Adapting the James Lind Alliance (JLA) methodology to suit local context: Why, how and what we learned

June 2017 Leah Lechelt, Director, Patient Partnerships Bernie Krewski, Patient Advisor

Overview • Intro to Alberta Cancer Foundation • JLA overview • Why modify? • Adaptations • What we learned

Alberta Cancer Foundation: Funder of $20-$25M annually in cancer research • “Our investments must have a direct line of sight to the patient and a clear and direct impact on patient outcomes.” • “Engagement of stakeholders and involvement of patients and families is a key consideration in our investment decisions.”

What are your top priorities for new research?

•Clinical/academic research •Industry •Clinicians

Patients •

“Mismatch” in priorities: patients vs research agenda

Talon, Chard & Dieppe, 2000

JLA Methodology Priority Setting Partnership (PSP) • Dr. Ian Chalmers – Cochrane Collaboration • Hundreds conducted worldwide • Mixed methods • Patients, caregivers & clinicians • Consensus on “Top 10” treatment uncertainties

Overview of JLA approach

•Raw uncertainties •Sort, PICO & verify •Add from literature •Initial ranking •Final workshop

“Please tell us, in your own words, the questions you have about the treatment or management of head and neck cancer. These should be questions that you think could be answered by new or better research.”

Unique Challenges

Desired outcomes

Geography

• Reduced project cost

Costs and resources

• < 1 year start to finish

Tight project timelines

• Manage recruitment risks • More inclusive data analysis

Patient participation barriers

• Maintain rigor/integrity & publish

Time-pressed FFS clinicians Depth of data interpretation

• Affiliation with JLA

Adaptation: “In association with JLA” • Modifications negotiated & agreed to in advance • Non-JLA lead – qualifications assessed by JLA • JLA Advisor as steering committee member & coach • “Spirit and intent” and “rigor” checks throughout

Time/intensity/format adaptations • From 18 to 9 mos • Meeting time cut by 80% • Patient-only meetings • From 4 to 1 admin staff • Videoconference + teleconference • Lit review out of sequence and split in 2

Time/intensity/format outcomes • “The pace worked very well.” • “We were all flying by the seat of our pants.” • “Needed more face-to-face meetings.” • “There is too much on the agenda for each meeting.” • Literature search in two stages (less efficient)

10 months to reach “Top 10 uncertainties”

818 72 •Raw uncertainties •Sort, PICO & verify •Add from literature •Initial ranking •Final workshop

77 27 Top 10

Recruitment concerns • UK study discontinued • Difficult to reach & mobilize patients • ~ 3,000 AB survivors

Sampling strategy adaptations

Results

• Snowball + convenience sampling

• REB & admin approval delayed project two months • 161 respondents • 73% patients/families • > 800 suggestions

• + Stratified random sample of 900 HNC patients from ACR (HREB approval rec’d) • 18 mos – 26 years since diagnosis

Extracted from narratives Extracted uncertainties 1.

“If you are young, female and don’t smoke it can be missed.”

2.

“The first doctor asked me if I smoke. I replied not for 15 years. So he told me I had swollen glands. Two months later I was in Stage 4.”

3.

“Why do some dentists and doctors just give the patient antibiotics, assuming that the patient has an abscess and not something more serious?”

Indicative question: How knowledgeable are community providers (such as family physicians, dentists, speech language pathologists) about the early signs and symptoms of head and neck cancer, and would provider education result in more routine screening or more thorough assessment of presenting signs/symptoms?

Data processing results • “Work took twice the suggested time because of unfamiliarity…” • “It is a huge learning curve.”

Bernie Krewski: Patient as participant in research Major themes: • Presence – just being there • Make participation attractive • The Process: life-long learning • Data Analysis – challenges & rewards

Presence • Imagine being in a customarily private meeting, discussing and debating a subject bound to bring forth disagreement and strong arguments from all sides. Ten minutes into the deliberations, a surprise to everyone, one of the attendees happens to be a news reporter. Think how that can immediately change the expression, tone and language in the room. • Patients like me bring three words to research-focused meetings: “we have cancer.” Little more needs to be said.

Make participation attractive • Obtaining insufficient information about their condition is a dominant theme and complaint from patients, well documented in the clinical literature. • “Nudge” theorists (Thaler & Sunstein, 2008) argue that attractive, simple & timely invitations to participate can be highly effective. • My participation in this project served as an “advanced cancer practicum” (non-credit?).

The Process • Cancer patients often feel alienated & lost. Since the need to learn was immediate & high during this project, I became a student once again. I thus had a new identity. I belonged. The project had such energizing momentum. • I could listen & absorb. I could receive coaching – like the PICO training in how to formulate research questions. I could raise the inevitable “why’s” that students ask, and not be embarrassed in doing so.

Data analysis • Reading 800 plus questions from survey respondents offered much insight. It provided a context for my 13 years of thinking about cancer. It was like flying over the head-and-neck cancer community in Alberta. • Spending several hours (one-to-one) doing data analysis with a research director and a clinician, skilled by his international experience, was so enriching. WE helped to finalize the short list of research questions. THEY provided me with another “crash course” in synthesizing information.

Core project team

Questions? [email protected] 780.643.7199

Bernie Krewski

Acknowledgements