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. Author manuscript; available in PMC: 2017 Sep 1.
Published in final edited form as: Mol Genet Metab. 2016 Jun 8;119(1-2):124–130. doi: 10.1016/j.ymgme.2016.06.006

Figure 6.

Figure 6

Dose-dependent correction of brain storage lesions in human IDUA tolerant dogs treated with intrathecal AAV9. Brains were sectioned and stained for LIMP2 and GM3 (A). Meningeal GAG accumulation was imaged using Alcian blue staining. Automated quantification of GM3 (B) and LIMP2 (C) positive cells was performed on cortical brain images (n = 10 per animal). Open symbols indicate animals tolerized with infusion of recombinant human IDUA. GM3 and LIMP2 were significantly reduced in the high dose cohort compared to untreated controls (p = 0.0002 and 0.0026, respectively, Kruskal-Wallis test followed by Dunn's multiple comparisons test).