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Immunoglobulin G4-related disease (IgG4-RD) is a recently recognized systemic condition. We report a patient presenting with progressive flaccid quadriplegia and thoracic pain. Urinalysis showed the presence of toxic granulation. Lumbar spine magnetic resonance imaging (MRI) revealed a symmetrical spinal dural thickening. The IgG4 staining and serum IgG4 levels were elevated. Histological examination of the pleura showed a characteristic diffuse fibrosis with storiform/brick-wall appearance and lymphoplasmacytic inflammation associated with elevated IgG4-positive plasma cells. We report the first patient with IgG4-related pleural disease. It is noteworthy that the IgG4 staining and serum IgG4 level were elevated.Systemic infusion of tirilazad mesylate in a human sepsis model.
Intravenous administration of tirilazad mesylate, an agent with potent free radical scavenging and iron chelating properties, was found to be effective in a human animal model of septic shock. For evaluation of tirilazad mesylate in septic shock, 15 subjects with gram-negative septic shock were enrolled. Subjects received open-label tirilazad mesylate 1 mg/kg bolus, followed by 0.5 mg/kg/hr infusion for 24 hrs. Patients received a variety of immune modulating drugs in the context of the septic shock. The primary endpoint was an improvement in mean arterial pressure. A number of secondary efficacy endpoints were assessed. Tirilazad mesylate was well tolerated with no episodes of hypotension or increased bleeding. There was no mortality. There was a significantly greater increase in mean arterial pressure (6.2+/-2.7 vs. 3.6+/-1.7 mm Hg, P=0.001) and a decrease in norepinephrine requirements (-0.4+/-1.3 vs. -0.2+/-1.0 microg/kg/min, P=0.02)
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