Fig. 1

Pmpcb deficiency impairs the development of central nervous system (CNS). A Western blot (WB) detection of Pmpcb protein level in wild type (WT) siblings and pmpcb−/− at 72 hpf (A, left), with graphs indicating the relative levels in different groups (A, right). B Phenotypes of WT siblings, pmpcb± and pmpcb−/− mutants at 4 h post fertilization (hpf), 5.3 hpf, and bud stages. Red arrows indicate the shield (5.3 hpf) and the brain domain (bud stages). C, D Hematoxylin & Eosin (HE) staining showed the cellular difference in mesencephalon (Me) of WT siblings and pmpcb−/− larvae at 72 hpf (C), with the calculation data (in black box and red box) (D). E Transmission Electron Microscope (TEM) analysis of the myelin structure in CNS in pmpcb−/− mutants at 72 hpf. The structure highlighted in red color is the axon. Red arrows indicate myelin sheath. a: axon, m: myelin sheath. F Behavioral assays in WT siblings, pmpcb± and pmpcb.−/− larvae at 96 and 120 hpf. B, lateral view, anterior to the up. Each experiment was repeated at least three times, and a representative result is shown. Data are analyzed using GraphPad Prism 9.5 for t-test, and are presented as mean ± SD. ***P < 0.001, **P < 0.01. Scale bar, 500 μm (B), 10 μm (C), 1 μm (E)