Chronic meningoencephalitis due to enterovirus A71 complicating rituximab therapy

Abstract

Background: Rituximab and other anti-CD20 therapies are increasingly used in the treatment of autoimmune and hematologic disorders. These treatments are associated with persistent immune impairment, potentially leading to severe infections. We describe a resolutive case of proven chronic enterovirus A71 (EV A71) meningoencephalitis complicating rituximab maintenance therapy for non-Hodgkin lymphoma.

Methods: This article combines an original case report and a literature review of cases of enteroviral meningoencephalitis complicating rituximab treatment.

Results: A 38-year-old man was treated with rituximab and chemotherapy for a mantle cell lymphoma. During maintenance treatment with rituximab, he developed a "hand-foot-mouth disease", and one month later severe neurological deterioration including quadriparesis and major neurocognitive disorder leading to a diagnosis of chronic enteroviral meningoencephalitis. A treatment associating monthly intravenous immunoglobulins (IVIg) and fluoxetine was initiated two months after neurological symptoms onset, resulting in dramatic clinical improvement within six months. A brief literature review shows that a treatment with high-dose IVIg often results in clinical improvement. Fluoxetine was added in recent reports based on in vitro evidence of anti-viral activity against enteroviruses.

Discussion: Enteroviral infection should be evoked in patients treated with rituximab presenting with an encephalitic symptomatology, and restoring humoral immunity with high-dose IVIg might improve their condition.

Keywords: Anti-CD20; Complication; Dementia; Enteroviral meningoencephalitis; Enterovirus A71; Hypogammaglobulinemia; Non-Hodgkin's lymphoma; Rituximab.