Alexandra Petiet1,2, Gonçalo C Vilhais-Neto3,4, Daniel Garcia-Lorenzo1, Stéphane Lehéricy1,2, and Olivier Pourquié3,4,5,6,7
1Center for Neuroimaging Research, Brain and Spine Institute, Paris, France, 2UPMC/Inserm UMRS1127 / CNRS UMR7225, Paris, France, 3Institut de Génétique et de Biologie Moléculaire et Cellulaire, Illkirch, France, 4Stowers Institute for Medical Research, Kansas City, MO, United States, 5Howard Hughes Medical Institute, Kansas City, MO, United States, 6Department of Anatomy and Cell Biology, University of Kansas Medical Center, Kansas City, MO, United States, 7Department of Genetics, Harvard Medical School and Department of Pathology, Brigham and Woman’s Hospital, Boston, MA, United States
Synopsis
While
humans show clear preference for right hand usage (90%), normal mice show
consistent right or left paw usage (50%). We used a Rere-deficient mouse model
(Rere+/om) that shows
clear right paw usage preference (80%) compared to wild-type (WT) mice (40%) to
evaluate structural connectivity changes in the cortico-spinal tract (CST)
using diffusion-based tractography. Our results showed significantly reduced
and more asymmetric FA along the CST of the dominant hemisphere in the dextral mutant
group compared to the WT group. These
results show Rere-dependent structural connectivity changes in the brain that
could be clinically relevant to human pathologies.Purpose
To study brain asymmetries in a Rere (Atrophin2)-deficient mouse model
1
using diffusion MRI-based tractography. The Rere gene affects the symmetry of vertebral precursors
2. Unlike humans who show clear preference for
right hand usage (90%), normal mice show consistent right or left paw usage
(50%). The Rere mouse model (
Rere+/om mutants,
Vilhais-Neto et al., submitted) shows
clear right paw usage preference (80%) compared to wild-type mice (WT, 40%) in
Mouse Reaching and Grasping (MoRaG) tests. In this study we aimed at evaluating
structural connectivity changes in the cortico-spinal tract (CST) of Rere-mutated
animals using diffusion-based tractography. We hypothesized that the Rere gene
had an effect on the symmetry of the CST in Rere-deficient animals.
Methods
A total of 44 mice (
Rere+/om:
N=22 and WT: N=22) were imaged after analysis with the MoRaG for paw usage (11-15
months old). All images were acquired on a Bruker Biospec 117/16 USR MRI system
(BGA-9S gradients, 750 mT/m, AVIII) running Paravision 5.1. A 72-mm resonator was
used for signal emission and a planar surface coil for mouse head was used for
signal reception (Bruker Biospin, Ettlingen, Germany). Structural T
2-weighted
images were acquired for all mice. A multi-slice turbo-RARE sequence was used
with TR=6500ms; TE=40ms; matrix=384x384; field-of-view(FOV)=23x23mm
2 (60x60μm
2 in-plane
resolution); 72 slices; slice thickness=0.22mm; number of excitations=4; scan
time=2h8min. After first and second order shimming with FASTMAP,
respiratory-gated diffusion-weighted images were acquired for all mice. A 3D
respiratory-gated EPI sequence was used with TR=1000ms; TE=25ms;
matrix=128x96x32; FOV=20x15x16mm
3 (resolution=156x156x500μm
3);
126 directions; b-value=1000s/mm
2; scan time=1h13min. A B
0
fieldmap was also acquired and used for distortion correction of the diffusion
data. A total of 12 sinistral WT mice (WT L-paw); 8 dextral WT mice (WT R-paw);
and 18 dextral
Rere+/om mutant
mice (
Rere+/om R-paw) were
included in the analyses. The
diffusion acquisitions were preprocessed with FSL software
3. We
corrected for motion with the Eddycor FSL function and we then used a fieldmap
acquisition to correct the EPI geometric distortions. The corrected mean B
0
volumes were then registered to the structural T
2 volumes. We
finally performed the diffusion tensor estimation (with FSL dtifit function) to
produce fractional anisotropy (FA) volumes for each mouse. The fiber-tracking analysis was performed with the MRtrix package (http://www.brain.org.au/software/). For the
corticospinal tract (CST) reconstruction, six mask ROIs were manually drawn in
the left and right motor cortex, internal capsule, and pyramidal tracts of the
normalized images, according to the Paxinos and Franklin mouse brain atlas
4
and with the MRView viewing tool. The ROIs were then denormalized and the
tracts were reconstructed from the native images using spherical deconvolution
and probabilistic tractography (Figure 1). Along-fiber FA analysis was performed similarly to the one presented
in Colby et al.
5. Each fiber was divided into 50 equidistant points;
the division was performed using the predefined ROIs to provide good alignment
across subjects. Measures were evaluated using a mixed-effects model to take
into account local FA and global subject effects. Statistical
unpaired t-tests were run for between-group comparisons.
Results
The
comparison between all groups (
Rere+/om
vs WT L-paw;
Rere+/om vs
WT R-paw;
Rere+/om vs WT
all; WT R-paw vs WT L-paw) did not show any global differences in FA along the
CST. Only fine local differences were found with reduced FA in the dominant
tract (left hemisphere) of the
Rere+/om
as compared with the WT R-paw (FA(WT-Rpaw)>FA(
Rere+/om), p<0.00005, Figure 2). The
Rere+/om group was found to
be locally more asymmetric than both the WT R-paw and L-paw groups (p<0.0005,
Figure 3).
Conclusion
The dextral
Rere+/om mutant
group showed reduced and more asymmetric FA in the dominant CST compared to the
matched dextral and sinistral WT groups. These results show Rere-dependent structural
connectivity changes in the brain that could be clinically relevant to human pathologies.
Acknowledgements
This
work was supported by the Howard Hughes Medical Institute, the Stowers
Institute for Medical Research, the European Research Council and the “Investissements
d'Avenir”, IHU-A-ICM, Paris Institute of
Translational neuroscience ANR-10-IAIHU-06.References
1. Zoltewicz
JS, Stewart NJ, Leung R et al. Atrophin 2 recruits histone deacetylase and
is required for the function of multiple signaling centers during mouse
embryogenesis. Development
2004;131(1):3-14.
2. Vilhais-Neto
GC, Maruhashi M, Smith KT et al. Rere controls retinoic
acid signalling and somite bilateral symmetry. Nature 2010;
463(7283):953-7. 3. Smith SM, Jenkinson M, Woolrich MW et al. Advances in functional and
structural MR image analysis and implementation as FSL. NeuroImage 2004;
23(S1):208-19. 3. “The
mouse brain in stereotaxic coordinates”, Paxinos G and Franklin KB, 2nd
Edition, Academic Press 2001. 4. Colby JB, Soderberg L, Lebel C et al.
Along-tract statistics allow for enhanced tractography analysis. Neuroimage
2012; 59(4):3227-42.