OR05-2 Development and Validation of a Short Version of the Quality of Life DSD Questionnaire (QoL-DSD) for Parents of Young Children with Disorders/Differences of Sex Development
Authors: Ahmed S Faisal , Flett Martyn , Gardner Melissa , Lee Boma , O’Toole Stuart , Sandberg David , Steven Mairi , Xin Yiqiao , Ali Salma Rashid 
Published: November 2022

Background

Disorders/Differences of sex development (DSD) may be associated with adverse psychosocial and psychosexual outcomes in adults. However, there is a paucity of information on health-related quality of life (QoL) outcomes in parents and children with DSD and a lack of instruments available for evaluating these outcomes. Recently, this has led to the development of Parent Self-Report and Proxy-Report QoL questionnaires (QoL-DSD), validated measures for parents of young children with DSD, comprising 63 items within 13 domains and 25 items within 5 domains, respectively.

Objective

While retaining the original domain structure of the QoL-DSD questionnaires, we aimed to develop short forms of the QoL-DSD, optimizing their use in routine clinic settings.

Methods

Short forms of the DSD-QoL Parent Self-Report (QoL-DSD Short PSR) and Parent Proxy-Report (QoL-DSD Short PPR) questionnaires were developed following exploratory factor analysis with maximum likelihood and varimax rotation, using previous QoL-DSD data from 132 parents. Long and resulting short form questionnaires were completed online by 18 parents of children with DSD, under 7 years of age, attending endocrine and urology clinics at one tertiary hospital in Scotland.

Results

Item selection for the short forms — QoL-DSD Short PSR and QoL-DSD Short PPR — based on item factor loadings of &gt;0.8, produced questionnaires containing 16 and 7 items, respectively. Eighteen parents completed both long and short forms of the Parent Self-Report for children aged &lt;7 years, and a subset of these (n=13) also completed long and short forms of the Parent Proxy-Report for children aged 2 to 7 years. Of the 18 target children, all were boys with a median age of 3.6 years (range 0.4, 6.6) and 10 (59%) had proximal hypospadias. Overall, agreement was achieved between the short and long questionnaires in 9 out of 12 (75%) domains on the Parent Self-Report and 4 out of 5 (80%) domains on the Parent Proxy-Report. Correlations between total scores on the short and long forms were 0.508 to 0.897 in 9 of 12 domains in the QoL-DSD Short PSR. Parental feedback (n=18) regarding the acceptability of the short versus long forms was evaluated using a 5-point Likert score: 83% (versus 66%) of parents agreed the length of time (less than 3 minutes) taken to complete short forms was acceptable, 39% preferred the short forms compared with 11% whom preferred the longer version, 45% (versus 22%) stated a preference to complete the short forms should they be implemented routinely at clinic visits in the future.

Conclusions

Short forms of the QoL-DSD for parents of young children with DSD may be more acceptable for use in a routine outpatient setting to evaluate psychosocial distress experienced by young children with DSD and their caregivers. Further psychometric validation in a larger cohort is warranted.

Presentation: Saturday, June 11, 2022 11:45 a.m. - 12:00 p.m.