Parent-Rated Severity of Illness and Anxiety among Caregivers of Children Born with a Disorder of Sex Development Including Ambiguous Genitalia

Abstract

Background/Aims: Parents of children born with disorders of sex development (DSD) often experience anxiety, but risk factors, including parental perception of the severity of their child’s DSD, have not been examined. We hypothesized that severity of illness (SOI) ratings would relate to parental anxiety, and would be higher for parents of children with a potentially life-threatening DSD (e.g., 21-hydroxylase deficiency). Methods: Eighty-nine parents (M_age = 33.0, 56.2% mothers) of 51 children (M_{age in months} = 8.7) with a DSD including ambiguous genitalia were recruited from 12 specialized DSD clinics. Parents completed questionnaires prior to genitoplasty, 6 months post-genitoplasty, and 12 months post-genitoplasty (if completed). Data were analyzed with linear mixed modeling. Results: Parental anxiety decreased over time, χ²(1) = 10.14, p < 0.01. A positive relationship between SOI and anxiety was found, with SOI being a strong predictor of anxiety (b = 0.53, p < 0.01; χ²[1] = 5.33, p < 0.05). An SOI by time interaction indicated SOI had an increasing effect on anxiety over time, b = 0.06, p < 0.05; χ²(1) = 6.30, p < 0.05. There was no diagnosis by SOI interaction. Conclusion: Parental anxiety decreased over time, but those with higher SOI ratings reported greater initial anxiety followed by slower resolution over time. Underlying etiology of DSD had no effect on the relationship between SOI and anxiety.

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

“…Compared with parents of children with other Endocrine conditions, parents of children with DSD reported greater Future Concerns with regards to their child's condition. These results highlight the need for ongoing parental support and effective communication between the multidisciplinary team and families [32] and the provision of early psychological input for parents of young children with DSD [12]. Despite these greater anxieties, fathers of children with DSD reported less stress with regards to attending for Clinic Visits and administering and managing Medication required for their child's condition.…”

“…A significant minority of parents of children with DSD experience symptoms suggestive of post-traumatic stress disorder with accompanying difficulties in communicating news of their child's condition with relatives and close friends, an independent risk factor for emotional distress [8,9]. Yet there is a paucity of studies evaluating the feasibility of routine, longitudinal screening for challenges to positive psychosocial adaptation in the context of usual care [10][11][12]. Such screening would ideally go beyond the heretofore restricted focus on psychosexual differentiation, i.e., gender identity, gender role, and sexual orientation [11,13].…”

Factor analysis of the Stigma Scale—Parent version in pediatric disorders/differences of sex development.

“…Data included in the present study are from the time point each caregiver first completed the stigma measures, which varied from baseline to the fourth annual visit. Portions of data from the larger, multisite study have been reported in previous publications with different aims and hypotheses (Perez et al, 2019, 2021; Roberts, 2020; Sharkey et al, 2018; Suorsa et al, 2015).…”

Prenatal detection and evaluation of differences of sex development: A qualitative interview study of parental perspectives and unmet needs

“…13,14 Algorithms have been proposed to coordinate the prenatal evaluation in these situations, [15][16][17] and much research has centered around the postnatal psychosocial functioning of families with children affected by DSD. [18][19][20][21] However, relatively little attention has been given to the psychosocial implications of the prenatal evaluation. 22,23 While the primary aim of prenatal diagnosis may be to optimize medical care of the infant, an important secondary aim is anticipatory guidance to optimize the family's emotional preparation.…”