Objective: The impact of parent-reported stigma due to their child's disorder/difference of sex development (DSD) on parent psychosocial adjustment is poorly understood. In other pediatric populations, perceived interference of medical conditions into daily activities (i.e., illness intrusiveness) mediates the relationship of stigma to adjustment. This study assessed relationships between parent-focused and child-focused stigma / illness intrusiveness / depressive and anxious symptoms. Exploratory analyses sought to identify patient characteristics associated with stigma. Method: Caregivers (59 women and 43 men) of 63 children diagnosed with a DSD up to age 4 years completed measures of demographics, parent-focused and child-focused stigma, illness intrusiveness, and depressive and anxious symptoms. Results: Increased parent-focused and child-focused stigma were associated with increased illness intrusiveness, which, in turn, was associated with increased depressive and anxious symptoms for parents nested within dyads. Among children with DSD family histories, parents reported greater childfocused stigma. Conclusion: Parents who experience DSD-related stigma report greater interference of their child's DSD into their daily activities, which is associated with poorer psychosocial adjustment. Findings support developing clinical interventions related to parents' perceptions of stigma and illness intrusiveness to improve parent adjustment.(J Dev Behav Pediatr 43:e473-e482, 2022) Index terms: disorders/differences of sex development, illness stigma, illness intrusiveness, parent psychosocial adjustment.Di sorders/differences of sex development (DSDs) refer to medical conditions in which a person's genetic, gonadal, and/or phenotypic sex are discordant. 1 When atypical genital development is present, diagnoses are often made in infancy. 2 As such, caregivers/parents (henceforth parents) face complex medical decisions for their child in the first years of life. Limited research exists regarding parents' psychosocial experiences of having a child with a DSD. 3 Importantly, parents report that stigma plays a role in medical decision making for their child's DSD and the likelihood of discussing their child's condition with others. 4 These concerns may interfere with parents' ability to engage in daily activities. Thus, understanding