ArticleMagnetic resonance in medicine2013
MRI analysis of cerebellar and vestibular developmental phenotypes in Gbx2 conditional knockout mice.
Article in Magnetic resonance in medicine, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 9 papers.
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Who cites it
9 citing papers in PubMed, 15 citations in OpenAlex.
- Cell specificity of Manganese-enhanced MRI signal in the cerebellum.NeuroImage · 2023Article
- Article
- Calcium-dependent molecular fMRI using a magnetic nanosensor.Nature nanotechnology · 2018Article
- An inactivating mutation in the histone deacetylase SIRT6 causes human perinatal lethality.Genes & development · 2018Article
- Multimodal Genetic Approach for Molecular Imaging of Vasculature in a Mouse Model of Melanoma.Molecular imaging and biology · 2017Article
- Consensus Paper: Cerebellar Development.Cerebellum (London, England) · 2016Review
- 4D MEMRI atlas of neonatal FVB/N mouse brain development.NeuroImage · 2015Article
- In vivo Mn-enhanced MRI for early tumor detection and growth rate analysis in a mouse medulloblastoma model.Neoplasia (New York, N.Y.) · 2014Article
- Pydpiper: a flexible toolkit for constructing novel registration pipelines.Frontiers in neuroinformatics · 2014Article
Corrections and comments
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Authors and funding
7 authors at 3 institutions in 2 countries.
Funding
Abstract
purposeOur aim in this study was to apply three-dimensional MRI methods to analyze early postnatal morphological phenotypes in a Gbx2 conditional knockout (Gbx2-CKO) mouse that has variable midline deletions in the central cerebellum, reminiscent of many human cerebellar hypoplasia syndromes.
methodsIn 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.
resultsIn 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.
conclusionThese results show the potential of three-dimensional MRI for detecting and analyzing developmental defects in mouse models of neurodevelopmental diseases.
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