Volume 224, January 2023, 107526Author links open overlay panelAbstract

Idiopathic spinal cord herniation (ISCH) most commonly occurs through a ventral dural defect at the midthoracic levels with a predilection to affect middle-aged females. It can have various presentations, the most common of which are Brown-Séquard syndrome and spastic paraparesis. Due to its rarity in clinical practice, the diagnosis of ISCH can be challenging to physicians unfamiliar with this entity. We report an exceedingly rare case of ISCH at the C7-T1 intervertebral disc level in a 44-year-old male presenting with eight months of isolated unilateral sensory symptoms. The diagnosis was made based on the findings on the patient’s magnetic resonance imaging of the spinal cord, including the presence of an extradural cerebrospinal fluid collection. Surgical reduction of the herniated segment and patching of the dural defect resulted in a remarkable clinical improvement beginning in the immediate postoperative period. Follow-up MRIs showed no sign of reherniation, and the patient remained asymptomatic after one year of follow-up. Early diagnosis and surgical intervention led to an excellent early outcome in this case. However, long-term follow-up is necessary to monitor for reherniation and relapse of the symptoms in ISCH patients.

Introduction

Spontaneous or idiopathic spinal cord herniation (ISCH) is a rare cause of spinal cord injury that most commonly occurs through a defect in the ventral or ventrolateral dura at the midthoracic levels with a predilection to affect middle-aged females [1]. ISCH leads to the gradual development of myelopathy, most frequently presenting as Brown-Séquard syndrome [1], [2]. Timely diagnosis of ISCH is pivotal in preventing or reversing the neurological deficits. Here we report an extremely rare case of ISCH at the C7-T1 level in a middle-aged man presenting with isolated unilateral sensory symptoms.

Section snippetsCase report

A 44-year-old male, without any history of spinal trauma, presented with an eight-month history of thermal hypoesthesia extending from his left axilla downward and a worsening burning sensation, most prominent in his left leg, with no sensory symptoms on the right side of his body and no complaint of muscle weakness or sphincter dysfunction.

Three months before presenting to our clinic, the patient had undergone spinal magnetic resonance imaging (MRI) and an electrodiagnostic study of the left

Discussion

ISCH most frequently occurs at the T2–T8 levels [1], a result of the physiologic kyphosis of the thoracic vertebrae and the relatively anterior position of the cord within the dural sac in this region, which facilitates the contact between the dura and the spinal cord. Here we described a case of spontaneous non-traumatic ventral herniation of the spinal cord at the C7-T1 level, which is an extremely rare phenomenon. Yang et al. recently reported a case of ISCH at the cervicothoracic junction

Conclusion

The most challenging aspect of the management of ISCH is its timely diagnosis since the diagnostic clues in imaging studies can be elusive at times and the clinical manifestations can be variable. Our case attests to the importance of early surgical intervention in the neurological recovery of the patients. Despite the favorable early outcome in our case, a longer follow-up period with further imaging studies is needed to fully delineate the long-term outcome and to monitor for the occurrence

Funding sources

This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Ethics

This study was approved by the Research Ethics Committee of Shiraz University of Medical Sciences.

Declarations of interest

None.

Acknowledgment

The authors would like to thank Ms. Hosseini from the Shiraz Neuroscience Research Center for her assistance in preparing this manuscript.

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