The online version of this article (https://doi.org/10.1186/s12891-017-1922-5) contains supplementary material, which is available to authorized users.
Persisting fatigue has been reported to be a common complaint by individuals with connective tissue disorders, including Osteogenesis imperfecta (OI). This controlled study evaluated in an adult OI population the subjective experience of fatigue, affecting daily life. Sleep disturbances and chronic pain were examined as hypothesized underlying factors.
This cross-sectional study analyzed the answers of 56 OI patients and 56 matched healthy controls to a questionnaire, designed to evaluate levels of experienced fatigue and bodily pain, as well as the presence or absence of symptoms related to sleep disturbances or sleep apnea. The relationships between fatigue, pain, and sleep disturbances were evaluated with correlation analysis and regression analysis.
Fatigue was reported by 96%, and daily pain by 87% of the individuals with OI. Notably, the level of fatigue was similarly experienced by patient respondents and controls. In total, 95% of the patients and 77% of the controls reported one to several sleep disturbance symptoms. These symptoms as well as previously diagnosed sleep apnea were statistically significantly more prevalent in the patient group than in the controls (p < 0.05). Likewise, the experienced bodily pain was statistically highly significantly more severe among the respondents with OI (p < 0.001), and correlated with the reported fatigue.
In comparison with age-matched controls, adults with OI do not differ in experienced fatigue, unlike hypothesized. Therefore, sleep disturbances, which based on the frequency of reported related symptoms and previous sleep apnea diagnoses appear to be common in OI patients, may remain undiagnosed.
Additional file 1: Fatigue and disturbances of sleep in patients with Osteogenesis imperfecta –survey questionnaire. (DOCX 102 kb)12891_2017_1922_MOESM1_ESM.docx
Sillence DO, Rimoin DL, Danks DM. Clinical variability in osteogenesis imperfecta-variable expressivity or genetic heterogeneity. Birth Defects. 1979;15:113–29. PubMed
Thomas IH, DiMeglio LA. Advances in the classification and treatment of osteogenesis imperfecta. Curr Osteopor Rep. 2016;14(1):1–9. CrossRef
Hill CL, Baird WO, Walters SJ. Quality of life in children and adolescents with osteogenesis imperfecta: a qualitative interview based study. Health Qual Life Out. 2014;12:54. CrossRef
Vanz AP, Félix TM, Rocha NS, Schwartz IVD. Quality of life in caregivers of children and adolescents with osteogenesis imperfecta. Health Qual Life Out. 2015;13:41. CrossRef
Borodulin K, Levälahti E, Saarikoski L, Lund L, Juolevi A, Grönholm M, et al. National Finriski health study. National Institute for Health and Welfare. Report. 2013;022:2.
Scheper MC, Juul-Kristensen B, Rombaut L, Rameckers EA, Verbunt J, Engelbert RH. Disability in adolescents and adults diagnosed with hypermobility related disorders: a meta-analysis. Arch Phys Med Rehab. 2016;97:2174–87. CrossRef
Pouliot-Laforte A, Veilleux L-N, Rauch F, Lemay M. Physical activity in youth with osteogenesis imperfecta type I. J Musculoskel Neuron Interact. 2015;15(2):171–6.
Morphy H, Dunn KM, Lewis M, Boardman HF, Croft PR. Epidemiology of insomnia: a longitudinal study in a UK population. Sleep. 2007;30(3):274–80. PubMed
Vgontzas AN, Bixler EO, Chrousos GP. Obesity-related sleepiness and fatigue: the role of the stress system and cytokines. Ann N.Y. Acad Sci. 2006;1083:329–44. CrossRef
- Fatigue and disturbances of sleep in patients with osteogenesis imperfecta – a cross-sectional questionnaire study
- BioMed Central
Neu im Fachgebiet Orthopädie und Unfallchirurgie
e.Med Kampagnen-Visual, Mail Icon II