Childhood Neurological Conditions PSP question verification form

The purpose of this Question Verification Form is to enable Priority Setting Partnerships (PSPs) to describe clearly how they checked that their questions were unanswered, before starting the interim prioritisation stage of the process.

The JLA requires PSPs to be transparent and accountable in defining their own scope and evidence checking process. This will enable researchers and other stakeholders to understand how individual PSPs decided that their questions were unanswered, and any limitations of their evidence checking.

Name of the PSP

The Top Ten UK research priorities for interventions in childhood neurological disorders: A British Paediatric Neurology Association and JLA Priority Setting Partnership.

Please describe the scope of the PSP

The PSP was limited to interventions for CYP age 0-25 years with Neurological disorders, rather than wider issues such as pathophysiology or epidemiology in order to focus on clinically based practical research priorities, meaningful to all stakeholders. Interventions were defined as medical and non-medical treatments, therapies, medical and surgical procedures. Conditions (e.g. autistic spectrum disorders) where there have been other recent other PSP were excluded.

Please provide a brief overview of your approach to checking whether the questions were unanswered

First Coding

Each question received was coded into a ‘theme’ based on the type of neurological disorder (e.g.: no specific neurological disorder; Motor disorders, epilepsies etc.), Then, each question was coded by two expert raters (members of the Information team) into one of three codes: Not a question (e.g., “please prioritize research”), Out of scope (e.g., “what can be done to ensure every young person is given fair treatment?”), or Keep for now (e.g., “What is the most effective treatment for migraine particularly in younger children?”).

Second Coding

Each question received was coded into ‘themes’ based on the topic of the question: a) To group with similar question, b) Too broad to formulate specific research question, c) Keep for Now, d) Out of Scope (not intervention, or not a neurological disorder,) e) To Discuss, f) Answered question, g) Move to other group of conditions h) Other ( included duplicate questions)

Coding of the questions were discussed and clarified between a pair of raters, disagreements were resolved through discussion, if not resolved the disagreement was brought to the wider team and resolved through further discussion. This also served as a means of quality control and rigour check. The original uncertainties were continuously referred to, to ensure we were staying true to the data.

A transparent audit trail in Excel accounted for the systematic examination at each level of analysis.

After the coding, draft research questions were composed from the uncertainties before the literature searches were completed.

Checking Uncertainties

The literature searches were completed by the information team as per the JLA guidance on levels of evidence.

Questions were deemed unanswered if there were no published systematic reviews, a recent, relevant and reliable review indicated an equivalent answer; or finally an out of date review indicated an equivocal answer.

Please list the type(s) of evidence you used to verify your questions as unanswered

The veracity of whether research questions were uncertain were checked as follows:

1. SR databases

2. Clinical trial registration

If 1 and 2 NEGATIVE

3. Pub med

Search of MEDLINE for systematic reviews pertaining to the research question. The paediatric age group will be defined by the MeSH terms “Neonatal”, “Newborn”, “Infant“, Child” and “Adolescent”. MeSH terms, where available, were used to identify the disease conditions of interest, whereby review articles published (Jan 2016 till current) will be reviewed to identify systematic reviews or RCTs published but not registered in publically available sites.

Please list the sources that you searched in order to identify that evidence

As above:

SR Databases
Clinical Trial Registration
PubMed:
Searches expanded as per information team knowledge of evidence base for specific condition, potential interventions and previous clinical trials etc.

What search terms did you use?

All searches commenced with hierarchy of:
Nervous system diseases.
Then specific mesh term according to type of childhood neurological condition and intervention suggested in the research question:

Examples include:

("Seizures/drug therapy"[Mesh]) AND "Infant, Newborn"[Mesh] Filters: Meta-Analysis, Randomized Controlled Trial, Systematic Review, Newborn: birth-1 month

("Migraine Disorders"[Mesh]) AND ( "Migraine Disorders/diet therapy"[Mesh] OR "Migraine Disorders/drug therapy"[Mesh] OR "Migraine Disorders/prevention and control"[Mesh] OR "Migraine Disorders/therapy"[Mesh] ) Filters: Meta-Analysis, Systematic Review, Child: birth-18 years, Young Adult: 19-24 years

("Cerebral Palsy"[Mesh]) AND ( "Cerebral Palsy/rehabilitation"[Mesh] OR "Cerebral Palsy/therapy"[Mesh] ) Filters: Meta-Analysis, Systematic Review, in the last 10 years, Child: birth-18 years, Young Adult: 19-24 years)

Please describe the parameters of the search (eg time limits, excluded sources, country/language) and the rationale for any limitations

Time limit: Scope of original search limited to January 2016 i.e. 5 years.

For the full age range – we used Child: birth-18years and Young adult: 19-24years; for particular questions the appropriate age range was used (as per example above for neonatal seizures)

Additional check to limit searches to 10 years – to ensure no key papers where there was recent significant paucity of evidence but previous known notable papers or literature,

Names of individuals who undertook the evidence checking

The information team – comprised of members of the steering committee and Expert Advisory Committee

Dr Jill Cadwgan
Dr Jane Goodwin
Professor Ming Lim
Dr Daniel Lumsden
Dr Sam Amin
Barbara Babcock
Professor JHC Cross

On what date was the question verification process completed?

Initial question verification completed by October 31st 2021 in preparation for prioritisation survey.

Any other relevant information

In view of delay in submission of JLA Engagement Documents and final manuscript all literature searches were reviewed and repeated by Dr Jill Cadwgan between May 2023-October 2023