Abstract
Objective
To quantify the cervical sagittal alignment in patients with Hirayama disease (HD) and to investigate the effect of loss of cervical sagittal alignment upon the cervical spinal lesions in HD.
Methods
Cervical sagittal alignments were measured in 253 HD patients and 63 healthy subjects by C2–C7 Cobb and a modified method of Toyama et al. Motor unit number estimation (MUNE) was performed in bilateral abductor pollicis brevis (APB) in all HD patients, and 31 patients further underwent cervical diffusion tensor imaging (DTI).
Results
Compared with healthy subjects, HD patients showed lower C2–C7 Cobb (P < 0.05), and 83.4% patients showed loss of cervical lordosis (cervical straight or kyphosis), which was greater than healthy subjects (55.6%, P < 0.05). Compared with lordotic/straight group, patients with cervical kyphosis showed lower MUNE values and greater single motor unit potential (SMUP) in bilateral APB, and higher apparent dispersion coefficient (ADC) and lower fractional anisotropy were observed at C4/C5 level in the latter than the former (P < 0.05). C2–C7 Cobb was associated with both C4/C5 ADC and bilateral SMUP (P < 0.05).
Conclusions
Most HD patients showed loss of cervical sagittal alignments, and both MUNE and DTI detections demonstrated a positive correlation between loss of cervical sagittal alignments and cervical spinal lesions in HD. These findings supported that loss of cervical sagittal alignments may worsen motor impairments in HD. Therefore, it is necessary for clinicians to be aware of restoring cervical sagittal alignments during HD treatment.
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Data availability
All data generated or analyzed during this study are included in this published article (and its supplementary material).
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Funding
This work was supported by Shanghai “Science and Technology Innovation Action Plan” Project (22s31900600) and the National Natural Science Foundation of China Science Foundation Project (82072488).
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ZCJ and JJY have made substantial contributions to conception and design; CKW, YY, and SC have made substantial contributions to acquisition of data, or analysis and interpretation of data; ZY, WHL, and LFZ have been involved in drafting the manuscript or revising it critically for important intellectual content; all authors have given final approval of the version to be published.
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This study protocol was approved by the Ethics Committee of Huashan Hospital (Fudan University, Shanghai, China) (KY2022-683), and informed consent was obtained from all participants. The procedures used in this study adhere to the tenets of the Declaration of Helsinki.
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Kaiwen Chen, Yang Yang, and Chi Sun contributed equally to this work and should be considered co-first authors.
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ESM 2
Supplementary Fig. 1: Correlation between HGS and MUNE measurements in the ipsilateral APB in HD patients. HGS was associated with CMAP amplitudes (A, D), average SMUP amplitudes (B, E) and MUNE values (C, F) in the ipsilateral APB. HD: Hirayama disease; MUNE: Motor unit number estimation; CMAP: Compound muscle action potential; SMUP: Single motor unit potential; HGS: Handgrip strength; APB: Abductor pollicis brevis. (PNG 3349 kb)
ESM 3
Supplementary Fig. 2: Correlation between DASH scores and MUNE measurements in HD patients. DASH was associated with CMAP amplitudes (A, D), average SMUP amplitudes (B, E) and MUNE values (C, F) on both sides. HD: Hirayama disease; MUNE: Motor unit number estimation; CMAP: Compound muscle action potential; SMUP: Single motor unit potential; DASH: The disabilities of the arm, shoulder and hand outcome measure.(PNG 2794 kb)
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Chen, K., Yang, Y., Sun, C. et al. Loss of cervical sagittal alignment worsens the cervical spinal lesions in patients with Hirayama disease. Neurol Sci 44, 2103–2111 (2023). https://doi.org/10.1007/s10072-023-06621-2
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DOI: https://doi.org/10.1007/s10072-023-06621-2