Henitsoa Rasoanandrianina1,2, Bertrand Audoin1,2,3, Aurélien Massire1,2, Lauriane Pini1,2, Claire Coste1,2, Maxime Guye1,2, Romain Marignier4, and Virginie Callot1,2
1Aix-Marseille Université, CNRS, CRMBM, Marseille, France, 2AP-HM, Hôpital Universitaire Timone, CEMEREM, Marseille, France, 3Department of Neurology, CHU Timone, AP-HM, Marseille, France, 4Department of Neurology, CHU Lyon, Hôpital Pierre Wertheimer, Lyon, France
Synopsis
The temporal pattern of spinal cord (SC) tissue changes in
Neuromyelitis optica spectrum disorder, a rare neuro-inflammatory condition,
was evaluated for the first time using a multi-parametric MR protocol covering the
whole cervical SC (high-resolution T2*-weighted anatomical imaging,
diffusion tensor imaging (DTI), 3D-MP2RAGE T1-mapping, conventional
and inhomogeneous Magnetization Transfer (MT/ihMT) imaging). Results
showed substantial pathological variations of all MR metrics at baseline in
agreement with the presence of a multi-level inflammatory lesion and a temporal
pattern suggesting progressive recovery to control values, with T1
values being the most sensitive to temporal changes and showing residual tissue
destructuration following the myelitis.
Introduction
Neuromyelitis optica spectrum
disorder (NMOSD) is a demyelinating inflammatory disorder driven by an
auto-antibody targeting the astrocytic water channel, aquaporin-4 (AQP4). NMOSD
is mainly affecting the spinal cord (SC) and optic nerves, resulting in functional
deficits often greater than that in multiple sclerosis1. Over the past decade, several cross-sectional
studies have applied both conventional (T1-weighted and T2-weighted)2,3 and non conventional MRI techniques,
particularly DTI and MT4–8 to investigate SC tissue changes in NMOSD
patients. Nonetheless, to our knowledge, no longitudinal studies using such
advanced MRI techniques have been reported. Our aim in this exploratory study was
thus to evaluate the potentiality of a multi-parametric MR protocol at 3T based
on axial 2D multi-slice multi-angle DTI9, MT/ihMT10,11, and high-resolution T2*-weighted
anatomical images, as well as a SC-optimized 3D MP2RAGE10,12 sequence, to investigate the temporal evolution
of SC microstructural tissue changes occurring in NMOSD.Methods
A 74-year-old male patient diagnosed with NMOSD and tested
positive for AQP4-lgG underwent the 3T MR protocol at the time of admission (M0)
and after three (M3), six (M6) and twelve (M12)
months. At each timepoint, the Expanded Disability Status Scale (EDSS)
including 8 functional system sub-scores (pyramidal, cerebellar, brainstem,
sensory, bowel and bladder, visual, cerebral (or mental), and
other)
were evaluated (cf. Table 1). Meanwhile, the
patient underwent plasmapheresis, oral steroids and rituximab.
Five healthy control (HC) subjects (2M/3F, age: 60.2±2.9 [57
- 64] years old) were additionally scanned for reference. The main sequence
parameters for the different acquisitions are presented in Table 2, together with the derived
MR metrics. Mean MR parameters were evaluated at each cervical level
considering the whole SC as a region-of-interest. Z-scores relative to HC were
computed for each MR metric at each cervical level and each timepoint. Results
An inflammatory hyperintense T2-w and gadolinium-enhanced
SC lesion extending from mid-C1 to mid-C5 was observed in
the patient at baseline (M0). It progressively dissolved, remaining gadolinium-enhanced
at M3 and M6 but not at M12. Tiny T2-w
abnormal fragments remained at C3 and C4 at M12.
Figure 1 illustrates the
sagittal T2-w and T1 maps at M0 allowing clear
depiction of the longitudinal extensive transverse myelitis. Slice-by-slice
plots of SC cross-sectional areas (CSA) and T1 values reflecting the
cord swelling and the resulting SC microstructural tissue changes with time are
presented as well. Illustration of the multi-parametric MR metrics, in
particular ADC and T1 maps from the patient’s SC (C1 to C7
levels) at all timepoints are shown in Figure 2 and Figure 3, respectively,
together with reference HC images. Finally, Figure 4 shows the temporal
and spatial evolution of ADC, T1, CSA and MT z-scores within the patient’s
SC.
CSA, ADC and T1 values within the patient’s SC were
found highly increased, and MTR decreased, particularly at the location of the
myelitis. Over the next timepoints (M3, M6 and M12),
CSA, MTR and T1 progressively recovered towards normal values,
whereas ADC exhibited a slight increase at C4 and C5 at M3
before decreasing towards HC values from M6. At M12, ADC
and MT were found in the range of HC values, whereas T1 and CSA
remained higher, particularly at C2-C4 and C4-C7,
respectively.Discussion / Conclusion
Altogether, all MR metrics showed important pathological
variations but T1 and ADC were the metrics of interest for this
AQP4-IgG-positive NMO. Indeed, in the presence of the myelitis, MTR and ihMTR were
substantially influenced by edema and inflammation, and therefore could not be exclusively
used as a myelin marker throughout the follow-ups. Nonetheless, at M12,
the values were in the range of HCs’ suggesting myelin preservation, while T1
values still demonstrate tissue suffering.
MTR, T1 et CSA exhibited similar spatio-temporal
variation dynamics reflecting the cord swelling induced by the myelitis. Afterwards,
all of them showed recovering trends as the myelitis progressively vanishes. ADC,
on the other hand showed different but interesting spatio-temporal trends,
which will be further explored on a larger cohort. Amongst all the explored MR metrics,
T1 was the most sensitive to microstructural tissue changes over times.
M12 exhibited substantial residual T1
increase relative to HC values between C2-C4,
which is possibly linked to the improved but still pathological EDSS score and
remaining pyramidal function deficits.
To summarize, this exploratory study showed the clinical
relevance of a multi-parametric MRI protocol providing different but
complementary insights into the spatial and temporal pattern of SC tissue
impairments in NMOSD patients, hence paving the way for the next coming multi-centric
longitudinal studies. Acknowledgements
No acknowledgement found.References
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