70 but also educational level and employment status influence the severity of fatigue. Bathmen et al. published on fatigue in Marfan syndrome, another hereditary connective tissue disorder. The authors concluded that occurrence of chronic pain and employment status influenced the severity of fatigue 22. Interestingly, a study in children with OI reported a decrease of the level of fatigue after a 12-week individual and supervised physical training program, and increase of the level of fatigue after the program had stopped 10, 23. Studies in other patient groups, including people with Marfan syndrome reported good effects of physical activity on fatigue 24–26. This is important knowledge since some OI patients or parents of OI patients tend to limit their physical activity when they become aware of the inherited bone fragility 23. Some age categories may benefit from an individual and supervised training program. Conclusion In this study the influence of fatigue on daily functioning was investigated in the largest cohort of OI patients to date and compared with control groups in particular a national control group. Although, there were several limitations of our study, based on the current data, there is sufficient evidence for increased severity of fatigue in our cohort of OI patients. An important direction for future research is performing longitudinal measurements using the FSS and exploring determinants of fatigue as this may be of importance for the quality of life in OI patients.
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