Hyperacusis in children: a scoping review.

BACKGROUND: Hyperacusis is a chronic condition commonly defined as a lowered tolerance or increased sensitivity to everyday environmental sounds. It has been viewed as a paediatric disorder which can cause significant impairment to a child's normal functioning. Although clinical guidance highlights the importance of identifying whether the child has intolerance to loud sounds and managing this appropriately, there are currently no assessment or treatment methods that have been designed and tested for use with children with hyperacusis. A review is therefore indicated to consider the profile of children with hyperacusis as a basis for future research into their assessment and treatment. METHOD: A scoping review methodology was followed with literature searches conducted in Embase, PsychINFO, PubMed CENTRAL, Scopus, Web of Science and Google Scholar. Research articles were included if they reported on research studies describing children diagnosed with hyperacusis, providing clinical profile information, and/or reporting on an assessment or management method for children with a primary complaint of hyperacusis. Data were charted on Excel and verified by a second researcher. Twenty-one research articles were included. RESULTS: Children with hyperacusis are typically described in terms of age at presentation, troublesome sounds, physical sensation, behavioural reactions, coping strategies, comorbid conditions and impact on daily life. Methods of assessing the children include semi-structured interviews, questionnaires, neurological assessment, observation and uncomfortable loudness levels. Management methods include psychological therapy, sound therapy, tinnitus retraining therapy, medication and neuro-rehabilitation. CONCLUSION: The information we catalogued on various elements of clinical profile, assessment and management can serve as a stepping stone in future research developing questionnaires for clinical measurement of the impact of hyperacusis on children, and the measurement of treatment related change in clinic and in trials. Positive outcomes were noted by the authors following all of the above treatments; future research must compare these and specify the parameters for optimal results.

An evaluation of paediatric tinnitus services in UK National Health Service audiology departments.

BACKGROUND: Whilst evidence indicates many children experience troublesome tinnitus, specialist services for children are far less established than those available for adults. To date, there is limited understanding of how paediatric tinnitus is managed in the UK, and to what extent current practice reflects what is recommended. This service evaluation aimed to 1) profile how tinnitus in children is managed in UK clinical practice, and assess to what extent care provided by services reflects advice included in the British Society of Audiology (BSA) Tinnitus in Children Practice Guidance, 2) collate clinician opinions on how services may be optimised, and 3) identify common problems experienced by children who present with bothersome tinnitus in clinic. METHODS: As part of a larger survey, eight questions regarding services for paediatric tinnitus were distributed to UK NHS audiology services via email and social media. Representatives from eighty-seven services responded between July and September 2017. RESULTS: Fifty-three percent of respondents reported that their department provided a paediatric tinnitus service. Among these services, there was widespread use of most BSA recommended assessments and treatments. Less widely used practices were the assessment of mental health (42%), and the use of psychological treatment approaches; cognitive behavioural therapy (CBT) (28%), mindfulness (28%), and narrative therapy (14%). There was varied use of measurement tools to assess tinnitus in children, and a minority of respondents reported using adult tinnitus questionnaires with children. Frequently reported tinnitus-related problems presented by children were sleep difficulties, concentration difficulties at school, situation-specific concentration difficulties, and emotional distress. CONCLUSIONS: Approaches used to manage children with troublesome tinnitus in UK NHS services are largely consistent and reflective of the current practice guidance. However, findings from this study indicate specialist staff training, access to child-specific tools, and the treatment and referral of children with tinnitus-related psychological problems represent key areas in need of optimisation.

A narrative synthesis of research evidence for tinnitus-related complaints as reported by patients and their significant others.

BACKGROUND: There are a large number of assessment tools for tinnitus, with little consensus on what it is important to measure and no preference for a minimum reporting standard. The item content of tinnitus assessment tools should seek to capture relevant impacts of tinnitus on everyday life, but no-one has yet synthesised information about the range of tinnitus complaints. This review is thus the first comprehensive and authoritative collection and synthesis of what adults with tinnitus and their significant others report as problems in their everyday lives caused by tinnitus. METHODS: Electronic searches were conducted in PubMed, Embase, CINAHL, as well as grey literature sources to identify publications from January 1980 to June 2015 in which participants were enrolled because tinnitus was their primary complaint. A manual search of seven relevant journals updated the search to December 2017. Of the 3699 titles identified overall, 84 records (reporting 86 studies) met our inclusion criteria and were taken through to data collection. Coders collated generic and tinnitus-specific complaints reported by people with tinnitus. All relevant data items were then analyzed using an iterative approach to narrative synthesis to form domain groupings representing complaints of tinnitus, which were compared patients and significant others. RESULTS: From the 86 studies analyzed using data collected from 16,381 patients, 42 discrete complaints were identified spanning physical and psychological health, quality of life and negative attributes of the tinnitus sound. This diversity was not captured by any individual study alone. There was good convergence between complaints collected using open- and closed-format questions, with the exception of general moods and perceptual attributes of tinnitus (location, loudness, pitch and unpleasantness); reported only using closed questions. Just two studies addressed data from the perspective of significant others (n = 79), but there was substantial correspondence with the patient framework, especially regarding relationships and social life. CONCLUSIONS: Our findings contribute fundamental new knowledge and a unique resource that enables investigators to appreciate the broad impacts of tinnitus on an individual. Our findings can also be used to guide questions during diagnostic assessment, to evaluate existing tinnitus-specific HR-QoL questionnaires and develop new ones, where necessary. TRIAL REGISTRATION: PROSPERO registration number: CRD42015020629 . Protocol published in BMJ Open. 2016;6e009171.

Tinnitus in Children.

This perspective reviews the current state of the art and literature on tinnitus in children, prevalence and risk factors, clinical management, and future priorities for healthcare provision and research. Most research in the field to date appears to be prevalence studies, which have reached dramatically different estimates; this reflects the lack of a standard language when asking about the presence of tinnitus, or how bothersome, distressing, or negatively impacting it is for the child. Estimates are also likely affected by a lack of awareness of tinnitus amongst children and parents. Children are less likely to spontaneously report tinnitus than adults, and parents are often unaware their child could even develop tinnitus, considering it a disease of older age for example. It is critical that children are asked and learn about tinnitus. In hearing clinics, clinicians should routinely ask about all children about tinnitus and offer tinnitus care and settings that are child- and family-friendly. As well as asking directly, clinicians should be alert to soft signs of tinnitus such as unexplained listening, speech perception, concentration difficulties, worry or anxiety, or difficulties completing hearing tests or using hearing aids. The recently developed impact of Tinnitus in Children Questionnaire (iTICQ) can then be used to assess problems that are most commonly core to children's experience of tinnitus. Clinical guidelines for tinnitus in children are few but provide recommendations for additional paediatric questionnaires and alternative assessments and for a range of treatment options. Of note, however, is the lack of clinical trials and, therefore, evidence of the effectiveness of any treatment for tinnitus in children. Significant and concerted work is therefore needed to raise awareness of tinnitus in children, understand the scale of clinical need, and standardise and evaluate clinical management options.

Natural history of otitis media with effusion-related hearing loss in children under 12 years: a systematic review.

OBJECTIVE: To assess natural history of otitis media with effusion (OME)-related hearing loss and OME causing hearing loss in children under 12 years. METHODS: Embase, MEDLINE, CINAHL, INAHTA database, CENTRAL, CDSR, Epistemonikos and PsycINFO were searched to identify observational single group studies, and comparative studies with untreated control arms published in English up to June 2022, reporting natural history of OME-related hearing loss and OME causing hearing loss. Risk of bias and overall quality of evidence were assessed using the JBI (Joanna Briggs Institute (JBI) checklist and GRADE (Grading of Recommendations Assessment, Development and Evaluation) methodology, respectively. RESULTS: Thirteen studies with 24-639 children were included. Resolution of OME-related hearing loss was 50% by 3 months, 60% by 6 months and 61-77% by 12 months. Resolution of OME causing hearing loss (OME of <1 month, >3 months, >6 months or unknown duration before follow-up) was 23-55% by 3 months, 20-50% by 6 months, 31% by 9 months and 21-93% by 12 months, depending on population and how resolution was defined. Resolution of chronic OME (OME of >12 months duration before follow-up) was only 7% by 1 month, 12% by 6 months and 6% by 12 months. Resolution was only 42% by 57 months in children with primary ciliary dyskinesia. CONCLUSIONS: There was greater resolution of OME-related hearing loss over longer follow-up periods. Resolution of OME causing hearing loss also showed a trend towards greater resolution over longer follow-up periods; however, this did not follow a linear pattern, potentially due to differences in populations and definitions of resolution across studies.

Modifiable risk factors for developing otitis media with effusion in children under 12 years in high-income countries: a systematic review.

OBJECTIVE: To systematically assess the modifiable risk factors for developing otitis media with effusion (OME) in children under 12 years. METHODS: We searched Embase, MEDLINE, INAHTA database, CENTRAL, CDSR and Epistemonikos for cohort studies with ≥40 children per arm/prognostic factor, published in English from 2000 to November 2022. We assessed risk of bias using the Quality in Prognosis Studies checklist, and overall evidence quality was assessed using Grading of Recommendations Assessment, Development and Evaluation (GRADE) methodology. Outcomes were analysed as risk ratio (RR), OR or Peto OR. RESULTS: Seven studies totalling 2 760 292 children were included. The evidence was very low quality. Fluid or pus discharge from ears (OR 2.1, 95% CI 1.01 to 4.35) and exposure to other children (RR 2.79, 95% CI 1.98 to 3.93) (OR 5.21, 95% CI 2.9 to 9.36) were strongly associated with development of OME. Coughs/colds ≥5 times (OR 1.91, 95% CI 1.22 to 2.99), breathing problems ≥5 times (RR 1.78, 95% CI 1.26 to 2.53) and ear infections (RR 1.95, 95% CI 1.39 to 2.72) in past year were associated with development of OME. Adenoid hypertrophy was strongly associated with development of fluctuating OME (recurrent OME) (OR 9.96, 95% CI 5.17 to 19.19). There was scare evidence for some potential modifiable risk factors, including breast feeding, household smoking, gastro-oesophageal reflux, dummy use and swimming. CONCLUSIONS: Upper respiratory tract infection, ear infection, adenoid hypertrophy and exposure to other children could be the predictors for development of OME. Further observational studies are needed to investigate other potential modifiable risk factors.

A scoping review to catalogue tinnitus problems in children.

BACKGROUND: The reported prevalence of tinnitus in children is similar to that in adults. However, unlike adults, there is relatively little understanding of the tinnitus-related problems children experience. Knowledge of the problems experienced by adults has led to the development of numerous clinical questionnaires used by health professionals in assessment and treatment practices; to date no child-specific questionnaire measure of tinnitus has been developed. To support development of a questionnaire measure of tinnitus in children, the aim of this scoping review was to catalogue the peer-reviewed and grey scientific literature according to 1) the methods used to identify problems experienced by children with tinnitus, 2) tinnitus-related problems observed in or reported by children, and 3) research recommendations suggested by investigators with regards to tinnitus in children. METHODS: A scoping review was conducted following an established methodological framework. Records were included where a tinnitus-related problem was reported in a child 18 years or younger, and tinnitus was reported as the primary complaint. Tinnitus problems were extracted and categorised into problem themes. RESULTS: Thirty-five records met the inclusion criteria for this review. Methods used to identify tinnitus-related problems in children, and the number and range of problems reported, varied across the records. Symptom impact was summarised according to six problem themes; Physical health, Cognitive health, Hearing and listening, Emotional health, Quality of life, and Feeling different/isolated. Identified research recommendations highlighted a demand for more standardised approaches. CONCLUSIONS: The findings evidence the detrimental impact tinnitus can have on a child's quality of life and emotional wellbeing. The current British Society of Audiology Tinnitus in Children Practice Guidance recommends management practices to address the most broadly reported problems identified in this review; sleep difficulties, emotional difficulties, and concentration and hearing problems at school. Given the finding of this review, we suggest problems relating to the impact of tinnitus on quality of life and feelings of isolation are also important problem domains to consider when managing a child who has tinnitus. Current variability in the approach to identifying children's tinnitus problems underlines the importance of developing a standardised and dedicated measure of tinnitus in children.

Identifying and prioritising unanswered research questions for people with hyperacusis: James Lind Alliance Hyperacusis Priority Setting Partnership.

OBJECTIVE: To determine research priorities in hyperacusis that key stakeholders agree are the most important. DESIGN/SETTING: A priority setting partnership using two international surveys, and a UK prioritisation workshop, adhering to the six-staged methodology outlined by the James Lind Alliance. PARTICIPANTS: People with lived experience of hyperacusis, parents/carers, family and friends, educational professionals and healthcare professionals who support and/or treat adults and children who experience hyperacusis, including but not limited to surgeons, audiologists, psychologists and hearing therapists. METHODS: The priority setting partnership was conducted from August 2017 to July 2018. An international identification survey asked respondents to submit any questions/uncertainties about hyperacusis. Uncertainties were categorised, refined and rephrased into representative indicative questions using thematic analysis techniques. These questions were verified as 'unanswered' through searches of current evidence. A second international survey asked respondents to vote for their top 10 priority questions. A shortlist of questions that represented votes from all stakeholder groups was prioritised into a top 10 at the final prioritisation workshop (UK). RESULTS: In the identification survey, 312 respondents submitted 2730 uncertainties. Of those uncertainties, 593 were removed as out of scope, and the remaining were refined into 85 indicative questions. None of the indicative questions had already been answered in research. The second survey collected votes from 327 respondents, which resulted in a shortlist of 28 representative questions for the final workshop. Consensus was reached on the top 10 priorities for future research, including identifying causes and underlying mechanisms, effective management and training for healthcare professionals. CONCLUSIONS: These priorities were identified and shaped by people with lived experience, parents/carers and healthcare professionals, and as such are an essential resource for directing future research in hyperacusis. Researchers and funders should focus on addressing these priorities.

Hyperacusis in children: A clinical profile.

OBJECTIVE: Hyperacusis is commonly seen in clinical paediatric practice and can be distressing for the children and their families. This paper looks at the clinical profile of children seen for hyperacusis in a paediatric audiology service and reviews the possible underlying mechanisms. METHODS: Retrospective study of case notes of 61 children with troublesome hyperacusis seen in the paediatric audiology service, looking at their clinical presentation and presence of other medical conditions. RESULTS: Hyperacusis was the main presenting complaint in more than half of the cases (n = 31, 51%). The commonest age at presentation with this problem was 3-4 years (n = 33, 54%). Hearing was normal in the majority of these children (n = 41, 67%). An active middle ear problem was observed in nearly half (n = 29, 48%) of all the children, of which glue ear (otitis media with effusion, OME) was the commonest. Presence of a neurodevelopmental condition was found in almost half (n = 28, 46%) of these patients of which autistic spectrum disorder was the commonest (8/61, 13%). In nearly one-fourth of the children (23%), presence of both middle ear problems and neurodevelopmental was noted. Tinnitus was an accompanying symptom reported in 11% of all the patients. CONCLUSION: Hyperacusis may commonly present at a very young age. Awareness of different clinical presentations, presence of other medical conditions and possible underlying pathomechanisms in children with hyperacusis can be helpful for clinicians in informing prognosis, counselling and in individualising management plan.

Dimensions of tinnitus-related complaints reported by patients and their significant others: protocol for a systematic review.

INTRODUCTION: Over 70 million people in Europe and >50 million people in the USA are reported to experience tinnitus (the sensation of noise in the absence of any corresponding sound source). Tinnitus is a multidimensional concept. Individual patients may report different profiles of tinnitus-related symptoms which may each require a tailored management approach and an appropriate measure of therapeutic benefit. This systematic review concerns the patient perspective and has the purpose to find what symptoms are reported by people who experience tinnitus and by their significant others. METHODS AND ANALYSIS: This protocol lays out the methodology to define what dimensions of tinnitus-related symptoms patients and their significant others report as being a problem. Methods are defined according to the Preferred Reporting Items for Systematic reviews and Meta-analyses for Protocols (PRISMA-P) 2015 and data will be collated in a narrative synthesis. Findings will contribute to the eventual establishment of a Core Domain Set for clinical trials of tinnitus. ETHICS AND DISSEMINATION: No ethical issues are foreseen. Findings will be reported at national and international ENT and audiology conferences and in a peer-reviewed journal. TRIAL REGISTRATION NUMBER: CRD42015020629.

Current-reported outcome domains in studies of adults with a focus on the treatment of tinnitus: protocol for a systematic review.

INTRODUCTION: In Europe alone, over 70 million people experience tinnitus. Despite its considerable socioeconomic relevance, progress in developing successful treatments has been limited. Clinical effectiveness is judged according to change in primary outcome measures, but because tinnitus is a subjective condition, the definition of outcomes is challenging and it remains unclear which distinct aspects of tinnitus (ie, 'domains') are most relevant for assessment. The development of a minimum outcome reporting standard would go a long way towards addressing these problems. In 2006, a consensus meeting recommended using 1 of 4 questionnaires for tinnitus severity as an outcome in clinical trials, in part because of availability in different language translations. Our initiative takes an approach motivated by clinimetrics, first by determining what to measure before seeking to determine how to measure it. Agreeing on the domains that contribute to tinnitus severity (ie, 'what') is the first step towards achieving a minimum outcome reporting standard for tinnitus that has been reached via a methodologically rigorous and transparent process. METHODS AND ANALYSIS: Deciding what should be the core set of outcomes requires a great deal of discussion and so lends itself well to international effort. This protocol lays out the first-step methodology in defining a Core Domain Set for clinical trials of tinnitus by establishing existing knowledge and practice with respect to which outcome domains have been measured and which instruments used in recent registered and published clinical trials. ETHICS AND DISSEMINATION: No ethical issues are foreseen. Findings will be reported at national and international ear, nose and throat (ENT) and audiology conferences and in a peer-reviewed journal, using PRISMA (Preferred Reporting Items for Systematic reviews and Meta-analysis) guidelines. TRIAL REGISTRATION NUMBER: The systematic review protocol is registered on PROSPERO (International Prospective Register of Systematic Reviews): CRD42015017525.

The impact of cochlear implants from the perspective of significant others of adult cochlear implant users.

OBJECTIVE: To explore the benefits and shortcomings of cochlear implants as perceived by a cochlear implant user's significant other in the context of the World Health Organization's International Classification of Functioning, Disability and Health (ICF). STUDY DESIGN: An open-ended questionnaire concerning the perceived shortcomings and benefits of the implant was completed by the significant others of cochlear implants users. SETTING: The study involved the participants of an adult cochlear implant program. SUBJECTS: Significant others as selected by the cochlear implant user. MAIN OUTCOME MEASURE: Nature of effects reported by the significant other relating to both the cochlear implant user and the significant other using the ICF domains. RESULTS: Benefits experienced by the significant others included a decrease in communication burden and an increased participation in life activities. Those affecting the cochlear implant user were related to psychological issues, particularly an increase in confidence. Improved quality of life was reported for both partners. Reported shortcomings predominantly involved the cochlear implant user and were device related. CONCLUSION: Because family and friends play an important role in how those with a profound hearing impairment cope with the impairment, it is important to acknowledge the impact that an intervention has on those supportive individuals. This study shows that a cochlear implant provides psychosocial and acoustic benefits for the significant other and the cochlear implant user. The ICF was able to accommodate both the auditory and nonauditory impact of the cochlear implant and the residual hearing difficulty.