Decisional regret and views about optimal timing of female genital restoration surgery in CAH: patient and parental perspectives
Konrad M. Szymanski, MD MPH, Benjamin Whittam, MD MS, Martin Kaefer, MD MS, Heather Frady, RN BSN, Jessica T. Casey, MD, Vi T. Tran, BSc BA, Mark P. Cain, MD, Richard C. Rink, MD.
Riley Hospital for Children, Indianapolis, IN, USA.
BACKGROUND: The role of female genital restoration surgery (FGRS) in girls with congenital adrenal hyperplasia (CAH) is controversial, with no long-term, patient-/parent-reported outcomes available. Decisional regret (DR) affects 40-92% of parents after treatments of their children for various pediatric conditions, including genital reconstruction for hypospadias in boys. DR after FGRS in CAH is unknown. We aimed to assess DR after FGRS in infancy and toddlerhood and explore optimal timing for surgery.
METHODS: Of 158 females with CAH undergoing FGRS at our institution (1980-2017), 38 adult women and 102 parents with contact information available were invited to enroll online. Higher Decision Regret Scale (DRS) scores indicated greater DR (range: 0-100). Participants reported preferred timing of FGRS relative to their surgery (earlier, same, later/delayed). Non-parametric tests were used for analysis.
RESULTS: Four women (median 29.0 years after FGRS) and 34 parents (median 4.7 years after FGRS) participated (10.5%, 33.3% response rates, respectively). Median age at FGRS was 8 months: 86.5% underwent vaginoplasty and 94.6% clitoroplasty. Median DRS scores were 0 for both women and parents (p=0.89, means: 3.8, 4.3, respectively). Overall, 25.0% of women and 20.6% of parents reported some regret (all mild-moderate). Fewer parents reported DR after FGRS compared to published DR after hypospadias repair (50-92%, p<=0.003) or adenotonsillectomy (41-45%, p<=0.04). No woman or parent preferred delayed FGRS. Six parents (17.7%) would have preferred earlier surgery.
CONCLUSIONS: Females with CAH and their parents report lower DR after FGRS in infancy and toddlerhood compared to reported genital and non-genital pediatric procedures. When present, DR is usually mild. Neither patients nor parents preferred delayed surgery, even among those with DR. Some preferred even earlier surgery.
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