Disorders of sex development (DSD) are a rare set of medical conditions with prevalence rates ranging between 1:00 to 1:5000 births. Secondary atypical genitalia is seen in a subset. Psychosocial aspects have until recently been understudied. We know that some parents are at risk for psychosocial distress following the diagnosis of DSD in their offspring, including symptoms of depression, anxiety and post-traumatic stress. Data thus far has been limited due to retrospective study design, small sample sizes and a failure to include fathers. Here, Perez et al. present the results of a prospective multisite study (12 DSD clinics across the United States). Participants (recruited between September 2013 and November 2017) included mothers (76) and fathers (63) of a child (78) diagnosed with DSD and having moderate to severe genital atypia. They demonstrate a difference between mothers and fathers in how they adapt to their child’s condition: mothers reported more anxious symptoms, depressive symptoms, post-traumatic stress symptoms and worse mental health, and rated greater dissatisfaction with the cosmetic appearance of their child’s genitals. Lower household income, increased medical care and travel expenses, and having no other children were linked to increased psychosocial distress. Knowledge of these findings is useful as they can be assessed in the multidisciplinary setting of the DSD clinic and allow holistic care to be provided to the family as a whole.