Abstract
Introduction The diagnosis of a rare congenital malformation with necessary surgical treatment
is an emotionally stressful event in a parent's life and may impact parental health-related
quality of life (HrQoL). We aimed to review the literature on HrQoL in parents and
caregivers of pediatric surgical patients with rare congenital malformations and summarize
the results.
Materials and Methods For this scoping review, a literature search in PubMed was conducted from inception
to November 21, 2019. Inclusion and exclusion criteria to select articles were defined
a priori.
Results Four articles (published 2014–2018) describing HrQoL in parents of pediatric surgical
patients with rare congenital malformations in comparison to population-based references
or healthy control groups were identified. Only European studies were found, and sample
sizes varied between 15 and 87 participants. Results for parental HrQoL yielded inconsistencies.
Conclusion So far, only a few studies with small sample sizes focus on parental HrQoL in pediatric
surgical patients with rare congenital malformations. To gain a comprehensive understanding
of the impact of a rare congenital malformation on HrQoL of all family members, it
is necessary to consider three main aspects: (1) to use both generic- and disease-specific
instruments measuring HrQoL of the young patient as well as his/her parents and siblings
in larger sample sizes, (2) to collect longitudinal data, and (3) to consider a mixed-method
approach.
Keywords health-related quality of life - parents - rare diseases - congenital malformations
- surgical treatment
