An unusual cause of chronic neuropathic pain: report of a case of multiple intradural spinal arachnoid cysts and review of the literature.
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SUBMITTER: El-Hajj VG
PROVIDER: S-EPMC10477091 | biostudies-literature | 2023 Sep
REPOSITORIES: biostudies-literature
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An unusual cause of chronic neuropathic pain: report of a case of multiple intradural spinal arachnoid cysts and review of the literature.
El-Hajj Victor Gabriel VG Edström Erik E Elmi-Terander Adrian A Fletcher-Sandersjöö Alexander A
Acta neurochirurgica 20230801 9
Spinal arachnoid cysts (SACs) arise either intra- or extradurally and are usually solitary, while cases of multiple SACs have been scarcely reported in the literature. Herein, we report on a rare case of multiple and recurring intradural spinal arachnoid cysts (SACs) causing severe spinal cord compression and neuropathic radicular pain, in a 35-year-old female with a 10-year follow-up. Two separate attempts at surgery were performed but only provided temporary relief since the cysts recurred and ...[more]
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Project description:Intradural spinal arachnoid cysts with cord compression are rare. When becoming symptomatic they cause variable symptoms involving gait disturbance, paraparesis or tetraparesis and neuropathic pain, decreasing significantly the patients' life quality. The extension of such cysts averages 3.7 vertebral bodies. The diagnosis is clinical and radiological with the use of MRI, CT myelography or a combination of both. The best treatment option is complete removal of the cyst. However, even when paresis is regressing there is no good recovery from neuropathic pain. Laminectomy approach can cause postoperative complications especially when the cyst(s) expand(s) in more than one level. Alternatively, a cyst fenestration can be performed, including the levels of the maximal spinal cord compression. The clinical outcome is as good as after the cyst resection enabling the patient to walk again. The neuropathic pain may persist and require medication. A clinical case is presented, and the literature is reviewed. In the present case we report a patient with intradural arachnoid cysts extending from T6 to L2 and causing severe gait ataxia as well as neuropathic pain and hypaesthesia. The spinal-cord was compressed at T8 and T12. Surgical treatment with partial cyst resection in the compressed levels with an interlaminar approach brought similar results as complete resection. The patient was able to walk without help which was not possible before surgery. The cysts' extension is impressive as well as the minimal operative procedure.
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Project description:Background/objectiveSpinal arachnoid cysts (SAC) are intradural lesions, which may provoke a compression of the spinal cord and roots. Endoscopic techniques are increasingly used to minimize the surgical access and the postoperative scar tissue. Shunts may also represent an option. The aim of this paper is to illustrate the technique of endoscopic-assisted fenestration and positioning of a cysto-peritoneal diversion in a thoracic SAC using a flexible endoscope and to perform a systematic literature review on this subject.Material and methodsWe reported our case and we performed a review of the literature, searching for all the adult cases of Type III SACs in English language treated through endoscopic procedures.ResultsWe found 5 articles matching our search criteria and we included 9 adult patients in our analysis. Six patients were females and the most common localization was the thoracic spine. Six patients underwent selective laminectomies followed by endoscopic fenestration without cyst wall resection. Three patients had a percutaneous endoscopic inspection of the cyst and in two cases a cysto-subarachnoid shunt space was performed. Improvement of pre-operative neurological deficit was reported in six patients, no patients experienced clinical deterioration. The mean follow-up was 22 months and no progression or recurrence was reported.ConclusionThe implementation of endoscopy allows a minimally invasive treatments with good visualization of cyst anatomy and precise shunt positioning under real-time guidance. Endoscopy is technically demanding but it can offer similar clinical outcomes when compared to microscopic procedures with a limited rate of post-operative complications.The long-term risk of recurrence should be established by prospective studies.
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Project description:BackgroundMesenchymal chondrosarcoma (MCS) is a rare malignant variant of chondrosarcoma with a high tendency of recurrence and metastasis. Intradural extramedullary spinal MCS is exceedingly rare and usually found in pediatric patients. Herein, we present an elderly patient with primary intradural extramedullary spinal MCS. Relevant literatures are reviewed to disclose characteristics of intradural extramedullary spinal MCS.Case presentationA 64-year-old female presented with urinary difficulty and tightness of upper back preceding progressive weakness of right lower extremity. Magnetic resonance imaging revealed an intradural extramedullary tumor at the level of 3rd thoracic vertebra. This patient underwent total tumor resection and then received adjuvant radiotherapy. Histopathological examination showed that the tumor composed of spindle and round cells with high nucleocytoplasmic ratio accompanied by scattered eosinophilic chondroid matrix. Along with immunohistochemical findings and the existence of HEY1-NCOA2 fusion transcript, the diagnosis of MCS was confirmed. Neurologic deficit recovered nearly completely after surgery. No evidence of local recurrence or distant metastasis was found 5 years after treatments. Including the current case, a total of 18 cases have been reported in the literature with only one case with local recurrence and one case of mortality. The current case was the eldest patient diagnosed with primary intraspinal MCS in the literature.ConclusionsMCS rarely appears in the intradural space of the spine. In contrast to classic MCS, treatment outcome of primary intradural extramedullary spinal MCS is usually excellent as total tumor resection is commonly achievable. Adjuvant radiotherapy may reduce local recurrence and chemotherapy may be associated with fewer recurrences especially for unresectable tumors.
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Project description:Background Mesenchymal chondrosarcoma (MCS) is a rare malignant chondrosarcoma with a high propensity for recurrence and distant metastasis. MCS usually arises from bone tissue, and rarely occurs outside the bone. MCS in the subdural and extramedullary regions of the spinal cord is especially rare. In this article, we report a case of spinal intradural extramedullary MCS with herpes virus infection, which is the first such case reported in East China. Case Description A 13-year-old male complained of intermittent low-grade fever, sweating, progressive constipation with weakness of both lower extremities and bilateral hypoesthesia after a 5-month history of herpes virus infection. Spinal magnetic resonance imaging (MRI) revealed a subdural-extramedullary solid nodular mass with isointensity on T1-weighted imaging and hyperintensity on T2-weighted imaging that was located behind the superior margin of the T5 vertebral body. The patient was initially diagnosed with thoracic meningioma and underwent spinal cord tumour resection followed by adjuvant chemotherapy. Histopathological examination revealed that the tumour was mainly composed of round or oval cells and mesenchymal chondroid matrix, and gene analysis showed the fusion of HEY1 exon 4 to NCOA2 exon 13. Both test results were consistent with the diagnosis of primary intraspinal MCS. At the 1-year follow-up, the patient received adjuvant chemotherapy, and the reexamination images revealed no evidence of tumour in situ tumour recurrence or distant metastasis. Conclusions As more research has been done on MCS, it has been found that the disease is more likely to occur in adolescents, but is often overlooked due to its lack of imaging characterization. Therefore, the misdiagnosis rate can be reduced only by closely considering clinical manifestations with pathology and imaging findings. Although MCS is a highly malignant tumour, early primary spinal intradural extramedullary MCS can cause neurological symptoms, early detection and treatment can achieve basic total surgical resection. Postoperative adjuvant chemoradiotherapy can further reduce recurrence.
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Project description:Neurenteric cysts (NECs) are rare congenital, benign lesions of the central nervous system (CNS), predominantly located within the spinal cord. However, they may also occur less frequently within the brainstem, fourth ventricle, or cerebellopontine angle (CPA). Originating from anomalous interactions between embryonic layers, NECs are recognized for their potential to compress adjacent structures. We report a unique case of disseminated NECs exhibiting few to absent symptoms, which represents an unusual presentation of this disease, with only six similar reports in the literature. A 22-year-old female presented to our institute with chronic headaches resistant to nonsteroidal anti-inflammatory drugs (NSAIDs). Initially treated for intracranial hypertension (ICH) secondary to a cyst in the quadrigeminal cistern at the age of 17 via neuroendoscopic surgery and subsequent ventriculoperitoneal shunting, she experienced transient relief. However, follow-up at 22 years of age revealed multiple cysts in the basal and spinal cisterns, with MRI findings suggestive of neurocysticercosis. Despite treatment with albendazole and corticosteroids, subsequent MRIs showed no change in the size or number of the cysts. Six years later, symptoms had worsened, previously identified cysts had grown, and the detection of new cysts prompted surgical intervention. Histopathological examination confirmed the presence of NECs. This case highlights the diagnostic challenges posed by NECs, especially in regions endemic for neurocysticercosis, where clinical and radiological findings may initially suggest this condition. It underscores the importance of considering NECs in the differential diagnosis of cystic lesions in the CNS, even in the absence of typical symptoms of spinal cord compression. The recurrence and spread of NECs post-treatment demand a comprehensive management approach, encompassing surgical intervention and close monitoring.
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Project description:PurposeThe formation mechanism of spinal extradural arachnoid cysts (SEACs) remains unclear. There are several hypotheses for the formation of SEACs, but none of them can fully explain its pathological findings and surgical procedures. In this study, we retrospectively analyzed the cases of SEACs, aiming to clarify the formation mechanism of SEACs. In addition, we summarize a concise method for locating dural defects preoperatively and formulate a putative explanation of this method.MethodsThe clinical data of 14 patients with SEACs underwent surgery in our hospital from January 2017 to December 2021 were retrospectively analyzed.ResultsFourteen patients were identified during the study period. The cysts all spanned the T12/L1 segment, and dural defects were also located at the T12/L1 level (2 cases not recorded) as well as the middle or the upper-middle level of the cysts. Nine cases were treated with total cyst excision, 2 cases were treated with dural defect closure only, and 3 cases were treated with total cyst excision and dural defect closure. Histopathological examination demonstrated that the cyst wall contained both the arachnoid epithelial and compact fibrous connective tissue. The symptoms were relieved in all patients, and no recurrence was observed.ConclusionsAccording to intraoperative and pathological findings, the dural outer layer cyst (DOLC) is a more reasonable hypothesis about SEACs formation. When CT myelography or cinematic MRI cannot determine the location of the dural defect preoperatively, it can be located according to the middle level of the SEACs with high accuracy.
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Project description:Chest pain is a common symptom culminating in hospital admissions and specialist referrals. Although cardiac work up is pursued in most of the cases, cardiac etiology is found to be the culprit in minority of the cases. Acute chest pain is a clinical syndrome that may be caused by almost any condition affecting the thorax, abdomen, or internal organs. On occasions this extensive and expensive diagnostic work up can be avoided with awareness of commoner and non-lethal reasons. We report a case of a woman with Bornholm disease secondary to Coxsackievirus B5 (CB5) infection and supplementary review of literature till date.
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Project description:IntroductionNeuropathic pain is a common complication of spinal cord injury (SCI), and is notoriously difficult to adequately treat. Gunshot wounds (GSW) near the spinal cord may cause intractable chronic pain through spinal/nerve root transection, or reactive tissue formation resulting in nerve root compression from retained bullet fragments (RBF).Case presentationThis case report describes a 30-year-old man with a T12 AIS B incomplete spinal cord injury with paraplegia secondary to multiple GSW who presented with severe bilateral lower extremity dysesthesias and muscle spasms. Symptoms failed to improve with oral antispasmodic medications. After being diagnosed with Complex regional pain syndrome (CRPS) type I secondary to an SCI via GSW, he underwent a spinal cord stimulator (SCS) trial, which improved his symptoms by greater than 80%.DiscussionNeuropathic pain refractory to conservative treatment may benefit from SCS. Effects of therapy go beyond gate-theory in SCI patients, and may benefit patients at the cellular and molecular level. Our case demonstrates the effectiveness of SCS treatment in a patient who developed CRPS type 1 after GSW resulting in SCI.
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Project description:The gene expression signature of seven surgically removed intracranial temporal fossa arachnoid cysts was generated with two normal arachnoid membrane samples as control. Briefly, we used the Qiagen RNeasy minikit (QIAGEN GmbH Germany, Qiagen) to extract total RNA. After RNA quality and quantity assessment, we then constructed cDNA with RT-PCR reagents (Applied Biosystems, U.S). Gene expression microarray analysis was performed on the ABI 1700 Expression Array System (Applied Biosystems, U.S) using the Applied Biosystems Chemo luminescent RT-IVT Labeling Kit and Human Genome microarray (Applied Biosystems, U.S). Signal intensities generated with the ABI 1700 Expression Array System (Applied Biosystems, U.S) were imported into the J-Express Pro 2.7 software (MolMine AS, Bergen, Norway), where inter-array quantile normalization was performed to minimize the effect of external variables into the data. All control spots and flagged spots were removed, leaving 33096 gene probes available for analysis. First, we performed an unsupervised hierarchical cluster analysis in which the group belonging of the samples was defined. Second, we used Significance Analysis of Microarrays (SAM) with 400 and 1000 permutations to compare AC and AM samples and generate gene lists of differentially expressed genes between these groups. With SAM the false discovery rate (FDR) of the gene lists was calculated. FDR returned the number of false positive genes present on the gene list. A measure of FDR is the Q value, which conveniently shows an estimation of the FDR in percent. In the current study only genes with a Q value <1.0 % were accepted as being differentially expressed.
2010-01-06 | GSE19727 | GEO
Project description:Appendiceal duplication is exceedingly rare, with a prevalence of 0.004% to 0.009% in appendectomy specimens. Appendiceal duplications can occur alone or in conjunction with cecal duplication. The persistence of the temporary embryologic second cecal appendix is hypothesized to cause appendiceal duplications. We present a case of appendiceal duplication in a 26-year-old Ethiopian female patient who had been experiencing abdominal pain in the right lower quadrant for 1 week. She developed anorexia, a loss of appetite, and a low-grade fever as a result of this. She reported direct and rebound mild discomfort in the right lower quadrant on abdominal examination. She was then operated on and she had an appendiceal duplication intraoperatively. As a result, an appendectomy was performed, and the patient was discharged with improved health. To avoid unfavorable patient outcomes and medicolegal difficulties, surgeons and surgical trainees who conduct several appendectomies throughout their training should be aware of the likelihood of appendiceal duplication.
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