Abstract
Context
Central congenital hypothyroidism (CH) requires lifelong medical treatment. The majority of children with central CH have multiple pituitary hormone deficiencies (MPHD), but in some cases central CH is isolated. Most pituitary hormone deficiencies are associated with impaired health-related quality of life (HRQoL). However, studies on HRQoL in central CH are lacking.
Objective
to evaluate HRQoL and fatigue in children and young adults with central CH, as well as parent perspectives.
Design
nationwide cross-sectional study comparing HRQoL between early-detected central CH patients and unaffected siblings with the Pediatric Quality of Life inventory (PedsQL™) and PedsQL Multidimensional Fatigue Scale. Participants ≥8 years old filled in self-reports; parents of participants aged 3-18 years filled in parent-reports. Isolated central CH patients, MPHD patients and siblings were compared using a linear mixed model and Tukey’s post hoc test.
Results
Eighty-eight patients and 52 siblings participated, yielding 98 self-reports and 115 parent-reports. Isolated central CH patients (n=35) and siblings showed similar scores on all subscales, both in the self-reports and parent-reports. For MPHD patients (n=53), self-reported scores were similar to those of siblings. Parent-reported total HRQoL and fatigue scores were significantly poorer in MPHD patients compared with siblings (mean differences -10.2 and -9.4 points; p<0.01), as were scores for physical functioning, social functioning and general fatigue.
Conclusion
Self-reported HRQoL scores in isolated central CH and MPHD patients were similar to siblings. However, parents reported significantly lower HRQoL and fatigue scores for MPHD patients, suggesting a difference in perceived limitations between MPHD patients and their parents.
Differences in Child versus Parent Reports of the Child's Health-Related Quality of Life in Children with Epilepsy and Healthy Siblings