Expectations and experience: Parent and patient perspectives regarding treatment for Severe Combined Immunodeficiency (SCID)

Clin Immunol. 2021 Aug:229:108778. doi: 10.1016/j.clim.2021.108778. Epub 2021 Jun 16.

Abstract

Introduction: Infants with SCID are treated with hematopoietic cell transplantation (HCT) or gene therapy (GT). Caregiver perceptions of pre-treatment counseling and understanding of durability of HCT/GT are poorly understood.

Methods: A survey was designed and distributed to families of patients with SCID. Topics in the questionnaire included SCID genotype and treatment, family recollections of pre-treatment counseling and present clinical status.

Results: 151 surveys were analyzed. 132 were treated with HCT, 19 with GT. From counseling received, 37% expected HCT/GT would lead to "cure"; 43% expected HCT/GT would last a lifetime. Of 136 living patients, 59% reported overall good health but 65% reported some persistent health challenges.

Conclusions: For some, interpretation of the word "cure" varied, leading to misunderstanding regarding need for continued medical evaluations and additional therapies. Clear communication regarding the importance of lifelong follow-up, no matter the treatment outcome, will help to optimize good health and quality of life.

Keywords: Gene therapy; Genetic counseling; Hematopoietic stem cell transplant; Patient perspective; Patient reported outcomes; Severe Combined Immunodeficiency.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, N.I.H., Intramural

MeSH terms

  • Adolescent
  • Adult
  • Child
  • Child, Preschool
  • Counseling
  • Female
  • Genetic Therapy
  • Health Status
  • Hematopoietic Stem Cell Transplantation
  • Humans
  • Infant
  • Infant, Newborn
  • Male
  • Parents / psychology
  • Pilot Projects
  • Quality of Life
  • Severe Combined Immunodeficiency / genetics
  • Severe Combined Immunodeficiency / immunology
  • Severe Combined Immunodeficiency / therapy*
  • Surveys and Questionnaires
  • Treatment Outcome
  • Young Adult