Fulminant chemical ventriculomeningitis following intrathecal liposomal cytarabine administration

Intrathecal liposomal cytarabine (ITLC) has shown prolonged time to neurological deterioration in some patients with leptomeningeal spread of breast cancer. We describe a patient with fulminant chemical ventriculomeningitis leading to cerebral edema and death following ITLC administration. A 56-year-old Caucasian female developed progressive headache, depressed level of consciousness and adventitious movements immediately following ITLC. Neurological examination was normal prior to injection. This progressed to loss of all brainstem function and a head CT scan demonstrated diffuse cerebral edema. Due to the absence of neurological function on examination, the family opted to withdraw care. To our knowledge we report the first patient with fulminant chemical ventriculomeningitis leading to cerebral edema and death following ITLC administration. Adjunctive glucocorticoids are recommended. Treatment options are limited and complicated by the liposomal formulation of intrathecal cytarabine.     

Neoplastic meningitis presenting with acute cerebellar ataxia

Acute cerebellar ataxia is a rare initial presenting feature of neoplastic meningitis (NM), particularly in gastric cancer. The authors report a 61-year-old woman with acute cerebellar ataxia secondary to NM from gastric cancer, which was not accompanied by other symptoms commonly associated with NM at initial presentation. It is suggested that NM should be considered in the differential diagnosis of cancer patients with acute cerebellar ataxia.     

Streptococcus salivarius meningitis: a case report and literature review

Twelve hours after spinal anaesthesia, a 61-year-old patient developed meningitis with fever, somnolence, headache and stiffness of the neck. The cerebrospinal fluid was found to contain 5.279/mm³ cells (95 granulocytes), 12.800 mg/l protein and 14.0 mmol/l lactate. Streptococcus salivarius was detected in the culture. The patient was treated with antibiotics (initially cefotaxime, fosfomycin and gentamycin, later piperacillin and sulbactam). Restitutio ad integrum took place after 7 days. Thirteen cases of Streptococcus salivarius meningitis over the past 40 years are described in the literature, nine of which occurred following spinal puncture. Streptococcus salivarius was probably communicated by the medical personnel from the oral cavity when working without masks. The outcome in each case was good. In our opinion suitable masks must be worn during spinal puncture. Although iatrogenic infection is uncommon, it cannot be ruled out.     

Mollaret's meningitis responding to phenylbutazonum. A case report

A patient with Mollaret's meningitis is presented, with some unusual features. Criteria for the diagnosis are discussed. Many therapies for this recurrent aseptic meningitis have been suggested. In our patient there was a good response to phenylbutazonum, which to our knowledge has not been reported before.     

A case of carcinomatous meningitis with papilledema as the only symptom: favorable response to intrathecal chemotherapy

A case of carcinomatous meningitis with papilledema as the only sympton improved after intraspinal chemotherapy with complete regression of the papilledema

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Sommario Gli AA. riportano un caso di meningite carcinomatosa monosintomatica (papilledema) migliorata dopo trattamento antiblastico per via endorachide, con regressione completa del papilledema.

     

Management of syringomyelia associated with tuberculous meningitis: A case report and systematic review of the literature

Determinants of tuberculosis (TB) syringomyelia, its management options and outcomes are still under investigation. The aim of this study is to present a case of TB syringomyelia with markedly improved symptoms status-post surgery and to understand the clinical characteristics and outcomes of 33 TB syringomyelia cases reported in the literature. Specifically, we examined the differences between patients who were managed medically and those who underwent surgical intervention. Inclusion criteria for the cases were (1) syringomyelia caused by TB infection rather than co-occurrence of these conditions, (2) management protocol described, and (3) post-treatment outcome described. The median age was 30 years (interquartile range (IQR): 23–40) with 55% males. The median time between TB onset to syringomyelia diagnosis was 2 years. Nineteen patients were surgically treated, 11 were medically treated, and 3 received no treatment. Twenty-one patients showed improvement in at least one prior symptom, but no patient experienced a full recovery. Those that underwent surgical intervention were more likely to have TB meningitis (95% vs. 64%, p < 0.05) upon initial TB presentation and have a greater interval between TB onset and syringomyelia presentation (median of 2.6 vs. 0.33 years, ns). A greater proportion of the surgically managed patients experienced improvement in any symptom (74% vs. 45%, ns). Future case-controlled studies with larger sample sizes are required to validate and further understand the outcomes of surgically-managed TB syringomyelia.     

Anti-N-methyl-D-aspartate receptor encephalitis associated with hepatic neuroendocrine carcinoma: A case report

Anti-N-methyl-D-aspartate receptor (Anti-NMDAR) encephalitis can present with and without tumor. Tumor associations are less common in older patients. We report a 65-year-old gentleman who presented with one week history of cough, chills, rigor and altered behavior, followed by florid visual and auditory hallucinations. Mini mental status examination score was 16/30. Both cerebrospinal fluid and plasma anti-NMDA receptor antibodies were detected. A course of intravenous methylprednisolone was given with partial symptom improvement. A hepatic neuroendocrine carcinoma was detected and confirmed on biopsy. Unfortunately, he developed several medical complications: non-ST elevation myocardial infarction, infected foot gangrene and peripheral vascular disease, which made him unsuitable for both surgery and chemotherapy. He passed away 6 months later due to the progression of the malignancy. This case illustrated that NMDAR encephalitis may be associated with an uncommon hepatic neuroendocrine carcinoma in an older person, which is responsive to early treatment.     

Acute aseptic meningitis during isotretinoin treatment for nodular acne solely presenting with headache: case report and brief review of the literature

Purpose/aim: We describe, in detail, the first case of isotretinoin-induced aseptic meningitis. A brief summary of the literature on drug-induced aseptic meningitis (DIAM) is also presented.

Materials and methods: A 20-year old female patient with probable (Naranjo adverse reaction probability score of 7) DIAM during treatment with isotretinoin therapy for nodular acne solely, presenting with headache. Pseudotumor cerebri was appropriately ruled-out.

Results: Summary of data altogether lead us suggest that isotretinoin triggered DIAM, possible due to a delayed hypersensitivity mechanism type III or IV.

Conclusion: We highlight a quite uncommon cause of DIAM that may be increasing in frequency due to the current increasing use of isotretinoin against nodular acne.     

脊髓脊膜膨出合并鳞状细胞癌1例

目的报告1例脊髓脊膜膨出合并鳞状细胞癌病例,探讨其形成原因及治疗方法。方法男性,11岁,生后即发现腰骶部有一囊性肿物并逐渐增大,6岁时曾破溃1次,入院前2个月发现肿物再次破溃并有恶臭及脓性分泌物。MRI显示脊髓脊膜膨出、脊髓拴系综合征。全面检查后行清创及脊髓脊膜膨出切除术,术中病理示鳞状细胞癌,扩大切除周围皮缘并行椎管内探查,直至冷冻病理未见癌细胞,缺损创面用轴形皮瓣旋转覆盖,供瓣区取同侧大腿中厚皮片覆盖。结果手术完整切除肿物,伤口及转移皮瓣愈合良好。结论脊髓脊膜膨出破溃后慢性感染或长期刺激可以继发癌变,尽早行脊髓脊膜膨出切除有助于预防其癌变的发生。     

Blood-brain-barrier and intrathecal immunoglobulin changes in tuberculous meningitis and demyelinating disorders: a preliminary report from a South Indian hospital

Sixty-three paired serum and CSF samples (15 controls and 48 patients) were studied for CSF albumin quotient, IgG index and synthesis rate. Control values for albumin quotient and IgG index were less than 10.0 and less than 0.6 respectively. IgG synthesis rate/day was calculated according to the Tourtellotte formula and a value of greater than 3.0 mg/day is considered high. Our findings in patients with central demyelinations, subacute sclerosing panencephalitis, Guillain Barré Syndrome and cerebrovascular accidents are comparable to earlier studies. Fifteen tuberculous meningitis patients were studied and could be separated into 2 groups, 9 with elevated albumin quotients and 6 with normal albumin quotients. Three patients with elevated albumin quotients and 4 with normal albumin quotients showed increased intrathecal IgG synthesis.     

原发性松果体区胚胎癌的诊断与治疗(附二例报告及文献复习)

目的 原发性松果体区胚胎癌非常罕见,本文报道2例,并结合文献探讨其诊断、治疗和预后.方法 2例经病理证实的原发性松果体区胚胎癌,术前例1患者血清甲胎蛋白(AFP)6 810μg/L,人绒毛膜促性腺激素(β-HCG)正常,例2患者血清AFP正常,β-HCG 5 260 mIU/ml.采用枕部经小脑幕入路显微手术切除肿瘤,术后联合放化疗.结果 2例患者肿瘤均达到镜下全切,术后肿瘤免疫组化示例1:CK、AFP阳性;例2:CK、β-HCG阳性.经放化疗后,血清AFP/β-HCG基本正常.术后1年随访2例患者仍然存活.结论 血清AFP、β-HCG对胚胎癌的诊断及预后判断有重要作用,免疫组化指标CK、AFP、β-HCG对病理诊断有鉴别意义,该病采用手术联合放化疗的综合疗法,可获得较好疗效.

Abstract：

Objective Primary embryonal carcinoma in pineal region is extremely rare Here we report two such cases and combined literatures review to discuss its diagnosis, treatment and prognosis. Method Two primary embryonal carcinoma in pineal region verified histologically were presented. In one cases, the preoperative serum alpha - fetoprotein (AFP) level reached significantly high level, 6 810μg/L, but showed negative staining forβ-human chorionic gonadotropin (β- HCG) . In the other one, accompanied by elevation of β-HCG level, 5 260 mIU/ml, the serum AFP was negative. The tumors were microsurgically removed by transeccipito-tentorial approach. Endoscopic third ventriculostomy was used after tumor removal because of recurrent hydrocephalus. Both patients received adjunctive treatments included radiation therapy (whole central nervous system 30 Gy and tumor bed 50 Gy) and 4 course of chemotherapy. Results In both patients, the tumors were totally removed. Postoperative tumor immunohistochemistry indicators were present in case 1 with positive reaction for cytokeratin(CK) and AFP, in case 2 with positive CK and β - HCG. After adjunctive treatments, the serum AFP and β - HCG of two patiants were decreased to normal ranges. After post - operative follow - up of one year,two patients still survived and the serum markers were almost normal. Conclusions Measurement of serum β-HCG and AFP is extraordinarily significant for the diagnosis and post - treatment monitoring of pineal region embryonal carcinoma. Pathological differential diagnosis requires immunohistochemical indicators including cytokeratin (CK),AFP andβ- HCG. With radically surgical removal and combined adjunctive therapy including radiation and chemotherapy, good consequence could be obtained.     

Paraneoplastic autoimmune encephalitis associated with pleomorphic lung carcinoma: An autopsy case report

A 64‐year‐old man was admitted with acute onset disturbed consciousness. Cerebrospinal fluid analysis revealed pleocytosis and elevated protein, with negative cultures and PCR. Serum antibodies for autoimmune encephalitis were also negative. Brain magnetic resonance imaging (MRI) was unremarkable, but whole‐body CT scan showed a tumor in the left lower lung lobe. Bronchial brush cytology demonstrated clusters of malignant cells, and ¹⁸F‐fluorodeoxyglucose positron emission tomography showed multiple lesions and increased uptake in the lung tumor. Clinically the patient had a stage IV lung carcinoma, graded as T3N3M1b (OSS). Steroid therapy had limited efficacy, but chemotherapy dramatically improved his neurological symptoms. Therefore, he was diagnosed with paraneoplastic autoimmune encephalitis based on the diagnostic criteria for paraneoplastic neurological syndromes. He died due to disease progression 14 months later. Subsequent postmortem examination revealed white ill‐defined nodules in the left lung, with similar nodules in other organs. The brain weighed 1500 g before fixation, and a nodule was observed in the right precentral gyrus. Microscopically, the lung tumor was a pleomorphic carcinoma with an adenocarcinoma component. Multiple areas of micro‐softening (≤500 μm) were identified in the cerebral cortex, gray–white matter junction and basal ganglia, and were distributed diffusely in both the limbic and non‐limbic systems. Mild lymphocytic infiltrates were observed involving few intraparenchymal vessels. Few tumor metastases were observed in the right precentral gyrus. The multiple micro‐softenings may reflect a chronic neuropathologic change of paraneoplastic autoimmune encephalitis. They were too small to be detected by brain MRI. However, these lesions may have the potential to cause the neurological symptoms in the acute phase because they were observed in many anatomical regions. We should pay attention to subtle findings such as micro‐softenings when estimating the neuropathology of autoimmune encephalitis. Further investigations are needed to understand the characteristic neuropathology of this condition.     

Subacute reversible toxic encephalopathy related to treatment with capecitabine: A case report with literature review and discussion of pathophysiology

IntroductionCapecitabine, a 5-fluorouracil (5FU) pro-drug, is increasingly used in breast and gastrointestinal cancers due to its more convenient oral route of administration when compared to 5FU. Despite its widespread use, there are only a few reports on capecitabine CNS toxicity, while the pathogenic basis of such toxicity remains unclear.CaseA 69-year-old male presented with recurrent generalized seizures 2.5 months after preoperative chemoradiotherapy with capecitabine in locally advanced rectal cancer. Brain MRI revealed a diffuse, subcortical white matter alteration suggestive of vasogenic edema. The diagnosis of toxic encephalopathy was supported after elimination of alternative causes of the neurological dysfunction and complete resolution of clinical and imaging findings after 3 months of no further chemotherapy.ConclusionsGiven the expanding use of capecitabine, physicians should be aware of this potential complication when a neurological worsening occurs during or after treatment with this chemotherapeutic agent. In our case, as in previously described cases encephalopathy was characterized by a favorable course after cessation of the drug. Vasogenic edema rather than cytotoxic edema may play a pivotal pathogenetic role in this form of encephalopathy.     

Intravascular carcinomatosis of central nervous system due to metastatic inflammatory breast cancer: A case report

Central nervous system (CNS) involvement by metastatic cancer is well‐recognized and typically presents with multifocal solid tumors within the brain parenchyma or leptomeningeal dissemination. We describe herein a histologically very rare case of CNS metastasis in a 52‐year‐old woman who presented with mental status changes. Post mortem examination revealed extensive CNS involvement by metastatic inflammatory breast carcinoma, characterized by the presence of single tumor cells diffusely present within capillaries without parenchymal or perivascular invasion, and acute ischemic changes/infarcts bilaterally involving multiple areas. The cancer cells were found predominantly in the cerebral cortices and deep gray matter structures. Pre‐mortem magnetic resonance and CT imaging of the brain did not identify metastatic cancer; however, widespread ischemic changes (i.e. brain infarcts) were identified. Inflammatory breast carcinoma is well‐known to have a predilection for spread through lymphovascular spaces. Post mortem examination revealed tumor involvement of bilateral lungs, heart and bladder, with sinusoidal spread in the liver and lymph nodes and prominent involvement of the splenic red pulp in addition to extensive vascular involvement of the brain and spinal cord without a discrete mass, despite the presence of widely metastatic disease. The tumor cells in the CNS were strongly immunoreactive for pancytokeratin, E‐cadherin, cytokeratin‐7, epithelial membrane antigen and CAM 5.2. This unique histologic pattern of tumor spread is considered to represent “intravascular carcinomatosis” in the CNS, and most likely the cause of the patient's widespread ischemic injuries.     

Myelin oligodendrocyte glycoprotein antibody-associated disease in a patient with symptoms of aseptic meningitis who achieved spontaneous remission: A case report and review of the literature

BackgroundA few case reports have described patients with myelin oligodendrocyte glycoprotein antibody (MOG-Ab)-associated demyelinating syndrome who presented with symptoms of aseptic meningitis. All such patients required immunotherapy. We report a patient with MOG-Ab-associated disorder (MOGAD) who presented with symptoms of aseptic meningitis and improved without treatment.CaseA 13-year-old girl presented with fever, headache, decreased appetite, and neck stiffness. Cerebrospinal fluid (CSF) analysis revealed pleocytosis and magnetic resonance imaging (MRI) showed leptomeningeal enhancement. The patient was diagnosed with aseptic meningitis at admission. However, there were no signs of recovery 4 days after admission (i.e., 8 days after disease onset). Therefore, we performed extensive investigations to identify the cause of the underlying infection and inflammation. On day 14 after admission, the serum MOG-Ab test performed at admission came back positive (1:128) and she was diagnosed with MOGAD. She was discharged on day 18 after admission, because her symptoms, CSF pleocytosis, and MRI findings had improved. About 6 weeks after discharge, MRI revealed hyperintensity without gadolinium enhancement. However, her serum MOG-Ab test was negative. We did follow-ups for 11 months but found no new neurological symptoms.Discussion and ConclusionTo the best of our knowledge, this is the first ever report of a pediatric patient with MOGAD experiencing spontaneous remission with no demyelinating symptoms during an extended follow-up period.