Defining successful outcomes and preferences for clinical management in differences/disorders of sex development: Protocol overview and a qualitative phenomenological study of stakeholders' perspectives

Published:October 29, 2021DOI:



      Utilizing a qualitative phenomenological design, the Defining Successful Outcomes and Trade-offs study examined stakeholder perspectives regarding optimal healthcare delivery and outcomes for individuals with a difference/disorder of sex development (DSD).


      We describe study methods and provide an overview of themes and subthemes.

      Study design

      Interviews were conducted with individuals with a DSD (n = 24), parents of those with a DSD (n = 19), healthcare providers (n = 37), and others (n = 30). Primary questions regarding clinical management of patients with DSD were: “What is a successful outcome?” and “How do you achieve it?”


      Themes included: understanding of DSD diagnosis and self-efficacy in management is necessary but complex; patient and family psychological well-being; support from others versus being stigmatized; affected person experiences physical health and accepts the implications of their condition; complexities in DSD decision making, roles and expectations; and knowledgeable providers and multidisciplinary teams are essential, notwithstanding persisting barriers. Participants recognized competing values potentially forcing trade-offs in decision making.


      Recognition of diverse and sometimes conflicting perspectives regarding optimal pathways of care and outcomes – both within and among those with DSD and their providers –promises to enhance shared decision making.


      Summary Figure
      Graphical AbstractIdentified Themes. Note. DSD = difference/disorder of sex development.


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        • Institute of Medicine
        • Committee on Quality Of Health Care in America
        Crossing the quality chasm: a new health system for the 21st century.
        The National Academies Press, Washington, DC2001
        • Creighton S.M.
        • Michala L.
        • Mushtaq I.
        • Yaron M.
        Childhood surgery for ambiguous genitalia: glimpses of practice changes or more of the same?.
        Psychology & Sexuality. 2013; 5: 34-43
        • Deans R.
        • Creighton S.M.
        • Liao L.M.
        • Conway G.S.
        Timing of gonadectomy in adult women with complete androgen insensitivity syndrome (CAIS): patient preferences and clinical evidence.
        Clin Endocrinol. 2012; 76: 894-898
        • Creighton S.M.
        • Wood D.
        Complex gynaecological and urological problems in adolescents: challenges and transition.
        Postgrad Med. 2012; 89: 34-38
        • Creighton S.
        • Chernausek S.D.
        • Romao R.
        • Ransley P.
        • Salle J.P.
        Timing and nature of reconstructive surgery for disorders of sex development - introduction.
        J Pediatr Urol. 2012; 8: 602-610
        • Rolston A.M.
        • Gardner M.
        • Vilain E.
        • Sandberg D.E.
        Parental reports of stigma associated with child's disorder of sex development.
        Int J Endocrinol. 2015; 2015: 980121
        • Crouch N.S.
        • Minto C.L.
        • Laio L.M.
        • Woodhouse C.R.
        • Creighton S.M.
        Genital sensation after feminizing genitoplasty for congenital adrenal hyperplasia: a pilot study.
        BJU Int. 2004; 93: 135-138
        • Minto C.L.
        • Liao L.-M.
        • Woodhouse C.R.J.
        • Ransley P.G.
        • Creighton S.M.
        The effect of clitoral surgery on sexual outcome in individuals who have intersex conditions with ambiguous genitalia: a cross-sectional study.
        Lancet. 2003; 361: 1252-1257
        • Diamond M.
        • Garland J.
        Evidence regarding cosmetic and medically unnecessary surgery on infants.
        J Pediatr Urol. 2014; 10: 2-6
        • Mouriquand P.
        • Caldamone A.
        • Malone P.
        • Frank J.D.
        • Hoebeke P.
        The ESPU/SPU standpoint on the surgical management of disorders of sex development (DSD).
        J Pediatr Urol. 2014; 10: 8-10
        • Mouriquand P.D.
        • Gorduza D.B.
        • Gay C.L.
        • Meyer-Bahlburg H.F.
        • Baker L.
        • Baskin L.S.
        • et al.
        Surgery in disorders of sex development (DSD) with a gender issue: if (why), when, and how?.
        J Pediatr Urol. 2016; 12: 139-149
        • Organization Intersex International
        OII intersex network.
        Date accessed: March 10, 2021
        • interACT
        Advocates for intersex youth.
        Date accessed: March 10, 2021
        • Human Rights Watch
        “I want to be like nature made me.” Medically unnecessary surgeries on Intersex children in the US.
        • Méndez J.E.
        • United Nations Human Rights Council
        Report of the Special Rapporteur on torture and other cruel, inhuman or degrading treatment or punishment.
        • Ghorayshi A.
        Born in between. Should doctors operate on intersex babies?.
        Buzzfeed News, 2015
        • Ghorayshi A.
        A landmark lawsuit about an intersex baby's genital surgery just settled for $440,000.
        BuzzFeed News, 2017
        • Louviere J.
        • Flynn T.
        • Marley A.
        Best-worst scaling: theory, methods and applications.
        Cambridge University Press, 2015
      1. Accord Alliance.; n.d. [accessed 10 March 2021].

        • Lee P.A.
        • Houk C.P.
        • Ahmed S.F.
        • Hughes I.A.
        The 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.
        Pediatrics. 2006; 118: e488-e500
        • Johnson T.P.
        Snowball sampling: introduction.
        Wiley StatsRef: Statistics Reference Online, 2014
        • Creswell J.W.
        • Poth C.N.
        Qualitative inquiry research design: choosing among five approaches.
        4th ed. Sage Publications, Thousand Oaks, CA2018
        • DeCuir-Gunby J.T.
        • Marshall P.L.
        • McCulloch A.W.
        Developing and using a codebook for the analysis of interview data: an example from a professional development research project.
        Field Methods. 2010; 23: 136-155
      2. Qualitative data analysis software. NVivo, 2020
        • Clayton P.E.
        • Miller W.L.
        • Oberfield S.E.
        • Ritzen E.M.
        • Sippell W.G.
        • Speiser P.W.
        • et al.
        Consensus statement on 21-hydroxylase deficiency from the European society for paediatric endocrinology and the lawson wilkins pediatric endocrine society.
        Horm Res. 2002; 58: 188-195
        • Lee P.A.
        • Nordenstrom A.
        • Houk C.P.
        • Ahmed S.F.
        • Auchus R.
        • Baratz A.
        • et al.
        • Global DSD Update Consortium
        Global disorders of sex development update since 2006: perceptions, approach and care.
        Horm Res Paediatr. 2016; 85: 158-180
        • Chivers C.
        • Burns J.
        • Deiros Collado M.
        Disorders of sex development: mothers' experiences of support.
        Clin Child Psychol Psychiatr. 2017; (0:1359104517719114)
        • 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: 10
        • Baratz A.B.
        • Sharp M.K.
        • Sandberg D.E.
        Disorders of sex development peer support.
        in: Hiort O. Ahmed S. Understanding differences and disorders of sex development (DSD) endocrine development. Karger, Basel2014: 99-112
        • Bennecke E.
        • Werner-Rosen K.
        • Thyen U.
        • Kleinemeier E.
        • Lux A.
        • Jurgensen M.
        • et al.
        Subjective need for psychological support (PsySupp) in parents of children and adolescents with disorders of sex development (dsd).
        Eur J Pediatr. 2015; 174: 1287-1297
        • Crerand C.E.
        • Kapa H.M.
        • Litteral J.L.
        • Nahata L.
        • Combs B.
        • Indyk J.A.
        • et al.
        Parent perceptions of psychosocial care for children with differences of sex development.
        J Pediatr Urol. 2019; 15: 522.e1-522.e8
        • Creighton S.
        • Minto C.
        Managing intersex. Most vaginal surgery in childhood should be deferred.
        Br Med J. 2001; 323: 1264-1265
        • Johnson E.K.
        • Rosoklija I.
        • Finlayson C.
        • Chen D.
        • Yerkes E.B.
        • Madonna M.B.
        • et al.
        Attitudes towards "disorders of sex development" nomenclature among affected individuals.
        J Pediatr Urol. 2017; 13: 608 e1-608 e8
        • Hemesath T.P.
        • de Paula L.C.P.
        • Carvalho C.G.
        • Leite J.C.L.
        • Guaragna-Filho G.
        • Costa E.C.
        Controversies on timing of sex assignment and surgery in individuals with disorders of sex development: a perspective.
        Front Pediatr. 2018; 6: 419
        • Diouf N.T.
        • Menear M.
        • Robitaille H.
        • Painchaud Guérard G.
        • Légaré F.
        Training health professionals in shared decision making: update of an international environmental scan.
        Patient Educ Counsel. 2016; 99: 1753-1758
        • Légaré F.
        • Politi M.C.
        • Drolet R.
        • Desroches S.
        • Stacey D.
        • Bekker H.
        Training health professionals in shared decision-making: an international environmental scan.
        Patient Educ Counsel. 2012; 88: 159-169
        • Boland L.
        • Lawson M.L.
        • Graham I.D.
        • Légaré F.
        • Dorrance K.
        • Shephard A.
        • et al.
        Post-training shared decision making barriers and facilitators for pediatric healthcare providers: a mixed-methods study.
        Academic Pediatrics. 2019; 19: 118-129
        • Stacey D.
        • Murray M.A.
        • Légaré F.
        • Sandy D.
        • Menard P.
        • O'Connor A.
        Decision coaching to support shared decision making: a framework, evidence, and implications for nursing practice, education, and policy.
        Worldviews Evidence-Based Nurs. 2008; 5: 25-35
        • Creswell J.W.
        Qualitative inquiry & research design.
        Sage Publications, Thousand Oaks, CA2007
        • Tong A.
        • Sainsbury P.
        • Craig J.
        Consolidated criteria for reporting qualitative research (COREQ): a 32-item checklist for interviews and focus groups.
        Int J Qual Health Care. 2007; 19: 349-357
        • Palinkas L.A.
        • Horwitz S.M.
        • Green C.A.
        • Wisdom J.P.
        • Duan N.
        • Hoagwood K.
        Purposeful sampling for qualitative data collection and analysis in mixed method implementation research.
        Adm Policy Ment Health. 2015; 42: 533-544
        • Phares V.
        • Lopez E.
        • Fields S.
        • Kamboukos D.
        • Duhig A.M.
        Are fathers involved in pediatric psychology research and treatment?.
        J Pediatr Psychol. 2005; 30: 631-643