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. 2019 Aug 16;30(8):957–966. doi: 10.1089/hum.2019.012

Figure 2.

Figure 2.

Sustained increased CSF IDUA activity in tolerized rhesus monkeys after a single AAV9.hIDUA intrathecal (i.t.) injection at 4 weeks of age. (A) A 4-methylumbelliferone-based artificial substrate assay was used to measure IDUA activity in CSF samples from monkeys aged 4 weeks to 3 years and 9 months. Results are expressed as nanomoles of fluorescent product released per milliliter every 3 h (the detection limit was 0.1 IU/mL/3 h). The gray area between the dotted lines represents the average ±1 SD of IDUA activity obtained at baseline in the CSF of 26 rhesus monkeys, including the pre-vector samples from infants (N = 4) and juveniles/adults unrelated to the study (N = 22). (B) ELISA measuring anti-hIDUA antibody levels in CSF (dilution 1:20). (C) ELISA measuring anti-hIDUA antibody levels in serum (dilution 1:1,000). CSF, cerebrospinal fluid; ELISA, enzyme-linked immunosorbent assay.