Psychosocial well-being in Dutch adults with disorders of sex development
Introduction
In individuals with disorders of sex development (DSD) the development of chromosomal, gonadal, and/or anatomic sex is atypical [1]. It is assumed that this incongruence puts them in a vulnerable position in society [2]. Current clinical management strategies therefore will include advice for early gender assignment, genital corrections, and hormonal treatments [1]. Lately, these early interventions have raised debate: it has been argued that they reflect society's intolerance to variance in sex and gender and major decisions are made without consent of children themselves [3], [4], [5], [6]. It has been suggested that postponement of gender assignment and genital surgery until the child is old enough to decide him/herself will benefit the child's well-being [7]. Randomized controlled comparison of the current treatment policy and the policy of delayed interventions is highly valued [8] but is difficult to conduct. The majority of parents living in Western countries choose gender assignment and genital surgery in early childhood [9], [10], [11].
Outcome studies on psychosocial well-being have been conducted. Due to differences in applied methodology and measures, findings are difficult to compare and show inconsistencies. These studies have mainly been carried out in females and focused on gender identity [12], [13], [14], [15], [16], [17], [18], sexual quality of life [19], [20], [21], [22], [23], and (psycho)sexual functioning [24], [25], [26], [27], [28], [29], [30], [31], [32], while studies on quality of life [33], social participation, self-esteem, and emotional problems are scarce. Studies addressing health related quality of life (HRQoL), emotional distress, and psychopathology in women with 46,XX congenital adrenal hyperplasia (CAH) revealed inconclusive outcomes, from reduced to a better HRQoL [20], [34], [35], [36], and from no substantial emotional distress to increases in emotional problems [28], [37], [38], [39], [40]. Women with complete androgen insensitivity syndrome (CAIS) reported to function psychologically well or even better than reference groups [20], [41]. In individuals with partial androgen insensitivity syndrome (PAIS), disorders in the biosynthesis of androgens, or gonadal dysgenesis, mental health problems have been reported [42], but findings had not been replicated in another study [16].
A few studies have been conducted in men and focused on sexual functioning [29]. Men with 46,XY CAH suffer from adrenal problems and testicular adrenal rest tumors and its consequences [43], [44]. These men reported more negative emotions [45], anxiety and depression [40], and psychiatric morbidity [46], [47]. Impairments in subjective health status have been reported [40], [48], but also a favorable health status compared to the general population [36].
In DSD there is a great variety in genital development between and within different diagnostic groups. In the current study we evaluated psychosocial well-being in relation to gender of rearing and degree of genital atypicality in Dutch individuals with DSD. In their prenatal development, persons with DSD have been exposed to atypical levels of androgens. We hypothesized that persons with DSD who underwent an atypical prenatal development leading to physical atypicality are more vulnerable to experiences that negatively affect their psychosocial well-being compared to persons with typical female or male genitalia [2].
Section snippets
Study design
The present study was embedded in a national follow-up audit on sexual well-being, gender identity development, and psychosocial well-being in persons with DSD [24], [25], [29], [30]. The study protocol was in line with the World Medical Association declaration of Helsinki and was approved by the boards of the ethical committees of the three medical centers that joined the study [49]. Data collection was carried out between 2007 and 2012. The Dutch patient support groups were involved in the
Statistical analysis
Before statistical analyses we evaluated the internal consistencies of the abovementioned measures for our sample using Cronbach's alpha. For most questionnaires, internal reliability values were good (Cronbach's α above .70). For three ASR-scales, internal reliability values were acceptable (Rule-breaking Behavior, Thought Problems and Antisocial Personality Problems; Cronbach α's respectively .64, .66, and .66).
For categorical variables, between-group differences were tested using Chi-square
Participant characteristics
Table 2 summarizes socio-demographic characteristics. Groups did not differ in median age (p = .49), ancestry (p = .09), marital status (p = .27), educational level (p = .52) or vocational status (p = .65).
Reasons for non-response remained unknown; most no-participation forms were not returned or were not filled out completely. Responders and non-responders did not differ with respect to diagnosis, medical treatment, age, living in urban or rural areas, or medical center [25], [29], [30].
Illness cognitions
Details on
Discussion
This study aimed to investigate psychosocial well-being in Dutch individuals with DSD. Overall, we conclude that participants reported good psychosocial well-being; they generally reported a good HRQoL, no serious emotional problems, a high self-esteem, and seemed to cope well with DSD compared to reference groups. In a recently published review [67] on this subject, seven out of 10 studies reported a (mildly) affected psychosocial well-being, in three studies such changes were not reported.
Conflicts of interest
The authors declare that there are no conflicts of interest.
Acknowledgements
We thank all participants who volunteered in this study for their carefulness and efforts. We thank Jan van der Ende and Erwin Birnie for statistical advices. We thank Ingrid van Slobbe, Joke Dunk, Karen Kwak, Jacqueline Knol, and Hanneke Kempes for their assistance in carrying out the study.
This study has been supported by the Edli Fund (Arianne B. Dessens), FWO Flanders Fund for scientific research (Nina Callens), and Fonds Swart – van Essen (Nita G.M. de Neve –Enthoven).
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2019, Journal of Pediatric UrologyCitation Excerpt :In addition, all studies are cross-sectional and do not allow the demonstration of causal effects. Concerning the recruitment procedure, 13 studies had recruited their samples from hospitals [4–7,10,12–15,18,19,21,22]; 3 studies had recruited their samples in hospital and through support groups, meeting, and conferences [2,3,9]; and 1 used national diagnostic registers [8]. For 1 study, this information was not provided [11].
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