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. 2023 Nov 21;101(21):e2103–e2113. doi: 10.1212/WNL.0000000000207878

Figure 2. Recreated Kaplan-Meier Curves for the Probability of Losing the Ability to Sit.

Figure 2

*The pooled curve across all 7 study arms (i.e., Kaneko et al., 2017 [types 2a and 2b], Russman et al., 1996 type 2, and Wadman et al., 2017 [types 2a, 2b, 3a, and 3b]), which is based on a total of 248 patients, is presented (along with the corresponding 95% CI). The follow-up length was variable across study arms, ranging from 1 year (Kaneko et al., 2017 [type 2a SMA]) to 79 years (Wadman et al., 2017 [type 3b]). The median time to no longer sitting in all pooled study arms was not reached. The pooled curve across all 5 study arms (i.e., Kaneko et al., 2017 [types 2a and 2b], Russman et al., 1996 [type 2], and Wadman et al., 2017 [types 2a and 2b]), which is based on a total of 182 patients, is presented (along with the corresponding 95% CI). The median time to no longer sitting based on pooled study arms was estimated to be 31.5 years (95% CI 31.5–NR). The pooled curve across all 4 study arms (i.e., Kaneko et al., 2017 type 2a, Kaneko et al., 2017 type 2b, Russman et al., 1996 type 2, and Wadman et al., 2017 type 2a), which is based on a total of 144 patients, is presented (along with the corresponding 95% CI). The median time for the sitter population based on the pooled curve across all 4 study arms was estimated to be 14.5 years (95% CI 14.1–31.5). In the sensitivity analysis (which excluded Kaneko et al., 2017 [types 2a and 2b SMA]), the median time to no longer sitting based on the pooled curve across the 2 study arms is estimated to be 19.5 years (95% CI 13.5–31.5). Compared with the base-case analysis, in the sensitivity analysis, the median increased by 5 years; however, the pooled curves seemed very similar both with and without the data from Kaneko et al., 2017 (types 2a and 2b SMA). §The pooled curve across both study arms, which is based on a total of 66 patients, is presented (along with the corresponding 95% CI). The data are immature for both type 3a and type 3b SMA, with no events observed in patients with type 3b SMA, and the median time to no longer sitting in type 3 SMA based on pooled study arms was not reached. NR = not reached; SMA = spinal muscular atrophy.