Bertwin Zhicheng Chen1, Reuben Chee Cheong Soh1,2, Septian Hartono2,3, Chu Ning Ann3, Ming-Ching Wen3, Weiling Lee1, Soo Lee Lim4, Julian Gan5, Yew Long Lo2,3, and Ling Ling Chan1,2
1Singapore General Hospital, Singapore, Singapore, 2Duke-NUS Medical School, Singapore, Singapore, 3National Neuroscience Institute, Singapore, Singapore, 4National Heart Centre Singapore, Singapore, Singapore, 5Siemens Healthineers, Singapore, Singapore
Synopsis
The
aetiopathogenesis of adolescent idiopathic scoliosis (AIS) remains elusive, with
studies suggesting a neurological basis. Using a case control, multimodal approach
incorporating volumetry and diffusion tensor imaging, we characterised brain
MRI changes in AIS. The pontine
volume was significantly larger while left insula was smaller in patients than controls.
Whole brain Tract-Based Spatial Statistics (TBSS) showed no difference between subject
cohorts. Tractography
of the corticoreticular pathway (CRP), which innervates the axial muscles,
showed significant differences in mean and axial diffusivity between the left
and right CRP only in the patient cohort, but no such asymmetry in the control
cohort.
Introduction
Adolescent Idiopathic
Scoliosis (AIS) is a progressive musculoskeletal disease characterised by a
lateral spinal deformity of ≥ 10° affecting up to 4% of adolescents, with females predilection. AIS progression
may lead to severe back pain requiring surgical intervention. Previous studies suggested
a neurological aetiopathogenesis.1 The corticoreticular pathway,
originating from the premotor cortex and terminating at the pontomedullary
reticular formation, innervates axial muscles and may play a role in truncal
muscular tone. In this study, we used a case control, multimodal quantitative approach
incorporating brain volumetry and diffusion tensor tractography (DTT) to characterise
brain and CRP changes on magnetic resonance imaging (MRI) in AIS. Methods
This study was
approved by the local Centralised Institutional Review Board. Sixteen AIS
patients and eighteen healthy controls (HC) were included in the study (Figure 1). A sex-
and age-matched case-control study design was adopted. All participants
underwent brain MRI using a 32-channel head coil on a 3T scanner including T1-weighted
MPRAGE and Diffusion Tensor Imaging (DTI) sequences. The T1-weighted MRPAGE
acquisition was performed with the following parameters: TR/TE/TI=2300/2.88/900
ms, matrix=256x256, resolution=1x1x1 mm3, GRAPPA factor=2, acquisition time=5:12
mins. DTI was performed with simultaneous multi-slice
(SMS) imaging, in conjunction with readout-segmented (RESOLVE) echo-planar
imaging (EPI)2 to reduce scan time and improve the quality of CRP tractography in
the posterior fossa. The following parameters were used: TR/TE=3000/70 ms,
RESOLVE factor=5, SMS factor=3, GRAPPA
factor=2, diffusion directions=64 with b-value=0, 1000 s/mm2,
matrix=128x128, resolution=2x2x2.5 mm3, acquisition time=16:10 mins.
Brain volumetry was
performed on MPRAGE images using the Siemens Morphobox prototype.3 Whole-brain
tract-based spatial statistics (TBSS) was performed on DTI images to assess
local changes in diffusion-based measures (fractional anisotropy-FA, mean,
axial and radial diffusivity-MD, AD and RD, respectively) in voxel-wise analysis.4
DTT of the left and right CRP using deterministic tracking algorithm was also performed
in DSI studio.5
CRP tractography was
performed by two experienced MR technologists under the supervision of a
neuroradiologist as per previously described protocol (Figure 2).6 A seed ROI
was placed on the reticular formation of the medulla, with the first target ROI
on the midbrain tegmentum and second target ROI on the pre-motor cortex. Student’s
t-tests were carried out to compare DTI indices of the CRP between AIS patients
and HC. Intra- and inter-rater reliability were assessed by intraclass
correlation coefficient (ICC). Results
There was no
significant difference between AIS patients and HC with regards to age, gender
composition, body mass index (BMI), handedness, and years of education (Figure 1).
Pons
volume was significantly bigger in AIS patients (1.42 ± 0.12%) compared to HC (1.31 ± 0.10%; p=0.006), whilst
left insula volume was slightly smaller (AIS: 0.55 ± 0.04%, HC: 0.57 ± 0.02%; p=0.041). No significant differences in DTI measures were found between AIS patients
and HC for all white matter tracts in TBSS.
Sample image of CRP vis-à-vis corticospinal tract
(CST) was shown in Figure 3. ICC for DTT of the CRP was excellent (> 0.9)
for both intra and inter-rater reliability. No significant difference was found
for all diffusion indices in both left and right CRP between AIS patients and HC
(Figure 4). However, there was a significant difference in mean (p=0.019) and
axial diffusivity (p=0.013) between the left and right CRP in the patient
cohort (Figure 5). Such CRP asymmetry was absent in the HC cohort (Figure 5).Discussion
Our findings of
relative pontine hypertrophy are consistent with findings from previous studies of a larger brainstem in AIS
patients than HC.7 The primary site of pathology in AIS has been postulated
to reside in the brainstem. Posture, proprioception, and equilibrium control
functions are integrated by structures in and around the brainstem. Abnormality
of the paramedian pontine reticular formation linking preocular motor nuclei
and vestibular nuclei are suspect in AIS patients. Preclinical studies have
also shown that creating lesions in the pons and periaqueductal grey matter
could induce scoliosis.
RESOLVE-DTI reduced geometric distortion from
susceptibility artefacts and improved spatial resolution in the posterior fossa,
affording better visualization of the small brainstem nuclei for accurate ROI placement
in the medullary reticular formation in our study. Our FA and MD values of the
CRP in HC are congruent with those reported in the literature.6,8 The
CRP is responsible for innervation of both axial and proximal muscles required
for gross motor skills such as postural control and locomotion.8 Diffusional
asymmetry between left and right CRP in our AIS cohort are also congruent with findings
of inter-side imbalance from intraoperative transcranial
electrical stimulation studies in AIS patients showing primary, simultaneous asymmetric
descending motor outputs and ascending sensory inputs, as opposed to that in adult
degenerative scoliosis.1 In contrast, whole brain approach using TBSS
analysis showed no differences between AIS patients and HC for all white matter
tracts. Conclusion
We
found relative pontine hypertrophy and asymmetry of diffusion indices in the CRP
in AIS patients, suggesting a primary brainstem origin to the source of
inter-side axial neuromuscular imbalance in AIS. Larger scale neuroimaging
studies could aid in further elucidating the complex aetiopathogenesis in AIS.Acknowledgements
We would like to thank National Neuroscience Institute, Singapore for their funding support.References
- Lo YL, Teo A,
Tan YE, et al. Motor and somatosensory abnormalities are significant etiological
factors for adolescent idiopathic scoliosis. Journal of Neurological Sciences. 2015; 359(1-2):117-123.
- Lim SL, Lee
W, Chan LL, et al. The Many-Sidedness in using RESOLVE. MAGNETOM Flash ASEAN Edition. 2018; 01(7/2018):1-10.
- Schmitter D,
Roche A, Maréchal B, et al. An
evaluation of volume-based morphometry for prediction of mild cognitive
impairment and Alzheimer’s disease. NeuroImage: Clinical. 2015; 7:7-17.
- Smith SM,
Jenkinson M, Johansen-Berg H, et al. Tract-based spatial statistics:
Voxelwise analysis of multi-subject diffusion data. NeuroImage. 2006; 31:1487-1505.
- Yeh F-C,
Verstynen TD, Wang Y, et al. Deterministic diffusion fiber tracking improved by
quantitative anisotropy." PLoS ONE. 2013; 8(11):e80713.
- Yeo SS, Chang
MC, Kwon YH, et al. Corticoreticular pathway in the human brain: Diffusion tensor
tractography study. Neuroscience Letters.
2012; 508(1):9-12.
- Liu T, Chu
WCW, Young, G, et al. MR Analysis of
Regional Brain Volume in Adolescent Idiopathic Scoliosis: Neurological
Manifestation of a Systemic Disease. Journal
of Magnetic Resonance Imaging. 2008; 27(4):732-736.
- Yeo SS, Jang SH and Son SM.
The
different maturation of the corticospinal tract and corticoreticular pathway in
normal brain development: diffusion tensor imaging study. Frontiers in Human Neuroscience. 2014; 8(573):1-6.