Literature DB >> 30591002

Couples coping with screening burden and diagnostic uncertainty in Li-Fraumeni syndrome: Connection versus independence.

Jennifer L Young1,2, Ashley Pantaleao2, Lori Zaspel3, Jessica Bayer1, June A Peters1, Payal P Khincha1, Renee C Bremer1, Jennifer T Loud1, Mark H Greene1, Maria Isabel Achatz1, Sharon A Savage1, Allison Werner-Lin1,3.   

Abstract

PURPOSE: Li-Fraumeni Syndrome (LFS) is an inherited tumor predisposition syndrome with lifetime cancer risks approaching 100% and evolving risk-management strategies. This study evaluated couples' coping with LFS-related burdens. RESEARCH APPROACH: Constructivist grounded theory and anticipatory loss frameworks guided design and analysis. SAMPLE AND METHODS: Twenty-six individuals enrolled in the NCI LFS Family Study completed semi-structured interviews with their partner during annual screening visits. An interdisciplinary team completed open and focused coding to identify patterns of coping and adaptation.
FINDINGS: Couples described living with ambiguous danger, a state of chronic apprehension resulting from LFS-associated uncertainties. Most couples communicated openly and alternated shouldering the burden, while others engaged in protective buffering to shield each other from distress and sustain the appearance of normalcy.
INTERPRETATION: Optimally, coping reduces shared psychosocial distress, yet some strategies may inadvertently increase disconnection. IMPLICATIONS: Mental health support is critical for both partners coping with LFS, together and separately.

Entities:  

Keywords:  Li-Fraumeni syndrome; coping; couples; family; hereditary cancer; qualitative

Mesh:

Year:  2018        PMID: 30591002      PMCID: PMC6584025          DOI: 10.1080/07347332.2018.1543376

Source DB:  PubMed          Journal:  J Psychosoc Oncol        ISSN: 0734-7332


  8 in total

1.  Family Health Leaders: Lessons on Living with Li-Fraumeni Syndrome across Generations.

Authors:  Ashley Pantaleao; Jennifer L Young; Norman B Epstein; Mae Carlson; Renée C Bremer; Payal P Khincha; June A Peters; Mark H Greene; Kevin Roy; Maria Isabel Achatz; Sharon A Savage; Allison Werner-Lin
Journal:  Fam Process       Date:  2019-10-24

Review 2.  [Update on Li-Fraumeni syndrome].

Authors:  C M Dutzmann; J Vogel; C P Kratz; K W Pajtler; S M Pfister; B B Dörgeloh
Journal:  Pathologe       Date:  2019-11       Impact factor: 1.011

3.  Cancer incidence, patterns, and genotype-phenotype associations in individuals with pathogenic or likely pathogenic germline TP53 variants: an observational cohort study.

Authors:  Kelvin César de Andrade; Payal P Khincha; Jessica N Hatton; Megan N Frone; Talia Wegman-Ostrosky; Phuong L Mai; Ana F Best; Sharon A Savage
Journal:  Lancet Oncol       Date:  2021-11-12       Impact factor: 41.316

4.  Family Identity and Roles in the Context of Li-Fraumeni Syndrome: "No One's Like Us Mutants".

Authors:  Catherine Wilsnack; Jennifer L Young; Shana L Merrill; Victoria Groner; Jennifer T Loud; Renee C Bremer; Mark H Greene; Payal P Khincha; Allison Werner-Lin
Journal:  Health Soc Work       Date:  2021-11-16

5.  Family communication challenges of adolescents and young adults with Li-Fraumeni syndrome: Implications for psychosocial care.

Authors:  Camella J Rising; Catherine Wilsnack; Patrick Boyd; Alix G Sleight; Sadie P Hutson; Payal P Khincha; Allison Werner-Lin
Journal:  Patient Educ Couns       Date:  2022-07-20

6.  Disclosure of genetic risk to dating partners among young adults with von Hippel-Lindau disease.

Authors:  Elysa Bond; Beverly Yashar; Tobias Else; Jenae Osborne; Monica Marvin
Journal:  Fam Cancer       Date:  2022-08-19       Impact factor: 2.446

7.  Specifications of the ACMG/AMP variant interpretation guidelines for germline TP53 variants.

Authors:  Cristina Fortuno; Kristy Lee; Magali Olivier; Tina Pesaran; Phuong L Mai; Kelvin C de Andrade; Laura D Attardi; Stephanie Crowley; D Gareth Evans; Bing-Jian Feng; Ann K M Foreman; Megan N Frone; Robert Huether; Paul A James; Kelly McGoldrick; Jessica Mester; Bryce A Seifert; Thomas P Slavin; Leora Witkowski; Liying Zhang; Sharon E Plon; Amanda B Spurdle; Sharon A Savage
Journal:  Hum Mutat       Date:  2020-12-25       Impact factor: 4.700

8.  Utility of interim blood tests for cancer screening in Li-Fraumeni syndrome.

Authors:  Leatrisse Oba; Ana F Best; Phuong L Mai; Maria Isabel Achatz; Paul S Albert; Sharon A Savage; Payal P Khincha
Journal:  Fam Cancer       Date:  2021-06-02       Impact factor: 2.446

  8 in total

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