A case of famotidine-induced aseptic meningitis]

We reported a 50-year-old woman with a history of mixed connective tissue disease. She had two episodes of meningitis-like symptoms after taking famotidine and tiquizium bromide for treatment of gastric ulcer. From CSF findings (elevated pressure, increase of protein, polymorphonuclear pleocytosis, negative culture) and result of famotidine challenge test, we diagnosed her as a drug induced aseptic meningitis. Because she had taken tiquizium bromide several times previously without any side effects, we concluded that famotidine was a causative drug. She was recovered without sequelae within a few days following cessation of these drugs. This is the first report of H2-blocker induced aseptic meningitis. When we encounter a patient with aseptic meningitis who presents polymorphonuclear pleocytosis in CSF, we should suspect drug induced aseptic meningitis and take a history of drug medication including H2-blocker.     

A case of recurrent aseptic meningitis caused by high-dose intravenous gammaglobulin therapy for chronic inflammatory demyelinating polyneuropathy]

We experienced a patient of CIDP who was twice complicated with aseptic meningitis following high-dose intravenous gammaglobulin therapy. The patient was a 29-year-old woman who first developed gait disturbance in September 1998. Neurological examination revealed muscle weakness and sensory disturbance in the distal parts of four extremities and decrease of deep tendon reflexes. Cerebrospinal fluid analysis revealed an elevated protein content and a normal cell count. Steroid therapy was effective in early stage, however, this effectiveness had been reduced gradually. She received high-dose intravenous gammaglobulin administrations in September 1999. On the fourth day after start of this therapy, she developed severe headache, nausea and nuchal rigidity without fever. Cerebrospinal fluid analysis revealed an increased cell count of mononuclear predominance and an elevated protein content. As bacterial culture remained negative and viral titers were not elevated, aseptic meningitis was diagnosed. The therapy was stopped, and thereafter her headache continued for 7 days. The muscle weakness and sensory disturbance were remarkably improved, but 9 months later, her symptoms became worse again. She received high-dose gammaglobulin administrations for 2 days in June 2000 and developed aseptic meningitis again. Over again, 9 months later, she received the same medication for only 1 day in March 2001 and she developed mild headache but not meningitis. Aseptic meningitis with CIDP following high-dose gammaglobulin therapy was rare, however, we should pay attention to this therapy in patients with CIDP and may prevent the occurrence of aseptic meningitis by reducing the total dose and shortening the administration periods of gammaglobulin.     

A case of urinary retention secondary to aseptic meningitis]

A case of a 37-year-old man with urinary retention secondary to aseptic meningitis is described. He was admitted to our hospital with complaints of fever, nausea, headache, and gait disturbance. He had begun treatment 1 week previously for high fever, headache and joint pain at an outpatient department. On admission, neurological examination revealed neck stiffness, spasticity and hyperreflexia of the extremities, hypesthesia of the lower extremities, and ataxic gait. A spinal fluid examination revealed aseptic meningitis. Disturbed consciousness, diplopia, aphasia, tetraparesis, and urinary retention appeared after admission. Computed tomography and magnetic resonance imaging (MRI) of the head revealed no abnormal lesions. On the 2nd day of hospitalization, a Foley catheter was inserted for urinary retention. The patient was treated with conservative therapy, and his symptoms gradually resolved. The Foley catheter was removed on hospital day 13, but bladder dysfunction was persisted. MRI of the lumbar spine revealed herniated lumbar discs at level L3-4, L4-5, and L5-S1, but the nerve roots were not compressed. The patient was managed with clean intermittent catheterization for more than two weeks. Cystometry revealed atonic bladder. Nineteen cases of urinary retention secondary to aseptic meningitis have been reported in the previous literature. We reviewed their clinical presentation and treatment.     

Case of relapsing polychondritis followed by cognitive impairment]

A 64-year-old man developed cognitive impairment. A neurological examination showed hypereactive tendon reflex. A cerebrospinal fluid (CSF) examination showed a mild increase in both the number of mononuclear cells (14 cells/mm3) and the protein level (45 mg/dl). An electroencephalogram showed diffuse slow wave activity. Brain MR images revealed multifocal lesions in the limbic area and white matter regions. He was suspected to have autoimmune encephalitis and his symptoms improved after treated with methylprednisolone (1 g/day for 3 days). Despite this treatment, his mental disorders later relapsed. His condition improved after undergoing repeated treatment. Thereafter, he developed bacterial meningoencephalitis which had been caused by Listeria monocytogenes during the period of the internal administration of steroids. Antibiotics successfully cured his Listeria meningoencephalitis. He thereafter developed conjunctivitis, polyarthritis, hearing difficulties, dizziness and auricular chondritis. Based on these findings, we diagnosed the patient to have relapsing polychondritis (RP). It was thus conceivable that his cognitive impairment has been caused by CNS vasculitis associated with RP. RP therefore should be carefully taken into consideration and be included in the differential diagnosis as a possible cause for CNS vasculitis.     

Meningoencephalitis or meningitis in relapsing polychondritis: four case reports and a literature review

Relapsing polychondritis (RP) is a rare autoimmune disease that affects cartilage throughout the body, causing episodic and progressive inflammation. Although rare, RP has diverse acute and subacute nervous system complications, which may sometimes precede systemic manifestations. Here, we report four patients with RP who presented with meningoencephalitis or meningitis without infectious aetiology. In addition, we review the literature for this disease with regard to clinical manifestations and treatment options.     

A case of relapsing polychondritis with oculobulbar symptoms and successful treatment of respiratory failure with BiPAP]

A 66-year-old man developed diplopia, ptosis, dysphagia, and acute respiratory failure. The initial diagnosis was myasthenia gravis and prednisolone had been administrated for three years. Because of recurrent upper respiratory infections, prednisolone was tapered off. Two months later, auricular chondritis, arthritis, and conjunctivitis appeared. He was diagnosed as having relapsing polychondritis on the basis of histological findings of the ear lobe biopsy. Reinstituted prednisolone had the effect on the auricular chondritis, arthritis, and conjunctivitis, but no effect on dysphagia, hoarseness, and respiratory failure caused by the deformity of the pharynx and airway. Tracheal collapse usually causes rapid death, so early tracheostomy and the use of endotracheal prostheses have been recommended in patients with airway obstruction from relapsing polychondritis, but such surgical management can only partially open up the large airways and has no effect on smaller airways. In this case tracheostomy and endoluminal stent placement have helped improve the patient's respiratory failure, but have had little effect on its aggravation at night in the supine position. The use of BiPAP after surgical management can be an effective treatment for airway involvement in relapsing polychondritis probably because it keeps the narrowed airways from collapsing, especially at night.     

An adult case of bacterial meningitis caused by penicillin-resistant streptococcus pneumoniae]

We report a patient of bacterial meningitis caused by penicillin-resistant streptococcus pneumoniae (PRSP). A 50-year-old Japanese man was admitted after developing a fever and quickly falling into unconsciousness. Neurological examination showed slightly consciousness disturbance and meningeal irritation. A lumbar puncture yielded turbid spinal fluid, with increased cell count (411/mm3), protein (685 mg/dl) and IgG (60.3 mg/dl) but decreased glucose (1 mg/dl). Bacterial meningitis was diagnosed and aminobenzylpenicillin (ABPC) and cefotaxime (CTX) were administered immediately, but they were ineffective. Penicillin-resistant streptcoccus pneumoniae (PRSP) was detected in the blood and spinal fluid, so antibiotics were changed to panipenem/betamipron (PAPM/BP) and vancomycin (VCM) with marked efficacy. With the increase in PRSP patients and documented failure in treatment of pneumococcal meningitis with ABPC and CTX, the need for alternative antibiotic therapy is critical. We emphasize the importance of initial therapy with PAPM/BP and VCM in patients with bacterial meningitis from streptcoccus pneumoniae.     

A case of subacute necrotizing lymphadenitis with recurrent aseptic meningitis 11 years after the first episode]

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.     

A case with bacterial meningitis caused by cerebrospinal fluid rhinorrhea 22 years after head trauma]

A 38-year-old man with past history of head injury at the age of 16 years was admitted to our hospital in April, 2000 because of bacterial meningitis. At the end of March 2000, he experienced massive cerebrospinal fluid (CSF) rhinorrhea when he rested in bed, and subsequently he developed high-grade fever, headache, and nuchal stiffness in 2 weeks. His symptoms and signs improved with the antibiotic therapy alone. He had no recurrence of meningitis since then. A defect of his right skull base was clarified by 3D-CT, and multiplanar reconstruction (MPR)-CT. MRI brain scan showed the dislocation of the right frontal lobe into the ipsilateral anterior ethmoidal sinus. The diagnosis of CSF rhinorrhea and bacterial meningoencephalitis were made according to his clinical manifestations and neuroradiological studies including 3D-CT, MPR-CT and MRI brain scans. These imaging modalities, which described his head anatomy precisely, helped us to clarify the cause of his illness. When CSF rhinorrhea is present, it is important to take detailed past history of head trauma, even if the event occurred more than 20 years previously.     

Stroke as initial manifestation of relapsing polychondritis

Relapsing polychondritis (RP) is a rare vasculitide characterized by recurrent inflammation of cartilagenous structures (auricular, nasal, laryngotracheal, articular) and specific sense organs (eyes and ear). Neurological complications are exceptional. We report the case of an elderly and previously well woman who presented with inflammatory syndrome and stroke as the first manifestations of this disease.

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Sommario La policondrite ricorrente è una rara forma di vasculite caratterizzata da ripetuti processi infiammatori delle strutture cartilaginee (auricolari, nasali, laringotracheali, articolari) e di organi di senso specifici (occhio, orecchio). Le complicanze neurologiche sono molto rare. Riportiamo un caso di una donna che ha sempre goduto di buona salute, che ha esordito con una sindrome infiammatoria e ictus come prima manifestazione della malattia.

     

A case of transverse myelopathy caused by acupuncture]

A 54-year-old man received insertion of an acupuncture needle into the region extending from the posterior neck to the back on two occasions for the treatment of shoulder stiffness. Two weeks after the second acupuncture, he developed fever, dysarthria and mictionary disturbance, finally reaching the condition of tetraplegia. He was immediately admitted to an emergency room in our hospital, and was diagnosed as sepsis with DIC, ARDS, heart failure, renal failure, liver failure, and myelitis. After one month, he recovered with transverse myelopathy as a residual deficit. Neurological findings showed transverse myelopathy below the level of Th2 at that time. Cervical CT revealed an irregular low density at the periphery of the cervical vertebra from the C2 to C4 level. Cervical MRI revealed an irregular swelling of his spinal cord from the C2 to C7 level. We explained the mechanism of transverse myelopathy in this case as follows. After the acupuncture, he suffered a focal infection of the region of needle insertion, and then the infection expanded to the cervical vertebra, thus causing osteomyelitis, sepsis, and finally cervical myelitis. Direct injury of the spinal cord and nerve roots as a complication of acupuncture was previously reported, but indirect injury of the spinal cord due to myelitis had not been reported except our present case. Careful attentions should be paid to the complications of acupuncture.     

A case of reversible encephalopathy caused by tegafur]

A case of reversible encephalopathy induced by tegafur: a masked compound of 5-Fluorouracil, is reported and discussed. A 67-year-old Japanese man suddenly began to show changes in mental status after 4.5 months of administration of tegafur for the peritonitis carcinomatosa. Alternation in consciousness supported by electroencephalographic evidence became apparent. The P100 latency in visual evoked potential was prolonged in both sides, and the conduction velocity in ulnar nerves in both sides was also slow. The patient made significant clinical recovery upon discontinuing tegafur, and the abnormal findings on electroencephalography, visual evoked potential and peripheral nerve conduction velocity also returned to normal. When the patients who are taking tegafur show mental symptoms, early diagnosis from both clinical findings and electrophysiological studies should be performed.