Predictors for complete surgical resection of posterior fossa neurenteric cysts: A case report and meta-analysis
- Department of Neurosurgery, Scientific Circle of Neurology and Neurosurgery, Pomeranian Voivodeship, Poland.
- Department of Neurosurgery, Medical University of Gdansk, Gdansk, Pomeranian Voivodeship, Poland.
Samuel D. Pettersson, Department of Neurosurgery, Scientific Circle of Neurology and Neurosurgery, Medical University of Gdansk, Gdansk, Pomeranian Voivodeship, Poland.
DOI:10.25259/SNI_723_2021Copyright: © 2021 Surgical Neurology International This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-Share Alike 4.0 License, which allows others to remix, tweak, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.
How to cite this article: Samuel D. Pettersson1, Shan Ali1, Pavlo Burmaka1, Justyna Fercho1, Tomasz Szmuda2, Ahmed Abuhaimed1, Yazeed Alotaibi1, Paweł Słoniewski2, Michał Krakowiak2. Predictors for complete surgical resection of posterior fossa neurenteric cysts: A case report and meta-analysis. 19-Oct-2021;12:530
How to cite this URL: Samuel D. Pettersson1, Shan Ali1, Pavlo Burmaka1, Justyna Fercho1, Tomasz Szmuda2, Ahmed Abuhaimed1, Yazeed Alotaibi1, Paweł Słoniewski2, Michał Krakowiak2. Predictors for complete surgical resection of posterior fossa neurenteric cysts: A case report and meta-analysis. 19-Oct-2021;12:530. Available from: https://surgicalneurologyint.com/?post_type=surgicalint_articles&p=11182
Background: Incomplete resection of neurenteric cysts (NCs) has been associated with increased recurrence rates in patients compared to complete resection (CR) and information on intracranial NCs appearance on diagnostic imaging is scarce. We sought to identify factors associated with CR and provide the largest up-to-date review of NCs appearances on various diagnostic images.
Methods: Data from Medline, EMBASE, and Web of Science were extracted. Univariate and multivariate logistic regression models were used to analyze factors associated with CR.
Results: A total of 120 publications reporting 162 original cases on posterior fossa NCs met the inclusion criteria for analysis. Eighty-nine (55.6%) of the patients were female, the mean (SD) age of the patients’ during operation was 34.3 (16.9) years, and CR was achieved in 98 (60%) of patients. Univariate analysis identified male sex as a statistically significant predictor for complete reaction (OR 2.13, 95% Cl 1.10–4.11, P = 0.02). The retrosigmoid approach (OR 1.89, 95% Cl 0.98–3.63, P = 0.06), far lateral approach (OR 0.46, 95% Cl 0.21–1.02, P = 0.06), and pediatric patient (OR 2.45, 95% Cl 0.94–6.56, P = 0.07) may be possible predictors for CR, however, they were not statistically significant. NCs are mainly hypodense on CT (32 [61.5%]), varied greatly in intensity on T1WI, hyperintense on T2WI magnetic resonance imaging (98 [67.1%]), and hyperintense on fluid-attenuated inversion recovery (17 [63.0%]).
Conclusion: We recommend utilizing various diagnostic imaging tests to help reduce misdiagnoses when identifying intracranial NCs. For patient safety, CR should be achieved when possible, to reduce risk of additional operations due to recurrence.
Keywords: Magnetic resonance imaging, Neurenteric cyst, Posterior fossa tumor, Resection
Neurenteric cysts (NCs) are extremely rare, benign, congenital endodermal lesions originating from the central nervous system. At present, the exact etiology of NCs is unknown.[
We present a case of a 36-year-old male who was admitted to the hospital due to a 1-month history of daily headaches within the occipital region. The headaches were reported to be worse at night and occasionally accompanied by nausea and dizziness. Neurological examinations revealed a right-sided exaggerated tendon reflex compared to the contralateral side. The patient was diagnosed with a positive pyramidal syndrome, Hoffmann’s reflex, and Babinski’s and Rossolimo’s sign on the right side. A CT scan revealed a 20 × 15 × 23 mm isodense focal lesion in the posterior fossa at the level of the foramen magnum [
Preoperative scans reveal a lesion located at the ventral craniocervical junction. The lesion is shown hyperintense on PD-weighted imaging (a, sagittal), heterogeneous on T1-weighted (b, sagittal; c, axial), isodense on CT (d, axial), and hyperintense on T2-weighted MRI (e, sagittal; f, axial).
The screening process was performed according to the guidelines of Preferred Reporting Items for Systematic Reviews and Meta-Analyses. The databases of Medline, Web of Science, and EMBASE were used to retrieve studies from inception to March 14, 2021, without language limits. The following keywords were used in all three databases: (“cyst”/exp OR cyst AND (neurenteric OR endodermal OR enterogenous OR enterogenous OR epithelial OR teratomatous OR enterogenic OR “foregut”/exp OR foregut OR respiratory OR bronchogenic) OR intestinoma OR gastrocytoma AND (foramen OR magnum OR craniocervical OR craniovertebral OR craniospinal OR posterior AND fossa OR cerebellopontine)).
The search was performed independently by three authors (SA, PB, and SP) and two additional authors (TS and JF) arbitrated any disagreements on inclusion or exclusion of the studies. All case reports related to NCs located specifically in the posterior fossa were evaluated. Studies that did not report the extent of the surgical resection of the cyst (complete or incomplete) were excluded from the study. The Joanna Briggs Institute Critical Appraisal Checklist for Case Reports was used to evaluate the quality of the reports. We considered the studies that included at least four of the eight criteria suitable to be used in our study.
The results of the surgical resection reported by the studies were classified into two groups: CR and IR. CR was only recorded if stated in the report. All other reported results such as near-total, subtotal, or partial resections were recorded as IR. The data from the first operation on a patient were included in our study. Therefore, any additional operations undergone by a patient due to cyst recurrence were excluded from the study. Readings from diagnostic images from the case reports collected were categorized into one of three groups by their intensity (H, hyperintense; G, isointense; and B, hypointense). Patient sex, age, cyst location, surgical approach, and NC type were also extracted.
Comparison of CR and IR was conducted using Chi-square or Fisher’s exact test for categorical variables. For continuous variables, Student’s t-test or Mann–Whitney U-test was used. To find the potential factors associated with CR, a univariate binary logistic regression model was used. For diagnostic images, data reporting either isointense (G) from MRI or isodense (G) from CT were excluded to dichotomize the variables. All variables with P < 0.25 from the univariate analysis were then used in a multivariate binary logistic regression model to calculate multivariate-derived odds ratios. P < 0.05 was considered statistically significant. Statistical analysis was conducted using IBM SPSS Statistics version 26 (IBM Corp.).
A total of 822 references were identified by the three databases, as shown in [
The collected data on patient characteristics are shown in [
Preoperative diagnostic imaging characteristics
A summary of the collected preoperative diagnostic images is shown in [
Due to cases frequently reporting only one MRI sequence type, [
Factors associated with CR
Variables tested in the univariate logistic regression model are shown in [
To the best of our knowledge, this is the first meta-analysis and systematic review on posterior fossa NCs. Cyst recurrence has been shown to occur greater in patients with IR,[
Cancerous tumors enhance strongly with gadolinium and are rarely located along the midline.[
Using various diagnostic imaging methods including the use of contrast agents can help narrow down and identify the correct intracranial abnormality. A CT scan is necessary to exclude congenital bony anomalies or bone malformation which is often observed with NCs.[
Surgical resection is currently the only effective treatment for patients with intracranial NCs and is recommended in symptomatic patients.[
When studying rare medical conditions, the main limitation often arises from the lack of data found in the literature. The extractable data on intracranial NCs were limited to single case reports and case series typically consisting of less than 10 patients. In addition, the data reported from each case differed in robustness, and therefore, statistical analyses were limited to only the variables commonly reported. This study analyzed only the first operation undergone by patients so statistical findings may not correlate to patients undergoing additional operations.
Incorporating additional diagnostic imaging methods such as DWI, DTI tractography, and the use of contrast agents may help to correctly diagnose NCs preoperatively and identify the cyst location with greater precision. Findings from our statistical analysis on NCs and general characteristics on diagnostic imaging may require additional reports in the future to strengthen these findings and to test other possible variables that may influence resection outcomes. As shown in [
CR of posterior fossa NCs is moderately achievable shown by 60% of all cases reported. The cyst is slightly more common in females, and the male sex is independently associated with CR. Diagnostic images show that NCs are mainly hypodense on CT, hyperintense on T2WI and show high variability on T1WI.
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