Advertisement

Exploring Factors Associated with Decisions about Feminizing Genitoplasty in Differences of Sex Development

Published:August 07, 2022DOI:https://doi.org/10.1016/j.jpag.2022.08.004

      ABSTRACT

      Study Objective

      Infants with genital development considered atypical for assigned female sex may undergo feminizing genitoplasty (clitoroplasty and/or vaginoplasty) in early life. We sought to identify factors associated with parent/caregiver decisions regarding genitoplasty for their children with genital virilization.

      Design

      Longitudinal, observational study

      Setting

      Twelve pediatric centers in the United States with multidisciplinary differences/disorders of sex development clinics, 2015-2020

      Participants

      Children under 2 years old with genital appearance atypical for female sex of rearing and their parents/caregivers

      Interventions/Outcome Measures

      Data on the child's diagnosis and anatomic characteristics before surgery were extracted from the medical record. Parents/caregivers completed questionnaires on psychosocial distress, experience of uncertainty, cosmetic appearance of their child's genitalia, and demographic characteristics. Urologists rated cosmetic appearance. For 58 patients from the study cohort with genital virilization being raised as girls or gender-neutral, we compared these data across 3 groups based on the child's subsequent surgical intervention: (i) no surgery (n = 5), (ii) vaginoplasty without clitoroplasty (V-only) (n = 15), and (iii) vaginoplasty and clitoroplasty (V+C) (n = 38).

      Results

      Fathers’ and urologists’ ratings of genital appearance were more favorable in the no-surgery group than in the V-only and V+C groups. Clitorophallic length was greater in the V+C group compared with the V-only group, with substantial overlap between groups. Mothers’ depressive and anxious symptoms were lower in the no-surgery group compared with the V-only and V+C groups.

      Conclusions

      Surgical decisions were associated with fathers’ and urologists’ ratings of genital appearance, the child's anatomic characteristics, and mothers’ depressive and anxious symptoms. Further research on surgical decision-making is needed to inform counseling practices.

      Key Words

      Abbreviations:

      DSDs (differences/disorders of sex development), V+C (vaginoplasty and clitoroplasty), V-only (vaginoplasty only)
      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 and Adolescent Gynecology
      Already a print subscriber? Claim online access
      Already an online subscriber? Sign in
      Institutional Access: Sign in to ScienceDirect

      References

        • Hughes IA
        • Houk C
        • Ahmed SF
        • Lee PA:
        Consensus statement on management of intersex disorders.
        J Pediatr Urol. 2006; 2: 148-162https://doi.org/10.1016/j.jpurol.2006.03.004
        • Mouriquand P
        • Caldamone A
        • Malone P
        • et al.
        The ESPU/SPU standpoint on the surgical management of disorders of sex development (DSD).
        J Pediatr Urol. 2014; 10: 8-10https://doi.org/10.1016/j.jpurol.2013.10.023
        • Ernst MM
        • Liao LM
        • Baratz AB
        • Sandberg DE:
        Disorders of sex development/intersex: gaps in psychosocial care for children.
        Pediatrics. 2018; 142https://doi.org/10.1542/peds.2017-4045
        • Yankovic F
        • Cherian A
        • Steven L
        • et al.
        Current practice in feminizing surgery for congenital adrenal hyperplasia; a specialist survey.
        J Pediatr Urol. 2013; 9: 1103-1107https://doi.org/10.1016/j.jpurol.2013.03.013
        • Bennecke E
        • Bernstein S
        • Lee P
        • et al.
        Early genital surgery in disorders/differences of sex development: patients’ perspectives.
        Arch Sex Behav. 2021; 50: 913-923https://doi.org/10.1007/s10508-021-01953-6
        • Baskin A
        • Wisniewski AB
        • Aston CE
        • et al.
        Post-operative complications following feminizing genitoplasty in moderate to severe genital atypia: results from a multicenter, observational prospective cohort study.
        J Pediatr Urol. 2020; 16: 568-575https://doi.org/10.1016/j.jpurol.2020.05.166
        • Finlayson C
        • Rosoklija I
        • Aston CE
        • et al.
        Baseline characteristics of infants with atypical genital development: phenotypes, diagnoses, and sex of rearing.
        J Endocr Soc. 2019; 3: 264-272https://doi.org/10.1210/js.2018-00316
        • Perez MN
        • Delozier AM
        • Aston CE
        • et al.
        Predictors of psychosocial distress in parents of young children with disorders of sex development.
        J Urol. 2019; 202: 1046-1051https://doi.org/10.1097/JU.0000000000000424
        • White PC
        • Speiser PW:
        Congenital adrenal hyperplasia due to 21-hydroxylase deficiency 1.
        Endocr Rev. 2000; 21: 245-291https://doi.org/10.1210/edrv.21.3.0398
        • Alpern AN
        • Gardner M
        • Kogan B
        • et al.
        Development of health-related quality of life instruments for young children with disorders of sex development (DSD) and their parents.
        J Pediatr Psychol. 2016; : jsw022https://doi.org/10.1093/jpepsy/jsw022
        • Suorsa-Johnson KI
        • Gardner MD
        • Baskin A
        • 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.e17)https://doi.org/10.1016/j.jpurol.2021.10.020
        • Sanders C
        • Carter B
        • Goodacre L:
        Searching for harmony: parents’ narratives about their child's genital ambiguity and reconstructive genital surgeries in childhood.
        J Adv Nurs. 2011; 67: 2220-2230https://doi.org/10.1111/j.1365-2648.2011.05617.x
        • Phares V
        • Lopez E
        • Fields S
        • et al.
        Are fathers involved in pediatric psychology research and treatment?.
        J Pediatr Psychol. 2005; 30: 631-643https://doi.org/10.1093/jpepsy/jsi050
        • Wolffenbuttel KP
        Timing of feminising surgery in disorders of sex development.
        Endocr Dev. 2014; 27: 210-221
        • Bougnères P
        • Bouvattier C
        • Cartigny M
        • Michala L:
        Deferring surgical treatment of ambiguous genitalia into adolescence in girls with 21-hydroxylase deficiency: a feasibility study.
        Int J Pediatr Endocrinol. 2017; 2017: 3-7https://doi.org/10.1186/s13633-016-0040-8
        • Jarrett OO
        • Ayoola OO
        • Jonsson B
        • et al.
        Country-based reference values and international comparisons of clitoral size in healthy Nigerian newborn infants.
        Acta Paediatr Int J Paediatr. 2015; 104: 1286-1290https://doi.org/10.1111/apa.13219
        • Kavanaugh GL
        • Mohnach L
        • Youngblom J
        • et al.
        Good practices” in pediatric clinical care for disorders/differences of sex development.
        Endocrine. 2021; 73: 723-733https://doi.org/10.1007/s12020-021-02748-4