MRI analysis of cerebellar and vestibular developmental phenotypes in Gbx2 conditional knockout mice

Gbx2条件性敲除小鼠小脑和前庭发育表型的MRI分析

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作者:Kamila U Szulc, Brian J Nieman, Edward J Houston, Benjamin B Bartelle, Jason P Lerch, Alexandra L Joyner, Daniel H Turnbull

Conclusion

These results show the potential of three-dimensional MRI for detecting and analyzing developmental defects in mouse models of neurodevelopmental diseases.

Methods

In vivo three-dimensional manganese-enhanced MRI at 100-µm isotropic resolution was used to visualize mouse brains between postnatal days 3 and 11, when cerebellum morphology undergoes dramatic changes. Deformation-based morphometry and volumetric analysis of manganese-enhanced MRI images were used to, respectively, detect and quantify morphological phenotypes in Gbx2-CKO mice. Ex vivo micro-MRI was performed after perfusion-fixation with supplemented gadolinium for higher resolution (50-µm) analysis.

Purpose

Our aim in this study was to apply three-dimensional MRI

Results

In vivo manganese-enhanced MRI and deformation-based morphometry correctly identified known cerebellar defects in Gbx2-CKO mice, and novel phenotypes were discovered in the deep cerebellar nuclei and the vestibulo-cerebellum, both validated using histology. Ex vivo micro-MRI revealed subtle phenotypes in both the vestibulo-cerebellum and the vestibulo-cochlear organ, providing an interesting example of complementary phenotypes in a sensory organ and its associated brain region.

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