Figure 3
Figure 3. Developmental abnormalities associated with Rps19 deficiency are alleviated with L-leucine treatment. (A-C) Brightfield images of live 2 dpf morphants, lateral view of head and yolk. rps19 morphants treated with control (B) displayed severe cranial (white arrow) and mild cardiac (black arrow) edema, as well as an underdeveloped eye, particularly the lack of a lens (gray arrow) compared with the control morphant (A). Cranial and cardiovascular edema was absent in rps19 morphants treated with L-leucine (white and black arrows, respectively) and eye development was markedly improved, most evident in the lens morphology (gray arrow). (D) Time course of body length measurements in rps19 morphants (red lines) and controls (black lines). L-leucine treated rps19 morphants (solid red line) had a significantly longer body length compared with rps19 morphants treated with control (dashed red line; ***P < .001, 2-way ANOVA). (E) Quantitation of eye defect in rps19 morphants using eye diameter. Filled shapes show eye diameter in embryos raised in egg water, open shapes show eye diameter in embryos raised in egg water with 100mM L-leucine (**P < .0001, 2-way ANOVA).

Developmental abnormalities associated with Rps19 deficiency are alleviated with L-leucine treatment. (A-C) Brightfield images of live 2 dpf morphants, lateral view of head and yolk. rps19 morphants treated with control (B) displayed severe cranial (white arrow) and mild cardiac (black arrow) edema, as well as an underdeveloped eye, particularly the lack of a lens (gray arrow) compared with the control morphant (A). Cranial and cardiovascular edema was absent in rps19 morphants treated with L-leucine (white and black arrows, respectively) and eye development was markedly improved, most evident in the lens morphology (gray arrow). (D) Time course of body length measurements in rps19 morphants (red lines) and controls (black lines). L-leucine treated rps19 morphants (solid red line) had a significantly longer body length compared with rps19 morphants treated with control (dashed red line; ***P < .001, 2-way ANOVA). (E) Quantitation of eye defect in rps19 morphants using eye diameter. Filled shapes show eye diameter in embryos raised in egg water, open shapes show eye diameter in embryos raised in egg water with 100mM L-leucine (**P < .0001, 2-way ANOVA).

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