Evidence map›Paper›PMID 34230208›Full record

ArticleNeurology2021

Enhancing Neurofibromatosis Clinical Trial Outcome Measures Through Patient Engagement: Lessons From REiNS.

Vanessa L Merker, Andrés J Lessing, Irene Moss, Maureen Hussey, Beverly Oberlander, Traceann Rose, Raquel Thalheimer, Tracy Wirtanen, Pamela L Wolters, Andrea M Gross and 2 more

Open access · bronzeAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Neurology, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.

0numbers the graph read from it
0cells of the map it votes in
6citing papers in PubMed
1.7field-weighted citation impact, top 14% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

6 citing papers in PubMed, 6 citations in OpenAlex.

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4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

12 authors at 1 institution in 1 country.

Vanessa L MerkerFrom the Center for Healthcare Organization and Implementation Research (CHOIR) (V.L.M.), Edith Nourse Rogers Memorial Veterans Hospital, Bedford; Department of Neurology and Cancer Center (V.L.M., R.T., S.R.P.), Massachusetts General Hospital, Boston; Neurofibromatosis Northeast (A.J.L.), Burlington, MA; Children's Tumor Foundation (I.M., M.H., T.R.), New York, NY; Neurofibromatosis Inc. California (B.O.), Vacaville; Neurofibromatosis Network (B.O.), Wheaton, IL; Littlest Tumor Foundation (T.W.), Appleton, WI; and Pediatric Oncology Branch (P.L.W., A.M.G.), National Cancer Institute, National Institutes of Health, Bethesda, MD. vmerker@mgh.harvard.edu.ORCID 0000-0002-4542-5227
Andrés J LessingFrom the Center for Healthcare Organization and Implementation Research (CHOIR) (V.L.M.), Edith Nourse Rogers Memorial Veterans Hospital, Bedford; Department of Neurology and Cancer Center (V.L.M., R.T., S.R.P.), Massachusetts General Hospital, Boston; Neurofibromatosis Northeast (A.J.L.), Burlington, MA; Children's Tumor Foundation (I.M., M.H., T.R.), New York, NY; Neurofibromatosis Inc. California (B.O.), Vacaville; Neurofibromatosis Network (B.O.), Wheaton, IL; Littlest Tumor Foundation (T.W.), Appleton, WI; and Pediatric Oncology Branch (P.L.W., A.M.G.), National Cancer Institute, National Institutes of Health, Bethesda, MD.ORCID 0000-0001-8250-0145
Irene MossFrom the Center for Healthcare Organization and Implementation Research (CHOIR) (V.L.M.), Edith Nourse Rogers Memorial Veterans Hospital, Bedford; Department of Neurology and Cancer Center (V.L.M., R.T., S.R.P.), Massachusetts General Hospital, Boston; Neurofibromatosis Northeast (A.J.L.), Burlington, MA; Children's Tumor Foundation (I.M., M.H., T.R.), New York, NY; Neurofibromatosis Inc. California (B.O.), Vacaville; Neurofibromatosis Network (B.O.), Wheaton, IL; Littlest Tumor Foundation (T.W.), Appleton, WI; and Pediatric Oncology Branch (P.L.W., A.M.G.), National Cancer Institute, National Institutes of Health, Bethesda, MD.
Maureen HusseyFrom the Center for Healthcare Organization and Implementation Research (CHOIR) (V.L.M.), Edith Nourse Rogers Memorial Veterans Hospital, Bedford; Department of Neurology and Cancer Center (V.L.M., R.T., S.R.P.), Massachusetts General Hospital, Boston; Neurofibromatosis Northeast (A.J.L.), Burlington, MA; Children's Tumor Foundation (I.M., M.H., T.R.), New York, NY; Neurofibromatosis Inc. California (B.O.), Vacaville; Neurofibromatosis Network (B.O.), Wheaton, IL; Littlest Tumor Foundation (T.W.), Appleton, WI; and Pediatric Oncology Branch (P.L.W., A.M.G.), National Cancer Institute, National Institutes of Health, Bethesda, MD.
Beverly OberlanderFrom the Center for Healthcare Organization and Implementation Research (CHOIR) (V.L.M.), Edith Nourse Rogers Memorial Veterans Hospital, Bedford; Department of Neurology and Cancer Center (V.L.M., R.T., S.R.P.), Massachusetts General Hospital, Boston; Neurofibromatosis Northeast (A.J.L.), Burlington, MA; Children's Tumor Foundation (I.M., M.H., T.R.), New York, NY; Neurofibromatosis Inc. California (B.O.), Vacaville; Neurofibromatosis Network (B.O.), Wheaton, IL; Littlest Tumor Foundation (T.W.), Appleton, WI; and Pediatric Oncology Branch (P.L.W., A.M.G.), National Cancer Institute, National Institutes of Health, Bethesda, MD.
Traceann RoseFrom the Center for Healthcare Organization and Implementation Research (CHOIR) (V.L.M.), Edith Nourse Rogers Memorial Veterans Hospital, Bedford; Department of Neurology and Cancer Center (V.L.M., R.T., S.R.P.), Massachusetts General Hospital, Boston; Neurofibromatosis Northeast (A.J.L.), Burlington, MA; Children's Tumor Foundation (I.M., M.H., T.R.), New York, NY; Neurofibromatosis Inc. California (B.O.), Vacaville; Neurofibromatosis Network (B.O.), Wheaton, IL; Littlest Tumor Foundation (T.W.), Appleton, WI; and Pediatric Oncology Branch (P.L.W., A.M.G.), National Cancer Institute, National Institutes of Health, Bethesda, MD.
Raquel ThalheimerFrom the Center for Healthcare Organization and Implementation Research (CHOIR) (V.L.M.), Edith Nourse Rogers Memorial Veterans Hospital, Bedford; Department of Neurology and Cancer Center (V.L.M., R.T., S.R.P.), Massachusetts General Hospital, Boston; Neurofibromatosis Northeast (A.J.L.), Burlington, MA; Children's Tumor Foundation (I.M., M.H., T.R.), New York, NY; Neurofibromatosis Inc. California (B.O.), Vacaville; Neurofibromatosis Network (B.O.), Wheaton, IL; Littlest Tumor Foundation (T.W.), Appleton, WI; and Pediatric Oncology Branch (P.L.W., A.M.G.), National Cancer Institute, National Institutes of Health, Bethesda, MD.
Tracy WirtanenFrom the Center for Healthcare Organization and Implementation Research (CHOIR) (V.L.M.), Edith Nourse Rogers Memorial Veterans Hospital, Bedford; Department of Neurology and Cancer Center (V.L.M., R.T., S.R.P.), Massachusetts General Hospital, Boston; Neurofibromatosis Northeast (A.J.L.), Burlington, MA; Children's Tumor Foundation (I.M., M.H., T.R.), New York, NY; Neurofibromatosis Inc. California (B.O.), Vacaville; Neurofibromatosis Network (B.O.), Wheaton, IL; Littlest Tumor Foundation (T.W.), Appleton, WI; and Pediatric Oncology Branch (P.L.W., A.M.G.), National Cancer Institute, National Institutes of Health, Bethesda, MD.
Pamela L WoltersFrom the Center for Healthcare Organization and Implementation Research (CHOIR) (V.L.M.), Edith Nourse Rogers Memorial Veterans Hospital, Bedford; Department of Neurology and Cancer Center (V.L.M., R.T., S.R.P.), Massachusetts General Hospital, Boston; Neurofibromatosis Northeast (A.J.L.), Burlington, MA; Children's Tumor Foundation (I.M., M.H., T.R.), New York, NY; Neurofibromatosis Inc. California (B.O.), Vacaville; Neurofibromatosis Network (B.O.), Wheaton, IL; Littlest Tumor Foundation (T.W.), Appleton, WI; and Pediatric Oncology Branch (P.L.W., A.M.G.), National Cancer Institute, National Institutes of Health, Bethesda, MD.
Andrea M GrossFrom the Center for Healthcare Organization and Implementation Research (CHOIR) (V.L.M.), Edith Nourse Rogers Memorial Veterans Hospital, Bedford; Department of Neurology and Cancer Center (V.L.M., R.T., S.R.P.), Massachusetts General Hospital, Boston; Neurofibromatosis Northeast (A.J.L.), Burlington, MA; Children's Tumor Foundation (I.M., M.H., T.R.), New York, NY; Neurofibromatosis Inc. California (B.O.), Vacaville; Neurofibromatosis Network (B.O.), Wheaton, IL; Littlest Tumor Foundation (T.W.), Appleton, WI; and Pediatric Oncology Branch (P.L.W., A.M.G.), National Cancer Institute, National Institutes of Health, Bethesda, MD.
Scott R PlotkinFrom the Center for Healthcare Organization and Implementation Research (CHOIR) (V.L.M.), Edith Nourse Rogers Memorial Veterans Hospital, Bedford; Department of Neurology and Cancer Center (V.L.M., R.T., S.R.P.), Massachusetts General Hospital, Boston; Neurofibromatosis Northeast (A.J.L.), Burlington, MA; Children's Tumor Foundation (I.M., M.H., T.R.), New York, NY; Neurofibromatosis Inc. California (B.O.), Vacaville; Neurofibromatosis Network (B.O.), Wheaton, IL; Littlest Tumor Foundation (T.W.), Appleton, WI; and Pediatric Oncology Branch (P.L.W., A.M.G.), National Cancer Institute, National Institutes of Health, Bethesda, MD.ORCID 0000-0002-6109-6419
REiNS International Collaboration
National Institutes of Health · US

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

objectiveAs part of an evaluation of the Response Evaluation in Neurofibromatosis and Schwannomatosis (REiNS) International Collaboration patient representative program, we surveyed REiNS members to (1) identify facilitators and barriers to involving patient representatives and (2) understand whether and how involving patient representatives affected recommendations for clinical trial outcomes.

methodsWe administered an anonymous online survey to all REiNS members. Facilitators and barriers to patient representative involvement were solicited using a modified free listing technique; responses were inductively grouped into higher-order categories and ranked based on saliency score (Smith

resultsA total of 63/172 (37%) members responded, including 18/30 (60%) patient representatives. Providing sufficient opportunities to meaningfully engage in research tasks and cultivating a respectful, inclusive atmosphere were key facilitators to patient representatives' satisfaction and ability to make an impact. Respondents perceived that patient representatives directly (through their input on research tasks) and indirectly (through effects on other stakeholders' knowledge and communication style) improved the organization's research, leading to selection of more meaningful, relevant, and feasible clinical trial outcome measures. Ongoing challenges to patient engagement include difficulty scheduling meetings and concerns about the level of scientific knowledge patient representatives needed to effectively engage.

conclusionsInvolving patient representatives in REiNS improved perceived quality of neurofibromatosis clinical trial outcome measures. Negotiating sufficient opportunities to engage, fostering an inclusive atmosphere, and navigating time pressures are key to effective patient engagement.

Indexed as

Clinical Trials as TopicCommunicationHumansNeurilemmomaNeurofibromatosesNeurofibromatosis 1Outcome Assessment, Health CarePatient ParticipationSkin Neoplasms

Identifiers

PMID34230208
PMCPMC8594004
OpenAlexW3179744631

What OpenQuestion holds

Textmetadata
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.