Koert Gooijer

36 A total of 190 (59%) of the 322 patients with OI in our cohort were women; 132 (41%) were men. The mean and median age of participants with OI at the first visit were, respectively, 38 and 35.5 years (interquartile range (IQR) 27 years). Skewness and kurtosis were, respectively, 0.343 and −1.119, confirming a normal distribution with a small overrepresentation of the middle group 12. There were 220 (66.7%) subjects who had a diagnosis of OI type 1, 40 (12.1%) were diagnosed with OI type 3, and 61 patients (18.5%) had OI type 4. Scores of all patients with OI across eight different SF-36 subscales Figure 1 shows the results of the eight different SF-36 subscales of the three OI types in comparison with two control groups in the Netherlands 5,11. Individuals with OI type 1, 3 and 4 had a significantly lower mean physical function score compared with the control groups (Table 1) 5,11. A significant difference between patients with OI and controls applied to all the subscales except for vitality in OI type 3, role limitations caused by emotional problems in OI types 3 and 4, and mental health in OI types 3 and 4 (Table 1). Comparison of SF-36 subscale scores in different age categories A complete overview of the results is available in the Supplementary Table S2. The comparisons have been made with the first control group 11 because in this control group participants were divided in age categories, suitable for making agematched comparisons. Physical functioning Physical function in the overall OI cohort was significantly lower compared with controls 5,11 in all different age categories, except for patients with OI type 4 and aged >55 years (Table 1 and Supplementary Table S2) 11. Individuals with OI type 3 had the lowest score on physical function, (Table 1), whereas individuals with OI type 1 in the age group 18 to 24 years had the highest score on physical function. The physical function of OI type 3 was significantly lower than OI type 1 and 4 in all age categories except when compared with OI type 4 in the age group 35 to 54 years (Supplementary Table S2). The physical function of individuals with OI types 1 and 4 in the different age categories were not significantly different from each other, except in the age category 18 to 24 years (Supplementary Table S2). In the control group 11, physical function declined during at least 30 years with 27.9 points (Δ; see Patients and Method section). The OI type 1 group followed that trend (Δ −21.72, p < 0.05), whereas patients with OI type 4 showed a climbing trend (Δ +18.7, p = 0.106), implying that several individ-

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