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Publications and Presentations

Presentations

NORD Rare Summit 2021 Presentation October 18-19

 

Shaunfield, S., Greene, G.J., Seebald, A., Chapman, R., Perschon, C., Peipert, J.D., Lai, J., Nowinski, C., Plymill, K., Guralnik, J., Carney, R., Neilan, E., Cella, D. (2021, October 18-19). Physical function clinical outcome assessments for rare disorders and sarcopenia: Scoping activities to plan core outcome sets with FDA [Conference presentation]. NORD Rare Summit 2021, Virtual. 

The 30th Annual ISOQOL Conference, 18-21 October 2023

Shaunfield S, Greene GJ, Peipert JD, Hurt CN, Perschon C, Jaeger E, Seebald A, Plymill K, Lai JS, Nowinski C, Guralnik J, Neilan E, Cella D. Stepwise Approach for Prioritizing Concepts for the Development of a Cross-Cutting Patient-Reported Outcome Measure of Physical Function. Poster presented at: The 30th Annual ISOQOL Conference; October 18-21, 2023: Calgary, Alberta, Canada. 

The 9th Annual PROMIS International Conference, 22-24 October 2023

Title: Developing a cross-cutting PROMIS physical function measure for sarcopenia and rare disorders drug registration trials

 

Authors (preliminary order):

Sara Shaunfield, PhD1

George J. Greene, PhD1

Courtney N. Hurt, MSW1

Devin Peipert, PhD1

Chelsea Perschon, BA1

Emilie Jaeger, MPH1

Allison Seebald2

Kayce Plymill, MSc1

Jin-Shei Lai, PhD1

Cindy Nowinski, MD, PhD1

Jack Guralnik, MD, PhD3

Edward Neilan, MD, PhD2

David Cella, PhD1

 

Institutional Affiliations:

1Northwestern University Feinberg School of Medicine

2National Organization for Rare Disorders

3University of Maryland School of Medicine

 

To be submitted to: The 9th Annual PROMIS International Conference, 22-24 October 2023, in Banff, Canada

Due: April 11, 2023

Title word count: 15/15 words

Abstract word count: 350/350 (excluding title, authors, affiliations)

Submission category: Using PROMs for population health initiatives

 

Objective: As part of a US Food and Drug Administration (FDA)-sponsored effort, we sought to develop a cross-cutting PROMIS® Physical Function (PF) PRO measure for older adults with sarcopenia and a subset of rare disorders. 

 

Methods: Concept elicitation interviews with individuals with systemic sclerosis (SSc), myositis, idiopathic pulmonary fibrosis (IPF), and older adults with muscle weakness were conducted to identify cross-cutting PF limitations (i.e., concepts) and quality of life (QOL) importance (rated on a 0-10 scale; 10=Extremely important). Transcripts and fieldnotes were analyzed to identify PF domains, concepts, and QOL importance for each concept and condition. Findings were entered into a concept mapping file containing results for each condition, including the number that reported each concept and QOL importance (Mean, Range). Next, concepts were prioritized via stepwise approach to identify core cross-cutting (reported by all conditions) and specialized cross-cutting (reported by a subset) concepts which were mapped to existing items from the PROMIS PF Item Bank. Finally, the item pool was refined and reduced with input from measure development experts, clinicians, patient and caregiver stakeholders, and FDA representatives. 

 

Results: Analysis of concept elicitation interviews with individuals diagnosed with SSc (n=10), IPF (n=10), myositis (n=11), and older adults with muscle weakness (n=24) resulted in the initial identification 62 PF concepts, of which 14 core and 17 specialized cross-cutting concepts were identified using the stepwise prioritization approach. Concept mapping yielded an initial item pool of 110 PROMIS PF items. This pool was reduced to 28 candidate items based on expert input; two items were added to enhance content coverage resulting from stakeholder input. The resultant draft 30-item PROMIS PF measure reflects cross-cutting limitations representing four PF domains: mobility/physical activity (n=13 items), upper extremity/dexterity (n=6 items), instrumental and activities of daily living (n=9 items), central/axial function (n=2 items).

 

Conclusions: This study demonstrates that PF is a “common denominator” impact across diseases and highlights the utility of the PROMIS PF Item Bank for the development of cross-cutting PRO measures to support clinical trials and research. Cognitive interviews are underway to further reduce items prior to psychometric evaluation in a longitudinal validation study.

NUCOAT NIH Toolbox Assessment Conference Presentation April 8-9

 

Kuharic M, Hurt C, Peipert JD, Shaunfield S, Greene GJ, Perschon C, Tang X, Jaeger E, Andrade M, Guzman SN, Plymill K, Lai JS, Nowinski C, Guralnik J, Neilan E, Seebald A, Cella D. Developing and Validating NIH Toolbox- and PROMIS-Based Clinical Outcome Assessments for Sarcopenia and Rare Diseases: Preliminary Results from the NUCOAT Project. Poster presented at: National Institutes of Health Natcher Conference Center; April 89, 2024; Bethesda, MD.