WHO definition of the pure tone average over 0.5, 1, 2 and 4 kHz in the better ear (PTA0.5–4) >25 decibel hearing level (dBHL) [1-4]. Its impact is substantial, as it is associated with social withdrawal, cognitive decline and depression [5-8]. The gold standard for measuring hearing loss is pure tone audiometry, which is not always available in large population-based studies. Self-reporting scales regarding hearing are more readily available and are therefore often used in the absence of pure tone audiometry [9, 10].

The assessment of subjective hearing loss in an ageing population through a single question or through questionnaires has been extensively investigated over the last two decades. Self-reporting is suitable for estimating the prevalence of hearing impairment, particularly for moderate to severe hearing loss [11]. The most commonly used, validated questionnaire is the hearing handicap inventory for the elderly (HHIE) [10, 12], and its screening version (HHIE-s) [13]. In addition, there are several studies that investigated a single question format. A review of 10 studies published between 1990 and 2004, concluded that a single question is able to identify hearing loss (especially moderate and severe) in adults over 60 years of age [14]. This review concludes that a single question is an acceptable substitute when audiometric measurements are not available, for example in epidemiological studies [14]. An overall linear relationship between self-reported hearing loss and pure tone audiometry is reported, although trends towards underestimation of hearing loss can be noted, especially in subjects younger than 60 years [15].

However, several issues remain to be addressed when using a question to estimate hearing loss in a general elderly population. First of all, many studies also included younger adults (from 20 years old), which has a significant influence on the outcome. At this young age, hearing is generally good and will be adequately reported as no hearing problems, in contrast to an elderly population characterized by a much higher prevalence of hearing loss. Secondly, most conclusions are based on the identification of (PTA0.5–4) ≥ 40 dBHL, thereby excluding slighter hearing loss. Slight hearing loss may already lead to communication problems in everyday situations, but is harder to identify as such on an individual level. It would therefore be of added value if a question could also differentiate between various degrees of hearing loss. A third point that deserves attention is how individual characteristics, including age and sex, may influence the outcome. Most studies report whether certain subgroups of individuals are better or worse at judging their hearing capacity. However, none of the studies have investigated how these easily obtained participant characteristics may actually be used to improve the estimation of hearing loss using a single question format.

The aim of our study was to investigate the reliability of a single question in assessing hearing loss in a large, ageing population. We used two thresholds for PTA0.5–4: >20 dBHL (mild or worse) and >35 dBHL (moderate or worse). Additionally, we investigated the ability of the question to assess hearing loss when adding individual characteristics, including age, sex and level of education.

Methods

Study subjects

This study was part of the Rotterdam Study, a prospective cohort study that has been ongoing since 1990, in which risk factors for common diseases are investigated in an ageing population [16, 17]. We report on data collected between February 2011 and June 2016. People aged 45 years and older were invited for participation via the population registry of Ommoord, a suburb of Rotterdam, The Netherlands. A total of 4,906 participants underwent both a home interview and pure tone audiometry by an experienced audiometrist in a dedicated research center in Ommoord. When individuals were screened at multiple time points, only the most recent one was taken into analysis.

The Rotterdam Study has been approved by the medical ethics committee of the Erasmus MC (registration number MEC 02.1015) and by the Dutch Ministry of Health, Welfare and Sport (Population Screening Act WBO, license number 1071272-159521-PG). The Rotterdam Study has been entered into the Netherlands National Trial Register (www.trialregister.nl) and into the World Health Organization International Clinical Trials Registry Platform (who.int/ictrp/network/primary/en/) under shared catalog number NTR6831. All participants provided written informed consent to participate in the study.

Interview

Each participant underwent an extensive home interview on health, background and environmental factors, before visiting the research center [16]. Participants were asked the following question (in Dutch), similar to that used in previous studies [18]: ‘Do you have any difficulty with your hearing (without hearing aids)?’ with answers ranging on a 4-point scale: ‘No, I always hear everything’, ‘Yes, sometimes I do not hear what is being said’, ‘Yes, I regularly do not hear what is being said’ or ‘Yes, I almost never hear what is being said’. Amongst many other parameters, highest achieved educational level was noted, using the UNESCO classification [19].

Pure tone audiometry

For all audiometric measurements, a clinical audiometer was used (Decos audiology workstation, version 210.2.6, with AudioNigma interface; Decos Audiology, Inc., Peachtree City, GA) with TDH-39P earphones. Measurements were performed in a professional soundproof booth. Hearing thresholds were set at the intensity level at which the tone was heard in 2 out of 3 ascents, according to the ISO standard 8253–1 [20]. If no response was obtained, even at maximum stimulation level for that given frequency, the threshold was set at 5 dB above maximum stimulation level. Air-conduction thresholds were obtained at octave frequencies 0.25, 0.5, 1, 2, 4 and 8 kHz. The subjectively better ear was measured first. For the analyses, we used a mid-frequency average at 0.5, 1, 2 and 4 kHz (PTA0.5–4) in the better ear and cut-offs proposed by the WHO [4, 21, 22].

Statistical analysis

Data management and analyses were done using IBM SPSS Statistics 24. A univariable linear regression model was created to investigate the association between the interview answers and PTA0.5–4 with forward selection of the independent variables sex, level of education and age. These three independent variables were chosen because they are easily assessed in a clinical situation. Independent variables were tested for their contribution to the regression with a Likelihood Ratio Test. The independent variable with the largest change in goodness of fit (R2) was first taken into the model and the other independent variables were added subsequently. The beta’s standardized regression coefficients were reported to show the effect sizes of the associations found.

For our next analyses, interview answers were dichotomized. For mild hearing loss (or worse), defined as PTA0.5–4 ≥20 dBHL in the better ear, only the first answer option was considered negative (‘No, I always hear everything’) and all other answers were considered positive. For moderate to severe hearing loss (or worse), defined as PTA0.5–4 ≥35 dBHL in the better ear, the first two answer options (‘No, I always hear everything’ and ‘Yes, sometimes I do not hear what is being said’) were considered negative and the other answers were considered positive [4]. With these dichotomized outcomes, we calculated sensitivity, specificity as well as negative and positive predictive values. Subsequently, a logistic regression model was fitted to calculate prediction estimates. Hearing loss was the outcome variable and the dichotomized interview answers were the investigated predictive values. The same independent variables from the final linear regression model were also used in the logistic regression model. The prediction estimates were used to plot receiver operating characteristic (ROC) curves. The area under the curve (AUC) was calculated to determine the discriminatory value of the question for hearing loss.

Secondary analysis

The full logistic regression model was repeated to calculate prediction estimates in a dataset stratified on age (<65 years of age, 65–80 years of age and >80 years of age). These prediction estimates were used to find the discriminatory value of self-reported hearing loss within each age category.

Results

Hearing loss as a continuous variable

Characteristics of the study population (n = 4,906) are listed in Table 1. We found a higher average hearing threshold in men, older participants and participants with only primary school level education (S1 Table). Distribution of the answers to the question was categorized per 20 dBHL hearing loss (Fig 1). As the hearing loss increased, the answers shifted from ‘no’ or ‘sometimes a problem’ to ‘often’ or ‘almost always a problem’. Above 40 dBHL (PTA0.5–4) hearing loss, almost all participants confirmed to have some degree of hearing problems.

Table 1

Characteristics of participants who had been asked the question: ‘Do you have any difficulty with your hearing?’.

Hearing thresholds were averaged over 0.5, 1, 2 & 4 kHz (PTA0.5–4).

N	4,906
Mean age, years (SD)	69.6 (9.8)
Age range, years	51.4–100.7
Female, %	56.3
Average hearing threshold, dBHL (SD)	24.5 (13.9)
Mild hearing loss or worse (PTA0.5–4), %
No (<20 dBHL)	47.4
Yes (≥ 20dBHL)	52.6
Moderate hearing loss or worse (PTA0.5–4), %
No (< 35 dBHL)	80.2
Yes (≥ 35 dBHL)	19.8
Level of education, %
Primary	7.8
Lower	38.9
Intermediate	30.0
Higher	23.3
Answer to the question, %
Never	48.7
Sometimes	33.6
Regularly	15.6
Often	1.9

Fig 1

Distribution of answers to the question: ‘Do you have any difficulty with your hearing?’.

Per 15dB hearing loss (PTA0.5–4) in the better ear.

Univariable linear regression analysis showed an increase of the PTA0.5–4 hearing threshold by 10.5 dB (95%CI 10.07, 10.85) for each step up in subjective difficulty in hearing (Table 2). Adding either sex, education or age led to a significant improvement of the regression (p<0.0001). The largest increase of the explained variance was seen for the factor “age” (R2 increase from 0.37 to 0.60, Fig 2). The subsequent addition of education and sex to the regression improved the model (p<0.0001), although the explained variance remained almost unchanged (R2 0.61).

Table 2

Results from the linear regression analysis for the question: ‘Do you have any difficulty with your hearing?’.

First, univariable analysis was done. Then each of the independent covariates were added, initially separately and later together. Beta’s reflect the number of decibel change in the PTA0.5–4 with each step up in degree of subjective hearing loss (never, sometimes, regularly, or often). R2 is given as a measure of the goodness of fit of the model.

	Intercept(95%CI)	Beta(95%CI)	R2
Univariable	17.05(16.64, 17.47)	10.46(10.07, 10.85)	0.37
+ sex	17.65(17.10, 18.21)	10.41(10.02, 10.80)	0.38
+ education	19.74(19.03, 20.45)	10.37(9.98, 10.75)	0.39
+ age(/ year)	-30.66(-32.45, -28.87)	8.26(7.94, 8.58)	0.60
+age + education	-29.02(-30.96, -27.09)	8.24(7.92, 8.57)	0.61
+ age + sex + education	-27.56(-29.54, -25.59)	8.16(7.84, 8.49)	0.61

Fig 2

Results from the linear regression analysis for the question: ‘Do you have any difficulty with your hearing?’.

First univariable analysis was done, then each of the independent covariates were added, initially separately and later together. R2 is given as a measure of the goodness of fit of the model.

Hearing loss as a dichotomous variable

Table 3 shows the 2x2 tables for the dichotomized answers and the presence of hearing loss (PTA0.5–4) (mild: ≥ 20 dBHL / moderate: ≥ 35dBHL). Prevalence of subjective mild hearing loss was 51.3% against 52.6% for objective mild hearing loss, while prevalence of subjective moderate hearing loss was 17.8% against 19.8% for objective moderate hearing loss.

Table 3

Crosstab for the dichotomized answers to the question ‘Do you have any difficulty with your hearing?’.

For defining mild hearing loss, all positive answers were included. For moderate hearing loss, “regularly” and “often” were the answer options included. These subjective measures were compared to the objective PTA0.5–4, mild: ≥20dBHL, and moderate: ≥35dBHL. Number of participants are depicted in each category.

		Whole population			< 65 years			65–80 years			> 80 years
Subjective hearing loss		No	Yes	Preva-lence (%)	No	Yes	Preva-lence (%)	No	Yes	Preva-lence (%)	No	Yes	Preva-lence (%)
Mild hearing loss ≥20dBHL	No	1,610	715	47.4%	911	468	77.5%	643	226	37.6%	56	21	9.4%
	Yes	777	1,804	52.6%	113	287	22.5%	481	960	62.4%	183	557	90.6%
Prevalence (%)		48.7%	51.3%		57.7%	42.3%		48.7%	52.3%		29.3%	70.7%
Moderate hearing loss ≥ 35 dBHL	No	3,593	340	80.2%	1,570	149	96.6%	1,705	151	80.3%	318	40	43.8%
	Yes	440	533	19.8%	17	43	3.4%	206	248	19.7%	217	242	56.2%
Prevalence (%)		82.2%	17.8%		89.2%	9.1%		82.7%	17.3%		65.5%	34.5%

The numbers in the 2x2 table were used to calculate sensitivity (mild hearing loss 69.9%, moderate hearing loss 54.8%), specificity (mild hearing loss 69.2%, moderate hearing loss 91.4%), positive predictive value (mild hearing loss 71.5%, moderate hearing loss 61.1%) and negative predictive value (mild hearing loss 67.4%, moderate hearing loss 89.1%).

A logistic regression model was used to identify the presence or absence of hearing loss at ≥ 20 dBHL and ≥ 35dBHL (PTA0.5–4), based on the question. Sex, education and age were taken into account as independent variables. The ability to identify mild hearing loss (PTA0.5–4 ≥ 20 dBHL), reflected in the AUC, was 0.70 (95% CI 0.68, 0.71). This increased when sex, education and age were taken into account (AUC: 0.86 (95% CI 0.85, 0.87)). The AUC for the identification of moderate hearing loss was 0.73 (95%CI 0.71, 0.75), which increased to 0.90 (95%CI 0.89, 0.91) with the addition of sex, education and age.

In a secondary analysis, a logistic regression model, adjusted for age, sex and education, was repeated in a dataset stratified in 3 age categories (<65 years of age, 65–80 years of age and >80 years of age). The AUC for mild hearing loss (PTA0.5–4 ≥ 20 dBHL) was highest for the oldest and lowest for the youngest age group (AUC <65 years 0.75 (95%CI 0.73, 0.78); 65–80 years 0.79 (95%CI 0.77,0.81); >80 years 0.81 (95%CI 0.76, 0.85)). For moderate hearing loss, the AUC was highest for the youngest age group and decreased with increasing age (AUC <65 years 0.86 (95%CI 0.80, 0.92); 65–80 0.83 (95%CI 0.81, 0.85); >80 years 0.79 (95%CI 0.76, 0.82)).

Discussion

In this study, we investigated the ability of the question ‘Do you have any difficulty with your hearing (without hearing aids)?’ to classify the severity of hearing loss measured by pure tone audiometry in an ageing population, using a four-category response. We have shown that this single question can be used to identify both mild and moderate hearing loss with reasonable accuracy. The ability of the question to identify hearing loss increases substantially when other factors are taken into account, with age being the most important one.

Our results are in line with the increasing support in literature for using a single question as an estimator for hearing loss in absence of pure tone audiometry [14, 15, 23–28]. There is a growing general interest in applying this concept to large population-based studies, for which time or other resources to perform audiometry are not available [29, 30]. We have shown that for these large population-based studies, a single question (adjusted for age, sex and highest achieved education) is a good surrogate for the actual hearing ability. Nevertheless, a single question might also be of value from a clinical perspective to identify populations at risk for hearing loss and should therefore be taken in consideration for screening purposes. Of course, one question testing is not meant to replace pure tone audiometry in the assessment of hearing in an individual.

In addition, we compared the ability of the same question to assess both mild (PTA0.5–4 ≥20 dBHL) and moderate hearing loss (PTA0.5–4 ≥35 dBHL). The predictive ability of the question for hearing loss (when taking age, sex and educational level into account) was 88% for mild hearing loss and 92% for moderate hearing loss. This is slightly higher than the previously reported ability of the HHIE-s to detect hearing loss (cut-off point at 8), where the AUC was 79% and 86% for mild and moderate hearing loss, respectively [31]. A single question is thought to be at least as good as or better than the HHIE-s, in detecting both mild and moderate hearing loss [12, 31]. One of the reasons is that HHIE-s has a broader scope than identifying hearing loss, as the HHIE(-s) also measures the possible impact of hearing loss on daily life [9]. Generally, a single question identifies moderate hearing loss better than mild hearing loss [14, 31, 32]. Nevertheless, we found that, when some participant characteristics are taken into account, the single question is able to also identify mild hearing loss. This might be explained by the fact that we used a 4-category response, instead of a simple yes/no, which allows for distinguishing between different grades of auditory-performance problems. Therefore, it is advisable to use more than two categories when it is also important to identify mild hearing loss.

Self-reporting always comes with the risk of misclassification bias, resulting in under- and overestimation. Age, sex and educational level, amongst other factors, are shown to be associated with identification of self-reported hearing loss [24, 25, 33] and objective hearing loss and are easily assessable in any situation [1, 14, 15, 24, 33, 34]. We found age, sex and educational level to increase the ability of the question to identify hearing loss, with age being the most important factor by far. When accounting only for age, the answer to our single question explained 61% of the hearing threshold, an increase of 24%-points. Older participants appear to be better at reporting their limitations in hearing ability than younger participants. An explanation might be that it is socially more acceptable for older people to experience hearing impairment. Compensation mechanisms for hearing loss do not function as well in older as they do in younger persons [35]. The role of age in the correct identification of hearing loss may also be attributable to the difference in prevalence of hearing loss, as reflected in the stratified analyses. As hearing is generally good in younger people, moderate hearing loss is uncommon, underestimation of hearing impairment and its symptoms is hardly possible. There might even be a bigger chance of overestimation of the hearing loss because the younger people often find themselves in more challenging listening environments. In older people with more prevalent hearing loss, both under- and overestimation are possible. The oldest group (>80 years) has the highest AUC for mild hearing loss, which is understandable as almost all people in that age group have some form of hearing loss.

Conclusion

Self-reported hearing loss, using the question ‘Do you have any difficulty with your hearing (without hearing aids)?’, has reasonable ability to detect both mild and moderate hearing loss in older adults, especially when the age of the individual is factored into the answer. This finding is mainly of importance for large population-based studies in which audiometry is absent but hearing loss still has to be quantified. A single question cannot substitute regular audiometry, but it is able to assess hearing on a population level with reasonable accuracy, adjusted for the age of the individuals.

Average hearing loss (in dB) for PTA0.5–4.

For 3 age categories, sex and highest achieved level of education.

(DOCX)

Click here for additional data file.

18 Aug 2019

PONE-D-19-19122

Assessing hearing loss in older adults with a single question and simple person characteristics; comparison with pure tone audiometry in the Rotterdam Study

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Reviewer #1: Interesting paper, I would recommend being clearer as to your purpose in refining a one question hearing test as a practical way to measure hearing impairment in large populations. You make that clear in the paper but not as clear in the abstract.

Comments are based on line of paper.

24-25rewrite HL is a frequent concern in the elderly population warranting investigation in numerous cohort studies. PT audiometry, the diagnostic gold standard for hearing impairment, is time consuming and costly for large population studies.

This statement clears up confusion that you are advocating for using one question to test hearing in individuals.

29-delete s on answer

38-39 sentence is missing words

46 rewrite ...assessed with reasonable accuracy...

50 which general population is growing older? western countries?

52 dB require a reference, in this case and for the entire paper use dBHL

55 depression making it imperative to identify hearing impairment at its onset

57 Self-reporting scales regarding hearing.... statement needs references

60 reference statement about extensive investigation

63-64 using a single question format.

75 ..increasing the positive correlation

77 ..which can lead to

78 change life to listening

83 hearing loss using a single question format.

93 45 years and older

97 recent result was taken

106 be clearer about what was asked in the extensive interview. What other questions were posed could impact how person felt about general body state, mental state and state of sensory abiliites

Analysis was well explained and your stats methods were clearly stated

150 primary education levels

184 Add a sentence explaining the impact of age of individual in predicting hearing loss

185 support in the literature

187 in which the time

188 audiometry are not practical or possible (delete sparse)

190 level. One question testing, however, wil...

192 In addition, we ...

203 delete distinct replace with distinguish

204 categories of answer foils when attempting to identify mild hearing loss

207 amongst other factors,

215 older people to experience hearing impairment.

216 older individuals as compared to younger persons

218 underestimation of hearing impairment and its symptoms would be rare.

226 when age of the individual is factored into the answer

226 delete additional

227.. there is a need to quantify hearing impairment.

228 substitute for audiometry, assess

229 on a population level, hearing can be assessed reasonably accurately with a single

230 question corrected for age of individual.

Reviewer #2: Using a large data set from the Rotterdam study, the authors aim to evaluate the effectiveness of estimating the magnitude of hearing loss based on the answer to a single question. Other attempts at achieving this same general goal and their relative success and failure is discussed. The authors specifically point out three deficits or complications of previous work that the current manuscript attempts to overcome. First, previous work included significant numbers of younger individuals with normal hearing, arguably inflating correlation. Second, previous work focused on finding the relationship between self-report and moderate or more severe hearing loss. And finally, these authors attempt to examine the improvement in single question performance when individual subject characteristics such as age are included in the prediction model.

The authors demonstrate considerable success in estimating hearing loss based on the four-layer answer to the single question. Performance of the single question improves on the inclusion of age as a variable. On average hearing loss increases by ~ 10.5 dB between categories in the answer. Diagnosis of both mild and moderate hearing loss is possible based on the answer to the single question, with performance being slightly better for moderate hearing loss.

Was sex, education, and age taken from participant reports? If that was the case, then perhaps sex should be referred to gender as it is reported but not biologically verified.

Why did the authors use 45 years as the cut off age for their sample?

Other items:

Line 80, the following sentence is difficulty to interpret: “Most studies report whether certain subgroups of individuals being better or worse at predicting hearing loss.”

The discussion regarding over and underestimation of hearing loss in the second half of page 12 is a bit confusing. The authors seem to argue at first that estimates are essentially one sided in younger individuals as objective hearing loss is limited. In contrast the estimates are two sided in older individuals. But the authors also argue that older individuals should be able to better estimate their objective hearing loss as compensation is more difficult and it is more socially acceptable to have hearing loss. Given the population sample that the authors have, the above hypotheses are verifiable. It may strengthen the paper if the authors attempted some secondary analyses after subdividing the sample into older and younger groups. In the limit, the error distributions could be estimated in five- or seven-year age windows.

Reviewer #3: This paper has some issues as there is a claim that the single question provides good estimation of hearing loss (PTA) in an older population, but in fact the variance accounted for by the answers to this question is 37% and it is only when other variables, particularly age, are used as additional predictors that the statistics show a "good" prediction. It is hardly a surprise that there would be some correlation between the question and PTA in the better ear and this has been shown before as noted in the introduction to the paper. In a population sense, age is quite a good predictor of hearing loss and this is certainly well known, so I am not sure that there is anything new here. I would concede that in a large population study with limitations of funding, the one question response plus the demographic factors (particularly age) will provide a good estimate of the prevalence of mild and moderate hearing loss, but the question by itself is not a very good "predictor" of the audiogram in an individual. It is only an estimator of prevalence. I think the manuscript needs to be more carefully written so that it is clear that we do not really have a prediction here, but only an estimator of prevalence. I also found some of the statistical discussion confusing. The analysis is named as a univariable regression but seems to be describing a multiple regression. Maybe I am misunderstanding the way the authors have used the term univariable. Additional specific comments are below.

p.4, l.50 "the general population grows older" - of course, but the issue is that people are living longer

p.4, l.55 "identify hearing loss in time" - in time for what?

p.11, l.193 This is not the predictive ability of the question, it is the predictive ability of the 4 variables. What would be the predictive ability of age, gender and education without the question?

p.12, l.203 "distinct" should be 'distinguish"

p.12, l.212 "in" should be "into"

p.12, l.216 "compared as in" should be "compared to"

p.12, l.217 "might as well be attributed" should be "may be attributable"

p.13, l.228 "asses" should be "assess"

p.13, l.229 "on population level' should be "on a population level"

p.13, l.229 "pretty accurately" - maybe "reasonably accurately" would be better

Reviewer #4: Assessing hearing loss in older adults with a single question and simple person characteristics; comparison with pure tone audiometry in the Rotterdam Study

The authors posed the question: whether a self-report using a single question of hearing loss and some additional data corresponds with audiometric approach to determining hearing loss. This is not a novel question, but important to ask none-the-less.

The analysis appears to be appropriate to the address the questions, although I have a query about the dichotomising. I also am not clear what data were used? Best ear, worst ear, mean of both?

The discussion and conclusions are properly drawn from the results, although a bit more clarity of the applicability of the findings would be good.

I would have also liked to know why this question was used? What was the basis or framework? Others have used another form of words. Why would one be better than another? Why are hearing difficulties related to severity of hearing loss?

On another note, the WHO and Global Burden of Disease Group have recommended a new approach to classification. Hume, IJA, https://doi.org/10.1080/14992027.2018.1518598

It would be very valuable, seeing this papers says the value of this approach is for population/epidemiology studies, for this classification system to be applied. Not >=25 and >=40. I can see that this needs some re-analysis, but it would make this study much more valuable. Maybe this paper will not report the prevalence of HL (just focussing on the use of a single question), but a subsequent paper should ideally use the WHO/GBD classifications.

A note on the English: the manuscript is quite readable, and the intent -if not always clear- can be assumed. However, there are traces of evidence that English is not the primary language of the authors, and the structure of the Dutch language is apparent at times. In some cases may come down to differences in choices of wording, and it does not affect the clarity of the manuscript. But there is also some loose phrasing that does need to be tightened up.

I would like to see this work published, but some attention needs to be paid to the writing. If the suggestion to use another set of classifications is not accepted, then the usefulness of the study to others in the long term will be restricted.

Specific comments:

Lines 38 and 39: spare space before the comma in line 38, and ‘are’ should be ‘area’

Line 45: asses should be assess; and ‘population’ should be ‘a population’.

Line 44: I’m not sure this statement (“never”) can be made: sure, this single question will not be a substitute. They are assess two different things. But it is possible that someone will develop a single question that will be better; but we do not know. In any case, I would leave this matter for the discussion, not a conclusion in the abstract.

Line 46: “pretty accurately” is not appropriate wording

Line 50: “The general population grows older” - taken literally, this can’t be otherwise. However, isn’t the point that the mean age of the population is increasing – and even then that is not in every country or population.

Line 57: references?

Line 67: What is ‘this’ review? Is it (14)? If so, the sentence needs some attention. This reference is a bit dated, and there have been more studies since then.

Line 75: Why is it easier? Is it easier to correct? Then this needs to be stated.

Line 82: ‘simple’ – this is a bit of a vague term. Why is one characteristic simple, and another characteristic not simple?

Line 84: ‘aim was’

Line 85: why these two levels? See earlier comment.

Line 97: “the most recent” meaning all the data (question and audiogram)? Adding the word ‘data’ after ‘recent’ (and changing ‘was’ to ‘were’) may be helpful.

Line 104: What additional data were obtained from treating physicians, and why. Treatment is not part of the study is it?

Line 107: perhaps it need not be stated, but I assume the questions and answers were in Dutch and not English.

Line 107: What is the basis for these answers? And why this particular question? This is not addressed in the introduction. I realise that this may be difficult to answer, and I doubt other similar studies have

provided a rationale either. (See comment made at the start.)

Line 117: maximum of the audiometer?

Line 118: why +5dB? And does that mean if no response was obtained at 50dB and testing continued to the maximum of the audiometer at that frequency (say 90dB) then the ‘threshold’ was set to 95dB? This is a tricky thing to manage. It is not a threshold, and who is to say they would not have heard a stimulus at 110dB? However, it’s good that this information is provided.

Line 120: ‘we used a mid-frequency average’ – ‘average’ implies one or the two ears; which ear? If both were in the analysis, then it should be ‘we used mid-frequency averages’. I did not find this clarified in the manuscript. Was the better ear data used? This needs to be stated, also on the figures.

Line 128: ‘independent variables’ is better.

Line 130: This raises the question: why did you then bother with four answers? Is it valid to combine the two pairs of answers?

Line 143: Is this needed here? I think I saw this mentioned earlier.

Line 150: “with primary education” this is hard understand without looking at the table (and even then it is not clear). Do you mean “with education only to a primary school level”?

Line 159: be clear that education and sex are added to the basic model, not in addition to age (second last row in Suppl table 2). By the way, this table is quite important for the main manuscript.

‘improved’ do you mean from 0.6 to 0.61? Because then you say it was almost unchanged.

Line 163: these figures are not shown in the table.

Line 170: I am not sure what is meant by ‘a clinical situation’

Line 179: the comma is not needed.

Line 180: ‘hearing loss’ should be ‘the severity of hearing loss’?

Line 182: What does ‘sufficient’ mean? This is undefined.

Line 190: this sentence needs some work. What is a ‘personal level’? I think what is meant that a single question may be a good proxy for hearing loss in population studies, but for clinical use it cannot replace audiometry. (However, doesn’t it have a place in clinical setting? See Swanepoel, JAAA, 2013)

Line 192: assess, not asses (also line 228)

Line 202: But weren’t the answers dichotomised when calculating sensitivity and specificity etc.?

Line 207: Do you mean on a personal level – or on a population level? I guess that the following references suggest this is dependent on the nature of the population?

Line 213: ‘recognise’ is different to being ‘socially acceptable’, isn’t it? Surely younger people are equally able to recognise that they have a hearing loss? And don’t you mean that it is more socially acceptable for older to admit that they have a hearing loss. ( I suggest dropping the word ‘suffer’ – the deaf community say that they do not ‘suffer’)

Line 218: Still have a bit of a problem with this. See previous comment. True, there is less chance that this will occur. This line with the next line needs to be tightened up a bit.

Line 219: I assume that males are not as reliable; but these data are not provided, and this sentence does not clarify it at all.

Figure 2: what does b. provide that a. does not, other than a different vertical axis? 2a: the purpose of the different symbols is not clear. I see not logic in the choice of shape or ‘colour’ or border.

Line 228: see earlier comment

Line 229: ‘pretty’ is not a word that should be used. See earlier comment.

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7 Nov 2019

The complete rebuttal letter was uploaded as a document with the manuscript.

Date: 27-09-2019

Subject: Revision of manuscript PONE-D-19-19122

Dear Dr. Fransesco Martines,

Thank you for reviewing our manuscript entitled ‘Assessing hearing loss in older adults with a single question and simple person characteristics; comparison with pure tone audiometry in the Rotterdam Study’ (PONE-D-19-19122).

We are grateful for all the detailed comments by the reviewers. In response to those comments, we have made changes to the manuscript. Regarding the language issues raised by the reviewers, we asked a native speaker to adjust the language. We feel that these changes have improved the manuscript and its readability.

Please find attached our point-by-point reply to the questions and comments of the reviewers. We hope you will consider this revised manuscript acceptable for publication in Plos One.

Yours sincerely,

On behalf of all authors,

Neelke Oosterloo, MD

Paul Nagtegaal, MD, PhD

André Goedegebure, Ir. PhD

Reviewer #1:

Interesting paper, I would recommend being clearer as to your purpose in refining a one question hearing test as a practical way to measure hearing impairment in large populations. You make that clear in the paper but not as clear in the abstract.

Response: We thank the reviewer for the comments. We have modified the abstract and hope this message is clearer now. We changed the abstract according the specific suggestions you made below.

Comments are based on line of paper.

24-25rewrite HL is a frequent concern in the elderly population warranting investigation in numerous cohort studies. PT audiometry, the diagnostic gold standard for hearing impairment, is time consuming and costly for large population studies.

This statement clears up confusion that you are advocating for using one question to test hearing in individuals.

Response: We have changed the statement as suggested by the reviewer.

38-39 sentence is missing words

Response: We hope the sentence is better this way.

50 which general population is growing older? western countries?

Response: We agree with the reviewer that this statement is unclear, therefor we changed it to:

‘Generally, world-wide life expectancy increases, resulting in an increase of age-related health problems, including hearing loss.’

52 dB require a reference, in this case and for the entire paper use dB HL

Response: We agree with the reviewer to add a reference to the dB. We have changed it throughout the manuscript when applicable.

55 depression making it imperative to identify hearing impairment at its onset

Response: We agree with the reviewer that the emphasis of this sentence is not correct. We changed the sentence.

57 Self-reporting scales regarding hearing.... statement needs references

Response: We changed the sentence and added the references.

60 reference statement about extensive investigation

Response: The requested references are listed in the sentences to follow.

106 be clearer about what was asked in the extensive interview. What other questions were posed could impact how person felt about general body state, mental state and state of sensory abilities

Response: We added extra information to the main text. However due to the extensiveness of the interview it is too much to explain it all in this paper, so a reference was added (1).

Analysis was well explained and your stats methods were clearly stated

184 Add a sentence explaining the impact of age of individual in predicting hearing loss

Response: We have altered the paragraph in the discussion section on this to add this explanation.

204 categories of answer foils when attempting to identify mild hearing loss

Response: We do not understand this comment. Is it possible to clarify?

Textual changes:

29-delete -s on answer

46 rewrite ...assessed with reasonable accuracy...

63-64 using a single question format.

75 ..increasing the positive correlation

77 ..which can lead to

78 change life to listening

83 hearing loss using a single question format.

93 45 years and older

97 recent result was taken

150 primary education levels

185 support in the literature

187 in which the time

188 audiometry are not practical or possible (delete sparse)

190 level. One question testing, however, wil...

192 In addition, we ...

203 delete distinct replace with distinguish

207 amongst other factors,

215 older people to experience hearing impairment.

216 older individuals as compared to younger persons

218 underestimation of hearing impairment and its symptoms would be rare.

226 when age of the individual is factored into the answer

226 delete additional

227.. there is a need to quantify hearing impairment.

228 substitute for audiometry, assess

229 on a population level, hearing can be assessed reasonably accurately with a single

230 question corrected for age of individual.

Response: We thank the reviewer for the suggestions to improve the quality of the language. We altered the text as suggested by the reviewer.

Reviewer #2:

Using a large data set from the Rotterdam study, the authors aim to evaluate the effectiveness of estimating the magnitude of hearing loss based on the answer to a single question. Other attempts at achieving this same general goal and their relative success and failure is discussed. The authors specifically point out three deficits or complications of previous work that the current manuscript attempts to overcome. First, previous work included significant numbers of younger individuals with normal hearing, arguably inflating correlation. Second, previous work focused on finding the relationship between self-report and moderate or more severe hearing loss. And finally, these authors attempt to examine the improvement in single question performance when individual subject characteristics such as age are included in the prediction model.

The authors demonstrate considerable success in estimating hearing loss based on the four-layer answer to the single question. Performance of the single question improves on the inclusion of age as a variable. On average hearing loss increases by ~ 10.5 dB between categories in the answer. Diagnosis of both mild and moderate hearing loss is possible based on the answer to the single question, with performance being slightly better for moderate hearing loss.

Response: We thank the reviewer for the comments. Please see our response to each comment below.

Was sex, education, and age taken from participant reports? If that was the case, then perhaps sex should be referred to gender as it is reported but not biologically verified.

Response: Sex was not taken from the participant report, it was objectified at the research center. We therefore choose to keep the term sex over gender.

Why did the authors use 45 years as the cut off age for their sample?

Response: This cut off point for age is due to the study design. The Rotterdam Study is designed to investigate determinants of health and disease in an ageing population(1). All participants of the Rotterdam Study were eligible to be included in the present study even though the youngest participant in the present study was 51.4 years old (table 1).

Other items:

Line 80, the following sentence is difficulty to interpret: “Most studies report whether certain subgroups of individuals being better or worse at predicting hearing loss.”

Response: We understand the difficulty, we adjusted the sentence hoping that it is more easily interpretable.

Introduction: ‘Most studies report whether certain subgroups of individuals are better or worse at judging their hearing capacity’.

The discussion regarding over and underestimation of hearing loss in the second half of page 12 is a bit confusing. The authors seem to argue at first that estimates are essentially one sided in younger individuals as objective hearing loss is limited. In contrast the estimates are two sided in older individuals. But the authors also argue that older individuals should be able to better estimate their objective hearing loss as compensation is more difficult and it is more socially acceptable to have hearing loss. Given the population sample that the authors have, the above hypotheses are verifiable. It may strengthen the paper if the authors attempted some secondary analyses after subdividing the sample into older and younger groups. In the limit, the error distributions could be estimated in five- or seven-year age windows.

Response: We agree with the reviewer that the proposed secondary analyses will strengthen the paper, we added them to the results section. However, we were not able to assess this in five or seven year age windows, as certain groups would contain a relatively low number of participants.(2)

Reviewer #3:

This paper has some issues as there is a claim that the single question provides good estimation of hearing loss (PTA) in an older population, but in fact the variance accounted for by the answers to this question is 37% and it is only when other variables, particularly age, are used as additional predictors that the statistics show a "good" prediction. It is hardly a surprise that there would be some correlation between the question and PTA in the better ear and this has been shown before as noted in the introduction to the paper. In a population sense, age is quite a good predictor of hearing loss and this is certainly well known, so I am not sure that there is anything new here. I would concede that in a large population study with limitations of funding, the one question response plus the demographic factors (particularly age) will provide a good estimate of the prevalence of mild and moderate hearing loss, but the question by itself is not a very good "predictor" of the audiogram in an individual. It is only an estimator of prevalence. I think the manuscript needs to be more carefully written so that it is clear that we do not really have a prediction here, but only an estimator of prevalence. I also found some of the statistical discussion confusing.

Response: We thank the reviewer for the comments on the manuscript. We agree that it is not new to show that age is a good predictor for hearing loss in a population sense, just like sex and hearing loss. Neither is it new that a single question format to assess hearing loss is able to estimate the prevalence. However, this study is new in a sense that we combine these 4 factors to assess hearing loss on a population basis. We agree that one of the possible applications of such a question is to estimate prevalence of hearing loss in a large population. However, the question could also be used to identify subgroups of a large population that could be compared, e.g. to analyze genetic susceptibility for hearing loss (GWAS).

The analysis is named as a univariable regression but seems to be describing a multiple regression. Maybe I am misunderstanding the way the authors have used the term univariable.

Response: The analyses started with a true univariable analysis (dependent variable: PTA0.5,1,2,4,, independent variable: the interview answers) , after that the covariates were added and we do not speak of a univariable analysis anymore.

Additional specific comments are below.

p.4, l.50 "the general population grows older" - of course, but the issue is that people are living longer

Response: This statement was changed into: ‘In the population of western countries the life expectancy increases’ we feel that with this change in sentence the issue of longevity and an increasing prevalence is captured.

p.4, l.55 "identify hearing loss in time" - in time for what?

Response: We see that the statement is unclear, the sentences were changed into:” Its impact is substantial, as it is associated with social withdrawal, cognitive decline and depression.”

p.11, l.193 This is not the predictive ability of the question, it is the predictive ability of the 4 variables. What would be the predictive ability of age, gender and education without the question?

Response: We know that these factors by itself have a high predictive ability for hearing loss, without the question the AUC is 0.82, which shows us that the question does contribute to the estimation of hearing loss (AUC:0.86).

Textual changes:

p.12, l.203 "distinct" should be 'distinguish"

p.12, l.212 "in" should be "into"

p.12, l.216 "compared as in" should be "compared to"

p.12, l.217 "might as well be attributed" should be "may be attributable"

p.13, l.228 "asses" should be "assess"

p.13, l.229 "on population level' should be "on a population level"

p.13, l.229 "pretty accurately" - maybe "reasonably accurately" would be better

Response: We thank the reviewer for the suggestions to improve the quality of the language. We altered the text as suggested by the reviewer

Reviewer #4:

Assessing hearing loss in older adults with a single question and simple person characteristics; comparison with pure tone audiometry in the Rotterdam Study

The authors posed the question: whether a self-report using a single question of hearing loss and some additional data corresponds with audiometric approach to determining hearing loss. This is not a novel question, but important to ask none-the-less.

The analysis appears to be appropriate to the address the questions, although I have a query about the dichotomising. I also am not clear what data were used? Best ear, worst ear, mean of both?

Response: We thank the reviewer for the comments. We can see that there was too little information, we used the best ear, this we also added to the method section to clarify.

The discussion and conclusions are properly drawn from the results, although a bit more clarity of the applicability of the findings would be good.

Response: We have added

I would have also liked to know why this question was used? What was the basis or framework? Others have used another form of words. Why would one be better than another? Why are hearing difficulties related to severity of hearing loss?

Response: In the time that the data collection on hearing was designed, around 2010/2011, a review of the literature was available on subjective hearing assessment by Chou et al. (2). This review showed a nice overview of several studies that used questions varying between ‘Would you say that you have any difficulty hearing? / Do you have difficulty hearing / Do you feel you have hearing loss?/ Do you have a hearing problem now?’

On another note, the WHO and Global Burden of Disease Group have recommended a new approach to classification. Hume, IJA, https://doi.org/10.1080/14992027.2018.1518598

It would be very valuable, seeing this papers says the value of this approach is for population/epidemiology studies, for this classification system to be applied. Not >=25 and >=40. I can see that this needs some re-analysis, but it would make this study much more valuable. Maybe this paper will not report the prevalence of HL (just focussing on the use of a single question), but a subsequent paper should ideally use the WHO/GBD classifications.

Response: We thank the reviewer for bringing up this discussion on the cut-off points. We agree with the reviewer that these cut-off points are better supported by literature. All analyses were rerun with these adjusted cut-off points, resulting in marginally different results.

A note on the English: the manuscript is quite readable, and the intent -if not always clear- can be assumed. However, there are traces of evidence that English is not the primary language of the authors, and the structure of the Dutch language is apparent at times. In some cases may come down to differences in choices of wording, and it does not affect the clarity of the manuscript. But there is also some loose phrasing that does need to be tightened up.

Response: We thank the reviewer for his/her comments and adjustments on the writing. The manuscript was redirected by a native speaking person.

I would like to see this work published, but some attention needs to be paid to the writing. If the suggestion to use another set of classifications is not accepted, then the usefulness of the study to others in the long term will be restricted.

Specific comments:

Textual changes:

Lines 38 and 39: spare space before the comma in line 38, and ‘are’ should be ‘area’

Line 45: asses should be assess; and ‘population’ should be ‘a population’.

Line 46: “pretty accurately” is not appropriate wording

Line 84: ‘aim was’

Line 128: ‘independent variables’ is better.

Line 150: “with primary education” this is hard understand without looking at the table (and even then it is not clear). Do you mean “with education only to a primary school level”?

Line 179: the comma is not needed.

Line 180: ‘hearing loss’ should be ‘the severity of hearing loss’?

Line 192: assess, not asses (also line 228)

Line 228: see earlier comment

Line 229: ‘pretty’ is not a word that should be used. See earlier comment.

Response: We thank the reviewer for the suggestions to improve the quality of the language. We altered the text as suggested by the reviewer.

Line 44: I’m not sure this statement (“never”) can be made: sure, this single question will not be a substitute. They are assess two different things. But it is possible that someone will develop a single question that will be better; but we do not know. In any case, I would leave this matter for the discussion, not a conclusion in the abstract.

Response: We agree that this a too strict statement, we changed ‘will never’ to ‘cannot’.

Line 50: “The general population grows older” - taken literally, this can’t be otherwise. However, isn’t the point that the mean age of the population is increasing – and even then that is not in every country or population.

Response: We agree that this statement is not correct, we changed it for ‘In the population of western countries the life expectancy increases’.

Line 57: references?

Response: We added the references to this sentence.

Line 67: What is ‘this’ review? Is it (14)? If so, the sentence needs some attention. This reference is a bit dated, and there have been more studies since then.

Response: We deleted the sentence between the reference and ‘this review’. A newer reference was added.

Line 75: Why is it easier? Is it easier to correct? Then this needs to be stated.

Response: It is generally easier because the hearing of younger participants is good in general, so if most do not have any objective hearing problems it is to be expected that they consider it subjectively good as well. We added ‘as this is generally good and will be adequately reported as no hearing problems.’ to the sentence in the main text, we hope this clarifies the statements.

Line 82: ‘simple’ – this is a bit of a vague term. Why is one characteristic simple, and another characteristic not simple?

Response: We agree that simple is a vague term, therefore we changed it to easily obtainable as this is what was meant with simple.

Line 85: why these two levels? See earlier comment.

Response: We agree with the reviewer and have changed this throughout the manuscript.

Line 97: “the most recent” meaning all the data (question and audiogram)? Adding the word ‘data’ after ‘recent’ (and changing ‘was’ to ‘were’) may be helpful.

Response: We agree with the reviewer and have added ‘results’ to clarify.

Line 104: What additional data were obtained from treating physicians, and why. Treatment is not part of the study is it?

Response: We removed this sentence from the manuscript as these data were not used in this current study.

Line 107: perhaps it need not be stated, but I assume the questions and answers were in Dutch and not English.

Response: The questions were in Dutch.

Line 107: What is the basis for these answers? And why this particular question? This is not addressed in the introduction. I realise that this may be difficult to answer, and I doubt other similar studies have

provided a rationale either. (See comment made at the start.)

Response: See explanation above.

Line 117: maximum of the audiometer?

Response: This depends on the frequency. We have changed this in the methods section to maximum stimulation level of the audiometer for that given frequency.

Line 118: why +5dB? And does that mean if no response was obtained at 50dB and testing continued to the maximum of the audiometer at that frequency (say 90dB) then the ‘threshold’ was set to 95dB? This is a tricky thing to manage. It is not a threshold, and who is to say they would not have heard a stimulus at 110dB? However, it’s good that this information is provided.

Response: This is a standard procedure in audiometry used in research, as a threshold is warranted to be able to execute the calculations. At a given frequency if the maximum stimulus in not heard the threshold is set 5dB above the maximum stimulus.

Line 120: ‘we used a mid-frequency average’ – ‘average’ implies one or the two ears; which ear? If both were in the analysis, then it should be ‘we used mid-frequency averages’. I did not find this clarified in the manuscript. Was the better ear data used? This needs to be stated, also on the figures.

Response: We added this to the manuscript and figures. The better ear data was used.

Line 130: This raises the question: why did you then bother with four answers? Is it valid to combine the two pairs of answers?

Response: Four answer categories were used to determine some degree of hearing loss. In the manuscript we show that considering these four answer categories one can differentiate between mild and moderate hearing loss by using different cut-off points. We think it is valid to combine the pairs of answers, this can also be derived from figure 1.

Line 143: Is this needed here? I think I saw this mentioned earlier.

Response: It should be added somewhere in the manuscript. We added a subheading to this section.

Line 159: be clear that education and sex are added to the basic model, not in addition to age (second last row in Suppl table 2). By the way, this table is quite important for the main manuscript.

‘improved’ do you mean from 0.6 to 0.61? Because then you say it was almost unchanged.

Response: We have added this table to the main manuscript and removed figure 2a, as these show the same results in a different way. We tested the fit of the regression model with a likelihood ratio test, this showed a significant improvement of the model although the explained variance (R2) remained almost unchanged, as stated in the manuscript.

Line 163: these figures are not shown in the table.

Response: We have shown the raw numbers in the table. We added the prevalence data to the table, sensitivity, specificity, negative predictive value and positive predictive value are described in the text.

Line 170: I am not sure what is meant by ‘a clinical situation’

Response: We agree with the reviewer that is unclear, we removed this part of the sentence.

Line 182: What does ‘sufficient’ mean? This is undefined.

Response: Sufficient was changed to reasonable.

Line 190: this sentence needs some work. What is a ‘personal level’? I think what is meant that a single question may be a good proxy for hearing loss in population studies, but for clinical use it cannot replace audiometry. (However, doesn’t it have a place in clinical setting? See Swanepoel, JAAA, 2013)

Response: We agree that we think that a single question can be used as a proxy in large population based studies, although it cannot replace pure tone audiometry in an individual. We do think it might have a place in a clinical setting for screening purposes, but it is (for now) not as accurate as pure tone audiometry, and therefore cannot replace pure tone audiometry in a diagnostic setting.

Line 202: But weren’t the answers dichotomised when calculating sensitivity and specificity etc.?

Response: Yes, the answers were dichotomized, however, we think that the dichotomization of the four answer categories contribute to the ability to detect mild or moderate hearing loss.

Line 207: Do you mean on a personal level – or on a population level? I guess that the following references suggest this is dependent on the nature of the population?

Response: This is correct, both on a personal and a population level.

Line 213: ‘recognise’ is different to being ‘socially acceptable’, isn’t it? Surely younger people are equally able to recognise that they have a hearing loss? And don’t you mean that it is more socially acceptable for older to admit that they have a hearing loss. ( I suggest dropping the word ‘suffer’ – the deaf community say that they do not ‘suffer’)

Response: We agree that recognize is not the best chosen word. We changed it for ‘report’ as we think that this covers the message better. We removed suffer from the text, thank you for pointing this out.

Line 218: Still have a bit of a problem with this. See previous comment. True, there is less chance that this will occur. This line with the next line needs to be tightened up a bit.

Response: We adjusted the two lines in: ‘As hearing is generally good in younger people, underestimation of hearing impairment and its symptoms would be rare. Whereas in older people with more prevalent hearing loss, both under- and over estimation are possible.’

Line 219: I assume that males are not as reliable; but these data are not provided, and this sentence does not clarify it at all.

Response: We realized that these data were not provided in the results section, we therefore dropped the sentences.

Figure 2: what does b. provide that a. does not, other than a different vertical axis? 2a: the purpose of the different symbols is not clear. I see not logic in the choice of shape or ‘colour’ or border.

Response: We removed figure 2a from the main text and substituted it with table 2.

References:

1. Ikram MA, Brusselle GGO, Murad SD, van Duijn CM, Franco OH, Goedegebure A, et al. The Rotterdam Study: 2018 update on objectives, design and main results. Eur J Epidemiol. 2017;32(9):807-50.

2. Chou R, Dana T, Bougatsos C, Fleming C, Beil T. Screening adults aged 50 years or older for hearing loss: a review of the evidence for the U.S. preventive services task force. Ann Intern Med. 2011;154(5):347-55.

Submitted filename: Rebuttal letter.docx

Click here for additional data file.

25 Nov 2019

PONE-D-19-19122R1

Assessing hearing loss in older adults with a single question and simple person characteristics; comparison with pure tone audiometry in the Rotterdam Study

PLOS ONE

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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

Reviewer #2: All comments have been addressed

Reviewer #3: (No Response)

Reviewer #4: All comments have been addressed

**********

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

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Reviewer #1: Yes

Reviewer #2: Yes

Reviewer #3: Partly

Reviewer #4: Yes

**********

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

Reviewer #1: Yes

Reviewer #2: Yes

Reviewer #3: Yes

Reviewer #4: Yes

**********

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Reviewer #1: Yes

Reviewer #2: No

Reviewer #3: Yes

Reviewer #4: No

**********

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Reviewer #1: Yes

Reviewer #2: Yes

Reviewer #3: Yes

Reviewer #4: Yes

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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: Authors have addressed all of my concerns. I believe the document should be published. I congratulate them on an interesting paper.

Reviewer #2: Thanks for addressing the reviewer concerns. The paper may still benefit from some editorial overview.

Reviewer #3: The authors have responded reasonably to my comments. I still feel that there is a tendency to overclaim the effectiveness of the "single question' in identifying hearing loss. In the author's response to one of my comments, it is noted that age, gender and education have high predictability for hearing loss (without the question) with an AUC (area under the ROC curve) of 0.82. This does improve to 0.86 with the use of the question but it suggests that the prediction is not greatly improved. i feel this information should be included in the paper.

Reviewer #4: A clean copy (not showing changes) has not been provided; the initially submitted version was instead. Furthermore, there were two versions of the figures.

The authors appear to have tackled all the comments and suggestions that I and the other reviewers made. However, I suggest another careful review of the English a few errors in wording and sentence structure are still there.

Line 77: capital S

Line 79: Not sure this captures the matter correctly. And the sentence swaps from the prevalence of hearing loss in younger adults (population) to their own hearing (an individual). I agree that ease of estimation is judged on a personal level. But the wording suggests that it is easier to estimate good hearing than poor(er) hearing. Is that the case? And that the ease translates to better estimation of prevalence. This section needs some work. I can see what the authors are getting at, but I am not convinced. And is this not complicated with the issue noted in the Discussion in Lines 267 onwards?

Line 84: Starting with ‘While’ leads the reader to expect more in the sentence. I suggest dropping the word.

Line 89: ‘have’ is correct, not ‘has’

Line 121: I know that for the previous review it was not necessary to point out that the question and answers were in Dutch. However, upon reflection it would be good to make that clear, in case someone uses the same wording, and then compares results; this would not be entirely appropriate as the English translation/version has not been validated.

Line 133: is it really not possible to reference this?

Table 1: The age range also needs to have ‘years’ in the first column

Line 177: ‘Hearing thresholds were’, not ‘Hearing loss was’

Line 181: ‘thresholds’? But actually it’s PTA.5-4 isn’t it?

Lines 188 to 195; two versions of the table caption.

Table 3: The placement of the words ‘Prevalence’ is a bit unhelpful when the 100% figures are seen. Can these 100% figures be removed?

Line 240: This needs to be more nuanced. To start with, I do not think anyone would think it would replace clinical assessment of individuals, which this implies. On the other hand, I think it may have some clinical value in identifying people who are at a higher risk of hearing loss, and so this information has potential for use as a screening tool.

Line 259 onwards: Comment/question: were males better or worse at estimation than females? This matter of self-reporting is complicated by things like denial of hearing problems and self-awareness which may be lower in males, but on the other hand males having a higher prevalence of HL. I think the authors have grappled with some of these issues.

Line 266: points?

**********

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Reviewer #1: No

Reviewer #2: No

Reviewer #3: Yes: Richard C Dowell

Reviewer #4: Yes: Robert Henry Eikelboom

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30 Dec 2019

Reviewer #1: Authors have addressed all of my concerns. I believe the document should be published. I congratulate them on an interesting paper.

Response: We thank the reviewer for his/her work.

Reviewer #2: Thanks for addressing the reviewer concerns. The paper may still benefit from some editorial overview.

Response: We thank the reviewer for his/her work and input, we have carefully gone through the text again.

Reviewer #3: The authors have responded reasonably to my comments. I still feel that there is a tendency to overclaim the effectiveness of the "single question' in identifying hearing loss. In the author's response to one of my comments, it is noted that age, gender and education have high predictability for hearing loss (without the question) with an AUC (area under the ROC curve) of 0.82. This does improve to 0.86 with the use of the question but it suggests that the prediction is not greatly improved. i feel this information should be included in the paper.

Response: We thank the reviewer for his work. We agree this is not an huge increase, however the scope of our paper is to investigate the question as the main outcome. The additional information in this paper is that the ability to identify hearing loss can be improved when age is taken into account on a population level.

Reviewer #4: A clean copy (not showing changes) has not been provided; the initially submitted version was instead. Furthermore, there were two versions of the figures.

The authors appear to have tackled all the comments and suggestions that I and the other reviewers made. However, I suggest another careful review of the English a few errors in wording and sentence structure are still there.

Response: We thank the reviewer for his careful review of the manuscript. We hope that we have sufficiently addressed the issues raised by the reviewer.

Line 77: capital S

Response: We have changed this.

Line 79: Not sure this captures the matter correctly. And the sentence swaps from the prevalence of hearing loss in younger adults (population) to their own hearing (an individual). I agree that ease of estimation is judged on a personal level. But the wording suggests that it is easier to estimate good hearing than poor(er) hearing. Is that the case? And that the ease translates to better estimation of prevalence. This section needs some work. I can see what the authors are getting at, but I am not convinced. And is this not complicated with the issue noted in the Discussion in Lines 267 onwards?

Response: We thank the reviewer for this point. What we aim to point out here is that statistics of self-reported hearing loss in a younger group is highly influenced by the relatively low prevalence of hearing loss, and should not be compared with the statistics of an older population. As this point has been addressed in more detail in the discussion, we shortened and rephrased the text in this section.

Line 84: Starting with ‘While’ leads the reader to expect more in the sentence. I suggest dropping the word.

Response: We have dropped the word while.

Line 89: ‘have’ is correct, not ‘has’

Response: We have changed has to have.

Line 121: I know that for the previous review it was not necessary to point out that the question and answers were in Dutch. However, upon reflection it would be good to make that clear, in case someone uses the same wording, and then compares results; this would not be entirely appropriate as the English translation/version has not been validated.

Response: We have added to the main text that the question was in Dutch.

Line 133: is it really not possible to reference this?

Response: Unfortunately not. This is done in all studies to be able to calculate with the hearing thresholds when the threshold of a participant is above the maximum stimulation level.

Table 1: The age range also needs to have ‘years’ in the first column

Response: We have added this to the table.

Line 177: ‘Hearing thresholds were’, not ‘Hearing loss was’

Response: We have changed this.

Line 181: ‘thresholds’? But actually it’s PTA.5-4 isn’t it?

Response: We have changed the sentence to ‘…increase of the PTA0.5-4 hearing threshold…’ as we feel this reflects the result best and meets the suggestion of the reviewer.

Lines 188 to 195; two versions of the table caption.

Response: We see what the reviewer means, but the second capitation is for figure 2.

Table 3: The placement of the words ‘Prevalence’ is a bit unhelpful when the 100% figures are seen. Can these 100% figures be removed?

Response: We have removed the 100% figure.

Line 240: This needs to be more nuanced. To start with, I do not think anyone would think it would replace clinical assessment of individuals, which this implies. On the other hand, I think it may have some clinical value in identifying people who are at a higher risk of hearing loss, and so this information has potential for use as a screening tool.

Response: We agree with the reviewer there are different perspectives on how the results can be interpreted. Therefore, we have chosen to emphasize the value of this tool in population based studies. The statement about the clinical value has been rephrased in a way that the perspective of screening has been taken in account, according to the reviewers comment.

Line 259 onwards: Comment/question: were males better or worse at estimation than females? This matter of self-reporting is complicated by things like denial of hearing problems and self-awareness which may be lower in males, but on the other hand males having a higher prevalence of HL. I think the authors have grappled with some of these issues.

Response: Males were better at the estimation than females, in spite of many other possible factors we think that the higher prevalence of hearing loss in males might have played an important role explaining this difference.

Line 266: points?

Response: We have changed point to points.

Submitted filename: Response to the reviewers.docx

Click here for additional data file.

14 Jan 2020

Assessing hearing loss in older adults with a single question and person characteristics; comparison with pure tone audiometry in the Rotterdam Study

PONE-D-19-19122R2

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With kind regards,

Francesco Martines, 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 #3: All comments have been addressed

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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 #3: Yes

**********

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

Reviewer #3: 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 #3: 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 #3: Yes

**********

6. Review Comments to the Author

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Reviewer #3: (No Response)

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Reviewer #3: Yes: Richard C Dowell

17 Jan 2020

PONE-D-19-19122R2

Assessing hearing loss in older adults with a single question and person characteristics; comparison with pure tone audiometry in the Rotterdam Study

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