Decision Support for Parents Receiving Information About Child's Rare Disease (DSD DST)
The birth of a child with a disorder of sex development (DSD) is stressful for parents and members of the healthcare team. The "right" decisions about gender assignment (is it a boy? a girl?) and the best course of action (e.g., should there be surgery? what kind? when?) are not obvious. While there have been large advances in diagnostic assessments like genetic and endocrine testing, the tests do not always show what caused the DSD. And, even when the tests do reveal an explanation for the DSD, knowing what happened genetically or hormonally does not usually lead to a single "correct" treatment plan. Instead, it is likely that there are different acceptable treatment options - and parents will need to make decisions based, in part, on their personal preferences, values, and cultural background. Adding more stress to the situation is knowledge that many of the decisions that need to be made by parents early in a child's life are irreversible and exert life-long consequences for the child and the family.
To support parents becoming actively involved in making such decisions, and to reduce the likelihood of future worry and regret about decisions that have been made, the investigators will create a decision support tool (DST). The DST will help educate families about typical and atypical sex development of the body, the process by which DSD are diagnosed (especially how to interpret genetic test results), and possible relationships between diagnostic/genetic testing, decisions about care, and known consequences of those decisions on their child and entire family. The DST will be used by parents of young children together with their child's health care provider.
The investigators will bring together a network of researchers, health care providers, representatives of patient support and advocacy organizations, and parents of children with DSD to share their experiences. Participants of this network will be involved at each stage of creating the DST, revising it, and putting it into practice. At the end of this project, the investigators will have a fully formed and tested DST that will be available for parents to use with their child's health care team as they are first learning their child may have a DSD.
Disorder of Sex Development
Congenital Adrenal Hyperplasia
Behavioral: Decision Support Tool
|Study Design:||Primary Purpose: Health Services Research|
|Official Title:||Decision Support for Parents Receiving Genetic Information About Child's Rare Disease|
- Qualitative Assessment of Focus Groups, Usual Care and DST Implementation [ Time Frame: 2.5 years ] [ Designated as safety issue: No ]To assess characteristics of communications during focus groups and between health care providers and parents before and during the implementation of the DST audiofiles will be coded and analyzed using SCCAP (Siminoff Communication Content & Affect Program). SCCAP© is a new computerized coding scheme used to facilitate, organize, and code health communication data. It is designed so that coders can code directly from audio recordings, allowing the coder to take advantage of the vocalic cues inherent in speech intonations. Transcriptions can also be uploaded and coded so specific text can be viewed and tagged for future use.
|Study Start Date:||June 2013|
|Estimated Study Completion Date:||March 2017|
|Estimated Primary Completion Date:||July 2016 (Final data collection date for primary outcome measure)|
No Intervention: Usual Care
After obtaining consent, we will audio-record standard clinical consultations with specialists represented on the DSD team. These appointments will not utilize the Decision Support Tool. A qualitative analysis of these recordings will assess quality assurance and provide guidance for the development of the Decision Support Tool.
Experimental: Use of Decision Support Tool
After obtaining consent, we will audio-record standard clinical consultations with specialists represented on the DSD team. These appointments will utilize the Decision Support Tool (DST). A qualitative analysis of these recordings will assess the practicality of use and possible benefits of the DST's implementation.
|Behavioral: Decision Support Tool|
No Intervention: Focus Groups
Focus group members, recruited from advocacy organizations and by medical chart review, will provide feedback on the content and appearance of a preliminary Decision Support Tool (DST). Their comments will guide the development and implementation of the DST.
|Contact: David E Sandberg, PhDfirstname.lastname@example.org|
|United States, California|
|University of California Los Angeles||Not yet recruiting|
|Los Angeles, California, United States, 90095|
|Contact: Eric Vilain, PhD, MD email@example.com|
|Principal Investigator: Eric Vilain, Phd, MD|
|United States, Michigan|
|University of Michigan||Not yet recruiting|
|Ann Arbor, Michigan, United States, 48104|
|Principal Investigator: David Sandberg, PhD|
|United States, Virginia|
|Virginia Commonwealth University||Not yet recruiting|
|Richmond, Virginia, United States, 23284|
|Contact: Laura Siminoff, PhD firstname.lastname@example.org|
|Principal Investigator: Laura Siminoff, PhD|
|United States, Washington|
|Seattle Children's Hospital||Not yet recruiting|
|Seattle, Washington, United States, 98105|
|Contact: Margarett Shnorhavorian, MD, MPH email@example.com|
|Principal Investigator: Margarett Shnorhavorian, MD, MPH|
|Principal Investigator: Patricia Fechner, MD|