Chao Li1,2,3, Shuo Wang3,4, Pan Liu3, Bart RJ van Dijken5, Carola-Bibiane Schönlieb3, and Stephen John Price1
1Department of Clinical Neurosciences, University of Cambridge, Cambridge, United Kingdom, 2Department of Neurosurgery, Shanghai Jiao Tong University School of Medicine, Shanghai, United Kingdom, 3The Centre for Mathematical Imaging in Healthcare, Department of Pure Mathematics and Mathematical Statistics, University of Cambridge, Cambridge, United Kingdom, 4Department of Radiology, University of Cambridge, Cambridge, United Kingdom, 5Radiology, Medical Imaging Center, University Medical Center Groningen, Groningen, Netherlands
Synopsis
The purpose of this study was to interrogate
the inter-dependence of perfusion and diffusion imaging, and further
investigate the clinical relevance of the inter-dependence of perfusion and
diffusion, using a glioblastoma cohort containing 115 patients. A statistical
method, the empirical copula transform, was applied obtain the joint
distribution of perfusion and diffusion imaging, which was then discretized to
extract second-order features for hierarchical patient clustering. Three
patient subgroups were identified which showed significantly different overall survival
and progression-free survival. The results showed that the inter-dependence
between perfusion and diffusion imaging may be useful in stratifying patients
and evaluating tumor invasiveness.
Introduction
Glioblastoma
represents the commonest malignant brain tumors, characterized by dismal
prognosis. A highly heterogeneous microstructure
and vasculature can be found within glioblastoma, which may create various
tumor microenvironment and lead to different phenotypes. A systematic method to
investigate the global mismatch between cellularity and vascularity is
important. The multi-parametric imaging may describe complementary information
including tumor microstructure and vasculature. However, due to the marginal
distribution of different imaging modalities, finding validated surrogates to
depict the inter-dependence between imaging modalities remains a challenge. Copula
transform is a multivariate probability distribution describing the
inter-dependence of random variables [1]. The purpose of this study was to
interrogate the inter-dependence of microstructure and vasculature using
perfusion and diffusion imaging using the copula transform, and to investigate
the utility of this approach in tumor invasiveness assessment.Methods
Patients
This
study was approved by the local institutional review board. Informed written
consent was obtained from all patients. We prospectively recruited 115 patients
(mean age 59.3 years, range 22 - 76 years, 87 males) with supratentorial de novo glioblastoma. Patients with
history of previous brain tumor, cranial surgery, radiotherapy/chemotherapy,
and contraindication for MRI scanning were excluded. All subjects received
maximal safe tumor resection and diagnosis was confirmed by pathology. After
surgery, temozolomide chemoradiotherapy was performed following the Stupp
protocol. The Response Assessment in Neuro-oncology criteria was used to
evaluate patient response [2].
Imaging processing and
regions of interest
All
MRI sequences were pre-operatively acquired using a 3T MRI scanner. MRI
sequences include T2-weighted, post-contrast T1-weighted and FLAIR images, as
well as dynamic susceptibility contrast (DSC), diffusion tensor imaging (DTI)
with inline ADC calculation using b values of 0–1000 sec/mm2. All
images were co-registered to T2W images. The relative cerebral blood volume
(rCBV) maps were generated from the DSC images after leakage corrected using
NordicICE. Contrast-enhancing (CE) regions were manually delineated and
cross-validated by three experienced researchers on the co-registered
post-contrast T1-weighted images.
Copula transform and patient clustering
The
study design is shown in Figure 1. Empirical copula transform was applied to
the ADC and rCBV maps on each patient individually. A discrete feature
extraction then was applied to the joint distribution of ADC and rCBV. The
extracted features included Energy, Contrast, Entropy, Homogeneity,
Correlation, SumAverage, Variance, Dissimilarity, and AutoCorrelation [3]. A
hierarchical clustering using complete method was then performed on the
patients, based on the extracted features. To find the most stable and
unambiguous patient clustering, we varied the number of clusters from 2 to 10.
The optimal number of clusters was selected according to the majority vote among
the 26 indices as implemented in the R package ‘Nbclust’ [4]. A leave-one-out
cross-validation (LOOCV) procedure was applied for constructing and validating
the patient clusters.
Statistical analysis
The
clinical characteristics of patient clusters were compared with Kruskal-Wallis
rank sum test. Kaplan-Meier analysis using Log-rank test was performed to
evaluate patient survival. Patients who were alive at the last known follow-up
were censored. The hypothesis of no effect was rejected at a two-sided level of
0.05.
Results
Three
patient subgroups were identified from the unsupervised clustering, containing
40 patients (35 %), 48 patients (42 %), and 27 patients (23 %) respectively
(Figure 2). The mean values of co-occurrence consensus clustering matrix from
LOOCV are 0.91, 0.95 and 0.98 respectively, suggesting that three patient
clusters generated from the unsupervised clustering were stable. The average
discretized matrix of ADC-rCBV joint distribution demonstrated that subtype I
displayed a most uniform joint distribution, and subtype Ⅲ displayed a most
diagonalized joint distribution (Figure 3). These subtypes showed no
significant differences in clinical characteristics, but were significantly
different in patient survivals. Kaplan-Meier analysis using Log-rank test
showed significantly different overall survival (P = 0.039) and progression-free survival (P = 0.025) for the three identified subtypes. Discussion
Tumor
microstructure estimated from diffusion imaging and vascularity estimated from
perfusion imaging can describe the key characteristics associated with tumor
pathogenesis. Although existing evidences suggest benefits from combining
imaging techniques to identify tumor subregions that are responsible for
treatment failure, systematic measures to integrate perfusion and diffusion
imaging are lacking. Previous studies have validated the robustness of the
copula transform in estimating non-linear correlation in multimodal
neuroimaging data [5]. Using this method, our study demonstrated that the
inter-dependence of ADC and rCBV and discrete feature extraction can be used to
characterize glioblastoma. The results showed that both high and low
inter-dependent tumor microvasculature and microstructure were associated with
worsened survival, suggesting that the inter-dependence between perfusion and
diffusion imaging may provide clinically relevant information. Acknowledgements
The research was
supported by the National Institute for Health Research (NIHR) Brain Injury
MedTech Co-operative based at Cambridge University Hospitals NHS Foundation
Trust and University of Cambridge; The views expressed are those of the
author(s) and not necessarily those of the NHS, the NIHR or the Department of
Health and Social Care (SJP, project reference NIHR/CS/009/011); Cambridge Trust and China Scholarship
Council (CL & SW); CBS acknowledges support
from the Leverhulme Trust project on ‘Breaking the non-convexity barrier’,
EPSRC grant Nr. EP/M00483X/1, the EPSRC Centre Nr. EP/N014588/1, the RISE
projects CHiPS and NoMADS, the Cantab Capital Institute for the Mathematics of
Information and the Alan Turing Institute.References
[1] Nelsen RB. An
introduction to copulas: Springer Science & Business Media; 2007.
[2] Wen PY, Macdonald DR, Reardon DA, et al. Updated Response Assessment Criteria
for High-Grade Gliomas: Response Assessment in Neuro-Oncology Working Group.
Journal of Clinical Oncology. 2010;28(11):1963-72.
[3] Haralick RM, Shanmugam K, Dinstein I. Textural Features for Image Classification. Ieee Transactions on
Systems Man and Cybernetics. 1973;Smc3(6):610-21.
[4] Charrad M, Ghazzali N, Boiteau V, Niknafs A. Nbclust: An R Package for Determining
the Relevant Number of Clusters in a Data Set. Journal of Statistical Software.
2014;61(6):1-36.
[5] Ince RA, Giordano BL, Kayser C, et al. A statistical framework for
neuroimaging data analysis based on mutual information estimated via a gaussian
copula. Hum Brain Mapp. 2017;38(3):1541-73.