Govind Nair1, Shila Azodi1, Tsemacha Dubuche1, Yair Mina1, Ikesinachi Osuorah1, Joan Ohayon1, Tianxia Wu1, Daniel S Reich1, and Steve Jacobson1
1National Institutes of Health, Bethesda, MD, United States
Synopsis
We sought to
better understand the relationship between atrophy along the entire spinal cord
and disease burden in multiple sclerosis using MRI. Towards this, we analyzed spinal
cord cross-sectional area in 48 healthy control and 250+ subjects clinically
diagnosed with various phenotypes of multiple sclerosis. Our results show cervical
cord atrophy early in the onset of the disease, which correlated with clinical
measures of disease severity. However, these correlations were reduced as the
disease progressed. Such studies may help in better understanding of disease
progression and can play a role as an imaging marker in clinical trials.
Introduction
Atrophy in the spinal
cord contributes to disability in patients with multiple sclerosis (MS).1,2 Its quantification from the entire spinal cord may be important to fully
understand the diseases.3 We compared spinal cord cross-sectional area (SCCSA)
along the entire cord in various MS phenotypes (primary progressive or PP;
relapsing remitting or RR; secondary progressive or SP) and healthy control
subjects (HC), and investigated correlation of SCCSA at various levels with
clinical markers of disability in MS.Methods
HC and
subjects clinically diagnosed with MS (by the 2010 McDonald criteria) underwent
MRI of the spinal cord (3T Siemens Skyra with spine matrix coil) which included
a 3D GRE sequence at 1mm isotropic resolution of the cervical (C)- and thoracic
(T)-spine (TR=8ms, TE=3ms, FA=18°). The C- and T-spine images were stitched
together using DICOM information, and SCCSA from C1-T10 levels were derived
using a semi-automatic method in Matlab® as previously described4,5 (Figure 1). The group-averaged SCCSA plots and average SCCSA from C2-3, C4-5,
and T4-10 regions (regional SCCSA) were compared between MS subtypes, and
correlated with standard clinical scores such as Expanded Disability Status
Scale (EDSS),6 Scripps Neurologic Rating Scale (SNRS)7, time to walk 25-feet (25FtW), and time to complete 9-hole peg test (9HPT), as
well as disease duration. MS cohorts were further subdivided based on z-scores of
regional SCCSA derived from the HC group (non-atrophic cord: z>=-1.5 and
atrophic cord: z<-1.5). Statistical analysis included analysis of variance, Tukey’s
multiple comparison, Pearson’s or Spearman's correlations, and analysis of covariance.
A p<0.05 was considered to be statistically significant.Results
The study
included 149 RRMS (age 44 ± 12 years, 70% female), 49 SPMS (age 55 ± 11 years,
65% female), 58 PPMS (age 54 ± 11 years, 55% female), and 48 HC subjects (age
45 ± 14 years, 55% female). Detailed demographic and clinical information for
the cohort is shown in Table 1. Compared to HC group, all MS groups had smaller
regional SCCSA except RRMS in the T4-10 region (Figure 2). Among the MS
cohorts, SPMS and PPMS (progressive forms of MS) had smaller SCCSA in all
regions compared to RRMS. Regional SCCSA did not differ significantly among the
progressive MS cohorts (Figure 2).
SCCSA
measures from all regions of the RRMS cohort were significantly correlated to
all clinical measures explored herein (|r| between 0.18 and 0.35, Table 2).
SCSSA from cervical regions of the progressive MS cohorts were significantly
correlated with EDSS, SNRS, 25FtW, and 9HPT (|r| between 0.32 and 0.45) except
with SCCSA from C4-5 region and 25FtW in the SPMS cohort. SCCSA from T-regions
explained about 10% of the variance in SNRS in SPMS cohorts and in 25FtW in
PPMS cohort. Disease duration was significantly correlated with SCCSA only in
the RRMS cohort, but this correlation was seen in all C- and T-regions.
Within the
RRMS cohort, C-regions were atrophic in 29 (19%) and T-region was atrophic in
18 subjects (12%), and those with atrophic cord had a significantly higher
disease burden and longer disease duration as compared to those having non-atrophic (Figure 3, top row), while these differences were
less severe in the SPMS cohort (Figure 3, bottom row). Finally, RRMS subjects
with atrophic cord had significantly higher magnitude of correlation with
clinical parameters such as SNRS and 9HPT compared to RRMS subjects with non-atrophic cord.Discussion and Conclusion
SCCSA profile
comparisons between HC, RRMS, and SPMS subjects in this cross-sectional study
reveals c-spine atrophy early in the disease process, which extends to thoracic
regions as disease progresses. Regional SCCSA were correlated with various
clinical measures of disability in all MS phenotypes. However, SCCSA measures
from RRMS phenotype had the stronger and broader correlations with clinical
measures of disability. Subgroup analysis of RRMS cohort based on z-scores
revealed that the subset of patients with atrophic-looking cord had a longer
disease duration and were more clinically disabled than subjects with non-atrophic cord. The results from this cross-sectional study suggest that cord
atrophy starts proximally and early in the disease, extending distally with
disease progression. As the disease progresses, this atrophy may decrease,
and the correlation with clinical measures drop off suggesting a floor-effect
for atrophy. It should be noted that the results could also be biased by the
limits of our imaging technique, specifically the resolution, to pick up small volume changes in a thin cord compared to a thick cord. Studying SCCSA patterns,
especially in a longitudinal setting, may help in better understanding of the
progression in the spinal cord. This study will be expanded to include
multivariable analysis, longitudinal observation of SCCSA changes in relation in
the context of progressive symptoms, as well as CSF immunological studies.Acknowledgements
Research
was funded by the Intramural Research Program at the National Institute of
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