Parental decisional regret and views about optimal timing of female genital restoration surgery in congenital adrenal hyperplasia

Purpose The role of female genital restoration surgery (FGRS) in girls with congenital adrenal hyperplasia (CAH) is controversial, with no long-term parent-reported outcomes available. Decisional regret (DR) affects most parents after their children's treatment of pediatric conditions, including hypospadias. We aimed to assess parental DR after FGRS in infancy or toddlerhood and explore optimal timing for surgery. Materials and methods One-hundred and six parents of females with CAH undergoing FGRS before 3 years old and followed at our institution (1999-2017) were invited to enroll online. Higher Decision Regret Scale (DRS) scores indicated greater DR (range 0-100). Participants also reported preferred FGRS timing relative to their surgery (earlier, same, later/delayed). Non-parametric statistical tests were used. Results Thirty-nine parents (median 4.4 years after FGRS) participated (36.8% response rate). Median age at FGRS was 9 months. Median DRS score was 0 (mean: 5.0). Overall, 20.5% of parents reported some regret (all mild-moderate) (Figure). Fewer parents reported DR after FGRS compared with published DR after hypospadias repair (50-92%, p <= 0.001) or adenotonsillectomy (41-45%, p <= 0.03). No parent preferred delayed FGRS. Seven parents (18.1%) preferred earlier surgery, especially when performed after birthday (80.0% vs. 8.8%, p = 0.004). Discussion We present the first report of validated long-term parent-reported outcomes after FGRS in infant and toddler girls with CAH. One limitation is that this is largely a single surgeon series. Reasons for the observed low levels of DR are likely multifactorial. Far from a definitive study, we aimed to provide parents willing to share about their experience an opportunity to do so. For that reason, selection bias may exist in our study. While parents with higher DR were potentially less likely to participate because of mistrust of the medical establishment, those with a negative experience may in fact be more likely to voice their opinions. A low participation rate was likely a result of the sensitive nature of FGRS, a desire for privacy, and inability to locate parents. A larger study will be required to assess how DR is affected by sexual function, genital appearance and complications, and DR among women with CAH. Conclusions Parents of females with CAH report low levels of DR after FGRS in infancy and toddlerhood. This appears to be lower than after other genital and non-genital pediatric procedures. When present, parental DR is usually mild. No parents preferred delayed surgery, even among those with DR. Some preferred earlier surgery.