Systematic assessment of outcomes following a genetic diagnosis identified through a large-scale research study into developmental disorders

Genet Med. 2021 Jun;23(6):1058-1064. doi: 10.1038/s41436-021-01110-3. Epub 2021 Feb 18.

Abstract

Purpose: The clinical and psychosocial outcomes associated with receiving a genetic diagnosis for developmental disorders are wide-ranging but under-studied. We sought to investigate outcomes from a subset of families who received a diagnosis through the Deciphering Developmental Disorders (DDD) study.

Methods: Individuals recruited through the Peninsula Clinical Genetics Service who received a confirmed genetic diagnosis through the DDD study before August 2019 (n = 112) were included in a clinical audit. Families with no identified clinical outcomes (n = 16) were invited to participate in semistructured telephone interviews.

Results: Disease-specific treatment was identified for 7 probands (6%), while 48 probands (43%) were referred for further investigations or screening and 60 probands (54%) were recruited to further research. Just 5 families (4%) opted for prenatal testing in a subsequent pregnancy, reflecting the relatively advanced maternal age in our cohort, and 42 families (38%) were given disease-specific information or signposting to patient-specific resources such as support groups. Six interviews were performed (response rate = 47%) and thematic analysis identified four major themes: reaching a diagnosis, emotional impact, family implications, and practical issues.

Conclusion: Our data demonstrate that receiving a genetic diagnosis has substantial positive medical and psychosocial outcomes for the majority of patients and their families.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Child
  • Developmental Disabilities* / diagnosis
  • Developmental Disabilities* / epidemiology
  • Developmental Disabilities* / genetics
  • Emotions
  • Female
  • Genetic Testing*
  • Humans
  • Pregnancy
  • Referral and Consultation