Medicine ® Clinical Case Report OPEN Therapeutic effects of acupuncture on sensory ataxia after a cerebral hemorrhage A case report Kuan-Yu Lu, MDa, Ka-Fai Yuen, MD, MSb, Jia-Yuan Luo, MDc, Chang-Zern Hong, MDd, ∗ Li-Wei Chou, MD, PhDe,f,g, Abstract Introduction: Sensory ataxia is a dysfunction of dynamic balance due to impairment of sensory input into the control of movement. The sequelae of stroke, such as hemiplegia, somatosensory impairment, and impaired balance may cause significant disability and may affect patients’ quality of life. In addition to rehabilitation programs, acupuncture therapy has been applied to stroke patients and is recommended as a complementary therapy in stroke rehabilitation. Patient concerns: A 70-year-old male had a sudden onset of conscious loss. The brain computed tomography showed intracerebral hemorrhage with subdural hemorrhage and subarachnoid hemorrhage. Diagnosis: Intracerebral hemorrhagic stroke was diagnosed. Downloaded from http://journals.lww.com/md-journal by BhDMf5ePHKbH4TTImqenVEQutYOvXT5zIGxqlNWmXPz2GsUU+3EqVWawo/90nx5d on 07/26/2020 Interventions: He received craniotomy with hematoma evacuation immediately and waked up 3 weeks with bilateral hemiparesis (right side weaker than left), impaired position sensation and tactile perception in the right lower limb. He then began to receive rehabilitation therapy and had significant improvement in muscle strength and static balance, but no improvement in tactile perception of position sense in the right lower limbs and reached plateau. Then he received acupuncture therapies to Yongquan (KI1), Tongtien (BL7) and Houxi (SI3). Outcomes: The patient’s walking ability recovered after receiving rehabilitation programs for 3 years, but the impairment in proprioception and dynamic balance persisted. The perception and dynamic balance had significantly improved after patient received acupuncture therapy, especially the acupuncture point of Yongquan (KI1). Conclusion: The clinical effect of acupuncture in combination with conventional rehabilitation therapy for neurological impairment recovery, improving activity of daily living performance and improving post-stroke imbalance was explored. We hope that this report can facilitate further well controlled quantitative objective studies on a big size of samples. Abbreviations: AcP = acupuncture, ADL= activity of daily living, CT = computed tomography. Keywords: acupuncture, cerebral hemorrhage, rehabilitation, sensory ataxia Editor: Maya Saranathan. LK and YK contributed equally to this work. No commercial party having a direct financial interest in the results of the research supporting this article has or will confer a benefit upon the authors or upon any organization with which the authors are associated. Patient has provided informed consent for publication of the case. The authors have no conflicts of interest to disclose. All data generated or analyzed during this study are included in this published article [and its supplementary information files]. The work was supported by China Medical University Hospital (CRS-108-047, DMR-109-095). a Department of Physical Medicine and Rehabilitation, Chang Gung Memorial Hospital, Chiayi, b Department of Rehabilitation, Ton-Yen General Hospital, Tsinchu County, c Department of Physical Medicine and Rehabilitation, Puzi Hospital, Ministry of Health and Welfare, Chiayi, Taiwan, d Retired professor, Department of Physical Medicine and Rehabilitation, University of California Irvine, CA, USA, e Department of Physical Medicine and Rehabilitation, China Medical University Hospital, f Department of Physical Therapy and Graduate Institute of Rehabilitation Science, China Medical University, g Department of Rehabilitation, Asia University Hospital, Taichung, Taiwan. ∗ Correspondence: Li-Wei Chou, Department of Physical Medicine and Rehabilitation, China Medical University Hospital, Address: No 2 Yuh-Der Road, Taichung, 40447, Taiwan (e-mail: chouliwe@gmail.com). Copyright © 2020 the Author(s). Published by Wolters Kluwer Health, Inc. This is an open access article distributed under the Creative Commons Attribution License 4.0 (CCBY), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. How to cite this article: Lu KY, Yuen KF, Luo JY, Hong CZ, Chou LW. Therapeutic effects of acupuncture on sensory ataxia after a cerebral hemorrhage: a case report. Medicine 2020;99:29(e21124). Received: 15 August 2019 / Received in final form: 20 May 2020 / Accepted: 4 June 2020 http://dx.doi.org/10.1097/MD.0000000000021124 1 Lu et al. Medicine (2020) 99:29 Medicine (2) the distant acupuncture points belonging the same channel passing through the lesion or the channels related to the group containing the lesion. 1. Introduction Sensory ataxia is a dysfunction of dynamic balance due to impairment of sensory input into the control of movement, but not cause by cerebellar dysfunction. Sensory ataxia is distinguished from cerebellar ataxia by the presence of near-normal coordination when the movement is visually observed. However, marked worsening of coordination when the eyes are closed (a positive Romberg’s sign). Sensory ataxia can be a manifestation of sensory large fiber peripheral neuropathies and conditions causing dysfunction of the dorsal columns of the spinal cord due to a variety of disorders: infectious, auto-immune, metabolic, toxic, vascular and hereditary diseases.[1–3] Acupuncture (AcP) is one valuable therapeutic intervention of the Traditional Chinese medicine. According to the theory of acupuncture, there are 14 channels of energy distributed in the whole human body. Inside these 14 channels, there is “Qi” circulated in the whole body regularly every day. Each channel contains many acupuncture points where more “Qi” accumulated and being more sensitive.[4] Acupuncture treatment on different acupuncture points may cure different various diseases, depending on the selection of different acupuncture points. In this case study, we applied two rules: The lesion according to theory of acupuncture is defined as the site where symptoms and signs are present.[5] A patient (a physiatrist, one of authors, CZH) with sensory ataxia due to subdural hemorrhage with sensory deficit at right sole area was selected for this case study. After therapeutic acupuncture, several subjective and objective evaluation were assessed. 2. Case presentation A 70 year-old male physiatrist (CZH) had a sudden onset of conscious loss on 2014-12-21. He was initially brought to a teaching hospital where computed tomography showed evidence of intra-cerebral hemorrhage with subdural hemorrhage and subarachnoid hemorrhage (Fig. 1A). He then received craniotomy with hematoma evacuation immediately (Fig. 1B). He waked up 3 weeks after craniotomy but found to have bilateral hemiparesis (right side weaker than left) with impaired position sensation and tactile perception in the right lower limb. He then began to receive rehabilitation therapy since after waked up. He discharged to a nursing home about 3 months later to continued rehabilitation therapy. He had significant improvement in muscle (1) the acupuncture points near the lesion, and Figure 1. (A) Brain CT showed intra-cerebral hemorrhage with subdural hemorrhage and subarachnoid hemorrhage on 2014-12-21. (B) After craniotomy with hematoma evacuation, brain CT showed some residual hematoma still in the cerebral and cerebellar area on 2014-12-22. CT = computed tomograpghy. 2 Lu et al. Medicine (2020) 99:29 www.md-journal.com strength and static balance. He was then able to control wheelchair by himself by 2015-02. Gradually, he was able to walk with a walker by 2016-01, then a cane by 2016-05, but still had impaired dynamic balance. There was no improvement in tactile perception of position sense in the right lower limbs. On 2017-12-30, he received acupuncture therapy to Yongquan (KI1). Size 36 acupuncture needles with 1 inch in length were inserted into the acupuncture points in sequence. Bilateral Yongquan (KI1) (in the kidney channel) were selected for near acupuncture point, and bilateral Tongtien (BL7) (in the bladder channel) and bilateral Houxi (SI3) (in the small intestine channel) for distant acupuncture point.[6] After each needling, electric stimulation with frequency of 2 Hz were applied for 30 minutes for each point. After only 1 treatment of acupuncture, he had significant improvement in perception, and dynamic balance. The case study was conducted after the first acupuncture therapy since the patient found the dramatic therapeutic effectiveness of acupuncture. The findings of evaluation before acupuncture was estimated by the patient depending on his memory. After therapeutic acupuncture, the following items were assessed: Table 1 The scales of assessment. Sd = Subjective evaluation of dynamic motor improvement (0∼5) Sm = Subjective evaluation of static motor improvement (0∼5) Ss = Subjective evaluation of sensory improvement (0∼5) 0 no improvement 1 less than 10% improvement 2 10% - 25% improvement 3 25% - 50% improvement 4 50% - 75% improvement 5 75% - 100% improvement Ws = Objective evaluation of walking speed (M/s) Wt = Objective evaluation of duration of walking tolerance (min) 2 LE = Objective evaluation of standing balance on 2 leg support (static balance) F+ Resist to some push force F OK without support FOK without support, but cannot resist to pushing P+ OK with partial support, but cannot resist to pushing 1 LE = Objective evaluation of duration of 1 leg support (static balance) 1 OK without support for only 1 s 0.75 OK without support for only 0.75 s 0.5 OK without support for only 0.5 s 0 Unable to do without support (1) Subjective feeling of dynamic stability (dynamic balance during walking) (Sd); (2) Subjective feeling of motor function (strength, static balance) (Sm); (3) Subjective feeling of sensory function (perception of position, tactile perception) (Ss); (4) Objective evaluation of walking speed (Ws); (5) Objective evaluation of walking tolerance (Wt); (6) Objective evaluation of static stability, 2 LE standing time with eye closed (2LE); (7) Objective evaluation of static stability, that is, the duration of 1-leg standing time with eye open (1LE). The scales of assessment are listed in Table 1. F = fair; P = poor. both motor improvement and to that of sensory improvement were both remarkable immediately after AcP treatment. But they were not sustained, but declined. Walking speed remained unchanged throughout the whole studying period. Walking tolerance improved after each AcP treatment, even in the last treatment. Regarding the objective balance, only little improvement in the 2 leg supported balance, but remarkable improvement in the 1 leg standing balance and became stable after 3 treatments of AcP. The results of assessment of functional improvement are listed in Table 2. The first row of the table showed the procedure (B = before AcP; R = resting period before the next AcP). The 2nd row listed the date of AcP, and the 3rd row showed the duration of AcP (30 minutes each AcP treatment, or the days between 2 AcP treatments. After the 4th to 10th rows are the functional improvement as explained in the legends of the table. The most remarkable findings include the subjective walking (dynamic balance) and objective assessment of 1 leg standing balance (static balance). It appears that the subjective feelings of 3. Discussion All the assessments in this case study were improved except for walking speed. It appeared that these improvements were related to the effectiveness of acupuncture. The patient was considered to reach a “plateau” of rehabilitation, until the physician applied the acupuncture to the point of Yongquan (KI1) to the patient. Yongquan (KI1) can treat conscious disturbance, cognitive function impairment, sole unable to touch the ground.[7] Quantitative and accurate measurements are crucial to evaluate Table 2 Assessment of functional improvement. Date Dur B 12/29 AcP 12/29 30m 2nd d 12/30 1d R 01/13 14d AcP 01/13 30m 2nd d 01/14 1d R 01/20 7d AcP 01/20 30m 2nd d 01/21 1d R 01/27 7d AcP 01/27 30m 2nd d 01/28 1d R 04/28 92d AcP 04/28 30m 2nd d 04/29 1d Sd Sm Ss Ws Wt 2LE 1LE 0 0 0 2 10 P+ 0 3 3 3–4 2 15 F+ 0.5 3 2 3 2 15 F/F 0 3 2 2 2 15 F/F 0 3 2 2 2 15 F/F 0.5 3 3 3 2 20 F+/F 0.5 3 2 2 2 15 F+/F 0.5 4 2 2 2 20 F+/F 0.5 4 3 3 2 30 F+/F 0.75 4 2 2 2 30 F+/F 0.75 4 2 2 2 40 F+/F 0.75 4 3 3 2 50 F+/F 0.75 4 2 2 2 50 F+/F 0.75 4.5 2 3 2 50 F+/F 0.75 4.5 3 3 2 60 F+/F 0.75 2nd d = assessments on the 2nd day after AcP, AcP = AcP into Yongquan, B = before AcP, Dur = duration of AcP (30 min each AcP, or duration between 2 AcP (days), R = resting period before the next AcP. Assessments item (Abbreviations; definition referred to Table 1): 1 LE = Objective evaluation of duration for 1 leg standing. 2LE = Objective evaluation of standing balance on 2 leg, Sd = Subjective evaluation of dynamic walking improvement (0∼5), Sm = Subjective evaluation of static motor improvement (0∼5), Ss = Subjective evaluation of sensory improvement (0∼5), Ws = Objective evaluation of walking speed (meters/s), Wt = objective evaluation of walking tolerance (min). 3 Lu et al. Medicine (2020) 99:29 Medicine the treatment outcomes. The most important findings in this case study include the remarkable improvement in the subjective assessment of dynamic balance (walking) and the objective assessment in 1 leg standing, an important indicator of static balance. Both improvement had continuous improvement even after cessation of acupuncture. In addition to subjective sensations of motor and sensory improvement, we also use several objective tests to evaluate the improvements after Yongquan (KI1) acupuncture treatment, including walking speed, walking tolerance and the standing stability with 1 leg and 2 legs support. The overall results indicate the positive effect of acupuncture treatment with Yongquan (KI1) except the walking speed. The walking speed remains a steady performance. According to the patient, the subjective feelings of walking balance (dynamic balance) was remarkable, unfortunately we did not design a quantitative and objective measurement of dynamic balance. The subjective feelings of motor and sensory improvement are both also remarkable and immediate increased after acupuncture. Though the effects declined gradually as time goes by, but compare with the baseline data on 2017-12-29, the improvement of subjective feelings of motor and sensory is still marked. Another important finding is the improvements in the walking tolerance. According to the above data, the patient could only walk without assistance for 10 minutes initially, but the walking time gained gradually with treatment. After complete the whole acupuncture treatment course, the patient can tolerate 60 minutes walking without assistance. Regarding to the dynamic balance, standing with 2 legs and 1 leg supported are chosen to evaluate the treatment results. The improvements of dynamic balance are observed after acupuncture with Yongquan (KI1), especially 1 leg standing balance. Though the patient can stand with 2 legs without assistance at initial assessment, he cannot resist to the pushing. After treatment, the patient feels subjectively that there is only little improvement in the 2 legs supported balance, but able to resist pushing force is noticed. As for the 1 leg standing balance, the patient can barely stand with 1 leg (0 sec) when first evaluate on 2017-12-29. After treatment, the duration of standing with 1 leg support can lasting for 0.75 sec. It’s worth noting that, the improvement of dynamic balance can be sustained after treatment without declined. Lower limbs somatosensory impairment has negative effects on walking tolerance and dynamic balance, etc. Whereas those factors: walking tolerance and dynamic balance play important roles of ambulation and are correlated with the activity of daily living (ADL) performance. Thus, when the patient’s foot sensory input regained after acupuncture therapy with Yongquan (KI1), the functional performance also improved. The sequelae of stroke, such as hemiplegia, somatosensory impairment, and impaired balance may cause significant disability and may affect patients’ quality of life.[8–10] Though patients may regain the ability to walk after undergoing rehabilitation, but still have higher risk to accidental falls due to impaired balance.[9,11] Balance deficits may also affect ADL functions such as transfers, ambulation, bath room activity, and so on. Balance can be influenced by many factors including motor function, sensory function, vestibular function, visual function, etc.[11–13] However, patients with sensory impairment seems tend to have worse functional recovery.[14] In this case, the patient’s walking ability recovered after receiving rehabilitation programs for 3 years, but the impairment in proprioception and dynamic balance persisted. The perception and dynamic balance had significantly improved after patient received acupuncture therapy, especially the acupuncture point of Yongquan (KI1). Previous reports about the prevalence of somatosensory deficits in chronic stoke patents varied in a wide range which probably related to the inadequate precise standard measurements of somatosensory deficits.[15–17] However, some authors believed that, somatosensory impairments are common in chronic stroke patients and which may influence the functional performance.[15,17] In chronic stroke patients, rehabilitation is often terminated when additional improvement has been reached “plateau” which may occur 6 months later after stroke.[17–19] However, the exact recovery course of somatosensory impairments is still not well documented, also, there are still no strong evidences can proof that the therapeutics of rehabilitation can effectively improve the somatosensory deficits.[17,20] In addition to rehabilitation programs, acupuncture therapy has been applied to stroke patients and is recommended as a complementary therapy in stroke rehabilitation.[8,21–24] The clinical effect of acupuncture in combination with conventional rehabilitation therapy for neurological impairment recovery, post-stroke shoulder pain, aphasia, improving ADL performance and improving post-stroke imbalance was explored.[8,21–26] Some researchers reported evidences of increase cerebral blood flow in sensorimotor area and activation of somatosensory cortex in stroke patients after acupuncture therapy, by using studies with images, single photon emission computed tomography (SPECT) perfusion and functional MRI to illustrate. Those studies demonstrated the therapeutic effect of acupuncture in stroke rehabilitation.[11,14,27,28] According to the theory of acupuncture in Traditional Chinese Medicine, acupuncture can direct Qi to the site that is lacking Qi, enhance the restoration of deficits.[4] Previous studies have indicated that acupuncture therapy is effective in improving balance function.[8,11,13,28,29] In some cases, the effectiveness can occur immediately after acupuncture. It also can affects the sensory perceptions. The selection of acupuncture points usually depends on the physicians’ personal experience in clinical practice, or the presenting symptoms of each individual patient.[22] Acupuncture points, Baihui (GV20), Yanglingquan (GB34) and Zusanli (ST36) are often chosen for treating the impairment of balance or coordination in stroke patients.[8,21,28,30] As for Yongquan (KI1), related studies are few,[30] but in ancient Chinese medicine theory, Yongquan (KI1) can treat conscious disturbance, cognitive function impairment, sole unable to touch the ground.[7] In a recent study conducted by Zhang’s colleagues,[31] they found that acupuncture at the Yongquan acupoints may induced stronger neuronal activity and increase synaptic activity in some areas of the brain. Long-term acupuncture may affect the quantity and function of synapses in brain area, leading the neural reorganization. 4. Conclusion This is just a single case study, and hopefully this study can facilitate more studies in this aspect. From this case, we have observed the effects and benefits of treating chronic stroke patient with Yongquan (KI1). From the pilot case study, more welldesigned investigations may be performed in the future. Furthermore, precise quantitative and objective measurements to evaluate treatment outcomes should be designed in the future studies. 4 Lu et al. Medicine (2020) 99:29 www.md-journal.com [15] Gorst T, Rogers A, Morrison SC, et al. The prevalence, distribution, and functional importance of lower limb somatosensory impairments in chronic stroke survivors: a cross sectional observational study. Disabil Rehabil 2018;41:1–8. [16] Connell LA, Lincoln NB, Radford KA. Somatosensory impairment after stroke: frequency of different deficits and their recovery. Clin Rehabil 2008;22:758–67. [17] Kessner SS, Bingel U, Thomalla G. Somatosensory deficits after stroke: a scoping review. Top Stroke Rehabil 2016;23:136–46. [18] Page SJ, Gater DR, Bach-Y-Rita P. Reconsidering the motor recovery plateau in stroke rehabilitation. Arch Phys Med Rehabil 2004;85: 1377–81. [19] Green JB. Brain reorganization after stroke. Top Stroke Rehabil 2003;10:1–20. [20] Pumpa LU, Cahill LS, Carey LM. Somatosensory assessment and treatment after stroke: an evidence-practice gap. Aust Occup Ther J 2015;62:93–104. [21] Chavez LM, Huang SS, MacDonald I, et al. Mechanisms of acupuncture therapy in ischemic stroke rehabilitation: a literature review of basic studies. Int J Mol Sci 2017;18(11.pii):E2270. [22] Wu P, Mills E, Moher D, et al. Acupuncture in poststroke rehabilitation: a systematic review and meta-analysis of randomized trials. Stroke 2010;41:e171–9. [23] Rabinstein AA, Shulman LM. Acupuncture in clinical neurology. Neurologist 2003;9:137–48. [24] Zhang JH, Wang D, Liu M. Overview of systematic reviews and metaanalyses of acupuncture for stroke. Neuroepidemiology 2014;42:50–8. [25] Zhang Y, Liu H, Fu C, et al. The biomechanical effect of acupuncture for poststroke cavovarus foot: study protocol for a randomized controlled pilot trial. Trials 2016;17:146. [26] Park J, Hopwood V, White AR, et al. Effectiveness of acupuncture for stroke: a systematic review. J Neurol 2001;248:558–63. [27] Lee JD, Chon JS, Jeong HK, et al. The cerebrovascular response to traditional acupuncture after stroke. Neuroradiology 2003;45:780–4. [28] Chang QY, Lin YW, Hsieh CL. Acupuncture and neuroregeneration in ischemic stroke. Neural Regen Res 2018;13:573–83. [29] Baeumler PI, Fleckenstein J, Takayama S, et al. Effects of acupuncture on sensory perception: a systematic review and meta-analysis. PLoS One 2014;9:e113731. [30] Li L, Zhang H, Meng SQ, et al. An updated meta-analysis of the efficacy and safety of acupuncture treatment for cerebral infarction. PLoS One 2014;9:e114057. [31] Zhang H, Sun X, Liu S, et al. Neuronal activation by acupuncture at Yongquan (KI1) and sham acupoints in patients with disorder of consciousness: a positron emission tomography study. Neural Regen Res 2014;9:500–1. Author contributions Methodology: Chang-Zern Hong. Supervision: Li-Wei Chou. Writing – original draft: Kuan-Yu Lu, Ka-Fai Yuen, Jia-Yuan Luo. Writing – review & editing: Chang-Zern Hong, Li-Wei Chou. References [1] Spinazzi M, Angelini C, Patrini . Subacute sensory ataxia and optic neuropathy with thiamine deficiency. Nat Rev Neurol 2010;6:288–93. [2] Sghirlanzoni A, Pareyson D, Lauria G. Sensory neuron diseases. Lancet Neurol 2005;4:349–61. [3] Moeller JJ, Macaulay RJ, Valdmanis PN, et al. Autosomal dominant sensory ataxia: a neuroaxonal dystrophy. Acta Neuropathol 2008; 116:331–6. [4] Wang Wei Shan: Science of Acupuncture. 1st Ed. Cheng Chung Bookstore, 1987:117–122. [5] Wang Wei Shan: Science of Acupuncture. 1st Ed. Cheng Chung Bookstore, 1987:474–475. [6] Sam Stuart: Anatomical Atlas of Chinese Acupuncture Points. 1st Ed. Shandong Science and Technology, 1982:26–40. [7] Wang Wei Shan: Science of Acupuncture. 1st Ed. Cheng Chung Bookstore, 1987:326. [8] Huang SW, Wang WT, Yang TH, et al. The balance effect of acupuncture therapy among stroke patients. J Altern Complement Med 2014;20:618–22. [9] Berg K, Wood-Dauphine S, J.I. Williams JI, et al. Measuring balance in the elderly: preliminary development of an instrument. Physiother Can 1989;41:304–11. [10] Feigenson JS, McDowell FH, Meese P, et al. Factors influencing outcome and length of stay in a stroke rehabilitation unit. Part 1. Analysis of 248 unscreened patients-medical and functional prognostic indicators. Stroke 1977;8:651–6. [11] Liu SY, Hsieh CL, Wei TS, et al. Acupuncture stimulation improves balance function in stroke patients: a single-blinded controlled, randomized study. Am J Chin Med 2009;37:483–94. [12] Niam S, Cheung W, Sullivan PE, et al. Balance and physical impairments after stroke. Arch Phys Med Rehabil 1999;80:1227–33. [13] Magnusson M, Johansson K, Johansson BB. Sensory stimulation promotes normalization of postural control after stroke. Stroke 1994;25:1176–80. [14] Li G, Jack CRJr, Yang ES. An fMRI study of somatosensory-implicated acupuncture points in stable somatosensory stroke patients. J Magn Reson Imaging 2006;24:1018–24. 5