Fig. 6
- ID
- ZDB-FIG-231212-74
- Publication
- Liu et al., 2023 - Identification of an α-l-iduronidase (IDUA) M1T mutation in a Chinese family with autosomal recessive mucopolysaccharidosis I
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Depletion of IDUA in zebrafish results in multiple developmental defects. (A) Representative stereomicroscopy images of control-MO, IDUA-MO, and IDUA-MO + WT-IDUA mRNA zebrafish larvae at 3 dpf. IDUA-MO zebrafish larvae had protracted jaws (red arrow), enlarged hearts, and pericardial edema (black arrow). Introduction of WT-IDUA mRNA in the morphant zebrafish fully rescued these phenotypes. (B) The enzymatic activities of IDUA in each group were assayed. IDUA enzymatic activity in the control group was normalized to 1, the enzyme activity from IDUA-MO and IDUA-MO + WT-IDUA mRNA zebrafish embryos was presented as fold induction above this level. Note: Data are representative of three individual experiments. (C) Quantitative analysis of the whole-body length of each group. As measured in millimeters (n = 15 per condition; Student's t-test; ***p<0.001; data are representative of three individual experiments). (D) Alizarin red bone staining of 5 dpf control-MO, IDUA-MO, and IDUA-MO + WT-IDUA mRNA zebrafish larvae. (E) Sagittal section images of hematoxylin and eosin-stained tissue from 5 dpf control-MO, IDUA-MO, and IDUA-MO + WT-IDUA mRNA zebrafish larvae. Abbreviations: C, cleithrum; IDUA, α-l-iduronidase; MO, morpholino; OP, operculum; OT, otoliths; WT, wild type. Student's t-test; *p<0.05, ***p<0.001. |