Introduction

Over the years, research on tinnitus has predominantly focused on adults, leaving studies on the pediatric population relatively scarce [1]. While estimates of tinnitus prevalence in children vary widely, studies indicate it could affect between 4.7% and 46% of those with normal hearing and between 23.5% and 62.2% of those with hearing loss, depending on the population, study design, and definition used [1]. There is still a significant gap in our understanding of tinnitus and its impact on children, but existing studies suggest that the sound children experience can be responsible for difficulties in concentrating as well as in hearing and listening, which in turn can cause problems at school and in other activities. Furthermore, tinnitus can impact their sleep, lead to a worsening of their mental health, and result in social isolation [2]. However, the assessment of the experienced tinnitus-related problems in these studies too date largely relied on open-ended questioning during clinical interviews and inconsistent use of structured questions, with much of the data reported by adult proxies [2]. Additionally, it is important to note that children are less likely to spontaneously report tinnitus and associated problems [3,4], and when they do, they often struggle with describing these abstract concepts [5]. This underscores the need for a more standardized, child-centered approach to measuring the impact of pediatric tinnitus in both clinical care and research.

Patient reported outcome measures (PROMs) have become an essential tool for understanding and quantifying the subjective experiences of individuals with varying health conditions. Generic PROMs, such as the Pediatric Quality of Life Inventory (PEDsQL) [6], provide a broad assessment of a child’s overall well-being and allow for comparisons across different conditions and populations. However, their broad nature may mean they lack sensitivity to measure the specific symptoms and challenges associated with particular conditions, such as tinnitus. While condition-specific PROMs lack the generalizability of generic PROMs, they are more sensitive to capture these experiences relevant to a particular condition or population [7]. For adults with tinnitus, multiple of these condition-specific PROMs are available, such as the Tinnitus Functional Index (TFI) [8] and the Tinnitus Handicap Inventory (THI) [9]. While these PROMs are sometimes used to assess tinnitus in children [2,10], they were not developed with the intention to be used in this population and have not been validated for this purpose [10]. Therefore, a condition-specific PROM for children with tinnitus, the Impact of Tinnitus in Children Questionnaire (iTiCQ) [11], has recently been developed. This self-report tool for children aged 8–16 years aims to address the impact of tinnitus in children, though further validation and translations are necessary to establish its effectiveness and accessibility across different populations.

A more comprehensive and innovative method for assessing health outcomes involves integrating generic and condition-specific approaches in PROMs. This method enables the development of tools that can accurately assess condition-specific impacts, while maintaining the ability to compare outcomes between different conditions and populations. This integrated approach is especially advantageous for conditions with multiple comorbidities, such as tinnitus [12,13], as the experiences of patients do not have to be attributed to a single health condition [14].

The Patient-Reported Outcomes Measurement Information System (PROMIS) offers a framework to enable this integration of generic and condition-specific approaches in health assessment. The PROMIS is a dynamic measurement system comprising generic item banks developed to assess physical, mental and social health outcomes in both adults and children [15,16]. An item bank is a collection of items that measure the same construct, or domain, such as sleep disturbance, depression, or peer relationships. These items are developed based on existing PROM items, which are evaluated, standardized and methodologically tested before they are included in the PROMIS item bank [17–19]. The constructs measured by these item banks can be assessed in a variety of languages using short forms or Computerized Adaptive Testing (CAT), with scores provided on a T-score metric. This allows for comparison with the general population, as well as between populations and conditions [16]. The dynamic character of the PROMIS enables flexibility in selecting item banks relevant to specific research goals or clinical applications, thereby facilitating a tailored approach to health assessment and providing the ability to use a generic framework to facilitate condition-specific assessment.

The objective of the proposed study is therefore to develop a PROM set for children with tinnitus using the PROMIS methodology. By combining input from patients with expert opinions, the study aims to create a list of outcomes that accurately reflect the impact of tinnitus on children of different ages and select relevant domains from the PROMIS framework to develop a validated PROM set. This approach ensures that the measure captures the specific impact of pediatric tinnitus while utilizing the generic framework of PROMIS for broader applicability and comparability.

Methods

Study design

The study is a monocenter, mixed-methods, multi-phase observational study conducted at the Wilhelmina Children’s Hospital (Wilhelmina Kinderziekenhuis; WKZ), part of the University Medical Center (UMC) Utrecht, a tertiary referral clinic in the Netherlands.

The study is organized into 6 consecutive steps (Fig 1), with the objective to develop a PROM set for children with tinnitus using the PROMIS framework. The study starts with identifying patient-important outcomes for pediatric tinnitus trough focus groups with children. The underlying constructs of these outcomes will then be defined and linked to relevant PROMIS domains. The defined outcomes and associated domains will be discussed with an expert panel as well as with the focus groups, after which necessary adjustments will be made to the measure. The last steps will consist of assessing the psychometric qualities of the PROM set by assessing the content validity in the expert panel and the reliability in a sample of children with tinnitus. The methodology for each step of the study is detailed in the following sections.

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Fig 1. Study design.

https://doi.org/10.1371/journal.pone.0334549.g001

Discussion

To address the need for more standardized, child-centered instruments for assessing the impact of pediatric tinnitus, this study proposes the development of a validated PROM set using the Patient-Reported Outcomes Measurement Information System. By identifying patient-important outcomes for different age groups and linking these outcomes to PROMIS domains, this approach ensures that the set is both comprehensive and sensitive to the diverse ways tinnitus might affect children of varying ages. While tailored to the specific domains impacted by tinnitus, the use of the generic PROMIS item banks ensures that the outcomes remain comparable across various health conditions and populations [16], and allows for an assessment of symptoms without requiring explicit attribution to a specific condition [14]. Additionally, the PROMIS item banks’ availability in multiple languages and their ease of administration and interpretation make this tool widely applicable and well-suited for both clinical care and research settings. The possibility to administer the item banks using Computerized Adaptive Testing provides the additional benefit of dynamically tailoring the assessment based on the respondent’s answers, thereby minimizing patient burden while increasing the accuracy of the results [16].

It is, however, important to also recognize potential challenges in using PROMIS to develop this PROM set for measuring the impact of tinnitus in children. There might be outcomes that are encountered during the creation of the patient-important outcomes list, that are not currently covered by existing PROMIS domains. In such cases, either a new PROMIS domain should be added, or additional items may need to be included within an existing item bank to comprehensively cover the domain. This process would involve collaboration with the PROMIS group, either to create a new domain with corresponding item bank or expand an existing one, both of which would require further validation for application. This process falls beyond the scope of the current study. However, the PROM set can be seen as a dynamic instrument which can be adjusted by adding or removing item banks/domains in future, also after finalization of this study and launching of the first concept of this measure. This adaptability ensures that the PROM set also remains relevant and responsive to emerging needs in health measurement.

Acknowledgments

The authors would like to thank Caroline Terwee for her valuable advice and insights regarding the study design.