Pleural effusion as the initial clinical presentation in disseminated cryptococcosis and fungaemia: an unusual manifestation and a literature review

Abstract

BackgroundCryptococcus neoformans infection usually presents as chronic meningitis and is increasingly being recognized in immunocompromised patients. Presentation with pleural effusion is rare in cryptococcal disease; in fact, only 4 cases of pleural effusion as the initial clinical presentation in cryptococcosis have been reported in English-language literature to date. We report the first case of pleural effusion as the initial clinical presentation in a renal transplant recipient who was initially misdiagnosed with tuberculous pleuritis but who then developed fungaemia and disseminated cryptococcosis. The examination of this rare manifestation and the accompanying literature review will contribute to increased recognition of the disease and a reduction in misdiagnoses.Case presentationWe describe a 63-year-old male renal transplant recipient on an immunosuppressive regimen who was admitted for left pleural effusion and fever. Cytological examinations and pleural fluid culture were nonspecific and negative. Thoracoscopy only found chronic, nonspecific inflammation with fibrosis in the pleura. After empirical anti-tuberculous therapy, the patient developed an elevated temperature, a severe headache and vomiting and fainted in the ward. Cryptococci were specifically found in the cerebrospinal fluid following lumbar puncture. Blood cultures were twice positive for C. neoformans one week later. He was transferred to the respiratory intensive care unit (RICU) immediately and was placed on non-invasive ventilation for respiratory failure for 2 days. He developed meningoencephalitis and fungaemia with C. neoformans during hospitalization. He was given amphotericin B liposome combined with 5-flucytosine and voriconazole for first 11 days, then amphotericin B liposome combined with 5-flucytosine sustained to 8 weeks, after that changed to fluconazole for maintenance. His condition improved after antifungal treatment, non-invasive ventilation and other support. Further pathological consultation and periodic acid-Schiff staining revealed Cryptococcus organisms in pleural sections, providing reliable evidence for cryptococcal pleuritis.ConclusionPleural effusion is an unusual manifestation of cryptococcosis. Cryptococcal infection must be considered in the case of patients on immunosuppressives, especially solid-organ transplant recipients, who present with pleural effusion, even if pleural fluid culture is negative. Close communication between the pathologist and the clinician, multiple special biopsy section stains and careful review are important and may contribute to decreasing misdiagnosis.

Cryptococcal Pleuritis Presenting with Lymphocyte-predominant and High Levels of Adenosine Deaminase in Pleural Effusions Coincident with Pulmonary Tuberculosis

“…First, both diseases may present with lymphocyte predominance and elevated ADA levels in the pleural effusion. Among reported cases of cryptococcal pleuritis, including our present case, 10/13 (77%) patients had lymphocyte-predominant pleural effusion ( Table ) ( 1 - 15 ). Because host defense against cryptococcus is associated with cell-mediated immunity, similar to tuberculosis, cryptococcal pleuritis may present with lymphocyte-predominant pleural effusion.…”

Pulmonary cryptococcosis coexisting with central type lung cancer in an immuocompetent patient: a case report and literature review

“…The organism may mainly invades lungs and CNS, but also can invade skin, bones and other parts of the body, which is closely related to the patient's immune status [3,11]. With a declining incidence of AIDS-related cryptococcosis in the highly active antiretroviral therapy (HAART) era and with increasing use of immunosuppresants worldwide, HIVnegative individuals may become the predominant group affected by cryptococcosis [10][11][12][13]. Currently, pulmonary cryptococcosis ranked as the third most common pulmonary fungal infection in China, and previous studies have shown that most of the patients with cryptococcosis of the lung in China were HIV-negative infection patients [14][15][16].…”

Pulmonary cryptococcosis coexisting with adenocarcinoma: a case report and review of the literature

“…Cavitation and halo sign are more frequently observed in immunocompromised patients [ 19 ]. Pleural effusion and lymphadenectasis sometimes occur as well [ 20 ]. Many case reports describe that pulmonary cryptococcosis mimicked lung cancer [ 3 – 5 , 21 ].…”