Evidence map›Paper›PMID 39554689›Full record

ArticleSSM. Qualitative research in health2024

A "rotating menu" of medical uncertainty for families affected by telomere biology disorders: A qualitative interview study.

Emily Pearce, Alina Majid, Toniya Brown, Catherine Wilsnack, Camella Rising, Ashley S Thompson, Rowan Forbes Shepherd, Arman Niknafs, Allison Werner-Lin, Melissa B Gilkey and 4 more

Abstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in SSM. Qualitative research in health, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
–field-weighted citation impact
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

2 citing papers in PubMed.

  1. Article
  2. Article
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

14 authors.

Emily PearceClinical Genetics Branch, Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health, Bethesda, MD 20892, USA.
Alina MajidOffice of the Associate Director, Healthcare Delivery Research Program, National Cancer Institute, National Institutes of Health, Bethesda, MD 20892, USA.
Toniya BrownTrans-Divisional Research Program, National Cancer Institute, National Institutes of Health, Bethesda, MD 20892, USA.
Catherine WilsnackClinical Genetics Branch, Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health, Bethesda, MD 20892, USA.
Camella RisingClinical Genetics Branch, Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health, Bethesda, MD 20892, USA.
Ashley S ThompsonClinical Genetics Branch, Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health, Bethesda, MD 20892, USA.
Rowan Forbes ShepherdClinical Genetics Branch, Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health, Bethesda, MD 20892, USA.
Arman NiknafsClinical Genetics Branch, Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health, Bethesda, MD 20892, USA.
Allison Werner-LinSchool of Social Policy and Practice, University of Pennsylvania, Philadelphia, PA 19104, USA, Rockville, MD 20850.
Melissa B GilkeyDepartment of Health Behavior, Gillings School of Global Public Health, University of North Carolina at Chapel Hill, 135 Dauer Drive, 302 Rosenau Hall, Chapel Hill, NC 27599-7440, USA.
Kurt M RibislDepartment of Health Behavior, Gillings School of Global Public Health, University of North Carolina at Chapel Hill, 135 Dauer Drive, 302 Rosenau Hall, Chapel Hill, NC 27599-7440, USA.
Sadie P HutsonClinical Genetics Branch, Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health, Bethesda, MD 20892, USA.
Paul K J HanBehavioral Research Program, Division of Cancer Control and Population Sciences, National Cancer Institute, National Institutes of Health, USA.
Sharon A SavageClinical Genetics Branch, Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health, Bethesda, MD 20892, USA.

Funding

Intramural NIH HHS Z99 CA999999
6 · The paper itself

Abstract

Background: Medical uncertainty may cause distress and challenge medical decision-making for patients with rare diseases and their caregivers. Few studies have examined the experience and management of medical uncertainty in rare disease and the dynamics of multiple medical uncertainty sources, issues, and management strategies. Objective: We explored the experience and management of uncertainty in individuals with telomere biology disorders (TBDs), a set of rare cancer-prone bone marrow failure syndromes, and their caregivers. Design: Participants (N=32) in this qualitative-descriptive study were individuals with a TBD (n=17) and/or their caregivers (n=15). We thematically analyzed transcripts to describe the presence and dynamics of medical uncertainty in TBDs using categories from a previously published taxonomy. Results: Individuals with TBDs and caregivers described medical uncertainty as a chronic burden embodied amidst a range of interrelated sources and issues. Conclusion: In TBDs, medical uncertainty manifests as a network of multiple, interrelated, sources and issues, which require evolving management strategies. Researchers must be mindful that complex, synergistic uncertainty networks contribute to psychosocial challenges in TBDs. Additional research is warranted to address scientific uncertainty in TBDs, including clinical manifestations and underlying biology, and to develop psychosocial interventions that recognize and anticipate evolving uncertainty.

Indexed as

dyskeratosis congenitagenetic diseasemedical decision makingpsychosocialqualitativetelomeretelomere biology disorderuncertainty

Identifiers

PMID39554689
PMCPMC11563169

What OpenQuestion holds

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Registered trials

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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.