Recurrent Aseptic Meningitis Research Articles

Posterior Fossa Dermoid Cyst With Sinus Tract and Meningitis in a Toddler

Intracranial dermoid cysts are rare, but can cause recurrent bacterial or aseptic meningitis, abscesses or mass effect. We describe a toddler who manifested an occipital "lump" for 6 months without further evaluation. He presented with lethargy and meningitis. A posterior fossa dermoid cyst with a sinus tract was detected by imaging studies. He underwent prompt surgical excision of the cyst and the sinus, and received a prolonged course of antibiotics, because anaerobes were isolated from the contents of the cyst and blood. His postoperative course was complicated, and required the placement of a lumboperitoneal shunt. A high index of suspicion for the detection of dermoid cysts is required when a congenital midline skin lesion such as a dimple or a scalp swelling is reported. Prophylactic excision is indicated to prevent serious sequelae.

Recurrent Benign Lymphocytic (Mollaret) Meningitis in Systemic Lupus Erythematosus

Aseptic meningitis is one of the most infrequent neuropsychiatric manifestations of systemic lupus erythematosus (SLE) with multifactorial etiologies including medications such as nonsteroidal anti-inflammatory drugs, azathioprine, and trimethoprim-sulfamethoxasole, as well as viruses and malignancy. Recurrent aseptic meningitis in SLE is rare, and remains a diagnostic challenge. We report a unique SLE patient with recurrent (10 episodes), benign (self-limited) lymphocytic aseptic meningitis, which suggests the diagnosis of Mollaret meningitis. There was no prior use of medications known to provoke meningitis. No infectious etiology was identified and chronic meningitis was not observed. The patient had spontaneous resolution of symptoms with no neurologic sequelae. Recurrent benign lymphocytic aseptic meningitis is recognized in this SLE patient. We propose that noninfectious Mollaret meningitis be classified as a feature of neuropsychiatric SLE syndromes.

１１年の経過を経て再発した無菌性髄膜炎をともなう亜急性壊死性リンパ節炎の１例

We report a 29-year-old man with subacute necrotizing lymphadenitis (SNL) associated with recurrent aseptic meningitis following an 11-year remission period. In both episodes, headache and fever were followed by lymphadenopathy, with increased serum IgE level. Although pleocytosis in cerebrospinal fluid was confirmed at admission in the first episode, it appeared at one week after admission in the second episode. Administration of glucocorticoid was effective for treating meningitis. The present case suggests a pathomechanism for SNL that involves both an immunological background and an acute viral infection as triggers of exacerbation of aseptic meningitis.

Is total excision of spinal neurenteric cysts possible?

Spinal neurenteric cysts are intradural cystic lesions. These represent a part of the spectrum of developmental anomalies. These are rare lesions. Most of the data in literature is review of case reports. This made us evaluate our results of 23 cases and review the literature. This is a retrospective study of 23 patients managed at our institute over 20 years. The slides were retrieved and histopathological features studied. Twenty patients were less than 30 years old and 21 were males. Follow-up was available for 21 patients with mean duration of 71 months (range 2 months to 23 years). The typical presentation was backache with progressive neurological deficits pertaining to the level. Atypical presentations as acute onset, recurrent episodic events and aseptic meningitis were also noted. The cervicothoracic region was the most common site. 16 patients had cyst in intradural extramedullary plane and seven had intramedullary location. Associated vertebral anomalies as hemivertebrae, Klippel – Feil and spina bifida were noted in seven patients. There were two histological types of cysts with no correlation between the type of cysts and associated vertebral anomalies and extent of outcome. Partial excision though had higher risk of recurrence, was not associated with poorer outcome. Dorsal approach is an acceptable route with reasonably good results for this lesion. Spinal neurenteric cysts present at younger age with varied clinical presentations. These are commonly located intradurally ventral to the cord. Histological types have no effect on the outcome. Total excision is the choice of treatment. However, partial excision is a feasible option in intramedullary lesions and when significant adhesions occur. Although associated with higher risk of recurrence, the outcome is still good in these patients on re-excision.

Mollaret Meningitis Associated With an Intraspinal Epidermoid Cyst

Mollaret meningitis, a benign recurrent aseptic disease, is known to be associated with intracranial epidermoid cysts. In this report, we describe a case of Mollaret meningitis caused by an intraspinal epidermoid cyst located at thoracic level 12. The patient's clinical manifestations and cerebrospinal fluid features were similar to those with bacterial meningitis characterized by predominant polymorphonuclear leukocytes. However, Mollaret cells, not bacteria, were identified in the patient's cerebrospinal fluid. The illness ceased after surgical resection of the cyst, and the cyst tissue was pathologically diagnosed as epidermoid. Therefore, an intraspinal epidermoid cyst can be etiologically associated with Mollaret meningitis and should be included in the differential diagnosis of recurrent aseptic meningitis.

Increased Detection Rate in Diagnosis of Herpes Simplex Virus Type 2 Meningitis by Real-Time PCR Using Cerebrospinal Fluid Samples

Efficient and sensitive diagnostic methods are needed in the management of virus infections in the central nervous system. There is a demand for an evaluation of the sensitivity of PCR methods for early diagnosis of meningitis due to herpes simplex type 2 (HSV-2) and varicella-zoster virus (VZV). The objective of this study was to evaluate real-time PCR in the detection of HSV-2 and VZV DNA from cerebrospinal fluid (CSF) for etiological diagnoses in clinically well-characterized cases of primary and recurrent aseptic meningitis. Samples from 110 patients, 65 of whom were diagnosed with or were strongly suspected of having HSV-2 meningitis and 45 with acute aseptic meningitis of unknown causes, were analyzed. Results were compared with the outcome of nested PCR for HSV-2 infection. Clinical parameters were analyzed in relation to CSF viral load. With real-time PCR, HSV-2 DNA was found in CSF from 80% (52/65) of patients with clinical HSV-2 meningitis compared to 72% (47/65) found by nested PCR. The sensitivity of real-time HSV-2 PCR was found to be 87% (33/38) in primary and 70% (19/27) in recurrent meningitis. The HSV-2 viral load was significantly higher in primary than in recurrent meningitis and correlated with the degree of inflammation. VZV DNA was detected in 2 of 45 samples (4.4%). Real-time PCR for the diagnosis of HSV-2 meningitis was evaluated in a large, clinically well-characterized sample of material and found to identify more cases than nested PCR in the group of patients with recurrent meningitis. Quantification of DNA enables further research of disease prognosis and treatment.

Recurrent aseptic meningitis: a new CSF complication of Sjogren’s syndrome

Sirs: Sjogren’s syndrome (SjS) is one of the major autoimmune disorders and is characterized by dry eyes and a dry mouth. A wide variety of extraglandular manifestations has been reported in SjS. Aseptic meningitis is a well-known manifestation of SjS; however, little is known about its pathomechanism [1, 3]. We detected a unique autoantibody in the serum and cerebrospinal fluid (CSF) of an SjS patient with recurrent aseptic meningitis. A 19-year-old female was readmitted to our hospital after the third recurrence of aseptic meningitis. Nine months and five months before this admission, she had experienced the same symptoms, i.e., headache, nausea, and fever. She was diagnosed with aseptic meningitis because of the presence of mononuclear pleocytosis in the CSF. The symptoms subsided spontaneously within a few days. On examination, there were no subjective symptoms of sicca syndrome, and Schirmer’s test was negative. Neurological examination revealed no abnormalities, except stiffness of the neck. Laboratory tests revealed elevated amylase (268 IU/dl) and serum IgG (2920 mg/dl) concentrations; positive rheumatoid factor (2+); accelerated erythrocyte sedimentation rate (30 mm/h); and positive serum antinuclear antibody (ANA) as well as the anti-SS-A and anti-SS-B antibodies at titers of 1:2560, 1:256, and 1:8, respectively. The CSF contained 153 mg/dl protein and 85 mm of cells (81% mononuclear cells); the anti-SS-A and anti-SS-B antibodies were absent. These findings led us to suspect SjS. A sialogram revealed the destruction of the sialoducts, and a salivary gland biopsy revealed abundant lymphocytic infiltration. The patient was diagnosed with SjS and recurrent aseptic meningitis, and she was treated with 50 mg of daily oral prednisolone. Based on the results of the physical and serological examinations, we inferred that the overlapping of other systemic autoimmune diseases with SjS is unlikely. The clinical symptoms resolved immediately without subsequent recurrence. The patient’s serum and CSF were analyzed by using western blotting (WB) and immunohistochemistry as reported previously [4]. WB of bovine tissue samples showed that the CSF reacted with two bands with apparent molecular weights of 56 kD and 44 kD in the cerebral cortex, meningeal tissue, and liver (Fig. 1a). Immunohistochemical analysis by the avidin-biotin-peroxidase method using rat cerebral slices showed that the CSF labeled the nuclei of the neurons and meningeal cells (Fig. 1b). We detected an identical autoantibody in the patient’s serum (data not shown). Mauch et al. reported that approximately 70% SjS patients had neurological complications; of these, one-sixth had aseptic meningitis [3]. Alexander et al reported that from a group of 25 consecutive SjS patients with CNS involvements, 5 (20%) had recurrent aseptic meningitis or meningoencephalitis [1]. Although aseptic meningitis is common in SjS, recurrent aseptic meningitis without other CNS involvements is rare, and its pathoetiology has not been elucidated thus far. Some autoantibodies may be related to the pathogenesis of the CNS involvement in SjS, for example, the possible association of the anti-SS-A antibody with cerebral angiitis [2]. However, the relationship between these autoantibodies and CNS involvement remains unknown. We demonstrated the presence of an autoantibody in the patient’s CSF and serum; this autoantibody recognizes 56and 44-kD nuclear antigens of the CNS neurons, meningeal cells, and cells of other systemic organs. The presence of this autoantibody in the patient’s CSF may imply its causative association with recurrent aseptic meningitis in SjS. We already reported some other autoantibodies in SjS patients with CNS involvements, but this is the first report K. Ishida, MD, PhD T. Uchihara, MD, PhD H. Mizusawa, MD, PhD Dept. of Neurology and Neurological Science, Tokyo Medical and Dental University Graduate School 1-5-45 Yushima, Bunkyo-ku Tokyo 113-8519, Japan

Recurrent aseptic meningitis associated with herpes simplex virus type 2

Recurrent aseptic meningitis associated with herpes simplex virus type 2

ISMP Adverse Drug Reactions - Intractable Epistaxis Associated with Topiramate; Nerve Injury Related to Implantable Hormonal Contraceptive; Cephalosporin Encephalopathy in a Patient with Normal Renal Function; Severe Hepatic Injury Associated with Orlistat; Methotrexate-Induced Brain Cyst as a Complication of Intraventricular Chemotherapy; Oculogyric Crisis Caused by Olanzapine; Burning Mouth Syndrome and Efavirenz; Recurrent Ibuprofen-Induced Aseptic Meningitis

ISMP Adverse Drug Reactions - Intractable Epistaxis Associated with Topiramate; Nerve Injury Related to Implantable Hormonal Contraceptive; Cephalosporin Encephalopathy in a Patient with Normal Renal Function; Severe Hepatic Injury Associated with Orlistat; Methotrexate-Induced Brain Cyst as a Complication of Intraventricular Chemotherapy; Oculogyric Crisis Caused by Olanzapine; Burning Mouth Syndrome and Efavirenz; Recurrent Ibuprofen-Induced Aseptic Meningitis

Recurrent ibuprofen-induced aseptic meningitis: Evidence against an antigen-specific immune response

Drug-induced aseptic meningitis (DIAM) may be difficult to differentiate from bacterial meningitis or from manifestations of the underlying disease.1,2 We report a patient with recurrent ibuprofen-induced aseptic meningitis, whose history suggests an antigen-specific T-cell mechanism. A 55-year-old man began a treatment of ibuprofen for osteoarthritis of the hip. After 3 months of nonspecific flu-like symptoms and muscle pain, he presented with an acute onset of headache, fever, meningism, and dizziness. CSF analysis disclosed an enhanced cell count (145 cells/mm3, predominantly polymorphonuclear cells and macrophages), enhanced proteinorachia (1,230 mg/L), but a normal glucose level. Blood and CSF cultures, viral serologies, and antinuclear factor were negative. Symptoms improved after ibuprofen was stopped. Subsequently the patient occasionally took aspirin and mefenamic acid without any adverse effect. A follow-up CSF analysis was normal. Nine months later, a single dose of 600 mg ibuprofen was administered, and within 90 minutes, the patient presented with fever, chills, headache, …

Characteristics of Meningitis Caused by Ibuprofen

Ibuprofen is a common nonsteroidal antiinflammatory drug that is the most frequent cause of aseptic meningitis induced by drugs. The incidence of this type of aseptic meningitis is increasing, mainly among patients with underlying autoimmune connective tissue disorder, but also among healthy people. We report 2 patients with recurrent meningitis caused by ibuprofen mimicking bacterial meningitis: the first patient a woman with dermatomyositis and the second patient a previously healthy woman who developed autoimmune thyroiditis a few months later. We then review 71 episodes of ibuprofen-related meningitis in 36 patients reported in the literature. Twenty-two patients (61%) presented with an autoimmune connective tissue disorder, mainly systemic lupus erythematosus, and 22 (61%) had recurrent episodes. Most episodes consisted of an acute meningeal syndrome with a predominance of neutrophils in cerebrospinal fluid (CSF) in 72.2% of episodes and elevated protein in the CSF, so the clinical presentation of this type of aseptic meningitis may be quite similar to that of acute bacterial meningitis. CSF glucose levels are usually normal, which may help to differentiate between these 2 types of meningitis. In some cases the clinical presentation is that of meningoencephalitis with neurologic focal deficits. Although based on the close relation between the administration of ibuprofen and the onset of symptoms, especially if previous episodes have occurred, the diagnosis of ibuprofen-induced aseptic meningitis is a diagnosis by exclusion. If the clinical picture is compatible with bacterial meningitis, empirical antibiotic therapy must be administered until negativity of cultures and other microbiologic tests is determined. Rechallenge to ibuprofen reproduces the symptoms and confirms the diagnosis, but is usually not advised. Whatever the clinical presentation, physicians must consider the possibility of ibuprofen-related meningitis or meningoencephalitis in patients taking ibuprofen, especially if they are suffering from an autoimmune connective tissue disorder. On the other hand, we think it would be appropriate to screen for autoimmune disease in previously healthy patients diagnosed with ibuprofen-related meningitis or meningoencephalitis. Finally, we propose that meningitis due to ibuprofen be included in the list of causes of recurrent aseptic meningitis.

Endodermal Cyst Ventral to the Lower Brain Stem-Case Report-

A 28-year-old woman presented with a rare case of endodermal cyst located ventral to the lower brain stem manifesting as recurrent aseptic meningitis. Computed tomography and magnetic resonance imaging demonstrated a cystic mass located ventral to the lower brain stem and extending from the prepontine to the upper cervical (C-2) cistern. The lesion was totally removed through a retrosigmoid craniotomy and C1-2 hemilaminectomy. Histological and immunohistochemical examination showed the cyst was derived from the endoderm. The histological diagnosis was endodermal cyst. Early diagnosis and surgical removal are important for patients with these cysts.

Primary Sjoegren's syndrome with recurrent aseptic meningitis.

Primary Sjoegren's syndrome with recurrent aseptic meningitis.

Meningitis recurrente aséptica secundaria a toma de ibuprofeno y ketorolaco

Aseptic meningitis is a process that is characterised by an inflammatory reaction of the meninges that is not due to any infectious agent. Its aetiology is varied and is most frequently caused by rheumatologic and/or autoimmune processes, chemical or medication-induced meningitis, the most notable drugs involved being antibiotics and non-steroidal anti-inflammatory drugs (NSAI). We report the case of a 70-year-old male, with no relevant history, who was admitted to hospital five times over a period of 16 months because of acute meningitis with polymorphonuclear pleocytosis, high protein levels in cerebrospinal fluid and normal glucose in cerebrospinal fluid. No evidence of an infectious causation, chemical meningitis, carcinomatosis or autoimmune disease was found and the patient was diagnosed with recurrent aseptic meningitis. It was found that the patient had taken ibuprofen or ketorolac on several occasions, a few hours before the appearance of symptoms. These episodes were quickly resolved after withdrawal of this medication. A number of NSAI have been reported as inducers of aseptic meningitis, one of the most notable being ibuprofen. We report the case of a patient who, as a consequence of taking ibuprofen and ketorolac, presented episodes of recurrent aseptic meningitis. To our knowledge this side effect of ketorolac has not been reported before. Its clinical features are impossible to differentiate from those of infectious meningitis. Diagnosis is reached by exclusion and a careful pharmacological study, including over-the-counter drugs like some of the NSAI, must be performed in patients with this condition, since it is a problem that can easily be solved by withdrawing the drug that causes it.

Recurrent Severe Aseptic Meningitis after Exposure to Lamotrigine in a Patient with Systemic Lupus Erythematosus

Recurrent Severe Aseptic Meningitis after Exposure to Lamotrigine in a Patient with Systemic Lupus Erythematosus

Complement C7 deficiency presenting as recurrent aseptic meningitis

Complement deficiency states are rare inherited disorders that may predispose affected individuals to angioedema, collagen vascular disease, or infection due to encapsulated organisms, especially Neisseria meningitidis. To report the case of a 36-year-old man of Irish descent with recurrent culture-negative neutrophilic meningitis, to offer potential reasons for the inability to recover a causative pathogen, and to review the genetics and prevalence of complement deficiency states, the methods of screening for such deficiencies, the features of meningococcal infection as they relate to such deficiencies, and management strategies for clinicians caring for patients with such deficiencies. The patient presented in 1988 and again in 2002 with culture-negative neutrophilic meningitis. His second episode was characterized by a rash suggestive of meningococcal infection, prompting immunologic evaluation. Immunologic evaluation revealed an undetectable CH50 level. Levels of C1, C2, and C5 through C9 were normal except for C7, which was undetectable. Further testing revealed that the patient's sister was also C7 deficient. Complement component deficiencies are relatively rare; individuals with collagen vascular disease and systemic neisserial infection should be screened using either the CH50 or the APH-50 assay. Key to the management of a late-complement component-deficient host is counseling, education about meningococcal infection, and discussions about the potential benefits of chemoprophylaxis and immunoprophylaxis. The ability to detect the bacterial cause of meningitis in such patients is organism dependent and may be influenced by factors such as cerebrospinal fluid bacterial concentration and previous antibiotic drug exposure.

Recurring aseptic meningitis after travel to the tropics: a case of Mollaret's meningitis? Case report with review of the literature

Recurrent aseptic meningitis in a 35-year-old caucasian woman is described. She had many attacks over a period of 9 years. The first attack occurred after travel in the tropics. In spite of extensive examinations no cause could be found for the recurrent attacks. Both the clinical presentation and characteristics of the cerebrospinal fluid are compatible with the diagnosis of Mollaret's meningitis. There is no known cure for this condition, although colchicine and indomethacin have been mentioned to relieve symptoms. In our patient, a treatment with indomethacin during the last attack resulted in a clear and rapid improvement of symptoms. Since this episode only mild relapses have occurred, all of which responded well to the same treatment. This case highlights the long time span in which attacks of Mollaret's meningitis can occur, and the spectacular benefit of indomethacin.

Analysis of candidate-host immunogenetic determinants in herpes simplex virus-associated Mollaret's meningitis.

Infection due to herpes simplex virus (HSV) is associated with recurrent aseptic meningitis (Mollaret's meningitis); however, the neuropathogenesis of this disease remains unknown. We collected 20 cerebrospinal fluid (CSF) specimens that were positive for HSV DNA by using polymerase chain reaction (PCR) assay from patients with a clinical diagnosis of Mollaret's meningitis. Patients were predominantly female (female:male, 22:1), with an average age of 32.8 years (range, 18-46 years). Using direct sequence analysis of HSV PCR products obtained from the CSF, we determined that all of the patients were infected with HSV type 2. In addition, we evaluated polymorphisms in 2 human genomic loci, which are associated with either severe or recurrent microbial infections (interferon-gamma receptor [IFN-gammaR] and mannose binding lectin [MBL]); these host genes were also amplified directly from the CSF specimens. No mutations were found in exons 2 or 3 of the IFN-gammaR gene (n=20). In contrast, there were 4 (20%), 4 (20%), and 0 mutations found in codons 52, 54, and 57, respectively, in exon 1 of MBL (n=20). A significantly higher frequency of codon 52 mutations (P=.04) was observed, compared with racially matched control patients.

Mollaret's recurrent aseptic meningitis and cerebral epidermoid cyst

A patient who initially presented with clinical and laboratory signs of bacterial meningitis developed four more similar episodes within a 6-month period. Immune system studies were unremarkable. A tentative diagnosis of Mollaret's meningitis was established after the third episode. Cranial computed tomography performed during the acute phase of the initial episode was normal, but, after the fourth scan, when the patient was asymptomatic, magnetic resonance imaging revealed a cyst in the anterior aspect of the medulla. Partial excision allowed for pathologic analysis, which established the diagnosis of epidermoid cyst. The differential diagnosis of recurrent aseptic meningitis should include Mollaret's meningitis and dermoid-epidermoid cysts. Neuroimaging studies, preferably magnetic resonance imaging, should be performed at a time when patients are asymptomatic.

P-5-677 - Recurrent aseptic meningitis in ruptured intracranial dermoid. Case report

Dermatofibrosarcoma protuberans (DFSP) is especially challenging to recognize and diagnose in children. Prompt and definitive treatment is essential based on the tumor's high rate of recurrence. Our purpose is to increase physician awareness and recognition of DFSP in the pediatric population with the largest case series of childhood DFSP to our knowledge thus reported in the literature. We also propose that magnetic resonance imaging (MRI) may be useful in the preoperative assessment. The records of 10 patients with primary DFSP between the ages of 8 months and 16 years were reviewed. Only 1 of 10 patients was given the correct diagnosis at the initial medical evaluation. Of the 10 cases, 5 were congenital. The average delay in diagnosis was 5 years. Of the patients, 5 had preoperative MRI to delineate the size and extent of the tumor. In the two cases with the largest tumors preoperative MRI significantly altered surgical planning and execution. Thus, preoperative MRI may be helpful in planning for successful surgical clearance of large tumors and preventing local recurrence. All 10 cases were treated with Mohs micrographic surgery and no recurrences were noted with a follow-up range of 6 weeks to 17 years.