Brown-Sequard syndrome after manual manipulation of the cervical spine: case report

Abstract

Introduction

Spinal cord injury after manual manipulation of the cervical spine is rare and has never been described resulting from a patient performing a manual manipulation on their own cervical spine. To the best of our knowledge, this is the first well-documented case of this association.

Case presentation

A healthy 29-year-old man developed Brown-Sequard syndrome immediately after performing a manipulation on his own cervical spine. Imaging showed large disc herniations at the levels of C4–C5 and C5–C6 with severe cord compression, so the patient underwent emergent surgical decompression. He was discharged to an acute rehabilitation hospital, where he made a full functional recovery by postoperative day 8.

Conclusion

This case highlights the benefit of swift surgical intervention followed by intensive inpatient rehab. It also serves as a warning for those who perform self-cervical manipulation.

Introduction

Brown-Sequard syndrome (BSS) was first described 1849 by Charles-Edouard Brown-Sequard, when he documented the constellation of symptoms a sea captain experienced after being stabbed in the neck [1]. BSS is caused by hemisection or hemi-compression of the spinal cord and presents with the following symptoms: ipsilateral loss of motor function, proprioception, and vibratory sense as well as contralateral loss of pain and temperature sensation [2]. Causes of BSS include traumatic injury, tumor, and hemorrhage with cervical disc herniation (CDH) being relatively rare [3]. We report a case of a man developing BSS from CDH after performing a manual manipulation of his own cervical spine.

Case report

A previously healthy 29-year-old African-American man presented to the emergency department with a sudden onset of right-sided weakness after “cracking” his neck. He denied any recent trauma, although he did report a remote history of a stabbing to the soft tissue of his neck as a child that only required closure of the wound with sutures. He did not have any neurological deficits after being stabbed, but he did report mild, persistent, neck stiffness since that point.

In order to manage this mild discomfort, he would perform manual manipulation of his own cervical spine. To do so, he would grasp his chin with one hand and place the other hand on the top of his head (Fig. 1). He would then forcefully bend his neck laterally while rotating his chin to the opposite shoulder until he elicited an audible “crack.” He would perform this maneuver twice: once bending his neck to the left while rotating his chin to the right, and once bending his neck right while rotating his chin to the left. He reports doing this several times per day for many years without any complications or pain.

Fig. 1. The author demonstrating how the patient performed manual manipulations.

The red arrows illustrate the rotary forces applied by each hand on the cervical spine.

On the date of his spinal cord injury, immediately after performing the maneuver, he felt a shooting pain go down his neck and instantly developed right-sided weakness prompting him present to the ED. His exam at that time was notable for right-sided hemiparesis and an abnormal sensory exam. Using the Medical Research Council Manual Muscle Testing scale, strength in right upper extremity was 3/5 shoulder abduction, 2/5 elbow flexion, 2/5 elbow extension, 0/5 wrist flexion, 0/5 wrist extension, and 0/5 grip strength. In his right lower extremity, his strength was 3/5 hip flexion, 3/5 knee extension, 3/5 knee flexion, 2/5 ankle dorsiflexion, and 2/5 ankle plantar flexion. The strength in his left upper extremity and left lower extremity were completely intact. His sensory exam was notable for decreased sensation to pinprick on left side of his body beginning at the C5 sensory level and decreased vibratory sense on right side of his body beginning at the C4 sensory level.

An MRI of the cervical spine revealed large disc herniations at the levels of C4–C5 and C5–C6 with severe cord compression and T2 cord signal change. Also noted were chronic degenerative changes including facet arthropathy and endplate changes greater than expected for the patient’s age (Fig. 2). The patient was started on dexamethasone and taken to the operating room for an emergent C4–C6 anterior cervical discectomy and fusion (ACDF), 16 h after symptom onset. Direct visualization intraoperatively was remarkable for the C4–5 disc herniation, which had ruptured through the annulus and posterior longitudinal ligament (PLL), leaving large free fragments of the disc in the space between the PLL and the thecal sac. There was a similar disc herniation at the C5–6 level, but with much smaller free fragments. Additionally, the PLL was noted to be quite thickened behind the C5 vertebral body, leading to mild stenosis of the spinal canal.

Fig. 2. Preoperative cervical MRI.

Sagittal (A) MRI shows large herniated discs at C4–5 and C5–6 levels. Axial (B) MRI shows a transligamentous disc rupture from the midline to the right at the C4–5 level with severe spinal cord compression and T2 signal change.

Routine MRI was done on postoperative day 1 which showed resolution of previously noted severe spinal canal stenosis and cord compression. There was increased focal signal abnormality within the right hemicord at the C4–C5 level, likely reflecting evolving edema/contusion (Fig. 3).

Fig. 3. Postoperative cervical MRI.

Sagittal (A) and axial (B) MRI show T2 hyperintensity in the right hemicord at the C4–C5 level.

His strength rapidly improved postoperatively, and he was discharged to an acute rehabilitation hospital on postoperative day 2 with the plan to compete a 14-day dexamethasone taper. On admission to the acute rehabilitation hospital, the patients Activity Measure for Post-Acute Care (AM-PAC) score was measured using the “6-Clicks” Inpatient Short Forms for activities of daily living (ADLs) and mobility were 19 and 13 respectively, for a total score of 32 out of 48 possible points. The strength in his right upper extremity was 5/5 shoulder abduction, 5/5 elbow flexion, 5/5 elbow extension, 4/5 wrist flexion, 4/5 wrist extension, and 3/5 grip strength. The strength in his right lower extremity was 5/5 hip flexion, 5/5 knee extension, 5/5 knee flexion, 3/5 ankle dorsiflexion, and 3/5 ankle plantar flexion. His sensory exam was unchanged from his preoperative exam, with decreased sensation to pinprick on left side of his body beginning at the C5 sensory level and decreased vibratory sense on right side of his body beginning at the C4 sensory level. He worked diligently with the therapy team and made significant improvements. After 6 days of inpatient rehabilitation (postoperative day 8), the only residual weakness found on exam was in his right-hand grip strength, which was 4/5. His sensation, however, had not improved as of yet. The patient was completely independent from a functional standpoint with an AM-PAC of 48, so he was discharged home with the plan to continue outpatient therapy. At follow up 2 months later, his strength had completely recovered, although he continued to note decreased sensation to light touch in the right upper extremity.

Discussion

BSS is classically characterized by ipsilateral loss of motor function, proprioception, and vibratory sense, as well as contralateral loss of pain and temperature sensation. Physical exam findings may vary in severity and character depending on the level of injury and size of the lesion, thus Brown-Sequard plus syndrome is used to describe non-classical cases [4]. For example, a patient with a hemicord lesion in the cervical spine could present with any of the following deficits:

• Motor deficits: Disruption of the anterior horn cells will lead to ipsilateral lower motor neuron weakness at the level of the lesion. Disruption of the corticospinal tracts will lead to ipsilateral upper motor neuron weakness below the level of the lesion.

• Sensation deficits: Disruption of the dorsal columns will lead to ipsilateral loss of conscious proprioception, light touch, and vibration sense at and below the level of the lesion. Disruption of the spinothalamic tracts will lead to contralateral loss of pain, temperature, and crude touch beginning 2–3 levels below the lesion, and ipsilateral loss of pain, temperature, and crude touch at the level of the lesion. Disruption of the dorsal spinocerebellar tract will lead to ipsilateral dystaxia. Disruption of the ventral spinocerebellar tract will lead to contralateral dystaxia.

• Autonomic deficits: Disruption of 2nd order sympathetic fibers descending from the hypothalamus will lead to ipsilateral Horner’s syndrome [2].

Spinal cord injury occurring after manual manipulation of the cervical spine is rare, with a recent systematic review only identifying 32 cases [5]. We performed a comprehensive review of the current literature to identify cases of BSS occurring after manual manipulation of the cervical spine and were able to identify 11 cases (Table 1). Of the 11 cases, only 4 were caused by CDH, while the others were caused by spinal epidural hematoma (SEH) or aberrant anatomy leading to a spinal cord contusion. The average age of those with CDH as the underlying pathology was 49, with 75% of them being female. 3 of the 4 cases reported no prior trauma while the 4th case did not comment on prior trauma. 2 of the 4 cases did not discuss time from symptom onset to surgical intervention, while the other 2 cases describe at least a few days having elapsed before surgery. All 4 cases of CDH were treated with ACDF. 2 of the 4 cases had a full return of strength at 12 weeks, while the other 2 cases had incomplete recovery. In all the cases that have previously been documented, the manual manipulation was performed another individual, presumably by a licensed practitioner [6–15].

Table 1.

Reported cases of BSS following manual manipulation of the cervical spine including the case described in this report.

N	Reference	Prior symptoms	History of trauma	Age	Sex	Level	Weakest motor group at presentation (MMT)	Tx	Weakest motor group upon recovery (MMT)	Time to recovery (Wks)	Dx
1	Our patient	MNP	No	29	M	C4–C5	0/5	ACDF	5/5	11	CDH
2	[6]	MNP	NR	61	F	C3–C4	3/5	ACDF	5/5	12	CDH
3	[7]	MNP w/ Rad	No	46	F	C4–C6	NR	ACDF	Incomplete	16	CDH
4	[8]	MNP	No	35	F	C5–C6	NR	ACDF	5/5	12	CDH
5	[8]	MNP	No	55	M	C4–C5	NR	ACDF	Incomplete	NR	CDH
6	[9]	MNP	Major	83	M	C1–C2	0/5	ORIF	3/5	8	Other
7	[10]	MNP	No	58	F	C4	3/5	Steroids only	Incomplete	15	Other
8	[11]	MNP	Minor	33	F	C4–C6	3/5	Lami	Incomplete	0.5	SEH
9	[12]	MNP	NR	67	F	C3–C5	1/5	Lami	5/5	52	SEH
10	[13]	MNP	No	55	M	C1–C5	1/5	Steroids only	4/5	0.2	SEH
11	[14]	None	No	86	M	C2–C7	0/5	Lami	5/5	12	SEH
12	[15]	MNP	No	52	F	C3–T1	NR	Lami	5/5	26	SEH

ACDF anterior cervical discectomy and fusion, CDH cervical disc herniation, Dx diagnosis, Lami laminectomy, MMT manual motor testing, MNP mild neck pain, MNP w/ Rad mild neck pain with radicular symptoms, NR not recorded, Wks weeks, SEH spinal epidural hematoma.

The other 4 case reports of CDH causing BSS don’t explicitly state the amount of time that elapsed from symptom onset to surgical intervention, nor the type of rehabilitation that was completed by patients after discharge from the acute care hospital, but our case highlights the potential benefit of swift surgical intervention followed by an intensive inpatient rehabilitation program [16, 17]. In our case, the time from symptom onset to surgical intervention was 16 h and the patient completed the most intensive form of physical rehabilitation after his procedure, an inpatient rehabilitation program at an acute rehabilitation hospital. Of the 4 other cases, our patient had the quickest return to full strength despite presenting with the most significant weakness (Table 1). His AM-PAC improved by 16 points over his 7-day inpatient rehabilitation course, going from 32 (needing “a little” to “a lot” of help with all ADLs and mobility) to 48 (independent with all ALDs and mobility) without the use of an assistive device, making his recovery even more remarkable.

To the best of our knowledge, the current case is the first reported case of a spinal cord injury resulting from a patient performing a manual manipulation on their own spine. Although this is exceedingly rare, it highlights the risk of aggressive manipulation of the cervical spine. The chronic degenerative changes seen throughout the patient’s cervical and upper thoracic spine, along with the thickened PLL noted intraoperatively, are quite pronounced for his age. Though highly speculative, these findings may indicate that he was causing subclinical trauma to his joints and discs every time he performed his manual manipulation, eventually leading to disc rupture and his presentation. Conversely, his chronic degenerative changes may have been leading to discomfort and thus the reason he performed cervical manipulations. Regardless, this case serves as a potential red flag to those who perform self-cervical manipulation, particularly in the setting of degenerative changes.

Patient perspective

The patient expressed gratitude to all who worked on his case and wanted it to be published so others who undergo frequent cervical spine manipulation know the potential risks involved. Regarding his treatment, he enjoyed his inpatient rehabilitation course and felt like his function greatly improved because of it.

Data availability

All data generated or analyzed during this study are included in this published article.

Competing interests

The authors declare no competing interests.

Consent for publication

The patient consented to the publication of this case report.

Supplementary information

The online version contains supplementary material available at 10.1038/s41394-022-00501-1.