Brain tumour diagnoses by hospital eye services: is there evidence of a Barker effect? ( ODR1920_288 )

University of Bristol requires access to NHS England data for the purpose of the following research project: Brain tumour diagnoses by hospital eye services: is there evidence of a Barker effect?

Papilloedema refers to swelling at the back of the eyes caused by increased pressure inside the head and can indicate life- or sight-threatening disease, e.g., a brain tumour, meningitis, or blood clot. In 2016, an optometrist Honey Rose was convicted of gross negligence manslaughter after failing to diagnose papilloedema in an 8-year-old boy called Vincent Barker who later died. As a result of the Honey Rose court case, the number of referrals from optometrists to hospital eye departments of people suspected to have papilloedema increased sharply. The aim of the project is to find out whether people with intracranial tumours were diagnosed earlier and more often by hospital ophthalmologists in England since 2016.

The following is a summary of the aims of the research project provided by University of Bristol.

To determine the time to diagnosis and outcome of patients with intracranial tumours before vs after the Honey Rose case in 2016.

1. To determine whether time to diagnosis depends on route to diagnosis before/after Honey Rose?

2. To determine whether outcomes (mortality/survival) depend on time and/or route to diagnosis for patients before/after Honey Rose.

3. To determine whether time and route to diagnosis and outcomes are influenced by tumour type, sex, ethnicity, smoking, age, geography and deprivation?

The current study is approved by the Health Research Authority (IRAS 286872; REC 20/LO/0948) and is sponsored by the University of Bristol (2020-4244).

University of Bristol aim to use the data requested to better understand the referral pathways for patients with suspected papilloedema/raised intracranial pressure from primary care (GP or optometrist). The results of this work will contribute to the wider outputs of the Improving the Diagnostic accuracy of referrals for Papilloedema (DIPP study), run by the University of Bristol by the same study team, which has the aim of "Improving the Diagnostic accuracy of referrals for PaPilloedema." One of the aims of the DIPP study is to develop evidence-based referral guidelines to ensure people with papilloedema are accurately identified in primary care, referred to the appropriate service in secondary care and without unnecessary delay. Currently, such referral pathways do not exist.

The DIPP study team and project are guided by the ‘Brain tumour diagnoses by hospital eye services: is there evidence of a Barker effect?’ steering group, comprised of an independent chair, independent clinical researcher, the Chief Investigators and two PPI representatives from the study.

The following NDRS data products are being requested previously and disseminated in 2021.

• NDRS Cancer Registry

• NDRS Cancer Wait times (CWT)

• NDRS Hospital Episode Statistics Admitted Patient Care (HES APC)

• NDRS Hospital Episode Statistics Accident & Emergency (HES A&E)

• NDRS Hospital Episode Statistics Outpatients (HES OP)

• NDRS Diagnostic Imaging Dataset (DIDs)

• NDRS Systemic Anti Cancer Dataset (SACT)

• NDRS Radiotherapy Dataset (RTDS)

The following NDRS data products are being requested as a refresh under this application.

• NDRS Cancer Registry (mortality fields only)

The level of the data is Pseudonymised. This level of data is necessary to capture true positive referrals (patients who do have intracranial tumours), cancer stage, treatment, and mortality (where relevant). Since the study team are investigating route to diagnosis of intracranial tumours, the plan is to work backwards from the date of diagnosis within the Cancer Registry, and cross reference this with the date of the diagnostic intracranial imaging in the DIDs dataset and the specialty of the clinician requesting the imaging from linked HES data (APC, A&E, OP). To compare the outcomes of people who underwent different routes to diagnosis, the study team will investigate their cancer wait times (CWT), and outcomes from the Radiotherapy (RTDS) and systemic anti-cancer treatment (SACT) datasets to establish if there are treatment differences between groups. The quantum of data could not be reduced without significantly affecting achievement of the project aims.

The data will be minimised and limited as follows to data for a study cohort identified by NHS England as meeting the following criteria:

• Limited to data between [2013- 2018];

• Benign and malignant intracranial tumours

• Limited to conditions relevant to the study identified by specific ICD or OPCS codes; C69-C72, C720-C725, C751-C753, D32, D42, D330-D334, D352-D354, D430-D434, D443-D445.

• Limited to the geographic area of England only.

University of Bristol is the research sponsor and the data controller as the organisation responsible for ensuring that the data will only be processed for the purpose described above.

The lawful basis for processing personal data under the UK GDPR is:

Article 6(1)(e) - processing is necessary for the performance of a task carried out in the public interest or in the exercise of official authority vested in the controller.

The lawful basis for processing special category data under the UK GDPR is:

Article 9(2)(j) - processing is necessary for archiving purposes in the public interest, scientific or historical research purposes or statistical purposes in accordance with Article 89(1) based on Union or Member State law which shall be proportionate to the aim pursued, respect the essence of the right to data protection and provide for suitable and specific measures to safeguard the fundamental rights and the interests of the data subject.

This project will determine how routes and time to diagnosis of people with intracranial tumours have been affected by the widespread media coverage of the Honey Rose/Vincent Barker case and the subsequent impacts of increased optometry referral numbers and hospital service demands in secondary care. It will highlight the strengths and weaknesses of different routes to diagnosis for people diagnosed with intracranial tumours.

The outputs from this work will be in the public interest. Firstly, the results of this analysis will contribute to the development of evidence-based referral guidelines by the DIPP study team that will improve the identification of people with intracranial tumours in primary care and reduce their time to diagnosis and risk of mortality. Secondly, these results will raise public awareness about the benefits of regular eye examinations to check on general health.

The study entitled "Brain tumour diagnoses by hospital eye services: is there evidence of a Barker effect?" is funded by educational grants from the University of Bristol. However, funding of the wider DIPP study is provided by two grants from the National Institute of Health Research (NIHR) that support parallel research to investigate geographical variations in referral practices and management of patients with papilloedema across England. This funding is in place until May 2024. The National Institute of Health Research (NIHR) will not have access to, nor process the NHS England data.

University of Bristol is the Sole Data controller and sole data processor who provide all IT support for the provision of this project.

The DIPP study has a steering group, comprised of an independent chair, independent clinical researcher, the Chief Investigators and two PPI representatives from the study. All members of the steering group, except the Chair, are based in Bristol. The Chair, who is based at the University of Manchester, is working in an advisory capacity and has no access to the data and no data controllership responsibilities.

Data will only be accessed by substantive employees of the data controller. This includes the supervision of postgraduate students at the University of Bristol who may be processing the data. Postgraduate students at the University of Bristol undergo mandatory training in Information Governance and Data Protection.

The DIPP study has a hospital PPI group comprising 6 volunteers from the Brain Tumour Support Group who were diagnosed with a brain tumour after presenting to their optician with visual problems. The study team asked the PPI group about their views on using data from NHS England to find out "whether the Honey Rose case affected the time to diagnosis of people with brain tumours and whether more brain tumours are diagnosed by eye specialists now compared with the time before the Honey Rose case".

There were 4 responses, which were all positive:

“I would be very happy to allow a team of research scientists access such information.”

“Personally, I would be happy for any information to be passed on, anything to help with the research, but not personal info! Eg, mobile numbers or actual addresses, other than that I certainly wouldn’t have a problem.”

“Please do use my information, as it is thanks to my optician that I was diagnosed.”

“I would have no problem with information about me being accessed by research scientists. Although my meningioma was removed in 1999, it was my optician who diagnosed that something was seriously wrong. I do hope that you are able to access the cancer registry data.”

In line with the National data opt-out policy, opt-outs are not applied because the data is not Confidential Patient Information as defined in section 251(10) and (11) of the National Health Service Act 2006

Where individuals have opted out of disease registration by the National Disease Registration Service (NDRS), their data has been permanently removed from the registry and therefore will not be disseminated under this Data Sharing Agreement (DSA). https://digital.nhs.uk/ndrs/patients/opting-out

No data will flow to NHS England for the purposes of this Agreement.

NHS England will provide the relevant records from the NDRS Cancer Registry, NDRS Cancer Wait times (CWT), NDRS Hospital Episode Statistics Admitted Patient Care (HES APC), NDRS Hospital Episode Statistics Accident & Emergency (HES A&E), NDRS Hospital Episode Statistics Outpatients (HES OP), NDRS Diagnostic Imaging Dataset (DIDs), NDRS Systemic Anti Cancer Dataset (SACT), NDRS Radiotherapy Dataset (RTDS) to University of Bristol. The data will contain no direct identifying data items but will contain a unique person ID which can be used to link the data with other record level data already held by the recipient previously disseminated. Data will not be linked to other data out of scope of this agreement.

The data will not be transferred to any other location.

University of Bristol will extract the disseminated pseudonymised data extracts using a secure electronic file transfer system (SEFT) and make this available to the study team based at University of Bristol.

The Data will be processed and stored within the University of Bristol’s secure Research Data Storage Facility. The University of Bristol’s Research Data Storage Facility (RDSF) is only accessible to members of the University of Bristol who have been added to the project's unique RDSF folder as users and have entered their user specific password.

The University of Bristol's Advanced Computing Research Centre (ACRC) is responsible for providing the University's advanced computing systems. Data that is stored in the University's Research Data Storage Facility is backed up to tape libraries housed in the ACRC.

The data will be accessed onsite at the premises of the University of Bristol only. The data will be accessed by authorised personnel via remote access conducted via secure VPN. The data will remain on the servers at the University of Bristol at all times. Personnel are prohibited from/ and not technically capable of downloading or copying data to local devices.

The data will not leave England.

Access is restricted to individuals within Bristol Medical School at the University of Bristol who have authorisation from the Principal Investigators. All such individuals are substantive employees of University of Bristol and postgraduate students enrolled at the University of Bristol who may be processing the data under supervision. One substantive employee of the University of Bristol will change roles during the term of the agreement and at such time will transfer to an honorary contract with the University of Bristol to undertake the research. Postgraduate students and substantive employees (and any individual working under honorary contract) at the University of Bristol undergo mandatory training in Information Governance and Data Protection.

There will be no requirement and no attempt to reidentify individuals when using the data, and no data will be transferred to third parties.

Analysts/researchers from Bristol Medical School at the University of Bristol will process/analyse the data for the purposes described above.

The processing of data includes interrupted time series analysis and the use of Cox proportional hazards models to investigate the impact of the Honey Rose case on time to diagnosis, the proportion of intracranial tumours diagnosed by hospital eye specialists, and the effect on patient outcomes (mortality/survival) whilst adjusting for age, sex, ethnicity, and geographical location.

The expected outputs of the processing will be:

• Submissions to peer reviewed journals aiming for at least 2-3 publications within high-value journals within 2 years.

• Presentations at Appropriate conferences. Such as the Association of Research in Vision and Ophthalmology conference in April 2023, Annual Congress of The Royal College of Ophthalmologists, Optometry Tomorrow or Society for Academic Primary Care.

The outputs will not contain NHS Digital data and will only contain aggregated information with small numbers suppressed as appropriate in line with the relevant disclosure rules for the dataset(s) from which the information was derived.

In 2022, the University of Bristol published the results of a study showing an immediate 5- to 6-fold increase in the number of referrals to Bristol Eye Hospital of patients suspected to have papilloedema after the Honey Rose case and these referrals have remained as high as this ever since. Most of these patients did not actually have papilloedema (>60-70%), suggesting that optometrists had a much lower threshold to refer patients to secondary care since the Honey Rose case. On the other hand, University Of Bristol found that more patients who did have papilloedema were picked up than before the Honey rose case but had to wait longer for their appointments.

The study’s intention is to use the data obtained from Public Health England’s ODR (now NHS England) to determine whether the Honey Rose case had a nationwide impact on the number of patients diagnosed with intracranial tumours by hospital eye specialists. This is because brain tumours are one of the most serious causes of papilloedema. Results so far are comparable to experiences in Bristol; in other words, more brain tumours are diagnosed by hospital eye specialists nationally than before the Honey Rose case, but these patients have had to wait longer for their appointments. The study also found that hospital eye specialists are diagnosing eye tumours more quickly (another type of intracranial tumour) and this is probably because optometrists are working more vigilantly since the Honey Rose case; however, there is some geographical variation in referral waiting times that reflect how primary eye care services are commissioned in different areas of England.

Given these temporal and geographical variations in referral waiting times across England for patients with intracranial tumours, University of Bristol now wish to investigate how this impacts patient outcome and survival. The study team plan to present preliminary findings at the Association of Research in Vision and Ophthalmology conference in April 2023 and to present analysis of patient outcomes in 2024. University Of Bristol also intend to publish these results in high impact peer-reviewed journals; given the time to prepare, submit, and revise manuscripts following peer review, the study team anticipate these publications will become available in 2023-2025.

University Of Bristol have also been successful in two grant applications to the National Institute for Health Research (NIHR) to fund the DIPP study: "Improving the Diagnostic accuracy of referrals for Papilloedema". The DIPP study team will further investigate geographical variations in referral practices and the management of patients with papilloedema across England by completing Freedom of Information requests to Integrated Health Boards about their service provision and surveying primary and secondary care health professionals about their practices. University Of Bristol intend to use the results from this data analysis project and the NIHR-funded research to develop some clinical guidelines and educational materials to improve referral practices, the quality of patient care, patient outcomes, and the experiences of patients navigating the healthcare system. For this purpose, the study team have created the DIPP study website and publicised the study through social media channels; media engagement; organisational newsletters; local, regional, and national networks established by the Centre for Academic Primary Care to draw attention to the project and disseminate our findings, guidelines and training resources. Furthermore, the study team shall host workshops with key stakeholder groups including optometrists, ophthalmologists, GPs, emergency care practitioners, neurologists, and patients. The study team shall publish and present DIPP guidelines at national meetings and conferences like the Annual Congress of The Royal College of Ophthalmologists, Optometry Tomorrow or Society for Academic Primary Care. The NIHR-funded research began in May 2022 and will continue until at least May 2024. Additionally, the University of Bristol plan to apply for a NIHR programme grant to continue this work.

High street optometrists currently provide an important primary care service for eye health. GPs often rely on high street optometrists to screen people for common eye conditions, like glaucoma and cataract, and to refer patients to them when they detect a problem.

Hospital eye specialists (ophthalmologists) also rely on the skill of high street optometrists to refer patients with serious eye problems to them and to reassure others who simply need glasses to improve their vision.

The study results to date suggest that optometrists are referring more people who they suspect to have papilloedema to secondary care than before the Honey Rose case. The impact is that more people with intracranial tumours are diagnosed by eye specialists than other health specialists following the Honey Rose case but they are having to wait longer for their appointments because so many more people are referred to secondary care overall who do or do not have anything wrong with them.

In future analysis of the data, the study team will determine the impact of increased waiting times for hospital appointments with eye specialists on patient outcomes and survival, but it is likely that people who wait longer for their diagnosis and treatment will have worse morbidity and mortality rates. The study team are conducting further research (as described above) to produce clinical guidelines and educational materials that will help community optometrists and GPs to diagnose people who do or do not have papilloedema so that patients with papilloedema are investigated and treated quickly and those who do not are reassured. The study team anticipate these guidelines will save the NHS money by avoiding the costs of missed or delayed diagnosis and false positive diagnostic cascades.

As a result, the use of the data could:

• help the system to better understand the health and care needs of populations.

• lead to the identification or improvement of health and care system design to improve health and care outcomes or experience.

• advance understanding of regional and national trends in health and social care needs.

• inform planning health services and programmes, for example to improve equity of access, experience and outcomes.

• inform decisions on how to effectively allocate and evaluate funding according to health needs.

• provide a mechanism for checking the quality of care. This could include identifying areas of good practice to learn from, or areas of poorer practice which need to be addressed.

• support knowledge creation or exploratory research (and the innovations and developments that might result from that exploratory work).

The results of this study will determine how the widely publicised Honey Rose court case have affected NHS service demands and patient outcomes for people in England diagnosed with intracranial tumours and they will highlight the strengths and weaknesses of different routes to diagnosis for people diagnosed with intracranial tumours. By understanding where these strengths and weaknesses lie, the study is hoped to help the design and planning of referral pathways and services to become more reliable, safe and ensure higher quality patient care and patient experience. It is also hoped the study can also target interventions and funding to identified geographical and service areas of need.

The study’s communication goals are: (i) to make eye care and other health professionals and GPs aware of the study and availability of DIPP guidelines and training; (ii) to inform patients and the public about what the study means for them; (iii) to make policy and commissioning audiences, e.g. NHS England, aware of the guidelines and training to improve the quality and cost-effectiveness of patient care; (iv) to gain support among funders and the academic community for further dissemination and implementation work.

Throughout the project, The University of Bristol will update the DIPP study website and publicise via key stakeholder groups, including the Royal Colleges (Ophthalmologists, GPs, Physicians, Emergency Medicine) College of Optometrists, third sector organisations like Specsavers opticians and patient groups like Brain Tumour Support with a view to disseminating the outputs nationally. The College of Optometrists also runs a very successful series of online webinars (1000+ delegates / session) and an annual conference, Optometry Tomorrow (800+ delegates).

As mentioned above, The University of Bristol will use media and social media channels; media engagement; organisational newsletters; local, regional, and national networks established by the Centre for Academic Primary Care to draw attention to the project and disseminate the findings, guidelines and training resources.

The University of Bristol will publish and present DIPP guidelines at national meetings and conferences like the Annual Congress of The Royal College of Ophthalmologists, Optometry Tomorrow or Society for Academic Primary Care. Additionally, then the study team will publish educational materials tailored to the following health professionals: optometrists, GPs, ophthalmologists, neurologists, emergency care practitioners, policymakers.

To date, the results of this data analysis project have supported two successful NIHR grant applications that aim to produce clinical guidelines and educational materials to help GPs and optometrists to make the diagnosis of papilloedema. The guidelines will also inform GPs, optometrists and their patients about what to expect when they refer/are referred to hospital and which secondary care services are the most appropriate destinations for these referrals. The continued analysis of the data from NHS England in this project will further inform these guidelines.

In addition, The University of Bristol have forged several new collaborations with other universities and representatives of major stakeholders to support our NIHR-funded research, including:

• University of Exeter (Sam Merriel)

• Queen Mary University London (Beth Stuart)

• College of Optometrists (Mike Bowen)

• Specsavers opticians (Frank Moore)

• Bristol North Somerset South Gloucestershire (BNSSG) integrated care board (Paul Roy)

• REACH emergency care hub (Jonathan Benger)

• NIHR Applied Research Centre-West (Teresa Redaniel)

Moreover, The University of Bristol have well-established patient, public, involvement (PPI) groups to represent patients and the public, who have been highly supportive of the ongoing work and engagement of our research team.