Case Report
 
Spinal cord injury without radiological abnormality manifested as self-limited brown-sequard syndrome
Yudha Mathan Sakti1, Marda Ade Saputra2, Tedjo Rukmoyo1, Rahadyan Magetsari3
1Staff of Orthopaedic & Traumatology Department, Gadjah Mada University, Sardjito Hospital, Yogyakarta
2Resident of Orthopaedic & Traumatology Department, Gadjah Mada University, Sardjito Hospital, Yogyakarta
3MD, PhD, Orthopaedic and Traumatology Department, Faculty of Medicine, Gadjah Mada University, Yogyakarta, Indonesia

Article ID: 100902Z01SA2018
doi: 10.5348/100902Z01SA2018CR

Corresponding Author:
Marda Ade Saputra,
Resident of Orthopaedic & Traumatology Department,
Gadjah Mada University, Sardjito Hospital, Yogyakarta

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How to cite this article
Sakti YM, Saputra MA, Rukmoyo T, Magetsari R. Spinal cord injury without radiological abnormality manifested as self-limited brown-sequard syndrome. Int J Case Rep Images 2018;9:100902Z01SA2018.


ABSTRACT

Introduction: Combination between spinal cord injury without radiological abnormality (SCIWORA) and Brown-Sequard syndrome in a patient is a rare condition. In SCIWORA, there is usually a delay in neurologic deficits which can potentially lead to misdiagnosis. Therefore, the clinician should have a good understanding of the course of the disease to make a good diagnosis and treatment.

Case Report: Reporting a case of female 20-year-old with chief complaint of severe neck pain and delayed limbs weakness. The mechanism of injury was fall with the head hit the ground in left lateral flexion position. The physical examination showed zero motor power of the right limbs and contralateral pain and temperature deficit one hour after the injury. We diagnosed the patient with incomplete spinal cord injury at C4 level with associated Brown-Sequard syndrome. We gave soft collar neck for immobilization, medication with NSAID for analgetic and Methylprednisolon. We found dramatic improvement in 10 hours after the injury with motor improvement from 0–5 and normal sensory function. The patient was then discharged with good functional outcome and with no sequelae.

Conclusion: Incomplete cervical spinal cord injury without radiological abnormality that manifested as Brown-Sequard syndrome is a rare case and potentially confusing condition. Better understanding of the course of the disease may help the clinician to make a right diagnosis and plan for management.

Keywords: Brown-Sequard syndrome, Spinal cord injury, Spinal cord injury without radiological abnormality


INTRODUCTION

Spinal cord injury without radiological abnormality (SCIWORA) is defined as the occurence of acute traumatic myelopathy despite normal plain radiographs and normal computed tomography (CT) studies. This occur predominantly among pediatric population with incidence range from 4–66% of all spinal cord injuries (10–20% of all pediatric spinal trauma) [1]. In young children, the pathogenesis of SCIWORA may be related to the mismatch in the elasticity of the tissue of the vertebral column and spinal cord [1], [2], [3]. The mechanism of injury could be direct or indirect spinal cord traction or compression and vascular or ischemic injury [1], [3], [4]. SCIWORA have a large spectrum of neurologic deficit, ranging from mild, transient until complete spinal cord injuries. The neurologic deficits can happen in delayed form, ranging from hours to days after the injury [1], [3],[5].

Brown-Sequard syndrome is an anatomic hemisection of the cord, resulting in ipsilateral motor and proprioception loss and contralateral pain and temperature deficit. It is a rare condition, as the trauma or something should damage the nerve fibers on just one half of the spinal cord. Fortunately, the prognosis for significant motor recovery is good and the most important prognostic variable relating to neurologic recovery is completeness of the lesion [6].

Combination betwen SCIWORA and Brown-Sequard syndrome is a rare condition that can lead to misdiagnosis. Therefore the clinicians should have a good awareness and understanding of the disease to make a right diagnosis and therapy for the patients.


CASE REPORT

A 20-year-old female presented with chief complaint of severe neck pain and delayed limbs weakness after suffering from sport injury in martial arts competition. The mechanism of injury was fall with the head hit the ground first in left lateral flexion position. No history of loss of consciousness. After the accident, the patient felt neck pain and about one hour after the accident she could not move her right limbs.

From physical examination, the general condition was good, the vital signs were within the normal limits. The motor functions of the right upper and lower extremities from C5 – S1 level were decreased to zero with no muscle tone and the sensory functions of the left side (contralateral side) below the C4 level were decreased with loss of pain and temperature sensation. The ASIA scores were 56 for sensory functions and 50 for motor functions and the functional score was 4 for Nurick scale that means unable to walk without assistance. We performed thorough diagnostic approach including X-ray, CT scan and MRI investigation (Figures 1 and 2). From plain X-ray and CT scan there was no visible fracture or dislocation and the alignment was good. The patient was diagnosed with incomplete spinal cord injury at C4 level with associated Brown-Sequard syndrome manifestation. The patient was observed and managed with soft collar neck for immobilization, medication with NSAID for analgesia and Methylprednisolon 30 mg/kg in 15 minutes followed by maintenance with 5,4mg/kg/h for the next 23 hours, and we found dramatic improvement in 10 hours after the injury with motor improvement from 0–5 in both affected extremity and normal sensory function in the contralateral side. The nurick scale improved to 1 that means the patient had no difficulty in walking. The patient was observed for the next 24 hours and given rehabilitation program. The patient was then discharged with good functional outcome and with no sequelae.


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Figure 1: Anterior-posterior and Lateral view of Cervical Spine X-ray. There was no fracture and dislocation. The alignment and pretracheal soft tissue even looks normal.


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Figure 2: Cervical CT scan and MRI. There was no fracture and dislocation found in the CT scan. From sagittal view of the cervical MRI the spinal cord looked good.


DISCUSSION

Spinal Cord Injury Without Radiographic Abnormality (SCIWORA) was first introduced by Pang and Wilberger and was first reported by Burke in 1974. It is used to define clinical symptoms of traumatic myelopathy with no radiographic or CT abnormality. The symptoms have a broad spectrum from mild and transient paresthesia in fingers to quadriplegia. The symptoms can appear at the time of injury but can also be delayed upto several days of injury. The main therapeutic treatment is external immobilization of the spine for up to 12 weeks [5]. Incomplete cervical spinal cord injury without radiological abnormality could be potentially confusing and frustating because there could be a delay in neurologic deficit and the course of the disease can be very dramatic. In this case, SCIWORA manifested as Brown-Sequard syndrome, which is a rare condition. The neurologic status and the imaging studies of the patient were initially normal but suddenly worsened very fast placing the patient and families in a big worry. And then, without any surgical intervention, there was surprisingly fast recovery time and excellent functional outcome. The delay in neurologic deficit needs full and thorough observation. We used soft collar neck for immobilization, high dose of intravenous corticosteroid, intravenous NSAID agents, and rehabilitation as the management approach. The prognosis is related to the severity of the spinal cord dysfunction. Outcome after incomplete injuries in older children is excellent [1]. The patient age and the first clinical presentation after the injury might be the clinical predictors for this patient to have a good recovery.


CONCLUSION

Incomplete cervical spinal cord injury without radiological abnormality that manifested as Brown-Sequard syndrome is a rare case and potentially confusing condition because of the delay in neurologic deficit and the course of the disease that was very dramatic. Better understanding of the course of the disease may help the clinician to make the right diagnosis and plan management with better explanation and education to the patient and the frustated families.


REFERENCES
  1. Dickman CA, Zabramski JM, Hadley MN, Rekate HL, Sonntag VK. Pediatric spinal cord injury without radiographic abnormalities: Report of 26 cases and review of the literature. J Spinal Disord 1991 Sep;4(3):296–305.   [PubMed]    Back to citation no. 1
  2. Pang D, Sahrarkar K, Sun PP. Pediatric spinal cord and vertebral column injuries. In: Youman JR, editor. Neurological Surgery. 4ed. Philadelphia: WB Saunders; 1996. p. 1991–2037.    Back to citation no. 2
  3. Pang D, Pollack IF. Spinal cord injury without radiographic abnormality in children: The SCIWORA syndrome. J Trauma 1989 May;29(5):654–64.   [CrossRef]   [PubMed]    Back to citation no. 3
  4. Slack SE, Clancy MJ. Clearing the cervical spine of paediatric trauma patients. Emerg Med J 2004 Mar;21(2):189–93.   [CrossRef]   [PubMed]    Back to citation no. 4
  5. Burke DC. Traumatic spinal paralysis in children. Paraplegia 1974 Feb;11(4):268–76.   [CrossRef]   [PubMed]    Back to citation no. 5
  6. Pollard ME, Apple DF. Factors associated with improved neurologic outcomes in patients with incomplete tetraplegia. Spine (Phila Pa 1976) 2003 Jan 1;28(1):33–9.   [PubMed]    Back to citation no. 6

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Author Contributions
Yudha Mathan Sakti – Substantial contributions to conception and design, Acquisition of data, Analysis and interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Marda Ade Saputra – Substantial contributions to conception and design, Acquisition of data, Analysis and interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Tedjo Rukmoyo – Substantial contributions to conception and design, Revising it critically for important intellectual content, Final approval of the version to be published
Rahadyan Magetsari – Substantial contributions to conception and design, Revising it critically for important intellectual content, Final approval of the version to be published
Guarantor of Submission
The corresponding author is the guarantor of submission.
Source of Support
None
Consent Statement
Written informed consent was obtained from the patient for publication of this case report.
Conflict of Interest
Author declares no conflict of interest.
Copyright
© 2018 Yudha Mathan Sakti et al. This article is distributed under the terms of Creative Commons Attribution License which permits unrestricted use, distribution and reproduction in any medium provided the original author(s) and original publisher are properly credited. Please see the copyright policy on the journal website for more information.