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. 2017 Feb 9;100(3):537–545. doi: 10.1016/j.ajhg.2017.01.019

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© 2017 The Author(s)

This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

PMC Copyright notice

Knockdown of inpp5ka in Zebrafish Causes Defective Eye Development and Muscle Formation

(A) General morphological abnormalities were observed in inpp5ka morphant embryos at 4 dpf, compared to control morpholino (MO) or uninjected control age-matched embryos. Scale bar represents 500 μm.

(B) Aberrant eye development was observed in inpp5ka morphant wax sections stained with H&E; eyes were observed reduced in size and downwardly oriented. Scale bar represents 200 μm.

(C) Structural analysis of the lens, by staining for α-crystallin, identifies disorganization in inpp5ka morphant embryos. In addition, abnormal medial nuclei retention was observed in inpp5ka morphant lens, as marked by DNA stain Hoechst. Scale bar represents 50 μm.

(D) Birefringence of inpp5ka morphant embryos reveals decreased muscle integrity compared to uninjected or control MO-injected embryos at 4 dpf. Scale bar represents 200 μm.

(E) Analysis of muscle fiber and neuromuscular junction formation using Phalloidin (Red, F-actin) and alpha-Bungarotoxin (Green, AChR), respectively, reveals misaligned myofibers and reduced NMJ arborization in morphants. Scale bar represents 100 μm.