A mixed‐methods study of the management of hearing loss associated with otitis media with effusion in children with Down syndrome

Objectives To scope current service provision across England for management of otitis media with effusion and hearing loss in children with Down syndrome; to explore professional decision‐making about managing otitis media with effusion and hearing loss; and to explore patient and public views on the direction of future research. Design Mixed methods including a service evaluation of NHS clinical practice through a structured telephone survey; a qualitative study of professional decision‐making with in‐depth interviews collected and analysed using grounded theory methods; patient/public involvement consultations. Participants Twenty‐one audiology services in England took part in the evaluation; 10 professionals participated in the qualitative study; 21 family members, 10 adults with Down syndrome and representatives from two charities contributed to the consultations. Results There was variation across services in the frequency of routine hearing surveillance, approaches to managing conductive hearing loss in infancy and provision of hearing aids and grommets. There was variation in how professionals describe their decision‐making, reflecting individual treatment preferences, differing approaches to professional remit and institutional factors. The consultations identified that research should focus on improving practical support for managing the condition and supporting decision‐making about interventions. Conclusions There is system‐level variation in the provision of services and individual‐level variation in how professionals make clinical decisions. As a consequence, there is inequity of access to hearing health care for children with Down syndrome. Future research should focus on developing core outcomes for research and care, and on improving decision support for families.

strategies in children with DS, with each having associated risks specific to children with DS. For example, grommet surgery may be difficult due to a narrow ear canal and children with DS are at increased risk of grommet associated ear discharge and eardrum perforation. 3 Air conduction hearing aids often do not fit well or exacerbate ear infections. Consequently, there is a need for further research regarding the effectiveness, acceptability and utilisation of management strategies for hearing loss associated with persistent OME, in children and young people with DS.
A recent project examined the feasibility and value of conducting research on management of OME in children with DS 1 : key findings were that parents had experienced a range of treatments and interventions for OME (as above), and perceive that management of their child's OME and hearing loss is inconsistent and based on uncertain foundations. 1 The top three interventions rated by clinicians to be most effective in children with DS were conventional hearing aids, bone-anchored auditory devices (softband or implanted) and grommets, 1 but the study did not investigate how professionals make decisions about these interventions or the preferred and actual patient pathway.
The objectives of this study are, for children with DS, OME and hearing loss, to: 1. Scope the range of current service provision across England; 2. Explore professional decision-making; and 3. Explore patient, parent and public views on the direction of future research.

| ME TH ODS
This was a mixed-methods study involving a service evaluation of NHS clinical practice for managing OME and hearing loss in children with DS and a qualitative investigation using grounded theory methods of professional decision-making in this patient group. Alongside, we ran patient, parent and public involvement groups to explore future research topics.

| Service evaluation
A telephone survey was conducted with clinicians in England who had responsibility for hearing services for children with DS. Clinicians were recruited through professional contacts as well as through a short online questionnaire to members of the Down Syndrome Medical Interest Group and British Academy of Audiology asking about the range of interventions for OME and hearing loss offered to children with DS in their department (air-bone conduction hearing aids, bone-anchored auditory devices on softbands, grommets) and willingness to take part in a 30-minute telephone survey. Services were sampled to include those providing bone-anchored auditory devices and air conduction hearing aids. The survey was structured to cover: protocols for hearing surveillance, criteria for intervention, types of interventions provided, outcome measures, the level of multidisciplinary involvement and parent involvement in decisionmaking in their service.

| Qualitative study of professional decisionmaking
We used grounded theory methods 4 to theorise the process by which professionals from different professional groups make decisions about managing hearing loss in children with DS. Professionals who work with children with DS and hearing loss were sent an invitation to participate in the study through regional audiology, ENT and child health networks and clinical contacts. The researcher initially interviewed those who responded first, and then selected from those who responded to provide contrast in profession, caseload and clinical population. 4 Professionals sampled included hearing support teachers, community paediatricians, audiologists and ENT surgeons. An optometrist was also sampled as it became apparent that this professional group is involved in decision-making about hearing aids in relation to eyeglasses. Volunteer participants contacted the researcher directly, and arrangements were made to conduct face-to-face interviews in the workplace where possible, or phone interviews.
The researcher (AH), who is also an audiologist, conducted indepth interviews guided by a topic guide (Table 1). They lasted between 30 and 60 minutes and were audio-recorded, transcribed verbatim and anonymised. Using an iterative process, data were gathered and analysed concurrently using the constant comparison method of grounded theory. Interviews were analysed using open and axial coding techniques; each statement was allocated a code and codes were linked from each data source into themes with variation in properties and dimensions. Reflective memos were kept Keypoints • Otitis media with effusion and hearing loss is common in children with Down syndrome and contributes to difficulties in listening and communication.
• The common interventions for otitis media with effusion and hearing loss have associated risks specific to children with Down syndrome and there is little evidence to guide clinical management for this group of children.
• It is not known how services are delivered or professionals make decisions about interventions.  during the process of data collection and analysis, and these were used to check emerging themes. Themes were grouped into a framework of decision-making. Transcripts were read by a second researcher (HP), an experienced qualitative researcher, who blindcoded a selection of data and checked derived codes.

| Patient and public involvement
Over the course of the study, PPI groups were held. These involved parents and carers of children with DS, people with DS, a charitable organisation representing people with DS and a charitable organisation representing children with hearing loss. The aim of the groups was to identify research topics and priorities relating to OME and hearing loss that are important to them as service users, to present the topics arising from the research and to discuss future research priorities. The findings which arose are grouped according to common topics and a narrative summary presented.

| Ethical considerations
The service evaluation was conducted with agreement from Univer- Three PPI groups were held and attended by a total of twelve mothers and one grandmother; additionally, eight parents provided feedback outside the groups. One group was held and attended by ten adults with DS. Consultation with representatives from two charities also took place.

| Variation in service delivery
There was variation in key aspects of service delivery for children with DS ( Table 2). All services report that parents are involved in decision-making and are provided with written information.

| Professional views on decision-making
There was variation in how professionals describe their decisionmaking about OME and hearing loss in children with DS. These variations reflect individual preferences, differing approaches to professional remit and institutional factors. The key theme, labelled "focus," refers to how the child's life is viewed by the professional and whether their focus is primarily on a child's ears and hearing or on their health, development, family and school life. This is influenced by how they viewed their professional remit, how the relative burdens and benefits of interventions and treatments were perceived and ultimately how decision-making was described ( Figure 1). Key themes are described below, with example quotations in Table 3.

| Impact of OME and hearing loss
There was variation in how the impact of OME and hearing loss on a child with DS was viewed. Some participants primarily focused on the ear and hearing impact of the condition, whereas others considered the impact on speech and communication and the wider impact on the child and family. Those who focused on ears and hearing described decision-making as primarily focused on alleviating the OME or hearing loss, viewing hearing aids or surgery as the solution.
This contrasted with those who took an approach to decision-making focused on developing listening, communication and participation.
Views on treatment and intervention success varied from a focus on curing OME or wearing hearing aids to those who viewed success as improving or maintaining quality of life or developmental outcomes, not always involving hearing aids or surgery and including sign language and other communication tactics.

| Professionals have treatment and intervention preferences
There was evidence of preferences amongst many of the professional participants for particular treatments and interventions for children with DS, with some preferring grommets and others hearing aids. This was not related to professional group, and there was variation in preference within groups. Participants described observing  Those who considered other aspects of the child, such as their sensory sensitivity or likely tolerance, were more nuanced in their discussion around decision-making.

| Context
The context in which decisions were made was influenced by availability of professional expertise, access to treatments and interventions, the health service set-up and the child's educational setting.
Having highly skilled professionals within a service provided a context in which decisions could be made effectively, such as having access to surgical expertise in children with DS and therefore ability to refer for grommet intervention or audiologists with experience of working with a range of hearing aids.

| Parent, patient and public involvement
Parents discussed their ideas for future research through relating their personal experiences, both positive and negative. Parents were asked to discuss their views on the importance of future research on improving decision support and improving service provision and management of OME and hearing loss. Adults with DS attended a group workshop on the topic of ears and hearing; they discussed their personal experiences and knowledge about hearing, hearing loss and audiology services. Charity representatives gave their perspectives (summarised in Table 4).

| Synopsis of key findings
Using mixed methods and involving a range of professionals as well as people with DS and families, we have identified that across England there is system-level variation in the provision of services for children with DS affected by OME and hearing loss, as well as individual-level variation in how professionals make clinical decisions. As

| Strengths of the study
Our approach enabled us to study the hearing services provided to children with DS from a range of perspectives, including that of the

| Comparison with other studies
In line with the model of candidacy for health care described by

| Clinical applicability
System constraints impact on adjudication of candidacy. These constraints were described in our data, such as availability of local clinical expertise, and these influenced treatment decisions. Given the lack of accepted national guidance about the management of OME and hearing loss in children with DS, practice is often decided at a local level and we observed variation in how services are delivered.
The NICE guideline on surgical management of OME for children 11 does not specify treatment criteria for children with DS, despite them being both at higher risk for OME and likely to be more adversely affected by its associated hearing loss. This may be due to the lack of evidence in children with DS and lack of consensus amongst professionals, and likely contributes to inequalities and unwarranted variations in care. 12 Based upon these findings, further work for children with DS affected by OME and hearing loss should include the following: