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Zhang et al., 2023 - A zebrafish model of Combined Saposin Deficiency identifies acid sphingomyelinase as a potential therapeutic target

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Fig. 5.

Disrupted mTORC1 signaling does not precede mbpa loss in psap^+63/+63 zebrafish. (A) Illustration of the major promyelinating signaling pathways examined in the current study. (B) Western blots of phosphorylated and unphosphorylated p70S6K and 4E-BP1 in psap^+63/+63 and WT sibling brains, demonstrating disrupted mTORC1 signaling at 4 mpf but no additional changes. Representative data from n=5-14 WT and n=4-14 psap^+63/+63 zebrafish; the signaling status was monitored over three independently propagated generations of fish to yield comparable results. Data for additional pathways illustrated in A are in Fig. S6. (C) Densitometry calculations of p-p70S6K, p70S6K, p-4E-BP1, 4E-BP1, and phosphorylated/non-phosphorylated protein ratios for 1 mpf, 2 mpf and 4 mpf psap^+63/+63 zebrafish and WT siblings. Data points for each dataset were normalized to the average of the WT samples in that set. Data show the mean±s.e.m. Two-tailed unpaired Student's t-test was used. *P<0.05, **P<0.01.

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