Defect of cerebellar development and malformation of Purkinje cells in Pex14ΔC/ΔC BL/ICR mice. Defect of cerebellar development and malformation of Purkinje.

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Defect of cerebellar development and malformation of Purkinje cells in Pex14ΔC/ΔC BL/ICR mice. Defect of cerebellar development and malformation of Purkinje cells in Pex14ΔC/ΔC BL/ICR mice. (A) Percentage of pups surviving at postnatal days. The survival days were based on the pups of 22 wild-type (+/+) and 25 Pex14ΔC/ΔC (ΔC/ΔC) BL/ICR mice. (B) Body weights of pups at postnatal days were plotted. (C) Hematoxylin and eosin staining of the sagittal sections of the cerebellum (P7). Arrowheads indicate the shallow cerebellar folia in the cerebellum of the Pex14ΔC/ΔC BL/ICR mouse (lower panel). Scale bar, 500 μm. (D) The sagittal section of the cerebellum at P7 was stained with hematoxylin and eosin. Scale bar, 20 μm. (E) Thickness of EGL was quantified (n = 3). (F, G) Confocal microscopy images of the sagittal sections of the cerebellum at P3 labeled with an antibody to calbindin-D28k, a Purkinje cell marker. Arrows indicate axons of wild-type Purkinje cells and arrowheads indicate swollen axons of Pex14 mutant Purkinje cells. Scale bar, 10 μm. (H) Percentage of swollen axons was quantified (+/+, n = 54; ΔC/ΔC, n = 52). (I) Confocal microscopy images of sagittal sections of the cerebellum at P7 labeled with an antibody to calbindin-D28k are shown. Arrows indicate axons of wild-type Purkinje cells and arrowheads indicate axonal reticular structures of Pex14 mutant Purkinje cells. Scale bar, 10 μm. ***P < 0.001, by t test (E) and χ2 test (H). EGL, external granular layer; IGL, internal granular layer; ML, molecular layer; PCL, Purkinje cell layer. Yuichi Abe et al. LSA 2018;1:e201800062 © 2018 Abe et al.