Hemorrhagic cervical juxta-facet cyst presenting with Brown-Séquard syndrome: illustrative case

Abstract

BACKGROUND

Intraspinal juxta-facet cysts of the spine are known to predominate at the lumbar level and is relatively rare at the cervical level. Most cervical spinal lesions are found incidentally, but they sometimes cause myelopathy or radiculopathy in a chronic course.

OBSERVATIONS

The authors present a rare case of hemorrhagic cervical juxta-facet cyst presenting with Brown-Séquard syndrome. An 86-year-old woman presented with acute-onset right hemiparesis following neck pain and was admitted to the local hospital. She was started on antithrombotic therapy with a suspected diagnosis of cerebral infarction, but quadriplegia progressed 2 days later. Cervical magnetic resonance imaging revealed an intraspinal mass at the C4–5 level and she was referred to the authors’ hospital. Her neurological findings on admission revealed right Brown-Séquard syndrome. In emergency surgery, the mass was resected with a posterior approach. Pathological findings showed hemosiderin deposition and fibroblast proliferation, consistent with a juxta-facet cyst with intracystic hemorrhage. The patient recovered well and returned to an independent daily life.

LESSONS

Rarely, juxta-facet cyst of the cervical spine can cause acute Brown-Séquard syndrome due to intraspinal hemorrhage. In a case of hemiparesis that develops following neck pain, hemorrhagic cervical juxta-facet cyst should be taken into consideration as a differentiation.

Intraspinal juxta-facet cysts of the spine occur mostly in the lumbar spine but have also been reported to be located in the cervical and thoracic level.¹ The development of the cysts is commonly reported to be related to mechanical stress on the facet joints.^2–4 Such cysts are often asymptomatic, but sometimes grow slowly and cause radiculopathy or myelopathy depending on their location. Only 10% of juxta-facet cysts are associated with intracystic bleeding, which is observed most often in the lumbar spine, and rarely at the cervical level.⁵ Here, we report a case of hemorrhagic cervical juxta-facet cyst presenting with acute Brown-Séquard syndrome and review the literature on the related case.

Illustrative Case

An 86-year-old woman experienced sudden-onset right hemiparesis following neck pain and was admitted to the local hospital. Although brain magnetic resonance imaging (MRI) showed no abnormality, a cerebral infarction was suspected and the patient was started on antithrombotic therapy. On the second day of admission, her neurological symptoms progressed to tetraparesis. Cervical MRI revealed an epidural mass lesion on the dorsal side of the spinal cord at the C4–5 level, and she was referred to our facility for treatment. Her self-reported symptoms were pain from the right neck to the shoulder, and neurologically, she presented with Brown-Séquard syndrome below the C6 segment. Dynamic imaging of the cervical spine revealed mild C4–5 instability. Cervical MRI revealed a 10 × 4 × 14–mm mass lesion compressing the cervical spinal cord on the right dorsal side of the spinal canal at the C4–5 level. The mass showed low signal intensity on a T1-weighted image, mixed signal intensity on a T2-weighted image, and a ring-like enhancement pattern on a contrast-enhanced T1-weighted image (Fig. 1). Based on these findings, a cervical juxta-facet cyst or epidural tumors were suspected preoperatively.

FIG. 1.

Preoperative sagittal T1-weighted image (A), contrast-enhanced T1-weighted image (B), T2-weighted image (C), axial T1-weighted image (D), contrast-enhanced T1-weighted image (E) and T2-weighted image (F), demonstrated the mass lesion compressing the spinal cord from the right dorsal side at the C4–5 level (arrows), with low signal on T1-weighted image, partially low signal in the interior on T2-weighted image, and a ring-like enhancement on contrast-enhanced T1-weighted image.

Emergency surgery was performed to remove the epidural lesion using a posterior approach. After creating the median and lateral groove of the lamina from C4 to C5 and opening the lamina, a cystic lesion with a dark-red capsule was seen between the dura and the yellow ligament (Fig. 2). During resection of the cyst, the cyst wall was ruptured and the hematoma within the cyst was drained. After complete excision of the cyst and confirmation of decompression of the spinal cord, laminoplasty was performed using a spacer.

FIG. 2.

Intraoperative pre-excision (A) and post-excision (B) view showing cystic lesion (arrowheads) with a dark red capsule between the dura (asterisks) and the yellow ligament (arrow).

Pathological examination demonstrated fibroblast proliferation in the capsule, consistent with a juxta-facet cyst. In addition, hemosiderin deposition and vitrification were observed, suggesting chronic inflammation and bleeding within the cyst wall (Fig. 3). Postoperative MRI confirmed complete excision of the cyst and decompression of the spinal cord (Fig. 4). The pain from the right neck to the shoulder was markedly improved from the early postoperative period. With continued rehabilitation, her neurological symptoms gradually recovered, and her left-sided sensory disturbance improved 3 months after the operation. Finally, she was able to walk independently 6 months after the operation.

FIG. 3.

Histopathological examination demonstrated fibroblast proliferation in the capsule, consistent with a synovial cyst (A). Vitrification (B) and hemosiderin deposition (C) were observed, suggesting chronic inflammation and hemorrhage. Hematoxylin and eosin, original magnification ×50 (A), ×125 (B), and ×400 (C).

FIG. 4.

Postoperative sagittal T2-weighted image showing the disappearance of the cyst (arrowhead) and the decompression of the spinal cord.

Discussion

Observations

Intraspinal juxta-facet cysts occur in the lumbar spine in more than 88% of the total reported cases. They are unusual in the cervical spine, occurring in less than 5% of cases, and most commonly located at the C7–T1 level (55%–57%).^2–4 Juxta-facet cysts at the cervical lesions have been more common in over 6 decades, and their clinical presentation varies according to the size, location, and relationship to adjacent structures, but they are often asymptomatic or present with neck pain and slowly progressive radiculopathy or myelopathy. The etiology of juxta-facet cysts remains unclear, but it is suggested that instability associated with spinal degeneration is involved in the pathogenesis of the disease.³

Hemorrhagic juxta-facet cysts often show rapid deterioration of neurological symptoms. It has been reported that approximately 10% of lesions in the lumbar spine are associated with hemorrhage, but hemorrhagic juxta-facet cysts in the cervical spine are rare, and only 7 cases including our case have been reported so far (Table 1).^6–11 The average age of the 7 patients was 71 years old (range, 58–86 years), and the most common location was at the C7–T1 level in 3 cases (43%). Neck or back pain was present in 5 of the 7 patients, and myelopathy progressed rapidly and required emergency decompression surgery in 6 cases. Only 2 cases including the present case have been reported with acute Brown-Séquard syndrome. Our case was important in suggesting that juxta-facet cyst should be considered as a rare cause of acute-onset hemiplegia.

TABLE 1.

Summary of cases of hemorrhagic cervical intraspinal juxta-facet cyst

Case No.	Authors & Year	Age (yrs), Sex	Location	Symptoms	Period from Onset to Op	Trauma	Antithrombotic	Treatment	Outcome
1	Jabre et al., 19876	60, M	C6–7	Myelopathy, neck pain	2 days	Yes	No	Surgery (laminotomy of C6–1)	GR
2	Conforti et al., 20147	75, M	C7–T1	Radiculopathy, neck pain	NA	No	Yes	Conservative management (steroid)	GR
3	Attwell et al., 20148	75, F	C4	Myelopathy	1 wk	No	Yes	Surgery, (cervical decompression & lesion excision)	GR
4	Kim et al., 20179	74, F	C7–T1	Myelopathy	2 wks	No	No	Surgery (removal of the lesion & C7–T1 fusion)	GR
5	Jitpun et al., 202010	58, F	C2–3	BSS, neck pain	3 wks	No	No	Surgery (laminotomy of C2–3 & surgical decompression)	GR
6	Radhouane et al., 202011	68, M	C7–T1	Myelopathy, back pain	1 wk	No	No	Surgery (laminectomy of C7–T1)	GR
7	Present case, 2022	86, F	C4–5	BSS, neck pain	1 wk	No	No	Surgery (laminoplasty of C4–5)	GR

BSS = Brown-Séquard syndrome; GR = good recovery; NA = not applicable.

The pathophysiology of intracystic hemorrhage is still unclear, but it has been suggested that chronic mechanical stress induces cyst enlargement and neovascularization of the cyst wall, and that additional triggers such as trauma and antithrombotic therapy lead to intracystic hemorrhage.¹⁰ In previous reports, the cause of intracystic hemorrhage was speculated to be trauma in 1 case⁶ and oral administration of antiplatelet agents in 2 cases.^7,8 Since the present case had no history of trauma or oral administration of antiplatelet agents, it was speculated that existing mild facet instability of a degenerative cervical spine was involved in the mechanism of cyst formation and intracystic hemorrhage.

As the present case shows, hemorrhagic cervical juxta-facet cysts may present as acute-onset hemiplegia mimicking stroke. Ischemic stroke, the most common cause of acute-onset hemiplegia, has recently required rapid therapeutic intervention, such as thrombolytic therapy and mechanical thrombectomy, requiring accurate diagnosis in a short time.¹² However, since commencement of thrombolytic therapy for bleeding cervical lesions without an accurate diagnosis can lead to serious consequences, careful diagnosis is necessary.¹³ Indeed, in our case, initiation of antithrombotic therapy may have contributed to the worsening of neurological symptoms. It has been reported that spinal cord lesions account for 0.7% of all stroke mimics, and 26% of patients with stroke mimics received thrombolytic therapy.¹⁴ Among them, spontaneous spinal epidural hematoma is known to be a representative disease, and the presence of neck pain at onset is important clinical information to distinguish it from ischemic stroke.¹⁵ Similarly, hemorrhagic juxta-facet cysts are characterized by neck or back pain due to the rapid enlargement of the epidural lesion.^6,7 In addition, since this lesion arises from a unilateral facet joint, it should be recognized that rapid enlargement, as in this case, can compress the spinal cord laterally and cause Brown-Séquard syndrome. In order to prevent misdiagnosis and provide appropriate treatment, it is important to determine the responsible lesion by paying attention to neck pain and the pattern of sensory disturbance as well as acute-onset hemiplegia.^13,15

Lessons

We report a rare case of hemorrhagic cervical juxta-facet cyst presenting with acute Brown-Séquard syndrome. In cases with acute-onset hemiplegia, it is important to accurately assess physical findings, including pain and sensory disturbance, and to consider cervical epidural lesions as a differential diagnosis.

Acknowledgments

We thank Dr. Trish Reynolds, MBBS, from Edanz for editing a draft of this manuscript.

Disclosures

The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper.

Author Contributions

Conception and design: Yagi, Shimura, Kinouchi. Acquisition of data: Yagi, Shimura, Kinouchi. Analysis and interpretation of data: Shimura. Drafting of the article: Yagi, Shimura, Kinouchi. Critically revising the article: Yagi, Saito, Yoshioka, Kinouchi. Reviewed submitted version of the manuscript: Kinouchi. Approved the final version of the manuscript on behalf of all authors: Yagi. Statistical analysis: Shimura. Study supervision: Yagi.