Pseudocystic form of neurocryptococcosis in pregnancy: case report

Abstract

-We report a case of neurocryptococcosis which is unique in the literature because the patient had a pseudocystic form of the disease during pregnancy and without any evidence of AIDS. The clinical picture was that of intracranial hypertension and the epidemiological background was highly suggestive of cysticercosis. CT showed multiple round hypodense lesions in the basal ganglia and cerebellum, without contrast enhancement. Since a scolex was not visible, the diagnosis of neurocysticercosis was considered probable. CSF examination was not performed in view of its high risk. The patient had progressive downhill course. Autopsy disclosed multiple gelatinous pseudocysts in the cerebral and cerebellar gray matter, containing abundant Cryptococcus neoformans. Meningeal involvement was minimal. The child was delivered by caesarean section and was free of infection, but died later of hyaline membrane disease. The neuroimaging appearances of this rare instance of the pseudocystic form of neurocryptococcosis mimicked closely neurocysticercosis and only postmortem examination allowed correct diagnosis. The pseudocystic form has so far only been reported in AIDS.KEY WORDS: neurocryptococcosis, neurocysticercosis, pregnancy, pseudocysts.Neurocriptococose na gravidez: forma pseudocística. Relato de Caso RESUMO -Relatamos o primeiro caso da forma pseudocística da neurocriptococose na gravidez, em paciente sem evidência de AIDS. O quadro clínico era de hipertensão intracraniana que se manifestou no segundo trimestre da gravidez. A tomografia computadorizada do crânio evidenciava múltiplas lesões hipodensas, císticas, não impregnadas por contraste, localizadas na substância cinzenta cerebral e cerebelar. Dados epidemiológicos, clínicos e tomográficos apontavam para o diagnóstico provável de neurocisticercose. A autópsia, porém, revelou múltiplos pseudocistos de material gelatinoso com Cryptococcus neoformans em abundância. Havia mínimo comprometimento meníngeo. A revisão da literatura mostrou que a forma pseudocística da neurocriptococose só foi até o momento relatada em pacientes com AIDS. Os casos de neurocriptococose na gravidez até hoje publicados foram todos da forma meningítica. PALAVRAS -CHAVE: neurocriptococose, neurocisticercose, gravidez, pseudocistos.Neurocryptococcosis is rare in pregnancy. Since the first report in 1936, 23 further cases not associated with acquired immunodeficiency syndrome (AIDS) have been recorded, all of which had the meningitic form [1][2][3][4] . The following case is unique in that the main feature was voluminous intraparenchymatous pseudocysts filled with parasites and leading to severe intracranial hypertension.

Puerperal brain cryptococcoma in an HIV-negative woman successfully treated with fluconazole: a case report

“…Although several cases of cryptococcal meningitis in immunocompetent pregnant women have been described 5,6 , to our knowledge, only two cases of cryptococcoma during pregnancy have been previously reported 7,8 . The presence of antecedent symptoms six years before this illness suggests the possibility of a prior infection that was spontaneously controlled until recrudescence and growth of the cryptococcoma during the relative immunosuppression of pregnancy, in which cell-mediated immunity is particularly affected.…”

Cerebellar cryptococcoma simulating metastatic neoplasm

“…Kanaly et al 7 also carried out surgical treatment of a patient with cerebellar pseudotumoral cryptococcosis masquerading as a metastatic brain tumor. On the other hand, Nucci et al 2 reported in Brazil a pseudocystic form of cryptococcosis affecting brainstem and cerebellum and ultimately causing the death of a pregnant HIV-negative patient.…”

“…Other rare forms of central nervous system cryptococcal infections are their solid-granulomatous appearance and abscesses, both being commonly named neurocryptococcomas, because of the mass effect they cause in surrounding tissue and their "tumoral aspect", many times simulating a brain tumor. Alternatively, Nucci et al 2 described the unique report of a pseudocystic form of neurocryptococcosis. These medical conditions were classically described in immunocompromised hosts, mainly in HIV/AIDS infected patients.…”

Cerebral Cryptococcomas: A Systematic Scoping Review of Available Evidence to Facilitate Diagnosis and Treatment

“…Though 40% (n = 19) of the patients did not report a past medical, surgical, or social history, of the 60% (n = 28) who did, 36% (n = 10) were HIV-infected, 21% (n = 6) had hypertension, and 18% (n = 5) had diabetes mellitus. One patient was in her second trimester of pregnancy with no other significant history [39], one patient had a history of polycythemia vera and monoclonal gammopathy of unknown significance [24], whereas two patients reported close contact with pigeons [41,46]. Of those with HIV, absolute CD4 count ranged from 0 to 157 cells/µL [9,28,36,42,43,50], whereas only three patients were noted to be on ART [36,42].…”