Stefan Paul Koch1,2, Felix Knab1, Ulrike Grittner1, René Bernard1, Susanne Mueller1,2, Marco Foddis1, Janet Lips1, Philipp Euskirchen1, Norman Zerbe3, Peter Hufnagl3, André Rex1, Ulrich Dirnagl1, Tracy Deanne Farr4, Christoph Harms1, and Philipp Boehm-Sturm1,2
1Department of Experimental Neurology and Center for Stroke Research Berlin, Charité – Universitätsmedizin Berlin, corporate member of Freie Universität Berlin, Humboldt-Universität zu Berlin, and Berlin Institute of Health, Berlin, Germany, 2Cluster of Excellence NeuroCure and Charité Core Facility 7T Experimental MRIs, Charité - Universitätsmedizin Berlin, Berlin, Germany, 3Institute of Pathology, Charité – Universitätsmedizin Berlin, Berlin, Germany, 4School of Life Sciences, University of Nottingham, Nottingham, United Kingdom
Synopsis
Post-stroke functional
recovery remains a poorly understood process. We aimed to identify anatomical
regions that statistically explain a deficit in the staircase test after stroke
in the mouse using two animal models (MCAO, photothrombosis). MATLAB toolboxes are
presented for co-registration of MRI and histology data to the Allen brain
atlas and implementation of atlas region-wise or voxel-wise mapping of behavioral deficits. Automatic techniques are shown for validation of MRI results on stained tissue
sections.
Introduction
Post-stroke functional
recovery remains a poorly understood process. Here, we introduce techniques adapted from voxel-based lesion symptom mapping1 in order to identify
anatomical regions whose damage statistically explain a functional deficit
and thus would be of highest interest for a targeted therapeutic intervention. To this end,
we present custom MATLAB toolboxes for Allen atlas registration of MRI and
histology data allowing brain region-based and voxel-based correlation of behavioral
deficits to tissue damage after stroke in the mouse.Methods
C57/Bl6 mice (n=60) were
trained in the staircase test2 and randomized to undergo either 45 min
middle cerebral artery occlusion (MCAO, final n=17), sham surgery (n=17) or photothrombosis
(PT, n=9). n=13 (MCAO) died before endpoint, n=1 (MCAO) did not learn the task, and n=1/1 (MCAO/PT) did not have a lesion. Experiments and analysis were performed blinded to the
condition of animals. 24 h after surgery, animals underwent T2-weighted (T2w) MRI (RARE, 32
contiguous 0.5 mm thick slices, FOV=(25.6 mm)2, matrix=256x196
zerofilled to 2562, TR/TEeff=4.2 s/36 ms, RARE factor 4,4 averages, TA=6:43 min) at 7 T (Bruker BioSpin, Ettlingen, Germany) using a 20 mm diameter Tx/Rx volume resonator coil (RAPID, Rimpar, Germany). A lesion mask was drawn by an
experienced researcher and images+masks were warped to the Allen-Brain-Atlas
using our publicly available MATLAB toolbox ANTX3 (Fig. 1). We correlated the
degree of initial deficit (mean performance 2-6 d post surgery) i) region-wise with percent lesion volume in atlas regions or ii) voxel-wise with image intensity
in the warped T2w image. 21 d post surgery, animals were sacrificed for histological validation. We performed DAB-enhanced NeuN staining on 20 µm thick slices (400 µm slice distance),
center of mass of NeuN nuclei were automatically identified using Cell
Profiler (http://cellprofiler.org/), and corresponding slices of the Allen brain atlas
were automatically determined and registered to each histological slice using a
custom ELASTIX-based (http://elastix.isi.uu.nl/) MATLAB toolbox
(Fig. 2).Results
MCAO animals had the largest difference in performance in the staircase test compared to shams (mean difference: 41.2%,
(30.7-51.7%), p<0.001) whereas the deficit was smaller for PT animals (mean
difference: 19.9% (6.8-33.0%), p=0.0049). We found a trivial correlation of initial deficit and total stroke
volume on T2w images at 24 h (R=0.73). However, region-based and voxel-based symptom mapping revealed similarly high correlation in very specific areas involved in somatosensory and fear processing. Damage in more caudal structures (e.g. thalamus and midbrain) also correlated well with the deficit (Fig. 4).Discussion
We developed a set of fully automated tools for data-driven analysis of anatomical substrates of functional recovery after stroke in the mouse. From the fact that lesions in MCAO animals are mainly subcortical and these animals performed worse than PT animals (primarily cortical damage), we conclude that the subcortex is important to perform well in a behavioral test of somatosensory and motor function. Region-based and voxel-based symptom mapping was used to explore and quantify the importance of anatomical lesion location, a comprehensive validation of these findings through correlation with neuronal cell densities is work in progress but the necessary tools have been established. One limitation of our study is the exclusion of animals that underwent MRI but died before the final endpoint, a systematic strategy to statistically deal with this cohort is work in progress. Another limitation is that correlation of a behavioral deficit with damage, e.g. in more caudal brain regions, may simply be due to effects related to overall lesion volume and not to loss of the specific function of the region. Therefore, in order to gain a more mechanistic insight, we aim to target most prominent candidates with pharmacogenetic excitation/inhibition in future studies.Conclusion
Atlas-based and voxel-based symptom mapping in the mouse is a powerful tool to explore mechanisms of functional recovery after stroke.Acknowledgements
Work was supported by the Federal Ministry of
Education and Research (BMBF) (grant number 01EO0801, Center for Stroke
Research Berlin) and Deutsche Forschungsgemeinschaft (DFG) (Excellence Cluster
NeuroCure).References
1 Bates E,
Wilson SM, Saygin AP, Dick F, Sereno MI, Knight RT et al. Voxel-based
lesion-symptom mapping. Nat Neurosci 2003; 6: 448–50.
2 Emmrich J V,
Neher JJ, Boehm-Sturm P, Endres M, Dirnagl U, Harms C. Stage 1 Registered
Report: Effect of deficient phagocytosis on neuronal survival and neurological
outcome after temporary middle cerebral artery occlusion (tMCAo) [version 1;
referees: 2 approved]. F1000Research 2017; 6.
doi:10.12688/f1000research.12537.1.
3 Koch S,
Mueller S, Foddis M, Bienert T, von Elverfeldt D, Knab F et al. Atlas
registration for edema-corrected MRI lesion volume in mouse stroke models. J Cereb
Blood Flow Metab 2017; : 0271678X1772663. https://github.com/philippboehmsturm/antx