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A qualitative exploration of patient perspectives on psychosocial burdens and positive factors in adults with osteogenesis imperfecta.
Rork, W Conor; Hertz, Alyssa G; Wiese, Andrew D; Kostick, Kristin M; Nguyen, Dianne; Schneider, Sophie C; Shepherd, Whitney S; Cho, Hannah; Murali, Chaya N; Lee, Brendan; Sutton, V Reid; Storch, Eric A.
Afiliação
  • Rork WC; Vagelos College of Physicians and Surgeons, Columbia University, New York, New York, USA.
  • Hertz AG; Department of Psychiatry and Behavioral Sciences, Baylor College of Medicine, Houston, Texas, USA.
  • Wiese AD; Department of Psychiatry and Behavioral Sciences, Baylor College of Medicine, Houston, Texas, USA.
  • Kostick KM; Center for Medical Ethics & Health Policy, Baylor College of Medicine, Houston, Texas, USA.
  • Nguyen D; Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas, USA.
  • Schneider SC; Department of Psychiatry and Behavioral Sciences, Baylor College of Medicine, Houston, Texas, USA.
  • Shepherd WS; Department of Psychiatry and Behavioral Sciences, Baylor College of Medicine, Houston, Texas, USA.
  • Cho H; Department of Psychiatry and Behavioral Sciences, Baylor College of Medicine, Houston, Texas, USA.
  • Murali CN; Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas, USA.
  • Lee B; Texas Children's Hospital, Houston, Texas, USA.
  • Sutton VR; Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas, USA.
  • Storch EA; Texas Children's Hospital, Houston, Texas, USA.
Am J Med Genet A ; 191(9): 2267-2275, 2023 09.
Article em En | MEDLINE | ID: mdl-37317786
Osteogenesis imperfecta (OI) is a pleiotropic, heritable connective tissue disorder associated with a wide range of health implications, including frequent bone fracture. While progress has been made to understand the spectrum of these physical health implications, the impact of OI on psychosocial well-being, as well as protective factors that buffer against adverse psychosocial outcomes, remain understudied. This present study relies on a qualitative approach to assess patient perspectives on both protective and adverse psychosocial factors specific to OI in 15 adults with varying disease status. Semi-structured interviews were conducted, subsequently coded, and themes extracted. Themes concerning psychosocial burdens (i.e., negative affective and behavioral impacts of disease status) and protective factors were identified from cooperatively-coded transcripts (two coders per transcript). Participants reported experiencing an increase in negative affect and disease-related distress after fracturing a bone and during recovery. Fear and concern specific to the uncertainty of future bone fractures and negative self-image was common. In contrast to these negative impacts, participants additionally described positive orientations toward their disease and attributed positive traits to their lived experience with a chronic disease. While limited due to small sample size and lack of ethno-racial diversity, findings highlight a need for continued research on the relationship between OI disease status and psychosocial outcomes, as well as the development of psychological interventions designed for OI populations. Findings have relevant clinical applications for healthcare providers working with those diagnosed with OI.
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Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Osteogênese Imperfeita / Fraturas Ósseas Tipo de estudo: Prognostic_studies / Qualitative_research Limite: Adult / Humans Idioma: En Revista: Am J Med Genet A Assunto da revista: GENETICA MEDICA Ano de publicação: 2023 Tipo de documento: Article País de afiliação: Estados Unidos

Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Osteogênese Imperfeita / Fraturas Ósseas Tipo de estudo: Prognostic_studies / Qualitative_research Limite: Adult / Humans Idioma: En Revista: Am J Med Genet A Assunto da revista: GENETICA MEDICA Ano de publicação: 2023 Tipo de documento: Article País de afiliação: Estados Unidos