Advertisement

A survey of healthcare professionals’ perceptions of the decisional needs of parents with an infant born with a disorder/difference of sex development

Published:September 27, 2022DOI:https://doi.org/10.1016/j.jpurol.2022.09.015

      Summary

      Introduction

      Parents of infants born with differences in sex development (DSD) face many difficult decisions. As part of a larger project designed to develop educational interventions to promote shared decision making, this study assessed healthcare professionals’ (HCPs) perceptions of parental decision-making needs when an infant is born with a DSD.

      Methods

      A cross-sectional web-based survey following the Ottawa Decision Support Framework was conducted in two waves, between October 2020 and June 2022. Survey domains included: common DSD decisions, indicators of parents’ decisional needs, and resources and approaches to support parental decision making. Eligible participants were HCPs working within interprofessional pediatric DSD centers in the USA. Up to three reminders were sent. Descriptive analysis was conducted.

      Results

      71 HCPs participated; most (>90%) reported parents experience signs of decisional conflict including feeling unsure, worrying about what could go wrong, and fear of choosing a “wrong,” irreversible option. The majority (90%) reported parents experience strong emotions interfering with their receptivity to information or deliberation. The majority (>70%) identified inadequate parental knowledge of the DSD as a barrier to decision making, coupled with information overload (>90%). HCPs rated several factors as “very” important, including: parents having information on benefits, harms, and other features of options (93%), having information about all the available options (87%), and having access to providers to discuss the options (84%). Providers endorsed using a variety of approaches to support parents’ decision making; however, access to decision aids was not universally rated as highly important (very, 44%; somewhat, 46%; a little, 10%).

      Implications

      Overall, HCPs expressed favorable attitudes toward supporting active parental participation in medical decision making. Opportunities for enhanced support of shared decision making included: a) recognizing and addressing parental emotional distress and informational overload at a time when parents need to consider complex options for their infant or young child; and b) the need for HCPs to encourage values clarification in decision-making encounters with parents.

      Keywords

      Abbreviations:

      DSD (Disorders/differences of sex development), HCP (Healthcare professionals), SDM (Shared decision making)
      To read this article in full you will need to make a payment

      Purchase one-time access:

      Academic & Personal: 24 hour online accessCorporate R&D Professionals: 24 hour online access
      One-time access price info
      • For academic or personal research use, select 'Academic and Personal'
      • For corporate R&D use, select 'Corporate R&D Professionals'

      Subscribe:

      Subscribe to Journal of Pediatric Urology
      Already a print subscriber? Claim online access
      Already an online subscriber? Sign in
      Institutional Access: Sign in to ScienceDirect

      References

        • Lee P.A.
        • Houk C.P.
        • Ahmed S.F.
        • Hughes I.A.
        International consensus conference on intersex organized by the lawson wilkins pediatric endocrine society, and the European society for paediatric endocrinology. Consensus statement on management of intersex disorders. International consensus conference on intersex.
        Pediatrics. 2006; 118: e488-e500https://doi.org/10.1542/peds.2006-0738
        • Human Rights Watch
        I want to be like nature made me.
        in: Medically unnecessary surgeries on Intersex children in the US. 2017 (accessed 02 July 2021)
        • Crissman H.P.
        • Warner L.
        • Gardner M.
        • Carr M.
        • Schast A.
        • Quittner A.L.
        • et al.
        Children with disorders of sex development: a qualitative study of early parental experience.
        Int J Pediatr Endocrinol. 2011; 2011: 10https://doi.org/10.1186/1687-9856-2011-10
        • Ellens R.E.H.
        • Bakula D.M.
        • Mullins A.J.
        • Scott Reyes K.J.
        • Austin P.
        • Baskin L.
        • et al.
        Psychological adjustment of parents of children born with atypical genitalia 1 year after genitoplasty.
        J Urol. 2017; 198: 914-920https://doi.org/10.1016/j.juro.2017.05.035
        • Siminoff L.A.
        • Sandberg D.E.
        Promoting shared decision making in disorders of sex development (DSD): decision aids and support tools.
        Horm Metab Res. 2015; 47: 335-339https://doi.org/10.1055/s-0035-1545302
        • Suorsa-Johnson K.
        • Gardner M.
        • Baskin A.
        • Gruppen L.
        • Rose A.
        • Rutter M.
        • et al.
        Defining successful outcomes and preferences for clinical management in differences/disorders of sex development: Protocol overview and a qualitative phenomenological study of stakeholders’ perspectives.
        J Pediatr Urol. 2022; 18: 36.e1-36.e17https://doi.org/10.1016/j.jpurol.2021.10.020
        • Timmermans S.
        • Yang A.
        • Gardner M.
        • Keegan C.E.
        • Yashar B.M.
        • Fechner P.Y.
        • et al.
        Does patient-centered care change genital surgery decisions? The strategic use of clinical uncertainty in disorders of sex development clinics.
        J Health Soc Behav. 2018; 59: 520-535https://doi.org/10.1177/0022146518802460
        • Streuli J.C.
        • Vayena E.
        • Cavicchia-Balmer Y.
        • Huber J.
        Shaping parents: impact of contrasting professional counseling on parents' decision making for children with disorders of sex development.
        J Sex Med. 2013; 10: 1953-1960https://doi.org/10.1111/jsm.12214
        • American Medical Association
        Association A.M. CEJA report 3-I-18. Report 3 on the council of Ethical and Judicial Affairs (1-I-19): Amendment to E-2.2.1, “Pediatric Decision Making” (resolution 3-A-16, “Supporting Autonomy for Patients with Differences of Sex Development [DSD]”) (resolution 13-A-18, “Opposing Surgical Sex Assignment of Infants with Differences of Sex Development”). CEJA Report 3-I-18. 2019 (Opinion 221): American Medical Association, Council on Ethical and Judicial Affairs) (accessed 02 July 2021)
        • Légaré F.
        • Witteman H.O.
        Shared decision making: examining key elements and barriers to adoption into routine clinical practice.
        Health Aff. 2013; 32: 276-284https://doi.org/10.1377/hlthaff.2012.1078
        • Weston W.W.
        Informed and shared decision-making: the crux of patient-centred care.
        Can Med Assoc J. 2001; 165: 438-439
        • Légaré F.
        • Adekpedjou R.
        • Stacey D.
        • Turcotte S.
        • Kryworuchko J.
        • Graham I.D.
        • et al.
        Interventions for increasing the use of shared decision making by healthcare professionals.
        Cochrane Database Syst Rev. 2018; 7: CD006732https://doi.org/10.1002/14651858.CD006732.pub4
        • Joseph-Williams N.
        • Abhyankar P.
        • Boland L.
        • Bravo P.
        • Brenner A.T.
        • Brodney S.
        • et al.
        What works in implementing patient decision aids in routine clinical settings? A rapid realist review and update from the International Patient Decision Aid Standards Collaboration.
        Med Decis Making. 2021; 41: 907-937https://doi.org/10.1177/0272989X20978208
        • Hoefel L.
        • O'Connor A.M.
        • Lewis K.B.
        • Boland L.
        • Sikora L.
        • Hu J.
        • et al.
        20th Anniversary Update of the Ottawa Decision Support Framework Part 1: a systematic review of the decisional needs of people making health or social decisions.
        Med Decis Making. 2020; 40: 555-581https://doi.org/10.1177/0272989X20936209
        • Jackson C.
        • Cheater F.M.
        • Reid I.
        A systematic review of decision support needs of parents making child health decisions.
        Health Expect. 2008; 11: 232-251https://doi.org/10.1111/j.1369-7625.2008.00496.x
        • Dayner J.E.
        • Lee P.A.
        • Houk C.P.
        Medical treatment of intersex: parental perspectives.
        J Urol. 2004; 172: 1762-1765https://doi.org/10.1097/01.ju.0000138519.12573.3a
        • Shippee N.D.
        • Domecq Garces J.P.
        • Prutsky Lopez G.J.
        • Wang Z.
        • Elraiyah T.A.
        • Nabhan M.
        • et al.
        Patient and service user engagement in research: a systematic review and synthesized framework.
        Health Expect. 2015; 18: 1151-1166https://doi.org/10.1111/hex.12090
        • O'Connor A.M.
        • Tugwell P.
        • Wells G.A.
        • Elmslie T.
        • Jolly E.
        • Hollingworth G.
        • et al.
        A decision aid for women considering hormone therapy after menopause: decision support framework and evaluation.
        Patient Educ Counsel. 1998; 33: 267-279https://doi.org/10.1016/s0738-3991(98)00026-3
        • Stacey D.
        • Legare F.
        • Boland L.
        • Lewis K.B.
        • Loiselle M.C.
        • Hoefel L.
        • et al.
        20th Anniversary Ottawa Decision Support Framework: Part 3 overview of systematic reviews and updated framework.
        Med Decis Making. 2020; 40: 379-398https://doi.org/10.1177/0272989X20911870
        • DSD - Translational Research Network
        (accessed 02 July 2021)
        • Jacobsen M.J.
        • O'Connor A.M.
        • Stacey D.
        Decisional needs assessment in populations: A workbook for assessing patients' and practitioners' decision making needs.
        2013 (accessed 02 July 2021)
        • Kogan B.
        • Gardner M.
        • Suorsa-Johnson K.
        • Lee P.
        • Mauger D.
        • Sandberg D.
        PD02-10 Changes in the approach of SPU members to DSD management.
        J Urol. 2022; 207: e39https://doi.org/10.1097/JU.0000000000002517.10
        • Gardner M.
        • Kogan B.A.
        • Suorsa-Johnson K.I.
        • Lee P.A.
        • Khorashad B.S.
        • Sandberg D.E.
        Changes in Perceptions of Pediatric Endocrinologists (PE) and Urologists (PU) to Infant sand Children regarding care of individuals with 46,XY DSD.
        2022 (Poster presented at ENDO 2022. Atlanta, GA)
        • Witteman H.O.
        • Ndjaboue R.
        • Vaisson G.
        • Chipenda Dansokho S.
        • Arnold B.
        • Bridges J.F.P.
        • et al.
        Clarifying values: an updated and expanded systematic review and meta-analysis.
        medRxiv. 2021; https://doi.org/10.1101/2021.01.21.21250270
        • Danon L.M.
        • Krämer A.
        Between concealing and revealing intersexed bodies: parental strategies.
        Qual Health Res. 2017; 27: 1562-1574https://doi.org/10.1177/1049732317697100
        • Stacey D.
        • Suwalska V.
        • Boland L.
        • Lewis K.B.
        • Presseau J.
        • Thomson R.
        Are patient decision aids used in clinical practice after rigorous evaluation? A survey of trial authors.
        Med Decis Making. 2019; 39: 805-815https://doi.org/10.1177/0272989X19868193
        • Perez M.N.
        • Clawson A.H.
        • Baudino M.N.
        • Austin P.F.
        • Baskin L.S.
        • Chan Y.M.
        • et al.
        Distress trajectories for parents of children with DSD: a growth mixture model.
        J Pediatr Psychol. 2021; 46: 588-598https://doi.org/10.1093/jpepsy/jsab004
        • Delozier A.M.
        • Gamwell K.L.
        • Sharkey C.
        • Bakula D.M.
        • Perez M.N.
        • Wolfe-Christensen C.
        • et al.
        Uncertainty and posttraumatic stress: differences between mothers and fathers of infants with disorders of sex development.
        Arch Sex Behav. 2019; 48: 1617-1624https://doi.org/10.1007/s10508-018-1357-6
        • Pasterski V.
        • Mastroyannopoulou K.
        • Wright D.
        • Zucker K.J.
        • Hughes I.A.
        Predictors of posttraumatic stress in parents of children diagnosed with a disorder of sex development.
        Arch Sex Behav. 2014; 43: 369-375https://doi.org/10.1007/s10508-013-0196-8

      Linked Article