Evidence map›Paper›PMID 32222840›Full record

ArticleFamilial cancer2020

Waiting and "weighted down": the challenge of anticipatory loss for individuals and families with Li-Fraumeni Syndrome.

Allison Werner-Lin, Jennifer L Young, Catherine Wilsnack, Shana L Merrill, Victoria Groner, Mark H Greene, Payal P Khincha

Open access · greenAbstract read
In one paragraph

Article in Familial cancer, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 14 papers.

0numbers the graph read from it
0cells of the map it votes in
14citing papers in PubMed
5.1field-weighted citation impact, top 4% 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

14 citing papers in PubMed, 27 citations in OpenAlex.

  1. Article
  2. Article
  3. Article
  4. Observational
  5. Article
  6. Article
  7. Article
  8. Article
  9. Young-onset colorectal cancer.Nature reviews. Disease primers · 2023
    Review
  10. Article
  11. Article
  12. Review
  13. Article
  14. 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

7 authors at 3 institutions in 1 country.

Allison Werner-LinSchool of Social Policy and Practice, University of Pennsylvania, 3701 Locust Walk, Philadelphia, PA, 19104, USA. awer@upenn.edu.ORCID 0000-0003-3133-820X
Jennifer L YoungStanford Center for Biomedical Ethics, Stanford, CA, USA.
Catherine WilsnackClinical Genetics Branch, Division of Cancer Epidemiology and Genetics, National Cancer Institute, Bethesda, MD, USA.
Shana L MerrillSchool of Social Policy and Practice, University of Pennsylvania, 3701 Locust Walk, Philadelphia, PA, 19104, USA.
Victoria GronerCenter for Genetic Medicine, Feinberg School of Medicine, Chicago, IL, USA.
Mark H GreeneClinical Genetics Branch, Division of Cancer Epidemiology and Genetics, National Cancer Institute, Bethesda, MD, USA.
Payal P KhinchaClinical Genetics Branch, Division of Cancer Epidemiology and Genetics, National Cancer Institute, Bethesda, MD, USA.
National Cancer Institute · USHospital of the University of Pennsylvania · USUniversity of Pennsylvania · US

Funding

Clinical Genetic Studies of Familial and Hereditary Cancer SyndromesZIACP010144 · NCI · DIVISION OF CANCER EPIDEMIOLOGY AND GENETICS · PI SAVAGE, SHARON A. · 2009 to 2025
$111.8M
The Stanford Training Program in ELSI ResearchT32HG008953 · NHGRI · STANFORD UNIVERSITY · PI Mildred K. Cho, Meghan Halley · 2016 to 2026
$3.1M
NCI NIH HHS Protocol 11-C-0255NHGRI NIH HHS T32 HG008953
6 · The paper itself

Abstract

Li-Fraumeni Syndrome (LFS) is characterized by risk of multiple primary malignancies in diverse sites, pediatric onset, near complete penetrance by age 70 years, limited options for prevention, and substantial uncertainty regarding disease manifestation and prognosis. Forty-five families, including 117 individuals aged 13-81 years, enrolled in the US National Cancer Institute's Li-Fraumeni Syndrome Study completed 66 interviews regarding their LFS experiences. An interdisciplinary team used modified grounded theory to examine family distress regarding expectations of loss and change due to likely cancer diagnoses, and the consequences of this likelihood across physical, social, and emotional domains. Disease-free periods were characterized by fearful anticipation of diagnosis or recurrence, uncertainty regarding post-treatment quality of life, and planning for shifts in family dynamics to enable caregiving. The chronicity of waiting for these changes incited dread and inhibited effective coping with the pragmatic, emotional, and existential challenges of the syndrome. Consequently, families reported high burden on roles and resources and limited guidance to prepare for, or achieve resolution with, grief. Anticipatory loss, the experience of bereavement prior to an expected change, distinguishes hereditary cancer risk from a sporadic diagnosis. Such grief is often incomplete in impact or meaning, subjected to rapid or profound change as conditions worsen, and poorly understood. In this study, losses were compounded by profound uncertainty, a chronic feature of LFS, which compromised mourning. Long-term engagement of mental health providers with bereavement training, in partnership with genetics providers, can provide invaluable educational and psychological support to families as they navigate these implacable challenges.

Indexed as

BereavementFamilyUncertaintyAdolescentAdultAgedAged, 80 and overAnticipation, PsychologicalChildChild, PreschoolData AnalysisEarly Detection of CancerFemaleGenes, p53Genetic CounselingGriefAnticipatory lossBereavementFamilyFamily therapyHereditary cancerLi-fraumeni syndrome

Identifiers

PMID32222840
PMCPMC7440840
OpenAlexW3013140165

What OpenQuestion holds

Textmetadata
LicenceTDM
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.