Microstructural Integrity of Peripheral Nerves in Charcot–Marie–Tooth Disease: An MRI Evaluation Study
Background Charcot–Marie–Tooth (CMT) disease is diagnosed through clinical findings and genetic testing. While there are neurophysiological tools and clinical functional scales in CMT, objective disease biomarkers that can facilitate in monitoring disease progression are limited. Purpose To investig...
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Published in | Journal of magnetic resonance imaging Vol. 53; no. 2; pp. 437 - 444 |
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Main Authors | , , , , , , , , , |
Format | Journal Article |
Language | English |
Published |
Hoboken, USA
John Wiley & Sons, Inc
01.02.2021
Wiley Subscription Services, Inc |
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Online Access | Get full text |
ISSN | 1053-1807 1522-2586 1522-2586 |
DOI | 10.1002/jmri.27354 |
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Abstract | Background
Charcot–Marie–Tooth (CMT) disease is diagnosed through clinical findings and genetic testing. While there are neurophysiological tools and clinical functional scales in CMT, objective disease biomarkers that can facilitate in monitoring disease progression are limited.
Purpose
To investigate the utility of diffusion tensor imaging (DTI) in determining the microstructural integrity of sciatic and peroneal nerves and its correlation with the MRI grading of muscle atrophy severity and clinical function in CMT as determined by the CMT neuropathy score (CMTNS).
Study Type
Prospective case–control.
Subjects
Nine CMT patients and nine age‐matched controls.
Field Strength/Sequence
3 T T1‐weighted in‐/out‐of phase spoiled gradient recalled echo (SPGR) and DTI sequences.
Assessment
Fractional anisotropy (FA), axial diffusivity (AD), radial diffusivity (RD), and mean diffusivity (MD) values for sciatic and peroneal nerves were obtained from DTI. Muscle atrophy was graded according to the Goutallier classification using in‐/out‐of phase SPGRs. DTI parameters and muscle atrophy grades were compared between CMT and controls, and the relationship between DTI parameters, muscle atrophy grades, and CMTNS were assessed.
Statistical Tests
The Wilcoxon Signed Ranks test was used to compare DTI parameters between CMT and controls. The relationship between DTI parameters, muscle atrophy grades, and CMTNS were analyzed using the Spearman correlation. Receiver operating characteristic (ROC) analyses of DTI parameters that can differentiate CMT from healthy controls were done.
Results
There was a significant reduction in FA and increase in RD of both nerves (P < 0.05) in CMT, with significant correlations between FA (negative; P < 0.05) and RD (positive; P < 0.05) with muscle atrophy grade. In the sciatic nerve, there was significant correlation between FA and CMTNS (r = –0.795; P < 0.05). FA and RD could discriminate CMT from controls with high sensitivity (77.8–100%) and specificity (88.9–100%).
Data Conclusion
There were significant differences of DTI parameters between CMT and controls, with significant correlations between DTI parameters, muscle atrophy grade, and CMTNS.
Level of Evidence 2
Technical Efficacy Stage 2
J. MAGN. RESON. IMAGING 2021;53:437–444. |
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AbstractList | Charcot-Marie-Tooth (CMT) disease is diagnosed through clinical findings and genetic testing. While there are neurophysiological tools and clinical functional scales in CMT, objective disease biomarkers that can facilitate in monitoring disease progression are limited.
To investigate the utility of diffusion tensor imaging (DTI) in determining the microstructural integrity of sciatic and peroneal nerves and its correlation with the MRI grading of muscle atrophy severity and clinical function in CMT as determined by the CMT neuropathy score (CMTNS).
Prospective case-control.
Nine CMT patients and nine age-matched controls.
3 T T
-weighted in-/out-of phase spoiled gradient recalled echo (SPGR) and DTI sequences.
Fractional anisotropy (FA), axial diffusivity (AD), radial diffusivity (RD), and mean diffusivity (MD) values for sciatic and peroneal nerves were obtained from DTI. Muscle atrophy was graded according to the Goutallier classification using in-/out-of phase SPGRs. DTI parameters and muscle atrophy grades were compared between CMT and controls, and the relationship between DTI parameters, muscle atrophy grades, and CMTNS were assessed.
The Wilcoxon Signed Ranks test was used to compare DTI parameters between CMT and controls. The relationship between DTI parameters, muscle atrophy grades, and CMTNS were analyzed using the Spearman correlation. Receiver operating characteristic (ROC) analyses of DTI parameters that can differentiate CMT from healthy controls were done.
There was a significant reduction in FA and increase in RD of both nerves (P < 0.05) in CMT, with significant correlations between FA (negative; P < 0.05) and RD (positive; P < 0.05) with muscle atrophy grade. In the sciatic nerve, there was significant correlation between FA and CMTNS (r = -0.795; P < 0.05). FA and RD could discriminate CMT from controls with high sensitivity (77.8-100%) and specificity (88.9-100%).
There were significant differences of DTI parameters between CMT and controls, with significant correlations between DTI parameters, muscle atrophy grade, and CMTNS. Level of Evidence 2 Technical Efficacy Stage 2 J. MAGN. RESON. IMAGING 2021;53:437-444. BackgroundCharcot–Marie–Tooth (CMT) disease is diagnosed through clinical findings and genetic testing. While there are neurophysiological tools and clinical functional scales in CMT, objective disease biomarkers that can facilitate in monitoring disease progression are limited.PurposeTo investigate the utility of diffusion tensor imaging (DTI) in determining the microstructural integrity of sciatic and peroneal nerves and its correlation with the MRI grading of muscle atrophy severity and clinical function in CMT as determined by the CMT neuropathy score (CMTNS).Study TypeProspective case–control.SubjectsNine CMT patients and nine age‐matched controls.Field Strength/Sequence3 T T1‐weighted in‐/out‐of phase spoiled gradient recalled echo (SPGR) and DTI sequences.AssessmentFractional anisotropy (FA), axial diffusivity (AD), radial diffusivity (RD), and mean diffusivity (MD) values for sciatic and peroneal nerves were obtained from DTI. Muscle atrophy was graded according to the Goutallier classification using in‐/out‐of phase SPGRs. DTI parameters and muscle atrophy grades were compared between CMT and controls, and the relationship between DTI parameters, muscle atrophy grades, and CMTNS were assessed.Statistical TestsThe Wilcoxon Signed Ranks test was used to compare DTI parameters between CMT and controls. The relationship between DTI parameters, muscle atrophy grades, and CMTNS were analyzed using the Spearman correlation. Receiver operating characteristic (ROC) analyses of DTI parameters that can differentiate CMT from healthy controls were done.ResultsThere was a significant reduction in FA and increase in RD of both nerves (P < 0.05) in CMT, with significant correlations between FA (negative; P < 0.05) and RD (positive; P < 0.05) with muscle atrophy grade. In the sciatic nerve, there was significant correlation between FA and CMTNS (r = –0.795; P < 0.05). FA and RD could discriminate CMT from controls with high sensitivity (77.8–100%) and specificity (88.9–100%).Data ConclusionThere were significant differences of DTI parameters between CMT and controls, with significant correlations between DTI parameters, muscle atrophy grade, and CMTNS.Level of Evidence 2Technical Efficacy Stage 2J. MAGN. RESON. IMAGING 2021;53:437–444. Charcot-Marie-Tooth (CMT) disease is diagnosed through clinical findings and genetic testing. While there are neurophysiological tools and clinical functional scales in CMT, objective disease biomarkers that can facilitate in monitoring disease progression are limited.BACKGROUNDCharcot-Marie-Tooth (CMT) disease is diagnosed through clinical findings and genetic testing. While there are neurophysiological tools and clinical functional scales in CMT, objective disease biomarkers that can facilitate in monitoring disease progression are limited.To investigate the utility of diffusion tensor imaging (DTI) in determining the microstructural integrity of sciatic and peroneal nerves and its correlation with the MRI grading of muscle atrophy severity and clinical function in CMT as determined by the CMT neuropathy score (CMTNS).PURPOSETo investigate the utility of diffusion tensor imaging (DTI) in determining the microstructural integrity of sciatic and peroneal nerves and its correlation with the MRI grading of muscle atrophy severity and clinical function in CMT as determined by the CMT neuropathy score (CMTNS).Prospective case-control.STUDY TYPEProspective case-control.Nine CMT patients and nine age-matched controls.SUBJECTSNine CMT patients and nine age-matched controls.3 T T1 -weighted in-/out-of phase spoiled gradient recalled echo (SPGR) and DTI sequences.FIELD STRENGTH/SEQUENCE3 T T1 -weighted in-/out-of phase spoiled gradient recalled echo (SPGR) and DTI sequences.Fractional anisotropy (FA), axial diffusivity (AD), radial diffusivity (RD), and mean diffusivity (MD) values for sciatic and peroneal nerves were obtained from DTI. Muscle atrophy was graded according to the Goutallier classification using in-/out-of phase SPGRs. DTI parameters and muscle atrophy grades were compared between CMT and controls, and the relationship between DTI parameters, muscle atrophy grades, and CMTNS were assessed.ASSESSMENTFractional anisotropy (FA), axial diffusivity (AD), radial diffusivity (RD), and mean diffusivity (MD) values for sciatic and peroneal nerves were obtained from DTI. Muscle atrophy was graded according to the Goutallier classification using in-/out-of phase SPGRs. DTI parameters and muscle atrophy grades were compared between CMT and controls, and the relationship between DTI parameters, muscle atrophy grades, and CMTNS were assessed.The Wilcoxon Signed Ranks test was used to compare DTI parameters between CMT and controls. The relationship between DTI parameters, muscle atrophy grades, and CMTNS were analyzed using the Spearman correlation. Receiver operating characteristic (ROC) analyses of DTI parameters that can differentiate CMT from healthy controls were done.STATISTICAL TESTSThe Wilcoxon Signed Ranks test was used to compare DTI parameters between CMT and controls. The relationship between DTI parameters, muscle atrophy grades, and CMTNS were analyzed using the Spearman correlation. Receiver operating characteristic (ROC) analyses of DTI parameters that can differentiate CMT from healthy controls were done.There was a significant reduction in FA and increase in RD of both nerves (P < 0.05) in CMT, with significant correlations between FA (negative; P < 0.05) and RD (positive; P < 0.05) with muscle atrophy grade. In the sciatic nerve, there was significant correlation between FA and CMTNS (r = -0.795; P < 0.05). FA and RD could discriminate CMT from controls with high sensitivity (77.8-100%) and specificity (88.9-100%).RESULTSThere was a significant reduction in FA and increase in RD of both nerves (P < 0.05) in CMT, with significant correlations between FA (negative; P < 0.05) and RD (positive; P < 0.05) with muscle atrophy grade. In the sciatic nerve, there was significant correlation between FA and CMTNS (r = -0.795; P < 0.05). FA and RD could discriminate CMT from controls with high sensitivity (77.8-100%) and specificity (88.9-100%).There were significant differences of DTI parameters between CMT and controls, with significant correlations between DTI parameters, muscle atrophy grade, and CMTNS. Level of Evidence 2 Technical Efficacy Stage 2 J. MAGN. RESON. IMAGING 2021;53:437-444.DATA CONCLUSIONThere were significant differences of DTI parameters between CMT and controls, with significant correlations between DTI parameters, muscle atrophy grade, and CMTNS. Level of Evidence 2 Technical Efficacy Stage 2 J. MAGN. RESON. IMAGING 2021;53:437-444. Background Charcot–Marie–Tooth (CMT) disease is diagnosed through clinical findings and genetic testing. While there are neurophysiological tools and clinical functional scales in CMT, objective disease biomarkers that can facilitate in monitoring disease progression are limited. Purpose To investigate the utility of diffusion tensor imaging (DTI) in determining the microstructural integrity of sciatic and peroneal nerves and its correlation with the MRI grading of muscle atrophy severity and clinical function in CMT as determined by the CMT neuropathy score (CMTNS). Study Type Prospective case–control. Subjects Nine CMT patients and nine age‐matched controls. Field Strength/Sequence 3 T T1‐weighted in‐/out‐of phase spoiled gradient recalled echo (SPGR) and DTI sequences. Assessment Fractional anisotropy (FA), axial diffusivity (AD), radial diffusivity (RD), and mean diffusivity (MD) values for sciatic and peroneal nerves were obtained from DTI. Muscle atrophy was graded according to the Goutallier classification using in‐/out‐of phase SPGRs. DTI parameters and muscle atrophy grades were compared between CMT and controls, and the relationship between DTI parameters, muscle atrophy grades, and CMTNS were assessed. Statistical Tests The Wilcoxon Signed Ranks test was used to compare DTI parameters between CMT and controls. The relationship between DTI parameters, muscle atrophy grades, and CMTNS were analyzed using the Spearman correlation. Receiver operating characteristic (ROC) analyses of DTI parameters that can differentiate CMT from healthy controls were done. Results There was a significant reduction in FA and increase in RD of both nerves (P < 0.05) in CMT, with significant correlations between FA (negative; P < 0.05) and RD (positive; P < 0.05) with muscle atrophy grade. In the sciatic nerve, there was significant correlation between FA and CMTNS (r = –0.795; P < 0.05). FA and RD could discriminate CMT from controls with high sensitivity (77.8–100%) and specificity (88.9–100%). Data Conclusion There were significant differences of DTI parameters between CMT and controls, with significant correlations between DTI parameters, muscle atrophy grade, and CMTNS. Level of Evidence 2 Technical Efficacy Stage 2 J. MAGN. RESON. IMAGING 2021;53:437–444. |
Author | Nawawi, Ouzreiah Shahrizaila, Nortina Fadzli, Farhana Cheah, Peng Loon Rozalli, Faizatul Izza Ramli, Norlisah Krisnan, Thiagu Rahmat, Kartini Wong, Jeannie Hsiu Ding Tan, Li Kuo |
Author_xml | – sequence: 1 givenname: Peng Loon orcidid: 0000-0002-4513-4219 surname: Cheah fullname: Cheah, Peng Loon organization: University of Malaya – sequence: 2 givenname: Thiagu surname: Krisnan fullname: Krisnan, Thiagu organization: University of Malaya – sequence: 3 givenname: Jeannie Hsiu Ding surname: Wong fullname: Wong, Jeannie Hsiu Ding organization: University of Malaya – sequence: 4 givenname: Faizatul Izza surname: Rozalli fullname: Rozalli, Faizatul Izza organization: University of Malaya – sequence: 5 givenname: Farhana surname: Fadzli fullname: Fadzli, Farhana organization: University of Malaya – sequence: 6 givenname: Kartini surname: Rahmat fullname: Rahmat, Kartini organization: University of Malaya – sequence: 7 givenname: Nortina surname: Shahrizaila fullname: Shahrizaila, Nortina organization: University of Malaya – sequence: 8 givenname: Li Kuo surname: Tan fullname: Tan, Li Kuo organization: University of Malaya – sequence: 9 givenname: Ouzreiah surname: Nawawi fullname: Nawawi, Ouzreiah organization: University of Malaya – sequence: 10 givenname: Norlisah surname: Ramli fullname: Ramli, Norlisah email: norlisahramli@gmail.com organization: University of Malaya |
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Keywords | diffusion tensor imaging Charcot-Marie-Tooth disease peripheral nervous system diseases peroneal nerve sciatic nerve |
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Charcot–Marie–Tooth (CMT) disease is diagnosed through clinical findings and genetic testing. While there are neurophysiological tools and clinical... Charcot-Marie-Tooth (CMT) disease is diagnosed through clinical findings and genetic testing. While there are neurophysiological tools and clinical functional... BackgroundCharcot–Marie–Tooth (CMT) disease is diagnosed through clinical findings and genetic testing. While there are neurophysiological tools and clinical... |
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SubjectTerms | Anisotropy Atrophy Biomarkers Charcot-Marie-Tooth disease Charcot-Marie-Tooth Disease - diagnostic imaging Correlation analysis Diffusion Tensor Imaging Diffusivity Field strength Genetic screening Humans Integrity Magnetic Resonance Imaging Medical imaging Muscles Parameters Peripheral nerves Peripheral Nerves - diagnostic imaging peripheral nervous system diseases Peripheral neuropathy peroneal nerve Prospective Studies Sciatic nerve Statistical analysis Statistical tests Tensors |
Title | Microstructural Integrity of Peripheral Nerves in Charcot–Marie–Tooth Disease: An MRI Evaluation Study |
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