Validation of a parent-reported outcome measure for young boys with a condition affecting sex development (PRO-CSD).

OBJECTIVE

To validate a short parent-reported outcome (PRO) questionnaire in young boys with conditions affecting sex development (PRO-CSD).

DESIGN

Consecutive sample cross-sectional study.

SETTING

A single tertiary children's hospital in Glasgow, UK.

PATIENTS

A total number of 98 parents of 98 affected boys with a median age of 3.1 years (10th, 90th: 0.7, 5.4).

INTERVENTIONS

Completion of recently developed brief PRO-CSD consisting of a parent proxy-report (PPR) and a parent self-report (PSR).

MAIN OUTCOMES

The study assessed test-retest reliability in 41 cases as intraclass correlation coefficients (ICCs) and group construct validity in 98 by exploring associations with seven demographic and clinical features.

RESULTS

The median ICC for all items was 0.8 (0.6, 0.9). On the PSR, parents of boys with an external masculinisation score (EMS) of less than 7 out of 12 were more concerned about their child's genitalia compared with those with an EMS of greater than 10.5 (p=0.01). An association with EMS was also observed for questions on future social concerns (p<0.01), future relationships (p<0.01) and stress on receiving the diagnosis (p=0.03). This association with EMS was maintained following adjustment for surgical history, parental age and education level. EMS was the only significant independent determinant of the overall PSR score. In the PPR analysis, parents in the lower socioeconomic grouping and those without tertiary education reported more concerns on the physical functioning of their children.

CONCLUSIONS

The psychometric properties of the PRO-CSD tool demonstrate its utility for the screening of parent-reported, condition-specific health-related quality of life outcomes in young boys with a CSD.