Effect of Angiofibromas on Quality of Life and Access to Care in Tuberous Sclerosis Patients and Their Caregivers

Pediatr Dermatol. 2016 Sep;33(5):518-25. doi: 10.1111/pde.12933. Epub 2016 Jul 19.

Abstract

Background/objectives: Facial angiofibromas (AF) have the potential to cause disfigurement in children with tuberous sclerosis complex (TSC). Facial disfigurement can impact the quality of life (QoL) of individuals and their families, leading to negative psychosocial outcomes. QoL has not been studied in TSC patients with AF.

Methods: We conducted a cross-sectional survey study to investigate QoL of TSC patients with AF and their caregivers and to explore the current state of access to treatment for AF. TSC patients and caregivers in TSC clinic at Boston Children's Hospital and through the Tuberous Sclerosis Alliance were recruited to complete QoL surveys including the CADIS, CDLQI, and Skindex-teen questionnaires, and a survey on access to treatment of AF.

Results: Fifty-eight patients with TSC and 161 caregivers participated in the study. Caregivers of patients with AF had significantly poorer QoL scores compared to caregivers of those without AF, as measured by a modified CADIS questionnaire (mean 31.7 vs. 11.7, p = 0.004). Among patients with AF, those who received treatment had significantly better QoL scores compared with those without treatment, as measured by the CDLQI (mean 3.8 vs. 9.5, p = 0.001). Forty-one and two-tenths percent of subjects reported never receiving treatment for AF. Forty-seven and three-tenths percent of subjects were prescribed topical rapamycin, 47.7% of whom experienced difficulty with insurance coverage.

Conclusions: Presence and lack of treatment of AF significantly impacts QoL in TSC patients and their caregivers. Access to care for AF is limited by multiple factors and should be addressed by clinicians working with this patient population.

Publication types

  • Comparative Study

MeSH terms

  • Adolescent
  • Angiofibroma / diagnosis*
  • Angiofibroma / etiology
  • Angiofibroma / nursing
  • Angiofibroma / psychology
  • Boston
  • Caregivers / psychology*
  • Child
  • Cross-Sectional Studies
  • Disease Management
  • Facial Neoplasms / diagnosis*
  • Facial Neoplasms / etiology
  • Facial Neoplasms / nursing
  • Facial Neoplasms / psychology
  • Female
  • Health Services Accessibility*
  • Hospitals, Pediatric
  • Humans
  • Male
  • Multivariate Analysis
  • Quality of Life*
  • Sickness Impact Profile
  • Statistics, Nonparametric
  • Tuberous Sclerosis / complications*
  • Tuberous Sclerosis / diagnosis