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. 2023:2:1020152.
doi: 10.3389/fruro.2022.1020152. Epub 2023 Jan 4.

Co-creating a suite of patient decision aids for parents of an infant or young child with differences of sex development: A methods roadmap

Sophie Lightfoot  1 , Meg Carley  2 , William Brinkman  3   4 , Melissa D Gardner  5 , Larry D Gruppen  6 , Noi Liang  7 , Kendra Pinkelman  7 , Phyllis W Speiser  8   9 , Kristina I Suorsa-Johnson  10 , Brian VanderBrink  11 , Jessica Wisniewski  7 , David E Sandberg  5   12 , Dawn Stacey  1   2
Affiliations

Co-creating a suite of patient decision aids for parents of an infant or young child with differences of sex development: A methods roadmap

Sophie Lightfoot et al. Front Urol. 2023.

Abstract

Introduction: Parents and guardians of infants and young children with differences of sex development (DSD) often face numerous health and social decisions about their child's condition. While proxy health decisions can be stressful in any circumstance, they are further exacerbated in this clinical context by significant variations in clinical presentation, parental lack of knowledge about DSD, irreversibility of some options (e.g., gonadectomy), a paucity of research available about long-term outcomes, and anticipated decisional regret. This study aimed to engage clinicians, parents, and an adult living with DSD to collaboratively develop a suite of patient decision aids (PDAs) to respond to the decisional needs of parents and guardians of infants and young children diagnosed with DSD.

Methods: We used a systematic co-development process guided by the Ottawa Decision Support Framework and the International Patient Decision Aids Standards (IPDAS). The five steps were: literature selection, establish the team, decisional needs assessment, create the PDAs, and alpha testing.

Results: We developed four PDAs to support parents/guardians of infants or young children diagnosed with DSD about four priority decisions identified through our decisional needs assessment: genetic testing, gender of rearing, genital surgery and gonadal surgery. All four PDAs include information for parents about DSD, the options, reasons to choose or avoid each option, and opportunities for parents/guardians to rate the importance of features of each option to clarify their values for these features. Qualitative feedback was positive from clinicians, parents and an adult living with DSD.

Conclusions: These PDAs are clinical tools designed to support parents/guardians and to promote making an informed and shared DSD-related decision. While these tools are specific to DSD, they contain themes and elements translatable to other pediatric populations.

Keywords: decision-making; decisional conflict; decisional needs assessment; differences of sex development; disorders of sex development; intersex; patient decision aids; shared decision-making.

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Figures

Figure 1.
Figure 1.
The PDA development process
Figure 2.
Figure 2.
Decisions identified during the decisional needs assessment.
Figure 3.
Figure 3.
Values clarification exercise in the PDA about genital surgery
See this image and copyright information in PMC

References

    1. Lee PA, Nordenström A, Houk CP, Ahmed SF, Auchus R, Baratz A, et al. Global disorders of sex development update since 2006: perceptions, approach and care. Horm Res Paediatr 2016; 85: 158–180. 2016/January/29. DOI: 10.1159/000442975. - DOI - PubMed
    1. Hughes IA, Nihoul-Fékété C, Thomas B and Cohen-Kettenis PT. Consequences of the ESPE/LWPES guidelines for diagnosis and treatment of disorders of sex development. Best Pract Res Clin Endocrinol Metab 2007; 21: 351–365. DOI: 10.1016/j.beem.2007.06.003. - DOI - PubMed
    1. Money J, Hampson JG and Hampson JL. Hermaphroditism: recommendations concerning assignment of sex, change of sex and psychologic management. Bull Johns Hopkins Hosp 1955; 97: 284–300. 1955/October/01. - PubMed
    1. Human Rights Watch. “I want to be like nature made me.” medically unnecessary surgeries on intersex children in the US. Washington, DC: 2017. https://www.hrw.org/sites/default/files/report_pdf/lgbtintersex0717_web_...
    1. Vavilov S, Smith G, Starkey M, Pockney P and Deshpande AV. Parental decision regret in childhood hypospadias surgery: a systematic review. J Paediatr Child Health 2020; 56: 1514–1520. 2020/September/05. DOI: 10.1111/jpc.15075. - DOI - PubMed

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