Skip to main page content
U.S. flag

An official website of the United States government

Dot gov

The .gov means it’s official.
Federal government websites often end in .gov or .mil. Before sharing sensitive information, make sure you’re on a federal government site.

Https

The site is secure.
The https:// ensures that you are connecting to the official website and that any information you provide is encrypted and transmitted securely.

Access keys NCBI Homepage MyNCBI Homepage Main Content Main Navigation
. 2020:2020:3.
doi: 10.1186/s13633-020-0073-x. Epub 2020 Feb 14.

Parent-reported outcomes in young children with disorders/differences of sex development

Salma R Ali #  1 , Zoe Macqueen #  1 , Melissa Gardner  2 , Yiqiao Xin  3 , Andreas Kyriakou  1 , Avril Mason  1 , M Guftar Shaikh  1 , Sze C Wong  1 , David E Sandberg #  2 , S Faisal Ahmed #  1
Affiliations

Parent-reported outcomes in young children with disorders/differences of sex development

Salma R Ali et al. Int J Pediatr Endocrinol. 2020.

Abstract

Background: There is a paucity of tools that can be used in routine clinical practice to assess the psychosocial impact of Disorders/Differences of Sex Development (DSD) on parents and children.

Objective: To evaluate the use of short Parent Self-Report and Parent Proxy-Report questionnaires that can be used in the outpatient setting.

Methods: Previously validated DSD-specific and generic items were combined to develop a Parent Self-Report questionnaire and a Parent Proxy-Report questionnaire for children under 7 years. Of 111 children approached at one tertiary paediatric hospital, the parents of 95 children (86%) with DSD or other Endocrine conditions completed these questionnaires.

Results: Questionnaires took under 10 min to complete and were found to be easy to understand. Compared to reference, fathers of children with DSD reported less stress associated with Clinic Visits (p = 0.02) and managing their child's Medication (p = 0.04). However, parents of children with either DSD or other Endocrine conditions reported more symptoms of Depression (p = 0.03). Mothers of children with DSD reported greater Future Concerns in relation to their child's condition (median SDS - 0.28; range - 2.14, 1.73) than mothers of children with other Endocrine conditions (SDS 1.17; - 2.00, 1.73) (p = 0.02). Similarly, fathers of children with DSD expressed greater Future Concerns (median SDS -1.60; - 4.21, 1.00) than fathers of children with other Endocrine conditions (SDS 0.48; - 2.13, 1.52) (p = 0.04).

Conclusion: DSD was associated with greater parental concerns over the child's future than other Endocrine conditions. Brief parent-report tools in DSD can be routinely used in the outpatient setting to assess and monitor parent and patient needs.

Keywords: DSD; PRO; Psychosocial adjustment; Screening.

PubMed Disclaimer

Conflict of interest statement

Competing interestsThe authors declare that they have no competing interests.

Figures

Fig. 1
Fig. 1
Cases and recruitment details. DSD, Disorder of Sex Development. aCaregivers not approached due to consultation running simultaneously (n = 7) bCaregivers not able to complete questionnaires as insufficient time after clinic (n = 8), other commitments (n = 7) or already completed a separate hospital questionnaire (n = 1)
Fig. 2
Fig. 2
Boxplots for Future Concerns, Clinic Visits and Depression. DSD, Disorder of Sex Development; Endocrine, Children with other Endocrine conditions. *p < 0.05 represents a significant difference compared to zero
See this image and copyright information in PMC

References

    1. Lee PA, Houk CP, Ahmed SF, Hughes IA. 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. doi: 10.1542/peds.2006-0738. - DOI - PubMed
    1. Ahmed SF, Achermann JC, Arlt W, Balen A, Conway G, Edwards Z, et al. Society for Endocrinology UK guidance on the initial evaluation of an infant or an adolescent with a suspected disorder of sex development (Revised 2015) Clin Endocrinol (Oxf) 2016;84:771–788. doi: 10.1111/cen.12857. - DOI - PMC - PubMed
    1. Suorsa KI, Mullins AJ, Tackett AP, Reyes KJ, Austin P, Baskin L, et al. Characterizing early psychosocial functioning of parents of children with moderate to severe genital ambiguity due to disorders of sex development. J Urol. 2015;194:1737–1742. doi: 10.1016/j.juro.2015.06.104. - DOI - PMC - PubMed
    1. Alpern AN, Gardner M, Kogan B, Sandberg DE, Quittner AL. Development of health-related quality of life instruments for young children with disorders of sex development (DSD) and their parents. J Pediatr Psychol. 2017;42:544–558. - PMC - PubMed
    1. Rolston AM, Gardner M, Vilain E, Sandberg DE. Parental reports of stigma associated with child’s disorder of sex development. Int J Endocrinol. 2015;2015:980121. doi: 10.1155/2015/980121. - DOI - PMC - PubMed

LinkOut - more resources