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The evaluation on neural status of cervical spinal cord in normal and Hirayama disease using diffusion tensor imaging

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Abstract

Purpose

To explore the changes in diffusion tensor imaging (DTI) parameters in cervical spinal cord in Hirayama disease (HD) patients and healthy volunteers and to compare these parameters between cervical flexion and neutral positions in HD patients.

Methods

Seventeen male patients with HD and eleven healthy young males were included to receive DTI scans in cervical flexion and neutral positions. The FA and ADC values of different levels were measured based on the region of interest drawn on the mid-sagittal plane. The dynamic compressed level’s parameters were defined as the lowest and the second lowest FA and the highest and the second highest ADC, respectively. The clinical assessment of patients was obtained using their disabilities of the arm, shoulder and hand (DASH) scores.

Results

For the HD patients, the FA values in the cervical flexion position were lower and the ADC values were much higher than those in the cervical neutral position. Compared with the controls, the ADC values were significantly higher in the lower levels (C5/6-C7/T1) and the FA values obviously lower at C7/T1 in HD patients in cervical neutral position. The FA and ADC values of the dynamic compressed level in HD patients deviated significantly from the average of the lower levels in controls. Both the FA and ADC values of the dynamic compressed level correlated with the DASH scores (FA, R2 = 0.520, P = 0.001; ADC, R2 = 0.421, P = 0.005).

Conclusions

DTI parameters can support a hypothesis of dynamic cervical flexion compression and noninvasively reveal the neural status of HD patients.

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References

  1. Hirayama K (2000) Juvenile muscular atrophy of distal upper extremity (Hirayama disease): focal cervical ischemic poliomyelopathy. Neuropathology 20(suppl):S91–S94

    Article  PubMed  Google Scholar 

  2. Huang YC, Ro LS, Chang HS, Chen CM, Wu YR, Lee JD, Lyu RK (2008) A clinical study of Hirayama disease in Taiwan. Muscle Nerve 37(5):576–582

    Article  PubMed  Google Scholar 

  3. Zhou B, Chen L, Fan D, Zhou D (2010) Clinical features of Hirayama disease in mainland China. Amyotroph Lateral Scler 11(1–2):133–139

    Article  CAS  PubMed  Google Scholar 

  4. Tashiro K, Kikuchi S, Itoyama Y, Tokumaru Y, Sobue G, Mukai E, Akiguchi I, Nakashima K, Kira J, Hirayama K (2006) Nationwide survey of juvenile muscular atrophy of distal upper extremity (Hirayama disease) in Japan. Amyotroph Lateral Scler 7(1):38–45

    Article  PubMed  Google Scholar 

  5. Pradhan S (2009) Bilaterally symmetric form of Hirayama disease. Neurology 72(24):2083–2089

    Article  PubMed  Google Scholar 

  6. Kang JS, Jochem-Gawehn S, Laufs H, Ferbert A, Vieregge P, Ziemann U (2011) Hirayama disease in Germany: case reports and review of the literature. Nervenarzt 82(10):1264–1272

    Article  PubMed  Google Scholar 

  7. Andreadou E, Christodoulou K, Manta P, Karandreas N, Loukaidis P, Sfagos C, Vassilopoulos D (2009) Familial asymmetric distal upper limb amyotrophy (Hirayama disease): report of a Greek family. Neurologist 15(3):156–160

    Article  PubMed  Google Scholar 

  8. Paredes I, Esteban J, Ramos A, Gonzalez P, Rivas JJ (2014) A severe case of Hirayama disease successfully treated by anterior cervical fusion. J Neurosurg Spine 20(2):191–195

    Article  PubMed  Google Scholar 

  9. Lewis D, Saxena A, Herwadkar A, Leach J (2017) A confirmed case in the United Kingdom of Hirayama Disease in a young white male presenting with hand weakness. World Neurosurg 105:1039.e7–1039.e12

    Article  Google Scholar 

  10. Kieser DC, Cox PJ, Kieser SCJ (2018) Hirayama disease. Eur Spine J 27(6):1201–1206

    Article  PubMed  Google Scholar 

  11. Zheng C, Zhu D, Lu F, Zhu Y, Ma X, Xia X, Jiang J (2017) A double determination of central motor conduction time in the assessment of Hirayama disease. Clin Neurophysiol 128(11):2369–2374

    Article  PubMed  Google Scholar 

  12. Lai V, Wong YC, Poon WL, Yuen MK, Fu YP, Wong OW (2011) Forward shifting of posterior dural sac during flexion cervical magnetic resonance imaging in Hirayama disease: an initial study on normal subjects compared to patients with Hirayama disease. Eur J Radiol 80(3):724–728

    Article  PubMed  Google Scholar 

  13. Lehman VT, Luetmer PH, Sorenson EJ, Carter RE, Gupta V, Fletcher GP, Hu LS, Kotsenas AL (2013) Cervical spine MR imaging findings of patients with Hirayama disease in North America: a multisite study. AJNR Am J Neuroradiol 34(2):451–456

    Article  CAS  PubMed  Google Scholar 

  14. Song J, Wang HL, Zheng CJ, Jiang JY (2017) Risk factors for surgical results of Hirayama disease: a retrospective analysis of a large cohort. World Neurosurg 105:69–77

    Article  PubMed  Google Scholar 

  15. Wang H, Sun C, Yang S, Jiang J, Lu F, Ma X, Xia X (2018) Dynamic cervical radiographs in patients with Hirayama disease: an unneglectable factor on the choice of surgery options. World Neurosurg 114:e433–e440

    Article  PubMed  Google Scholar 

  16. Zheng C, Nie C, Lei W, Zhu Y, Zhu D, Wang H, Lu F, Weber R, Jiang J (2018) CAN anterior cervical fusion procedures prevent the progression of the natural course of Hirayama disease? An ambispective cohort analysis. Clin Neurophysiol 129(11):2341–2349

    Article  PubMed  Google Scholar 

  17. Lu F, Wang H, Jiang J, Chen W, Ma X, Ma X, Xia X, Wang L (2013) Efficacy of anterior cervical decompression and fusion procedures for monomelic amyotrophy treatment: a prospective randomized controlled trial. J Neurosurg Spine 19(4):412–419

    Article  PubMed  Google Scholar 

  18. Wang HL, Wu YW, Song J, Jiang JY, Lu FZ, Ma XS, Xia XL (2018) Cortical activation changes in Hirayama disease after anterior cervical decompression and fusion. World Neurosurg 116:e588–e594

    Article  PubMed  Google Scholar 

  19. Wang X, Wang H, Sun C, Zhou S, Meng T, Lv F, Ma X, Xia X, Jiang J (2018) Analysis of radiological parameters associated with decreased fractional anisotropy values on diffusion tensor imaging in patients with lumbar spinal stenosis. Eur Spine J. https://doi.org/10.1007/s00586-018-5562-8

    Article  PubMed  Google Scholar 

  20. Song T, Chen WJ, Yang B, Zhao HP, Huang JW, Cai MJ, Dong TF, Li TS (2011) Diffusion tensor imaging in the cervical spinal cord. Eur Spine J 20(3):422–428

    Article  PubMed  Google Scholar 

  21. Lee JW, Kim JH, Park JB, Park KW, Yeom JS, Lee GY, Kang HS (2011) Diffusion tensor imaging and fiber tractography in cervical compressive myelopathy: preliminary results. Skelet Radiol 40(12):1543–1551

    Article  Google Scholar 

  22. Jones JG, Cen SY, Lebel RM, Hsieh PC, Law M (2013) Diffusion tensor imaging correlates with the clinical assessment of disease severity in cervical spondylotic myelopathy and predicts outcome following surgery. AJNR Am J Neuroradiol 34(2):471–478

    Article  CAS  PubMed  Google Scholar 

  23. Boelmans K, Kaufmann J, Schmelzer S, Vielhaber S, Kornhuber M, Münchau A, Zierz S, Gaul C (2013) Hirayama disease is a pure spinal motor neuron disorder—a combined DTI and transcranial magnetic stimulation study. J Neurol 260(2):540–548

    Article  PubMed  Google Scholar 

  24. MacDermid JC, Kramer JF, Woodbury MG, McFarlane RM, Roth JH (1994) Interrater reliability of pinch and grip strength measurements in patients with cumulative trauma disorders. J Hand Ther 7(1):10–14

    Article  CAS  PubMed  Google Scholar 

  25. Ghosh PS, Moodley M, Friedman NR, Rothner AD, Ghosh D (2011) Hirayama disease in children from North America. J Child Neurol 26(12):1542–1547

    Article  PubMed  Google Scholar 

  26. Rajasekaran S, Yerramshetty JS, Chittode VS, Kanna RM, Balamurali G, Shetty AP (2014) The assessment of neuronal status in normal and cervical spondylotic myelopathy using diffusion tensor imaging. Spine (Phila Pa 1976) 39(15):1183–1189

    Article  CAS  Google Scholar 

  27. Kuhn FP, Feydy A, Launay N, Lefevre-Colau MM, Poiraudeau S, Laporte S, Maier MA, Lindberg P (2016) Kinetic DTI of the cervical spine: diffusivity changes in healthy subjects. Neuroradiology 58(9):929–935

    Article  PubMed  Google Scholar 

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Acknowledgements

We gratefully thank all the participants in this research study together with our technicians for their dedicated efforts in scanning standardization and quality assurance. This work was supported by Shanghai Municipal Science and Technology Commission (16411964100), Youth Science Project of National Natural Science Foundation of China (81501909), and National Natural Science Foundation of China (81772388).

Funding

This study was funded by Science and Technology Commission of Shanghai Municipality (16411964100), National Natural Science Foundation of China (81501909), and National Natural Science Foundation of China (81772388).

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Author notes

  1. Chi Sun and Shuyi Zhou are co-first authors: Chi Sun and Shuyi Zhou have contributed equally to this work.

Authors and Affiliations

  1. Department of Orthopaedics, Huashan Hospital, Fudan University, Shanghai, China

    Chi Sun, Zhongyi Cui, Yuxuan Zhang, Hongli Wang, Jianyuan Jiang, Feizhou Lu & Xiaosheng Ma

  2. Department of Radiology, Huashan Hospital, Fudan University, Shanghai, China

    Shuyi Zhou

  3. The Fifth People’ s Hospital of Shanghai, Fudan University, Shanghai, China

    Feizhou Lu

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  1. Chi Sun
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  5. Hongli Wang
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Correspondence to Hongli Wang or Jianyuan Jiang.

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We declare that all human and animal studies have been approved by university and hospital and have therefore been performed in accordance with the ethical standards laid down in the 1964 Declaration of Helsinki and its later amendments.

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Sun, C., Zhou, S., Cui, Z. et al. The evaluation on neural status of cervical spinal cord in normal and Hirayama disease using diffusion tensor imaging. Eur Spine J 28, 1872–1878 (2019). https://doi.org/10.1007/s00586-019-06013-1

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  • DOI: https://doi.org/10.1007/s00586-019-06013-1

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