caa a22 4500 605544948 CHVBK 20210128100933.0 cr unu---uuuuu 210128e20151001xx s 000 0 eng 10.1007/s10897-014-9807-3 doi (NATIONALLICENCE)springer-10.1007/s10897-014-9807-3 Genetic Information-Seeking Behaviors and Knowledge among Family Members and Patients with Inherited Bone Marrow Failure Syndromes [Elektronische Daten] [Jada Hamilton, Sadie Hutson, Amy Frohnmayer, Paul Han, June Peters, Ann Carr, Blanche Alter] Inherited bone marrow failure syndromes (IBMFS) including Fanconi anemia, dyskeratosis congenita, Diamond-Blackfan anemia, and Shwachman-Diamond syndrome are rare genetic disorders characterized by hematologic complications and increased risk of cancer. Patients and their families likely experience obstacles in obtaining sufficient health information given their disorders' rarity. To investigate this possibility, we examined information-seeking behaviors and levels of general and disorder-specific genetic knowledge among 315 members of 174 families with an IBMFS, and how information-seeking behaviors and socio-demographic factors may be associated with their genetic knowledge. Cross-sectional survey data indicated that participants were most likely to have ever used the Internet or healthcare providers for genetic information. On average, participants correctly answered 57% of items assessing general genetic knowledge and 49-59% of disorder-specific knowledge items. Greater knowledge was associated with greater education and ever experiencing genetic counseling, attending a scientific meeting, and seeking information from the Internet and scientific literature. Among families with Fanconi anemia (whose family support organization has the longest history of providing information), greater disorder-specific genetic knowledge was also associated with seeking information from support groups and other affected families. Results suggest that families with IBMFS have uncertainty regarding genetic aspects of their disorder, and highlight potential channels for delivering educational resources. Springer Science+Business Media New York (Outside the USA), 2014 Genetic counseling nationallicence Information seeking nationallicence Genetic literacy nationallicence Inherited bone marrow failure syndromes nationallicence Fanconi anemia nationallicence Dyskeratosis congenita nationallicence Diamond-Blackfan anemia nationallicence Shwachman-Diamond syndrome nationallicence Hamilton Jada Department of Psychiatry & Behavioral Sciences, Memorial Sloan Kettering Cancer Center, 641 Lexington Avenue, 7th floor, 10022, New York, NY, USA aut Hutson Sadie College of Nursing, University of Tennessee-Knoxville, Knoxville, TN, USA aut Frohnmayer Amy Clinical Mental Health Counseling Program, Oregon State University - Cascades, Bend, OR, USA aut Han Paul Center for Outcomes Research and Evaluation, Maine Medical Center Research Institute, Scarborough, ME, USA aut Peters June Clinical Genetics Branch, Division of Cancer Epidemiology and Genetics, National Cancer Institute, Rockville, MD, USA aut Carr Ann Westat, Rockville, MD, USA aut Alter Blanche Clinical Genetics Branch, Division of Cancer Epidemiology and Genetics, National Cancer Institute, Rockville, MD, USA aut Journal of Genetic Counseling Springer US; http://www.springer-ny.com 24/5(2015-10-01), 760-770 1059-7700 24:5<760 2015 24 10897 https://doi.org/10.1007/s10897-014-9807-3 text/html Onlinezugriff via DOI BK010053 XK010053 XK010000 Metadata rights reserved Springer special CC-BY-NC licence nationallicence 1 research-article jats NATIONALLICENCE NATIONALLICENCE NL-springer NATIONALLICENCE 856 40 https://doi.org/10.1007/s10897-014-9807-3 text/html Onlinezugriff via DOI NATIONALLICENCE 700 1- Hamilton Jada Department of Psychiatry & Behavioral Sciences, Memorial Sloan Kettering Cancer Center, 641 Lexington Avenue, 7th floor, 10022, New York, NY, USA aut NATIONALLICENCE 700 1- Hutson Sadie College of Nursing, University of Tennessee-Knoxville, Knoxville, TN, USA aut NATIONALLICENCE 700 1- Frohnmayer Amy Clinical Mental Health Counseling Program, Oregon State University - Cascades, Bend, OR, USA aut NATIONALLICENCE 700 1- Han Paul Center for Outcomes Research and Evaluation, Maine Medical Center Research Institute, Scarborough, ME, USA aut NATIONALLICENCE 700 1- Peters June Clinical Genetics Branch, Division of Cancer Epidemiology and Genetics, National Cancer Institute, Rockville, MD, USA aut NATIONALLICENCE 700 1- Carr Ann Westat, Rockville, MD, USA aut NATIONALLICENCE 700 1- Alter Blanche Clinical Genetics Branch, Division of Cancer Epidemiology and Genetics, National Cancer Institute, Rockville, MD, USA aut NATIONALLICENCE 773 0- Journal of Genetic Counseling Springer US; http://www.springer-ny.com 24/5(2015-10-01), 760-770 1059-7700 24:5<760 2015 24 10897