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Interdisciplinary Neurosurgery: Advanced Techniques and Case Management 2 (2015) 57–60

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Case Reports & Case Series (CRP) Cervical selective dorsal rhizotomy for treating spasticity in upper limb neurosurgical way to neurosurgical technique☆

Yu Duan a, Xiaoqi Luo b, Xing Gao b, Chengyan Sun a,b,⁎

a Department of Cerebral Palsy Center, Sichuan Rehabilitation Hospital, Chengdu, Sichuan, China b Department of Functional Neurosurgery, Shanghai Tong Ren Hospital, 1111, Xianxia road, Changning District, Shanghai 200336, China

article info abstract

Article history: Selective dorsal rhizotomy is an effective method to reduce spasticity of the lower limbs. However, functional Received 16 September 2014 outcomes in the upper limb following selective dorsal rhizotomy at the cervical level have not been reported. Revised 20 December 2014 Here we report the clinical course after selective dorsal rhizotomy at the cervical level in a patient with Accepted 20 December 2014 hemiplegic spasticity caused by brain injury. The selective dorsal rootlets at the cervical level were sectioned Available online xxxx under electrophysiological monitoring. The patient was followed for 1 year to evaluate the outcome of Keywords: surgery. The spasticity in the upper limb was reduced and the passive range of motion and function of Selective dorsal rhizotomy movement improved. However, the effectiveness and the safety of operation should be studied further in Spasticity clinical trials. Upper limb © 2014 The Authors. Published by Elsevier B.V. This is an open access article under the CC BY-NC-ND license Functional neurosurgery (http://creativecommons.org/licenses/by-nc-nd/4.0/).

Introduction dorsal rhizotomy (CSDR) could reduce spasticity in the upper limb and improve the function. In this case report, we performed CSDR for a Spasticity is a component of the upper motor neuron syndrome, 23-year-old patient with serious hemiplegic spasticity after brain and has been defined as a velocity-dependent increase in resistance to injury and had 12-months of follow-up. the passive stretch of muscles [1]. Some patients suffering from mild to moderate spasticity can be treated by conservative methods Clinical history including functional training, oral antispastic , and botulinum toxin injections. However, a substantial number of patients The patient was a 23-year-old male. He underwent a severe traffic have been resistant to these conservative interventions. accident which resulted in left temporoparietal hematoma and brain Selective dorsal rhizotomy (SDR), a neurosurgical approach to contusion three years ago. The patient received craniotomy to evacuate sever sensory nerve roots, is thought to decrease both the excessive the hematoma and contusion tissue 12 h after the injury. The patient afferent input to intramedullary neurons and the excitatory output of recovered from the operation in a short period while right hemiplegic α-motor neurons. SDR is a well-known and efficacious therapeutic paralysis took out gradually. Stretch and functional training for the patient option for reducing spasticity in children with cerebral palsy or other were started one month after the brain injury in our hospital. Six months diseases [2,3]. It also has been demonstrated to improve motor after the brain injury, the patient was treated with series botulinum toxin function [4] and the performance of the activities of daily living [5], type A injections, which showed limited effectiveness for the function of and decrease the need for orthopedic surgery [6]. right limbs. One year after the brain injury, the spasticity in his right lower Thirty years ago, cervical SDR was mentioned in treating a patient limb was reduced through SDR at the lumbar level and the equinus with hemiplegia caused by cerebral vascular accident. The spasticity deformity was corrected subsequently by an Achilles tendon lengthening of upper limb was reduced obviously, but the function worsened and procedure in our hospital. Rehabilitation for the patient lasted for 2 years, hardly recovered [7]. After SDR at lumbar level, patients sometimes while severe spasticity and limited ROM still remain in the right upper would gain the “suprasegmental benefits” such as reduction in the limb. Both active and passive functions of his right upper limb were spasticity in upper limb and the unexpected improvement in the affected; to the patient, picking up a glass or holding a book to read was function of upper limb [8,9]. It makes that cervical selective impossible; raising or abducting cleaning the right upper limb or putting the right arm in sleeve was very difficult. The patient was willing to receive CSDR to relieve the spasticity in the upper limb. ☆ Conflict of interest statement: The authors declare that they have no competing financial interests and personal relationships with other people or organizations that Evaluation can inappropriately influence our work. ⁎ Corresponding author. Tel.: +86 13512135189; fax: +86 28 21 62908857. The patient was evaluated 1 week before surgery and 1 month, 6 E-mail address: [email protected] (C. Sun). months, and 12 months after surgery. The modified Ashworth Scale http://dx.doi.org/10.1016/j.inat.2014.12.003 2214-7519/© 2014 The Authors. Published by Elsevier B.V. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/). 58 Y. Duan et al. / Interdisciplinary Neurosurgery: Advanced Techniques and Case Management 2 (2015) 57–60

(Fig. 2). The C5, C6 and C7 rootlets were sectioned at 35%, 40% and 35%, respectively. Patient was not instrumented or fused at time of the surgery. The incision was covered with pressure dressing for 12 h and cervical fixation was used for three weeks after surgery.

Postoperative rehabilitation The postoperative rehabilitation items were similar to those received preoperatively. After CSDR, the patient got intensive inpatient training for three months, including passive stretch, CMIT and bilateral arm training. The home-CMIT was guided by our therapists during the following nine months. One year postoperatively, the cervical spine X-rays (posterior, flexion and extension) were done to look for instability. The stability of vertebral body was not affected (Fig. 3).

Fig. 1. Intraoperative photographs of cervical selective dorsal rhizotomy. DR = dorsal rootlets, AR = anterior rootles, CSC = cervical . Results

(MAS), the passive range of motion (PROM) assessment, and the Overall, the shoulder and elbow Ashworth scores were obviously Fugl–Meyer assessment in the upper limb were performed by an decreased. Preoperatively, the patient had a MAS score of 3 out of 4 in independent senior occupational therapist. muscle groups of both shoulder and elbow. One month following the surgery, the MAS scores for shoulder adduction, abduction, and extension had decreased to 1 (out of 4) and the MAS sores for the Surgical procedure elbow decreased to 2 (out of 4). After twelve months of training, only fl For general anaesthesia, midazolam, penehyclidine hydrochloride the MAS score for elbow exion was stable with a score of 2; the other and granisetron hydrochloride were used during anesthetic induction. muscle groups at the shoulder and elbow decreased to scores of 1. fl During the surgery, sevoflurane and remifentanil were sustained. Both wrist exion and extension were initially scored at 4 (of 4). fl Specially, any muscle relaxants were not used to keep the excitability Following the CSDR, the MAS for wrist extension and wrist exion of the muscle. The patient was fixed in left-lateral position. The decreased to 3 at one month postoperatively. Since then, the scores hemilaminectomies from the fifth cervical vertebra to the seventh had not changed (Table 1). cervical vertebra were performed. The cervical spinal cord and the C5 The score results of PROM of the shoulder, elbow, and wrist are to C7 dorsal roots were exposed and divided into rootlets. The shown in Fig. 4. After 12 months of functional training, the PROM in – fl – procedure involved the C5 to C7 dorsal roots. The dorsal roots were shoulder adduction abduction and shoulder exion extension in- – separated from the anterior roots. C5, C6, and C7 were identified creased by 6° and 8° respectively. The PROM of elbow extension fl –fl according to their foramen intervertebrales (Fig. 1). The neurophy- exion increased by 11° and wrist extension exion by 4° (Fig. 4). – siologist monitored muscular responses to electrical stimulations of The Fugl Meyer assessment showed a gradual increase in scores at the sensory rootlets during the operation. The electromyographic three months after surgery, especially at shoulder function, forearm recording was performed using needle electrodes applied to the pronation, and elbow extension (Fig. 5). After 12 months of CSDR, the pectoralis major, subscapularis, biceps, pronator teres, flexor carpi patient began to pick up a glass or hold a book to read, even though fi ulnaris, and flexor carpi radialis. Intraoperative electromyography with dif culty; raising, abducting, cleaning the right upper limb or was recorded after a one second 50 Hz stimulation at 3 mA and the putting the right arm in sleeve was easier than before. pulse duration at 3 s of individual dorsal rootlets. Generally, clonic or bilateral responses were considered abnormal [10]. Because the Complications contralateral limb was not monitored, the sustained and extensive responses in electromyography and clonic responses in muscle groups One day after surgery, the patient felt numb in the radial forearm of the right upper limb were defined “abnormal” in this surgery and shoulder and the sensation disappeared at 3 weeks. There was no

Fig. 2. Intraoperative electromyography recordings after stimulation of individual dorsal rootlets. (A) Sustained and extensive muscle responses occurred when “abnormal” C6 dorsal rootlet was stimulated. (B) The electromyography of “normal” C6 dorsal rootlet. FCR = flexor carpi radialis, PT = pronator teres, PM = pectoralis major, SS = subscapularis, BI = biceps, FCU = flexor carpi ulnaris. Y. Duan et al. / Interdisciplinary Neurosurgery: Advanced Techniques and Case Management 2 (2015) 57–60 59

Fig. 4. The passive range of motion in the upper limb. SAA = shoulder adduction to adbuction, SFE = shoulder flexion to extension, WFE = wrist flexion to extension.

multiple neurorehabilitation techniques were conducted [11,12]. For instance, bilateral arm training, one of the active neurorehabilitation treatments, shows less effectiveness when being compared with other treatments [13]. Stretch can prevent secondary deformities in certain degree, and robot can provide the high-intensity, accuracy and task-specific movements with repetitions, both of which might contribute to reduce the spasticity and improving the joint mobility [14,15], although some clinic studies showed inconsistencies in results [16,17]. In our case, the patient got little improvement from regular rehabilitation including passive stretch, Constraint-induced movement therapy (CMIT) and bilateral arm training. The therapeutic effects of botulinum toxin have been studied extensively in the upper limb [18]. Botulinum toxin injections are effective in managing adult spasticity in the upper limb and improving passive function [19]. However, botulinum toxin injections have been commonly used for focal upper limb spasticity [20], especially for the tone in the distal muscle groups; meanwhile, its effectiveness in improving active function has been quite controversial [21].Inourcase,thepatientacceptedtwo years of systematic rehabilitation for his spastic right upper limb, including CMIT, bilateral arm training and botulinum toxin injections, but the effectiveness was still limited. Some studies have reported that intrathecal baclofen (ITB) therapy as a reversible way can be applied to the patients with hemiplegic paralysis; it can reduce the spasticity in the lower limb and improve the walking ability [22,23] and also impacts the spasticity in upper limb equally as in lower limb [24]. However, baclofen for intrathecal injection is not available in China; thus, ITB was not considered in this case. SDR has been widely performed to reduce spasticity in the lower limbs and effectively improve mobility, particularly in patients with cerebral palsy, hereditary spastic paraparesis [25], traumatic brain injury [26], spinal cord injury [27],andstroke[28]. In adult patients after SDR, the passive range of motion improved, the tone in the muscle groups in the upper limb reduced, and the activities of daily living Fig. 3. The cervical spine X-rays after one year of CSDR. (A) Posterior position, the opening of improved [29]. SDR at the lumbar level also can reduce spasticity in the hemilaminectomy in the black grid. (B) Flexion position. (C) Extension position. upper limb, and more than 90% of patients with spastic quadriplegia showed the improvement in the upper limb tone in 2 or more muscle change on other proprioceptive or superficial sensory, and the groups after surgery [8]. function of the right limb did not worsen. Cervical dorsal rhizotomy or dorsal root entry zone operation (DREZ) is performed to relieve the in patients with . Discussion After the dorsal rootlets on C1, C2, C3, and C4 were sectioned, patients with refractory occipital neuralgia could feel pain relief and Spasticity is considered to be the important factor hampering improvement in function [30,31]. Dorsal root entry zone lesion is an patients’ functional recovery, especially in the upper limbs, even if effective intervention for the pain of brachial plexus avulsion [32,33].

Table 1 The tone of the muscle groups in upper limbs before and after CSDR.

MAS Pre 1 M 3 M 6 M 12 M

shoulder adduction 3 1 1 1 2 shoulder abduction 3 1 1 2 2 shoulder flexion 3 3 2 2 2 shoulder extension 3 1 1 1 1 elbow flexion 3 3 3 2 2 elbow extension 3 2 2 3 2 wrist flexion 4 3 3 3 4 wrist extension 4 3 3 3 3

Pre: Pre-operation, M: months. Fig. 5. The Fugl–Meyer assessment in upper limb. 60 Y. Duan et al. / Interdisciplinary Neurosurgery: Advanced Techniques and Case Management 2 (2015) 57–60

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