The role of spinal fusion in the treatment of cervical synovial cysts: a series of 17 cases and meta-analysis

Clinical article

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Object

This study was undertaken to compare surgical outcomes between patients with atlantoaxial versus subaxial cervical synovial cysts (CSCs) and to compare outcomes between patients who underwent decompression alone versus decompression and fusion for the treatment of CSCs.

Methods

The authors present a series of 17 cases involving patients treated at their institution and report the surgical outcomes. Due to the rarity of CSCs, a meta-analysis was conducted, and results of the literature search were combined with the case series to enhance the power of the study.

Results

Seventeen patients underwent surgical treatment for CSCs at our institution: 3 patients (17.6%) had atlantoaxial cysts and 14 (82.3%) had subaxial cysts. Of the 17 patients, 16 underwent a decompression and fusion; most patients experienced symptom resolution at last follow-up, and there were no cyst recurrences. A total of 54 articles (including the current series) and 101 patients were included in the meta-analysis. The mean age at presentation was 64 ± 13.9 years, and the most common symptoms were motor and sensory deficits. Forty-one patients (40.6%) presented with atlantoaxial cysts, and 60 (59.4%) with subaxial cysts. There were no significant differences between groups in terms of presenting symptoms, Nurick scores, surgical treatment, or surgical outcomes. Fifty-two patients (51.4%) underwent surgical decompression without fusion, while 49 patients (48.6%) underwent fusion. The preoperative Nurick scores were significantly lower in the fused group (p = 0.001), with an average score of 1.32 compared with 2.75 in the nonfused group. After a mean follow-up of 16.5 months, a difference of means analysis between final and preoperative Nurick scores revealed that patients who received a decompression alone improved on average 1.66 points (95% CI 1.03–2.29) compared with 0.8 points (95% CI 0.23–1.39) in the fused group (p = 0.004). However, there was no statistically significant difference in symptom resolution between the groups, and the rate of cyst recurrence was found to be 0%.

Conclusions

In this study, patients with CSCs had similar outcomes regardless of cyst location and regardless of whether they underwent decompression only or fusion. In the authors' institutional experience, 16 of 17 patients underwent fusion due to underlying spinal instability. While there were no reports of cyst recurrence in their series or in the literature in patients who only received decompression, this is likely due to the limited follow-up time available for the study population. Longer follow-up and prospective and biomechanical studies are needed to corroborate these findings.

Abbreviation used in this paper:CSC = cervical synovial cyst.

Object

This study was undertaken to compare surgical outcomes between patients with atlantoaxial versus subaxial cervical synovial cysts (CSCs) and to compare outcomes between patients who underwent decompression alone versus decompression and fusion for the treatment of CSCs.

Methods

The authors present a series of 17 cases involving patients treated at their institution and report the surgical outcomes. Due to the rarity of CSCs, a meta-analysis was conducted, and results of the literature search were combined with the case series to enhance the power of the study.

Results

Seventeen patients underwent surgical treatment for CSCs at our institution: 3 patients (17.6%) had atlantoaxial cysts and 14 (82.3%) had subaxial cysts. Of the 17 patients, 16 underwent a decompression and fusion; most patients experienced symptom resolution at last follow-up, and there were no cyst recurrences. A total of 54 articles (including the current series) and 101 patients were included in the meta-analysis. The mean age at presentation was 64 ± 13.9 years, and the most common symptoms were motor and sensory deficits. Forty-one patients (40.6%) presented with atlantoaxial cysts, and 60 (59.4%) with subaxial cysts. There were no significant differences between groups in terms of presenting symptoms, Nurick scores, surgical treatment, or surgical outcomes. Fifty-two patients (51.4%) underwent surgical decompression without fusion, while 49 patients (48.6%) underwent fusion. The preoperative Nurick scores were significantly lower in the fused group (p = 0.001), with an average score of 1.32 compared with 2.75 in the nonfused group. After a mean follow-up of 16.5 months, a difference of means analysis between final and preoperative Nurick scores revealed that patients who received a decompression alone improved on average 1.66 points (95% CI 1.03–2.29) compared with 0.8 points (95% CI 0.23–1.39) in the fused group (p = 0.004). However, there was no statistically significant difference in symptom resolution between the groups, and the rate of cyst recurrence was found to be 0%.

Conclusions

In this study, patients with CSCs had similar outcomes regardless of cyst location and regardless of whether they underwent decompression only or fusion. In the authors' institutional experience, 16 of 17 patients underwent fusion due to underlying spinal instability. While there were no reports of cyst recurrence in their series or in the literature in patients who only received decompression, this is likely due to the limited follow-up time available for the study population. Longer follow-up and prospective and biomechanical studies are needed to corroborate these findings.

Synovial cysts of the spine are a known cause of back pain and radiculopathy. They are most commonly located in the lumbar region, but in approximately 4% of cases they occur in the cervical spine and may cause neck pain, radiculopathy, myelopathy, and/or neurological deficits.13,61 Cervical synovial cysts (CSCs) have been associated with advanced age and both osteoarthritis and rheumatoid arthritis,46 but the precise pathogenesis of their formation is obscure. Some authors have advocated for segmental instability as a potential cause, similar to what occurs in the lumbar region.7,55

Management of spinal synovial cysts depends on their specific location, but it mainly focuses on decompressing the nerve root and/or spinal cord and preventing cyst recurrence, which has been found to be 1.8% in the lumbar region when fusion is not performed.7 However, the role of instrumentation to improve symptomatology and prevent cyst recurrence has not been well established in the cervical spine.

In this article, we first present a series of 17 cases of surgically managed CSCs from our institutional experience. We also report the results of a meta-analysis of case reports and case series involving CSCs, which we conducted in order to compare outcomes in patients with atlantoaxial versus subaxial cysts and in patients treated with and without spinal fusions.

Methods

Current Series

We performed a retrospective electronic medical record review of cases of surgically treated symptomatic cervical spine and cervicothoracic junction (C-1 to T-1) synovial cysts either proven by pathology or (when not excised) identified intraoperatively at our institution from 1991 to 2013. Data were gathered from electronic records of clinical interviews and examinations, surgical narratives, radiology reports, pathology reports, and follow-up visits. Variables collected included patient age, sex, relevant medical history, imaging findings, presenting symptoms, surgical intervention performed, and follow-up times. Our main outcome variables were symptom resolution at last followup, last reported Nurick scores, and cyst recurrences.

Meta-Analysis

The PRISMA guidelines were followed in preparation of this article.44

A comprehensive online search was conducted via PubMed on August 1, 2013, using the key words “cervical spine synovial cyst,” “cervical spine ganglion cyst,” “cervical spine degenerative cyst,” and “cervical spine juxtafacet cyst.” The results are presented in Fig. 1. Duplicates and articles written in a language other than English were excluded.

Fig. 1.
Fig. 1.

Flow chart of methodology for identifying articles reporting on CSCs following the PRISMA guidelines. PRISMA flow diagram template obtained from Moher et al: Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. PLoS Med 6(6):e1000097, 2009.

Abstracts were reviewed and included if they reported patients with a proven diagnosis of cervical spine or cervicothoracic junction (C-1 to T-1) synovial cyst by either pathology or intraoperative findings. This selection process yielded a total of 84 articles. The articles were reviewed by hand and excluded from our meta-analysis if: 1) no individual patient information (age, sex, presenting symptom, treatment, and outcome) was given, or 2) no follow-up time was mentioned. We also cross-referenced articles to reduce publication bias. This reduced our selection to 53 articles that were included quantitatively in our meta-analysis. The same variables as recorded in our series were collected in our review whenever possible. The primary end points were symptoms at last follow-up, last follow-up Nurick scores, and cyst recurrences.

Statistical Analysis

Statistical analysis was performed using STATA12 (StataCorpLP). Demographic variables are reported as mean ± standard deviation when appropriate. The Student t-test and the c2 test were used to compare continuous variables and nominal variables, respectively, between subgroups. Statistical significance was set at p < 0.05.

Results

Current Series

Seventeen patients were identified; their characteristics are summarized in Table 1. The patients' mean age at presentation was 63.7 ± 13.7 years, and 52.9% of patients were male. The most common relevant preoperative finding was rheumatoid arthritis (present in 3 patients [17.64%]), and the most common associated radiographic finding was degenerative disc disease (present in 16 [94.1%]). A total of 3 patients presented with atlantoaxial cysts, whereas 14 patients presented with subaxial cysts. The most common presenting symptoms were radiculopathy (88.2% of cases) and motor deficits (70.5%). The mean duration of symptoms before surgery was 5.1 months (range 1–12 months). The average preoperative Nurick score was 1.35. Sixteen patients (94.2%) were treated with decompression, cyst excision, and fusion; 1 patient underwent decompression only. After a mean follow-up duration of 25.4 months, the majority of patients had improvement in their symptoms, there were no cyst recurrences, and the mean value for the last reported Nurick score was 0.58.

TABLE 1:

Characteristics of 17 patients undergoing surgical management of CSCs at our institution*

CharacteristicValue
mean age ± SD (yrs)63.7 ± 13.72
male sex9 (52.9)
relevant history
 rheumatoid arthritis3 (17.64)
 spine trauma0 (0)
 prior cervical fusion2 (11.8)
 congenital cervical fusion0 (0)
radiographic findings
 degenerative disc disease16 (94.1)
 spondylolisthesis10 (58.8)
cyst location
 atlantoaxial3 (17.6)
 subaxial14 (82.4)
 C2–31 (5.9)
 C3–42 (11.8)
 C4–50 (0)
 C5–60 (0)
 C6–71 (5.9)
 C7–T110 (58.8)
presenting symptoms
 neck pain8 (47.1)
 radiculopathy15 (88.2)
 motor deficit12 (70.6)
 sensory deficit10 (58.8)
 myelopathy6 (35.3)
 gait disturbance4 (23.5)
 bowel/bladder dysfunction3 (17.6)
mean symptom duration (mos)5.1
mean preop Nurick score1.35
surgery
 decompression only1 (5.9)
 decompression & fusion16 (94.1)
improvement of
 neck pain7 (87.5)
 radiculopathy14 (93.3)
 motor deficit11 (91.7)
 sensory deficit8 (80)
 myelopathy5 (83.3)
 gait disturbance3 (75)
 bowel/bladder dysfunction2 (66.7)
mean Nurick score at last follow-up0.58
cyst recurrence0 (0)
mean duration of follow-up (mos)25.4

Values represent numbers of patients (%) unless otherwise indicated.

Percentage values for improvement are based on the number of patients who originally presented with the given symptom.

Meta-Analysis

A total of 53 previous publications that reported on 84 cases were included in our meta-analysis and are summarized in Table 2.1–4,6,8–12,15,17,19–22,25–31,33,34,36,38,39,42,43,45–54,56–58,60–64,66–69 We also included our own 17 cases from the present study in the analysis, bringing the total number of articles to 54 and the total number of patients to 101. There were 10 case series (including the present study) and 44 case reports. The patients' mean ages at presentation ranged from 55.3 to 75.4 years, and the mean followup times ranged from 1.5 to 40.2 months.

TABLE 2:

Case reports and case series reporting patients with cervical degenerative cysts and outcomes*

Authors & YearNo. of PatientsAge (mean yrs)LocationInterventionFollow-Up (mean mos)
present series1763.7C1–2 (3), C2–3 (1), C3–4 (2), C6–7 (1), C7–T1 (10)decompression only (1), decompression & fusion (14)26.4
Birch et al., 1996471C1–2decompression only (2), decompression & fusion (2)7
Cudlip et al., 1999365.6C3–4 (1), C7–T1 (2)decompression only8.1
Cai et al., 2001272.5C1–2decompression & fusion6.8
Yamamoto et al., 2001273C3–4decompression only1.5
Zorzon et al., 2001279C1–2decompression only21
Nojiri et al., 2009269C2–3 (1), C7–T1 (1)decompression only24.3
Van Gompel et al., 20111075.4C1–2decompression only (1), decompression & fusion (9)40.2
Uschold et al., 20131263.4C4–5 (2), C5–6 (1), C6–7 (3), C7–T1 (6)decompression only (5), decompression & fusion (7)9.2
Bisson et al., 2013355.3C7–T1 (3)decompression only (1), decompression & fusion (2)16.3
case reports4461C0–1 (2), C1–2 (18), C3–4 (3), C4–5 (5), C5–6 (2), C6–7 (4), C7–T1 (11)decompression only (33), decompression &/or fusion (11)11.7

Values in parentheses represent numbers of cases.

One patient had cysts in both C4–5 and C5–6.

A total of 101 patients underwent surgical management of CSCs (mean age 64 ± 13.9 years) (Table 3). Of these 101 patients, 56 (55.4%) were male, and the most common relevant history findings were cervical spine trauma (10.9% of cases) and congenital fusion (7.9%). Radiographically, 26.7% of patients had degenerative disc disease, and 18.8% had spondylolisthesis. The most common presenting symptoms were motor deficits (84.2% of patients) and sensory deficits (61.4%). The mean duration of symptoms was 4.9 months and the mean preoperative Nurick score was 2.05 ± 1.9. The most common cyst location was C7–T1 (42 cases) (Fig. 2). A total of 52 patients (51.4%) underwent decompression only and 49 (48.6%) were treated with instrumented fusion with or without decompression. Most patients experienced improvement in their symptoms, and no cysts recurred. The mean Nurick score at last follow-up was 0.81. Patients were followed for a mean duration of 16.5 months (range 1–66 months).

TABLE 3:

Characteristics of the 101 patients included in the meta-analysis*

CharacteristicValue
mean age (yrs)64 ± 13.9
male sex56 (55.4)
relevant history
 rheumatoid arthritis5 (5.0)
 spine trauma11 (10.9)
 prior cervical fusion7 (6.9)
 congenital fusion8 (7.9)
radiographic findings
 degenerative disc disease27 (26.7)
 spondylolisthesis19 (18.8)
presenting symptoms
 neck pain51 (50.5)
 radiculopathy42 (41.6)
 motor deficit85 (84.2)
 sensory deficit62 (61.4)
 myelopathy deficit59 (58.4)
 gait disturbance52 (51.5)
 bowel/bladder dysfunction15 (14.9)
mean symptom duration (mos)4.9
mean preop Nurick score2.05 ± 1.9
surgery
 decompression only52 (51.5)
 decompression and fusion49 (48.5)
improvement of
 neck pain45 (88.2)
 radiculopathy40 (95.2)
 motor deficit76 (89.4)
 sensory deficit55 (88.7)
 myelopathy51 (86.4)
 gait disturbance47 (90.4)
 bowel/bladder dysfunction14 (93.3)
mean Nurick score at last follow-up0.8 ± 1.3
cyst recurrence0 (0)
mean duration of follow-up (mos)16.5

Values represent numbers of patients (%) unless otherwise indicated. Means are presented with SDs. The 101 cases include the 17 reported in this article as well as 84 identified in our review of the literature.

Percentage values for improvement are based on the number of patients who originally presented with the given symptom.

Fig. 2.
Fig. 2.

Graph showing CSC locations.

Atlantoaxial Versus Subaxial Cysts

Patients were first divided according to cyst location into atlantoaxial (n = 41) and subaxial (n = 60) groups (Table 4). The subaxial group included a significantly higher number of male patients (p = 0.019), patients who had previously undergone cervical fusion (p = 0.023), and patients with associated degenerative disc disease (p = 0.004). On the other hand, the atlantoaxial group had a significantly higher number of patients with congenital cervical fusions (p = 0.039). In terms of presenting symptoms, a significantly higher number of patients in the subaxial cyst group presented with radiculopathy (p < 0.001). Surgical outcomes were similar in the 2 groups, with no significant differences in Nurick scores, symptom resolution, or cyst recurrences. Patients in the atlantoaxial cohort were followed for a significantly longer time (p = 0.04).

TABLE 4:

Characteristics and surgical outcomes in 101 cases based on cyst location*

CharacteristicAtlantoaxial CystSubaxial Cystp Value
no. of cases4160
mean age (yrs)67 ± 17.362.6 ± 10.80.138
male sex17 (41.5)39 (65)0.019
relevant history
 rheumatoid arthritis1 (2.4)4 (6.7)0.336
 spine trauma5 (12.2)6 (10)0.480
 prior cervical fusion0 (0)7 (11.7)0.023
 congenital fusion6 (14.6)2 (3.3)0.039
radiographic findings
 degenerative disc disease5 (12.2)22 (36.7)0.004
 spondylolisthesis5 (12.2)14 (23.3)0.150
presenting symptoms
 neck pain21 (51.2)30 (50)0.904
 radiculopathy5 (12.2)37 (61.7)<0.001
 motor34 (82.9)51 (85)0.779
 sensory deficit24 (58.5)38 (63.3)0.752
 myelopathy28 (68.3)31 (51.7)0.096
 gait disturbance22 (53.7)30 (50.0)0.718
 bowel/bladder dysfunction2 (4.9)12 (20)0.303
mean symptom duration (mos)5.24.20.385
mean preop Nurick score1.92 ± 1.862.15 ± 1.980.560
surgery
 decompression only18 (43.9)34 (56.7)0.208
 decompression and fusion23 (56.1)26 (43.3)0.208
improvement of
 neck pain19 (90.5)26 (86.7)0.709
 radiculopathy5 (100)35 (94.6)0.238
 motor deficit28 (82.4)48 (94.1)0.095
 sensory deficit19 (79.2)36 (94.7)0.085
 myelopathy24 (85.7)27 (87.1)0.572
 gait disturbance20 (90.9)27 (90)0.978
 bowel/bladder dysfunction1 (50)12 (100)0.224
mean Nurick score at last follow-up0.90 ± 1.460.75 ± 1.220.580
cyst recurrence0 (0)0 (0)
mean duration of follow-up (mos)20.813.60.040

Values represent numbers of patients (%) unless otherwise indicated. Means are presented with SDs. The 101 cases include the 17 reported in this article as well as 84 identified in our review of the literature.

Boldface indicates significance at p < 0.05.

Percentage values for improvement are based on the number of patients who originally presented with the given symptom.

Decompression Only Versus Decompression and Fusion

Lastly, patients were categorized into those who underwent decompression alone (n = 52) and those who underwent decompression and fusion (n = 49) (Table 5). The 2 cohorts did not significantly differ in preoperative characteristics, including age, sex, relevant history, and radiographic findings. Presenting symptoms were also similar, with the exception of a higher incidence of radiculopathy in the fusion cohort (p = 0.023). Preoperative Nurick scores were significantly lower in the fusion group (p = 0.001), with an average score of 1.32 compared with 2.75 in the decompression-only group.

TABLE 5:

Characteristics and surgical outcomes of 101 patients from our series and literature review based on surgical management*

CharacteristicDecompressionDecompression & Fusionp Value
no. of cases5249
mean age (yrs)66 ± 1162.9 ± 16.50.300
male sex32 (61.5)24 (49)0.204
relevant history
 rheumatoid arthritis1 (1.9)4 (8.2)0.078
 spine trauma4 (7.7)7 (14.3)0.180
 prior cervical fusion2 (3.8)5 (10.2)0.118
 congenital fusion3 (5.8)5 (10.2)0.254
radiographic findings
 degenerative disc disease11 (21.2)16 (32.7)0.191
 spondylolisthesis7 (13.5)12 (24.5)0.154
presenting symptoms
 neck pain26 (50)25 (51)0.910
 radiculopathy16 (30.8)26 (53.1)0.023
 motor deficit46 (88.5)39 (79.6)0.220
 sensory deficit35 (67.3)27 (55.1)0.208
 myelopathy31 (59.6)28 (57.1)0.801
 gait disturbance31 (59.6)21 (42.9)0.092
 bowel/bladder dysfunction12 (23.1)5 (10.2)0.084
mean symptom duration (mos)4.45.50.776
improvement of
 neck pain24 (92.3)21 (84)0.739
 radiculopathy16 (100)24 (92.3)0.061
 motor deficit40 (87)36 (92.3)0.688
 sensory deficit33 (94.3)22 (81.5)0.061
 myelopathy26 (83.9)25 (89.3)0.918
 gait28 (90.3)19 (90.5)0.129
 bowel/bladder function12 (100)4 (80)0.040
mean preop Nurick score2.75 ± 1.881.32 ± 1.70.001
mean Nurick score at last follow-up1.09 ± 1.380.51 ± 1.190.020
difference of means for Nurick scores (95% CI)1.66 (1.03–2.29)0.81 (0.23–1.39)0.004
cyst recurrence (%)0 (0)0 (0)
mean duration of follow-up (mos)13.619.60.050

Values represent numbers of patients (%) unless otherwise indicated. Means are presented with SDs. Bold type indicates statistical significance. The 101 cases include the 17 reported in this article as well as 84 identified

Percentage values for improvement are based on the number of patients who originally presented with the given symptom.

Improvement in symptoms at last follow-up differed significantly with respect to only one measure: a higher percentage of patients in the decompression-only cohort had improvement in bowel/bladder function (p = 0.40) when compared with patients who additionally underwent a fusion. Comparison of preoperative and postoperative Nurick scores showed significant improvement in both groups. A difference of means analysis between final and preoperative Nurick scores revealed that scores for patients who received decompression alone improved on average 1.66 points (95% CI 1.03, 2.29) compared with 0.8 points (95% CI 0.23, 1.39) in the fusion group (p = 0.004).

Cyst recurrence was not observed in either cohort. The mean follow-up time was 13.6 months in the decompression-only group and 19.26 months in the fusion group, but this difference did not reach statistical significance (p = 0.05).

Discussion

Synovial and ganglion cysts may arise in joints of the upper and lower extremities and also in the spine. The differentiation of these lesions relies on histopathological examination,4 with synovial cysts (“true cysts”) presenting a synovial lining, direct attachment to the joint, and clear or xanthochromic fluid.30 Ganglion cysts, in contrast, have been called “pseudo-cysts” and are not directly connected to the joint; they have a fibrous connective tissue capsule instead of synovial tissue, and a gelatinous fluid.56 Nonetheless, the presenting symptoms, management, and prognosis of these 2 types of lesions are the virtually the same.23 In the spine, these cysts may occur at the juxtafacet joints, median atlantoaxial joints, atlantoocciptal joints, and/or ligamentous structures.38

The etiology and pathogenesis of cervical synovial cysts (CSCs) is poorly understood, and it is hypothesized that these cysts develop in the degenerated and segmentally unstable spine.15,41 Spinal degeneration is thought to cause synovial membrane protrusion through small joint capsule defects, forming a para-articular cavity.5 Furthermore, current evidence suggests that the mechanical stress on the spine induces an inflammatory cascade (upregulation and release of interleukin-1, interleukin-6, platelet-derived growth factor, and vascular endothelial growth factor, among other cytokines) that ultimately leads to neovascularization and hyperplasia of the synovium and exudation of fluid.16,59 Other theories that attempt to explain cyst formation include the myxoid degeneration,14,20 trauma,9,28 and chronic joint capsule weakness theories.52,58 Our meta-analysis revealed associated degenerative disc disease and spondylolisthesis in 26.7% and 18.8% of patients, respectively; this supports, in particular, the mechanical stress theory,15,41 but does not account for all cases. Other associated findings in our meta-analysis were a history of spine trauma in 10.9% of patients, congenital fusion in 7.9%, and rheumatoid arthritis in 4.9%. These findings suggest that CSC pathogenesis is most likely multifactorial in nature.

The currently accepted treatment for CSCs is surgical intervention (Fig. 3).4 However, there is no consensus on the optimal technique. Although the most common procedure is decompression and excision, instrumented fusion is often added for cases with underlying instability or potential iatrogenic instability or to eliminate the risk of recurrence. In line with this query, we compared patients who underwent decompression only versus fusion and assessed their outcomes.

Fig. 3.
Fig. 3.

Illustrative case of a CSC at the C6–7 level in an 86-year-old woman. A and B: T2-weighted sagittal (A) and axial (B) MR images showing a hyperintense lesion causing severe spinal cord compression. C and D: Postoperative T2-weighted sagittal (C) and axial (D) MR images showing opening of the spinal canal.

We found 52 patients who underwent decompression1–4,6,9,11,12,15,17,19,20,22,25,26,28–31,33,34,36,38,42,43,45,47,48,50,52,54,56,58, 60–64,68,69 and 49 patients who underwent fusion.3,4,8,10, 21,27,39,46,49,51,53,57,61,62,66,67 Our analysis revealed no significant difference between groups in terms of age, sex, or medical history. Symptom resolution was found to be the same in both groups, except that a higher number of patients treated only with decompression experienced resolution of their bowel/bladder dysfunction (Table 4). Moreover, we found no reported cyst recurrences in surgically treated patients irrespective of fusion status.

Because these cysts are thought to arise from abnormal movement and/or segmental instability, stabilization is often employed to eliminate the source of the cyst (diseased facet joint) and prevent recurrence. In our meta-analysis, we found 4 publications citing treatment with only instrumented fusion (without cyst excision) for atlantoaxial cysts, and interestingly, all patients experienced symptom resolution and cyst regression.10,27,46,66 The authors of these 4 papers attributed cyst development to abnormal segmental motion and emphasized the benefits of fusion. In the systematic review of lumbar synovial cysts by Bydon et al.,7 risk of recurrence was higher in patients who did not undergo instrumented fusion (1.8%) compared with patients who did undergo fusion. However, these results cannot be currently extrapolated to the cervical spine, where we found no difference in cyst recurrence between the fusion and decompressiononly groups. However, it should be stated that this finding may be a function of the length of follow-up time.10,27,46,66 Indeed, synovial cyst recurrences have been reported to occur up to 60 months after initial decompression.32

Patients who present with a symptomatic CSC may benefit from fusion if they also have facet disease, multilevel degenerative disc disease, sagittal deformity, spondylolisthesis, or iatrogenic instability due to the decompression. 35,40 In determining whether a fusion is warranted, preoperative flexion-extension radiographs may be helpful in revealing occult mechanical instability (Fig. 4).24 Additionally, extensive drilling of the facet joints intraoperatively may result in iatrogenic instability. Several studies have shown that resection of more than 50% of the facet joint is associated with increased instability postoperatively.18,65 Voo et al. found that facet resections exceeding 50% in the cervical spine result in significant increases in annulus stresses and segmental mobility.65

Fig. 4.
Fig. 4.

Example of segmental instability revealed by flexion-extension radiographs. Left: Moderate facet joint narrowing at C2–3 and C3–4 causing 3-mm anterior subluxation of C-2 over C-3 and 5-mm anterior subluxation of C-3 over C-4 in flexion. Right: Anterior subluxation of C-2 over C-3 decreases slightly in extension to approximately 1 mm.

In our case series, 94.1% of patients presented with additional degenerative disc disease and 58.8% with spondylolisthesis, and all patients except one underwent a fusion procedure accordingly. These findings, combined with the fact that synovial cysts are typically a disease of the elderly and oftentimes seen at the cervicothoracic junction, point to underlying spinal instability as a culprit. 15,41 While surgical outcomes in cases of antlantoaxial and subaxial cysts were similar in the current meta-analysis, these lesions differ in their typical presenting symptoms and treatments.37 Patients with atlantoaxial cysts more often present with myelopathy, whereas those with subaxial cysts may present with myelopathy and/or radiculopathy. Moreover, atlantoaxial cysts can be treated via either a posterior or a transoral approach, while treatment of subaxial cysts is exclusively done via a posterior approach. The similar outcomes reported herein could be attributed to a relatively small sample size, which might result in the study being underpowered.

When taking into account only symptom resolution and cyst recurrence after decompression for CSCs, we found no difference between fused and nonfused patients. Patients who did not undergo a fusion were found to have a greater improvement in Nurick score than patients treated with fusion; however, the patients in the decompression-only group had higher preoperative Nurick scores and thus more room for improvement. The difference in severity of presenting symptoms was likely due to underlying variations in the pathogenesis of CSCs in the presence versus absence of concurrent spinal disease requiring fusion.

Limitations

In this manuscript, we present the first meta-analysis of cases of CSCs specifically comparing surgical outcomes in patients treated with decompression alone versus outcomes in those treated with decompression and fusion. However, retrospective studies have inherent limitations, and additional objective measures such as the Japanese Orthopaedic Association or SF-36 health survey scores could not be assessed. Additionally, average follow-up time was also a significant limitation; while the mean follow-up time in our series was 25.4 months, overall mean follow-up time in the meta-analysis was 16.5 months. Prospective, randomized studies may help to broaden our understanding of the optimal treatment of CSCs.

Conclusions

The occurrence of synovial cysts in the cervical spine is rare but well documented. Our institutional case series included 17 patients, and all but one underwent decompression and fusion. The literature review revealed that symptom resolution and cyst resolution were similar whether or not fusion was performed. However, the lack of cyst recurrence in the cervical spine may be a function of follow-up time (mean 16.5 months). Additionally, the most common location was the cervicothoracic junction, and 18.8% of patients presented with underlying spondylolisthesis, both factors pointing to instability as a likely etiological factor. Biomechanical and prospective studies are needed to further understand and corroborate our findings.

Disclosure

Dr. Ali Bydon is the recipient of a research grant from DePuy Spine and serves on the clinical advisory board of MedImmune, LLC. Dr. Gokaslan is the recipient of research grants from DePuy Spine, AOSpine North America, Medtronic, NREF, Integra Life Sciences, and K2M; receives fellowship support from AOSpine North America; and holds stock in Spinal Kinetics and US Spine. Dr. Witham is the recipient of a research grant from Eli Lilly and Company. Dr. Sciubba is the recipient of a research grant from DePuy Spine and has consulting relationships with Medtronic, NuVasive, Globus, and DePuy. The remaining authors have nothing to disclose.

Author contributions to the study and manuscript preparation include the following. Conception and design: all authors. Acquisition of data: Lin, De la Garza-Ramos. Analysis and interpretation of data: M Bydon, De la Garza-Ramos. Drafting the article: all authors. Critically revising the article: all authors. Reviewed submitted version of manuscript: all authors. Approved the final version of the manuscript on behalf of all authors: A Bydon. Statistical analysis: De la Garza-Ramos.

This article contains some figures that are displayed in color online but in black-and-white in the print edition.

References

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    Akhaddar AQamouss OBelhachmi AElasri AOkacha NElmostarchid B: Cervico-thoracic juxtafacet cyst causing spinal foraminal widening. Joint Bone Spine 75:7477492008

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    Birch BDKhandji AGMcCormick PC: Atlantoaxial degenerative articular cysts. J Neurosurg 85:8108161996

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    Boviatsis EJStavrinou LCKouyialis ATGavra MMStavrinou PCThemistokleous M: Spinal synovial cysts: pathogenesis, diagnosis and surgical treatment in a series of seven cases and literature review. Eur Spine J 17:8318372008

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    Brotis AGKapsalaki EZPapadopoulos EKFountas KN: A cervical ligamentum flavum cyst in an 82-year-old woman presenting with spinal cord compression: a case report and review of the literature. J Med Case Reports 6:922012

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    • Export Citation
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    Bydon AXu RParker SLMcGirt MJBydon MGokaslan ZL: Recurrent back and leg pain and cyst reformation after surgical resection of spinal synovial cysts: systematic review of reported postoperative outcomes. Spine J 10:8208262010

    • Search Google Scholar
    • Export Citation
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    Cai CYPalmer CAParamore CG: Exuberant transverse ligament degeneration causing high cervical myelopathy. J Spinal Disord 14:84882001

    • Search Google Scholar
    • Export Citation
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    Cartwright MJNehls DGCarrion CASpetzler RF: Synovial cyst of a cervical facet joint: case report. Neurosurgery 16:8508521985

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    Cheng WYShen CCWen MC: Ganglion cyst of the cervical spine presenting with Brown-Sequard syndrome. J Clin Neurosci 13:104110452006

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    Cheng YYChen CCYang MSHung HCLee SK: Intraspinal extradural ganglion cyst of the cervical spine. J Formos Med Assoc 103:2302332004

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    Christophis PAsamoto SKuchelmeister KSchachenmayr W: “Juxtafacet cysts”, a misleading name for cystic formations of mobile spine (CYFMOS). Eur Spine J 16:149915052007

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    • Export Citation
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    • Export Citation
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    • Search Google Scholar
    • Export Citation
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    • Search Google Scholar
    • Export Citation
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    Ito THayashi MOgino T: Retrodental synovial cyst which disappeared after posterior C1-C2 fusion: a case report. J Orthop Surg (Hong Kong) 8:83872000

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    • Export Citation
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    • Export Citation
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    Kirk HJPik JH: A novel operative technique to manage a symptomatic synovial cyst associated with an os odontoideum. J Clin Neurosci 16:8228242009

    • Search Google Scholar
    • Export Citation
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    • Export Citation
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    • Search Google Scholar
    • Export Citation
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    • Export Citation
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    • Export Citation
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    • Export Citation
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    • Export Citation
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    • Export Citation
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    Muzii VFTanganelli PSignori GZalaffi A: Ganglion cyst of the ligamentum flavum: a rare cause of cervical spinal cord compression. A case report. J Neurol Neurosurg Psychiatry 81:9409412010

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    • Export Citation
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If the inline PDF is not rendering correctly, you can download the PDF file here.

Article Information

Dr. M. Bydon and J. A. Lin contributed equally to this work.

Address correspondence to: Ali Bydon, M.D., The Johns Hopkins Hospital, 600 North Wolfe St., Meyer 5-109, Baltimore, MD 21287. email: abydon1@jhmi.edu.

Please include this information when citing this paper: published online September 26, 2014; DOI: 10.3171/2014.8.SPINE13897.

© AANS, except where prohibited by US copyright law.

Headings

Figures

  • View in gallery

    Flow chart of methodology for identifying articles reporting on CSCs following the PRISMA guidelines. PRISMA flow diagram template obtained from Moher et al: Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. PLoS Med 6(6):e1000097, 2009.

  • View in gallery

    Graph showing CSC locations.

  • View in gallery

    Illustrative case of a CSC at the C6–7 level in an 86-year-old woman. A and B: T2-weighted sagittal (A) and axial (B) MR images showing a hyperintense lesion causing severe spinal cord compression. C and D: Postoperative T2-weighted sagittal (C) and axial (D) MR images showing opening of the spinal canal.

  • View in gallery

    Example of segmental instability revealed by flexion-extension radiographs. Left: Moderate facet joint narrowing at C2–3 and C3–4 causing 3-mm anterior subluxation of C-2 over C-3 and 5-mm anterior subluxation of C-3 over C-4 in flexion. Right: Anterior subluxation of C-2 over C-3 decreases slightly in extension to approximately 1 mm.

References

  • 1

    Aizawa TOzawa HKusakabe TNakamura TChanplakorn PItoi E: C1/2 facet cyst revealed by facet joint arthrography. J Orthop Sci 15:6036072010

    • Search Google Scholar
    • Export Citation
  • 2

    Akhaddar AQamouss OBelhachmi AElasri AOkacha NElmostarchid B: Cervico-thoracic juxtafacet cyst causing spinal foraminal widening. Joint Bone Spine 75:7477492008

    • Search Google Scholar
    • Export Citation
  • 3

    Birch BDKhandji AGMcCormick PC: Atlantoaxial degenerative articular cysts. J Neurosurg 85:8108161996

  • 4

    Bisson EFSauri-Barraza JCNiazi TSchmidt MH: Synovial cysts of the cervicothoracic junction causing myelopathy: report of 3 cases and review of the literature. Neurosurg Focus 35:1E32013

    • Search Google Scholar
    • Export Citation
  • 5

    Boviatsis EJStavrinou LCKouyialis ATGavra MMStavrinou PCThemistokleous M: Spinal synovial cysts: pathogenesis, diagnosis and surgical treatment in a series of seven cases and literature review. Eur Spine J 17:8318372008

    • Search Google Scholar
    • Export Citation
  • 6

    Brotis AGKapsalaki EZPapadopoulos EKFountas KN: A cervical ligamentum flavum cyst in an 82-year-old woman presenting with spinal cord compression: a case report and review of the literature. J Med Case Reports 6:922012

    • Search Google Scholar
    • Export Citation
  • 7

    Bydon AXu RParker SLMcGirt MJBydon MGokaslan ZL: Recurrent back and leg pain and cyst reformation after surgical resection of spinal synovial cysts: systematic review of reported postoperative outcomes. Spine J 10:8208262010

    • Search Google Scholar
    • Export Citation
  • 8

    Cai CYPalmer CAParamore CG: Exuberant transverse ligament degeneration causing high cervical myelopathy. J Spinal Disord 14:84882001

    • Search Google Scholar
    • Export Citation
  • 9

    Cartwright MJNehls DGCarrion CASpetzler RF: Synovial cyst of a cervical facet joint: case report. Neurosurgery 16:8508521985

  • 10

    Chang HPark JBKim KW: Synovial cyst of the transverse ligament of the atlas in a patient with os odontoideum and atlantoaxial instability. Spine (Phila Pa 1976) 25:7417442000

    • Search Google Scholar
    • Export Citation
  • 11

    Cheng WYShen CCWen MC: Ganglion cyst of the cervical spine presenting with Brown-Sequard syndrome. J Clin Neurosci 13:104110452006

    • Search Google Scholar
    • Export Citation
  • 12

    Cheng YYChen CCYang MSHung HCLee SK: Intraspinal extradural ganglion cyst of the cervical spine. J Formos Med Assoc 103:2302332004

    • Search Google Scholar
    • Export Citation
  • 13

    Christophis PAsamoto SKuchelmeister KSchachenmayr W: “Juxtafacet cysts”, a misleading name for cystic formations of mobile spine (CYFMOS). Eur Spine J 16:149915052007

    • Search Google Scholar
    • Export Citation
  • 14

    Cohen-Gadol AAWhite JBLynch JJMiller GMKrauss WE: Synovial cysts of the thoracic spine. J Neurosurg Spine 1:52572004

  • 15

    Cudlip SJohnston FMarsh H: Subaxial cervical synovial cyst presenting with myelopathy. Report of three cases. J Neurosurg 90 :1 Suppl1411441999

    • Search Google Scholar
    • Export Citation
  • 16

    Eguchi KMigita KNakashima MIda HTerada KSakai M: Fibroblast growth factors released by wounded endothelial cells stimulate proliferation of synovial cells. J Rheumatol 19:192519321992

    • Search Google Scholar
    • Export Citation
  • 17

    Elhammady MSFarhat HAziz-Sultan MAMorcos JJ: Isolated unilateral hypoglossal nerve palsy secondary to an atlantooccipital joint juxtafacet synovial cyst. Case report and review of the literature. J Neurosurg Spine 10:2342392009

    • Search Google Scholar
    • Export Citation
  • 18

    Enyo YYamada HKim JHYoshida MHutton WC: Microendoscopic lateral decompression for lumbar foraminal stenosis: a biomechanical study. J Spinal Disord Tech 27:2572622014

    • Search Google Scholar
    • Export Citation
  • 19

    Eustacchio STrummer MUnger FFlaschka G: Intraspinal synovial cyst at the craniocervical junction. Zentralbl Neurochir 64:86892003

    • Search Google Scholar
    • Export Citation
  • 20

    Fonoff ETDias MPTarico MA: Myelopathic presentation of cervical juxtafacet cyst: a case report. Spine (Phila Pa 1976) 29:E538E5412004

    • Search Google Scholar
    • Export Citation
  • 21

    Found EBewyer D: Cervical synovial cyst: case report. Iowa Orthop J 31:2152182011

  • 22

    Fransen PPizzolato GPOtten PReverdin ALagier Rde Tribolet N: Synovial cyst and degeneration of the transverse ligament: an unusual cause of high cervical myelopathy. Case report. J Neurosurg 86:102710301997

    • Search Google Scholar
    • Export Citation
  • 23

    Freidberg SRFellows TThomas CBMancall AC: Experience with symptomatic spinal epidural cysts. Neurosurgery 34:9899931994

  • 24

    Greenberg MS: Initial management of spinal cord injury. Handbook of Neurosurgery ed 7New YorkThieme2010. 933943

  • 25

    Harries AWasserberg J: Synovial cyst presenting as a C1/2 tumour. Br J Neurosurg 24:5955962010

  • 26

    Hatem OBedou GNégre CBertrand JLCamo J: Intraspinal cervical degenerative cyst. Report of three cases. J Neurosurg 95 :1 Suppl1391422001

    • Search Google Scholar
    • Export Citation
  • 27

    Ito THayashi MOgino T: Retrodental synovial cyst which disappeared after posterior C1-C2 fusion: a case report. J Orthop Surg (Hong Kong) 8:83872000

    • Search Google Scholar
    • Export Citation
  • 28

    Jabre AShahbabian SKeller JT: Synovial cyst of the cervical spine. Neurosurgery 20:3163181987

  • 29

    Kahiloğullari GTuna HAttar A: Management of spinal synovial cysts. Turk Neurosurg 18:2112142008

  • 30

    Kao CCWinkler SSTurner JH: Synovial cyst of spinal facet. Case report. J Neurosurg 41:3723761974

  • 31

    Kaufmann AMHalliday WCWest MFewer DRoss I: Periodontoid synovial cyst causing cervico-medullary compression. Can J Neurol Sci 23:2272301996

    • Search Google Scholar
    • Export Citation
  • 32

    Khan AMSynnot KCammisa FPGirardi FP: Lumbar synovial cysts of the spine: an evaluation of surgical outcome. J Spinal Disord Tech 18:1271312005

    • Search Google Scholar
    • Export Citation
  • 33

    Kirk HJPik JH: A novel operative technique to manage a symptomatic synovial cyst associated with an os odontoideum. J Clin Neurosci 16:8228242009

    • Search Google Scholar
    • Export Citation
  • 34

    Kotilainen EMarttila RJ: Paraparesis caused by a bilateral cervical synovial cyst. Acta Neurol Scand 96:59611997

  • 35

    Ludwig SCKramer DLVaccaro ARAlbert TJ: Transpedicle screw fixation of the cervical spine. Clin Orthop Relat Res 35977881999

  • 36

    Lunardi PAcqui MRicci GAgrillo AFerrante L: Cervical synovial cysts: case report and review of the literature. Eur Spine J 8:2322371999

    • Search Google Scholar
    • Export Citation
  • 37

    Lyons MKBirch B: Transoral surgical approach for treatment of symptomatic atlantoaxial cervical synovial cysts. Turk Neurosurg 21:4834882011

    • Search Google Scholar
    • Export Citation
  • 38

    Machino MYukawa YIto KKato F: Cervical degenerative intraspinal cyst: a case report and literature review involving 132 cases. BMJ Case Rep 2012 bcr20120071262012

    • Search Google Scholar
    • Export Citation
  • 39

    Marbacher SLukes AVajtai IOzdoba C: Surgical approach for synovial cyst of the atlantoaxial joint: a case report and review of the literature. Spine (Phila Pa 1976) 34:E528E5332009

    • Search Google Scholar
    • Export Citation
  • 40

    McCullen GMGarfin SR: Spine update: cervical spine internal fixation using screw and screw-plate constructs. Spine (Phila Pa 1976) 25:6436522000

    • Search Google Scholar
    • Export Citation
  • 41

    McGuigan CStevens JGabriel CM: A synovial cyst in the cervical spine causing acute spinal cord compression. Neurology 65:12932005

  • 42

    Miller JDal-Mefty OMiddleton TH III: Synovial cyst at the craniovertebral junction. Surg Neurol 31:2392421989

  • 43

    Miwa MDoita MTakayama HMuratsu HHarada TKurosaka M: An expanding cervical synovial cyst causing acute cervical radiculopathy. J Spinal Disord Tech 17:3313332004

    • Search Google Scholar
    • Export Citation
  • 44

    Moher DLiberati ATetzlaff JAltman DG: PRISMA Group: Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. Int J Surg 8:3363412010. (Erratum in Int J Surg 8: 658 2010)

    • Search Google Scholar
    • Export Citation
  • 45

    Moon HJKim JHKim JHKwon THChung HSPark YK: Cervical juxtafacet cyst with myelopathy due to postoperative instability. Case report. Neurol Med Chir (Tokyo) 50:112911312010

    • Search Google Scholar
    • Export Citation
  • 46

    Morio YYoshioka TNagashima HHagino HTeshima R: Intraspinal synovial cyst communicating with the C1-C2 facet joints and subarachnoid space associated with rheumatoid atlantoaxial instability. Spine (Phila Pa 1976) 28:E492E4952003

    • Search Google Scholar
    • Export Citation
  • 47

    Mujic AHunn ALiddell JTaylor BHavlat MBeasley T: Isolated unilateral hypoglossal nerve paralysis caused by an atlanto-occipital joint synovial cyst. J Clin Neurosci 10:4924952003

    • Search Google Scholar
    • Export Citation
  • 48

    Muzii VFTanganelli PSignori GZalaffi A: Ganglion cyst of the ligamentum flavum: a rare cause of cervical spinal cord compression. A case report. J Neurol Neurosurg Psychiatry 81:9409412010

    • Search Google Scholar
    • Export Citation
  • 49

    Nijensohn ERussell EJMilan MBrown T: Calcified synovial cyst of the cervical spine: CT and MR evaluation. J Comput Assist Tomogr 14:4734761990

    • Search Google Scholar
    • Export Citation
  • 50

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