Emilie K Johnson1, Ilina Rosoklija2, Angela Shurba2, Anthony D'Oro2, Elisa J Gordon3, Diane Chen4, Courtney Finlayson5, Jane L Holl6. 1. Division of Urology, Ann & Robert H. Lurie Children's Hospital of Chicago, Chicago, IL, USA; Department of Urology, Northwestern University Feinberg School of Medicine, Chicago, IL, USA; Center for Healthcare Studies, Northwestern University Feinberg School of Medicine, Chicago, IL, USA. Electronic address: ekjohnson@luriechildrens.org. 2. Division of Urology, Ann & Robert H. Lurie Children's Hospital of Chicago, Chicago, IL, USA. 3. Center for Healthcare Studies, Northwestern University Feinberg School of Medicine, Chicago, IL, USA; Department of Surgery, Northwestern University Feinberg School of Medicine, Chicago, IL, USA. 4. Division of Adolescent Medicine, Ann & Robert H. Lurie Children's Hospital of Chicago, Chicago, IL, USA; Department of Psychiatry and Behavioral Sciences, Northwestern University Feinberg School of Medicine, Chicago, IL, USA; Department of Pediatrics, Northwestern University Feinberg School of Medicine, Chicago, IL, USA; Division of Child and Adolescent Psychiatry, Ann & Robert H. Lurie Children's Hospital of Chicago, Chicago, IL, USA. 5. Division of Endocrinology, Ann & Robert H. Lurie Children's Hospital of Chicago, Chicago, IL, USA; Department of Pediatrics, Northwestern University Feinberg School of Medicine, Chicago, IL, USA. 6. Center for Healthcare Studies, Northwestern University Feinberg School of Medicine, Chicago, IL, USA.
Abstract
BACKGROUND: Children, adolescents, and young adults (children/youth) with differences/disorders of sex development (DSD) face challenges related to future fertility; this may be due to variations in gonadal development, and, for some, gonadectomy performed to reduce the risk of malignancy. Childhood may be the only time for preservation of biological fertility potential for children/youth who undergo gonadectomy or have early gonadal failure. Fertility-related decision-making for these patients is particularly complicated, due to the need for parental proxy decision-making, potential discordance between gender identity and gonadal type, and uncertain future assisted reproductive technologies. OBJECTIVE: This study aimed to assess: (1) attitudes regarding future fertility, and (2) healthcare needs for fertility-related decision-making among parents of children/youth with DSD. STUDY DESIGN: Semi-structured qualitative interviews about future fertility were conducted with parents of children/youth with DSD. Parents who had never discussed fertility with a healthcare provider were excluded. Grounded theory methodology was used to identify emergent themes and patterns. Demographics and clinical characteristics were assessed via survey and medical chart review. RESULTS: Nineteen parents were interviewed (participation rate: 60%, 14 mothers/5 fathers, median patient age at diagnosis 6 months (range 0-192), eight DSD diagnoses). The most common emergent themes are summarized in the Summary Table. Most parents identified fertility as a key concern, both at time of diagnosis and throughout development. Parents expressed difficulty with timing of disclosure about potential infertility to their children. Multiple preferences related to medical decision-making about future fertility and fertility preservation were expressed, including: a desire for step-by-step decision-making, and use of medically vetted information and research to guide decisions. DISCUSSION: This qualitative study provided new information about the perspectives of parents of children/youth with DSD regarding future fertility. Previous studies have suggested that the possibility of biological parenthood is important to many individuals with DSD. This study provided an in-depth parental perspective. This is important because many decisions that affect future fertility are made in childhood, and require parents to make decisions on behalf of their children. The study sample was limited in its geographic diversity. Strengths of the study included diversity in age of the child/youth, ethnic backgrounds, and the DSD diagnoses that were represented. CONCLUSIONS: Future fertility was a concern for many parents of children/youth with DSD. Parents expressed multiple priorities and preferences related to making difficult fertility-related medical decisions for their children. Many of the study findings could be incorporated into future best practices for discussions about fertility with families of children/youth with DSD.
BACKGROUND:Children, adolescents, and young adults (children/youth) with differences/disorders of sex development (DSD) face challenges related to future fertility; this may be due to variations in gonadal development, and, for some, gonadectomy performed to reduce the risk of malignancy. Childhood may be the only time for preservation of biological fertility potential for children/youth who undergo gonadectomy or have early gonadal failure. Fertility-related decision-making for these patients is particularly complicated, due to the need for parental proxy decision-making, potential discordance between gender identity and gonadal type, and uncertain future assisted reproductive technologies. OBJECTIVE: This study aimed to assess: (1) attitudes regarding future fertility, and (2) healthcare needs for fertility-related decision-making among parents of children/youth with DSD. STUDY DESIGN: Semi-structured qualitative interviews about future fertility were conducted with parents of children/youth with DSD. Parents who had never discussed fertility with a healthcare provider were excluded. Grounded theory methodology was used to identify emergent themes and patterns. Demographics and clinical characteristics were assessed via survey and medical chart review. RESULTS: Nineteen parents were interviewed (participation rate: 60%, 14 mothers/5 fathers, median patient age at diagnosis 6 months (range 0-192), eight DSD diagnoses). The most common emergent themes are summarized in the Summary Table. Most parents identified fertility as a key concern, both at time of diagnosis and throughout development. Parents expressed difficulty with timing of disclosure about potential infertility to their children. Multiple preferences related to medical decision-making about future fertility and fertility preservation were expressed, including: a desire for step-by-step decision-making, and use of medically vetted information and research to guide decisions. DISCUSSION: This qualitative study provided new information about the perspectives of parents of children/youth with DSD regarding future fertility. Previous studies have suggested that the possibility of biological parenthood is important to many individuals with DSD. This study provided an in-depth parental perspective. This is important because many decisions that affect future fertility are made in childhood, and require parents to make decisions on behalf of their children. The study sample was limited in its geographic diversity. Strengths of the study included diversity in age of the child/youth, ethnic backgrounds, and the DSD diagnoses that were represented. CONCLUSIONS: Future fertility was a concern for many parents of children/youth with DSD. Parents expressed multiple priorities and preferences related to making difficult fertility-related medical decisions for their children. Many of the study findings could be incorporated into future best practices for discussions about fertility with families of children/youth with DSD.
Authors: Diane Chen; Moira A Kyweluk; Afiya Sajwani; Elisa J Gordon; Emilie K Johnson; Courtney A Finlayson; Teresa K Woodruff Journal: LGBT Health Date: 2019-04 Impact factor: 4.151
Authors: Jaclyn L Papadakis; Jonathan L Poquiz; Cindy L Buchanan; Yee-Ming Chan; Canice E Crerand; Jennifer Hansen-Moore; Hillary M Kapa; Leena Nahata; Keeley J Pratt; Amy C Tishelman; Diane Chen Journal: Clin Pract Pediatr Psychol Date: 2020-12-03
Authors: Erin E Rowell; Timothy B Lautz; Krista Lai; Erica M Weidler; Emilie K Johnson; Courtney Finlayson; Kathleen van Leeuwen Journal: Semin Pediatr Surg Date: 2021-09-04 Impact factor: 1.900
Authors: William Byne; Dan H Karasic; Eli Coleman; A Evan Eyler; Jeremy D Kidd; Heino F L Meyer-Bahlburg; Richard R Pleak; Jack Pula Journal: Transgend Health Date: 2018-05-01