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Post-infectious Inflammatory Response Syndrome after Cryptococcal Meningitis in a Patient with Sarcoidosis

Year: 2024
Type: article
Study design: Case Report
Abstract: Objective We describe a case of cryptococcal meningitis (CM) followed by postinfectious inflammatory response syndrome (PIIRS). Background CM presents a severe opportunistic infection which may affect immunocompromised and immunocompetent individuals with high morbidity and mortality even when timely diagnosed and treated. Besides direct morbidity from the CM, post-treatment paradoxical inflammatory response syndromes have been described in CM which are life-threatening as the infection itself. Design/Methods N/A. Results A 56-year-old male with personal medical history of hypertension, vestibular migraine, pulmonary sarcoidosis in remission and not on treatment presented with two days of confusion, urinary incontinence, gait impairment. MRI showed obstructive hydrocephalus, ependymal thickening and T2 hyperintensities favoring neurosarcoidosis. CSF studies were remarkable for xanthochromia, lymphocytic pleocytosis and markedly elevated protein while infectious studies were negative including cryptococcal antigen in serum with CSF not tested for cryptococcus. Patient had marked improvement with pulse steroids and was discharged on oral taper with follow up in sarcoid clinic where he was started on methotrexate, but readmitted within 3 weeks post-discharge with intractable headaches. CSF studies were significant for elevated opening pressure, xanthochromia, persistently elevated protein, low glucose and positive CSF and serum cryptococcal antigen with high titers. Antifungal therapy was initiated, patient required lumbar drain and eventually ventriculoperitoneal shunt due to persistently elevated intracranial pressure. His course was complicated by multiple shunt revisions and several re-admissions for lethargy and encephalopathy with persistently elevated protein while cryptococcal antigen titers decreased to very low levels and fungal culture was negative. Patient had improvement of symptoms after initiation of high dose steroids with return of symptoms during taper suggestive of PIIRS. Conclusions PIIRS is a rare paradoxical inflammatory response mediated by activated T-cells which has been described in CM as well as some parasitic infections. Early recognition and initiation of prompt treatment is key to better functional outcomes.
Source: Neurology
Language: English
Subfield: Epidemiology
Field: Medicine
Open Access status: bronze
OpenAlex ID: W4403206847
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