For families, the birth of a child with a DSD and the attendant uncertainty about the child's gender and future psychological and psychosexual development is believed to be extraordinarily stressful . High levels of stress are likely to arise from both the unfamiliarity and perceived stigma associated with DSD and from the controversies surrounding current clinical management of these conditions. Despite parents having had access to services delivered at reputable tertiary care facilities, parents' experiences suggest a need to continue to strengthen the Consensus Statements' call for comprehensive and integrated long-term care for families affected by DSD . The findings suggest, in particular, a need to improve provider-family/patient communication and to increase the availability of psychosocial support as an integral component in the delivery of care. While the parents in this study reported that early surgical interventions eased some of their immediate concerns, the findings suggest varied opportunities for enhancing education, shared decision-making, and linking families of affected children to others sharing common challenges .
Study participants expressed certainty about their child's sex and gender-whether announced at birth or subsequently assigned. For those parents that referenced diagnostic tests as a source of their certainty, it is unclear if such confidence stemmed from their understanding of test findings (e.g., karyotype, studies of internal reproductive anatomy), or if these results were selectively called upon to reinforce their intuitions. Several parents' reports suggested that healthcare providers framed clinical and laboratory findings to either generate or reinforce parents' beliefs that these indicators of sex development are determinative of gender identity. Until the mid-1950s, medical management of persons affected with DSD was guided by the belief that an individual's ''true sex'' could be revealed through examination of internal anatomy and that the person's identification as boy or girl would naturally conform to their ''true sex" . We now know that individual markers of biological sex can be associated with a range of gender outcomes [29–33]. Given the potential effect of this information on parental decision-making, this prompts the question to what degree and with how much detail should healthcare providers, in the promotion of the principles of shared decision-making, educate parents about the nuances of somatic sex development and their inconsistent relationship with gender identity in DSD if, by doing so, they potentially enhance cognitive and emotional conflict in the parents? Understandably, providers may fear alienating those parents who already have a strong conviction about their child's sex and gender by presenting a contrary viewpoint. However, without providing comprehensive information, providers risk breaching the ethics of informed consent for clinical interventions and the possibility that the parents will later learn about the withheld information and interpret provider's selectivity as a shortcoming or even a deception.
Delivering information that aligns with the ethics of informed consent is particularly critical when interventions are elective, non-urgent, controversial, and associated with potentially serious risks [34, 35]. To enhance transparency and diminish the likelihood of decisional regret, Karkazis and colleagues recently outlined a 6-step model for shared decision-making in DSD as it pertains to genital surgery in young children .
Parents in our study also reported frustration over gaps in information about their child's condition. This could be due to a number of factors: (1) uncertainty about the diagnosis in the early stages which creates difficulties when discussing the condition, its course, and early management, (2) lack of educational tools that make complex medical concepts accessible to the general public, (3) parents' potentially diminished capacity to process complex medical concepts and make decisions during a time of stress, and (4) the existing gaps in medical literature surrounding DSD and DSD care. Parenting with uncertainties regarding the child's future is common in pediatric chronic illness and disease specific parent-to-parent support has been shown to be particularly useful in helping parents to cope with uncertainties and their frustrations during early decision-making; the use of support groups is additionally endorsed by the Consensus Statement . Development of high quality DSD-specific educational content that adheres to the principles of health literacy  may also facilitate improved communication and knowledge sharing between provider and family. One exemplary sample of such content are the Sex Development pages of the AboutKidsHealth website edited by faculty and staff at the Hospital for Sick Children in Toronto . Providers may also integrate HRQoL assessments into clinical care as a means of better identifying and addressing parent and child needs and concerns .
Parents recalled events surrounding their child's genital surgery with particular salience. Consistent with other reports, parents viewed genital surgery as a necessary and obvious "fix" for their child's DSD [9, 13, 38]. They justified early surgery as a means of averting negative consequences, such as stigmatization, that are associated with atypical genital appearance or function [13, 39, 40]. While satisfied with surgical outcomes, they continued to be concerned about the child's future experiences. Parents' worries primarily pertained to uncertainty about changes in their child's genital appearance or function associated with the onset of puberty, whether spontaneous or by hormone replacement, and the renewed risk of stigmatization or rejection by potential sexual/romantic partners and peers. These findings point out that early surgery reduces early parental concerns regarding genital appearance, but does not eliminate worries about their child's future sex development or function. Accordingly, there is an important need to maintain contact with families in order to monitor parents' expectations and address unresolved anxiety about the child's future, even in those cases in which early surgery was considered an unqualified success.
Previous research regarding disclosure in DSD has focused on harm to affected individuals by being either uninformed or misinformed about their condition [1, 6, 15]. However, parents of affected individuals also appear to grapple with issues of information sharing. For the parents in the present study, withholding information from other adults was motivated by a desire to protect the child's privacy and to prevent stigmatization. However, parents varied markedly in the degree to which they disclosed details. Those who maintained fairly strict privacy experienced this approach as very stressful, whereas those who chose to share information with trusted others reported experiencing less strain. The current findings suggest that in addition to parents educating their child in a developmentally-appropriate way about their condition, they may benefit from more explicit and extensive discussions about sharing information with their usual social support system (family and friends). The extent to which such discussions between parents and providers are currently occurring is unclear. The results suggest, however, that the status quo is inadequate with respect to the counseling of parents on the challenges of information sharing and support seeking. The child's right to privacy should be balanced against the risks associated with secrecy, promoting a sense of shame, and limiting opportunities for social support. Failure to achieve this balance could contribute to unresolved parental feelings of guilt and possibly to a negative self-concept, shame or isolation for the DSD-affected person [9, 41, 42]. The Consensus Statement  identifies the timing and content of information management as warranting targeted study.
The majority of interactions between parents and their child's healthcare providers were described as positive. However, several negative interactions were noted in the context of genital examinations which parents felt were unnecessary or exploitative. There is reason to be concerned that repeated genital examinations and medical photography can have lasting and severe negative psychological consequences [43–45]. Responses to genital examinations in DSD and strategies to perform them in a way that reduces the likelihood of harm is another area in which systematic information is missing. In the interim, providers should continue to communicate openly with the patient and family, describe the purpose of the exams, ask for consent (and when appropriate, assent), and minimize patient exposure. Input from child life specialists who are trained to mitigate distress associated with medical procedures may be helpful .
Study Strengths and Limitations
This study presents the experiences of a relatively large sample of mothers and fathers of diagnostically diverse young children with DSD. Parent participants were identified through a systematic review of the children's medical records; only a small proportion of those contacted refused participation. An additional strength of this study was the extent of fathers' participation (35%). Frequently, studies of children with medical conditions rely exclusively on maternal reports [47, 48].
All children had undergone genital surgery. It is possible that this high rate of genital surgery is related to our sampling process which identified participants via chart review at academic medical centers. This sampling approach may be suboptimal for ascertaining cases in which surgery had not been performed. The proportion of children with DSD who have not undergone surgical interventions is unknown. Accordingly, it is difficult to determine the extent to which our findings can generalize to patients and families who have elected not to have surgery performed. Research by Warne and Raza  encourage investigators in this area to be sensitive to variability across cultural and socioeconomic contexts.
Interviews were guided by the standardized probes to evaluate the quality of items to be incorporated into HRQoL questionnaires for parents of young children with DSD. Accordingly, although parents were asked to talk about areas of importance to them and their family that were not specifically covered in the HRQoL instrument, there may be topics of importance that did not emerge due to the interview structure. Tempering this concern is that novel questions were added to the HRQoL questionnaires based on ideas parents generated during the interviews, demonstrating that parents explored experiences beyond the confines of the cognitive interview structure. Finally, because interviews were conducted at varying intervals after some of the events being described, the potential exists for distorted recall.