Brainstem encephalitis caused by Coxsackie A16 virus in a rituximab-immunosuppressed patient

Smail, Ruaridh Cameron; O’Neill, John; Andresen, David

doi:10.1136/bcr-2019-230177

Cited by 6 publications

(5 citation statements)

References 18 publications

(18 reference statements)

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Section: Resultsmentioning

confidence: 99%

“…According to numerous reports, encephalitis has emerged as the most prevalent sequela of enterovirus infection and is typified by neurological symptoms such as vomiting, headache, and convulsions. [22][23][24] F I G U R E 5 Viral tissue tropism in coxsackievirus B1 (CVB1)-infected rhesus macaques. (A) Pathological damage and viral antigen expression in the visceral tissues of CVB1-infected rhesus macaques.…”

Section: Suspected Viral Meningitis-like Changes In Cvb1-infected Rhe...mentioning

confidence: 99%

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A 3‐month‐old neonatal rhesus macaque HFMD model caused by coxsackievirus B1 infection and viral tissue tropism

Suqin,

Yongjie,

Wei

et al. 2024

Journal of Medical Virology

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Coxsackievirus B1 (CVB1), an enterovirus with multiple clinical presentations, has been associated with potential long‐term consequences, including hand, foot, and mouth disease (HFMD), in some patients. However, the related animal models, transmission dynamics, and long‐term tissue tropism of CVB1 have not been systematically characterized. In this study, we established a model of CVB1 respiratory infection in rhesus macaques and evaluated the clinical symptoms, viral load, and immune levels during the acute phase (0–14 days) and long‐term recovery phase (15–30 days). We also investigated the distribution, viral clearance, and pathology during the long‐term recovery period using 35 postmortem rhesus macaque tissue samples collected at 30 days postinfection (d.p.i.). The results showed that the infected rhesus macaques were susceptible to CVB1 and exhibited HFMD symptoms, viral clearance, altered cytokine levels, and the presence of neutralizing antibodies. Autopsy revealed positive viral loads in the heart, spleen, pancreas, soft palate, and olfactory bulb tissues. HE staining demonstrated pathological damage to the liver, spleen, lung, soft palate, and tracheal epithelium. At 30 d.p.i., viral antigens were detected in visceral, immune, respiratory, and muscle tissues but not in intestinal or neural tissues. Brain tissue examination revealed viral meningitis‐like changes, and CVB1 antigen expression was detected in occipital, pontine, cerebellar, and spinal cord tissues at 30 d.p.i. This study provides the first insights into CVB1 pathogenesis in a nonhuman primate model of HFMD and confirms that CVB1 exhibits tissue tropism following long‐term infection.

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Section: Resultsmentioning

confidence: 99%

Section: Suspected Viral Meningitis-like Changes In Cvb1-infected Rhe...mentioning

confidence: 99%

A 3‐month‐old neonatal rhesus macaque HFMD model caused by coxsackievirus B1 infection and viral tissue tropism

Suqin,

Yongjie,

Wei

et al. 2024

Journal of Medical Virology

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“…The inhibition of the humoral immune response puts the patient at increased risk for viral infections, as evidenced by the black box warnings for reactivation of hepatitis B virus and infection with the John Cunningham (JC) virus [4]. There have been few reported cases of enteroviral meningoencephalitis associated with rituximab, the majority of which occurred in the treatment of hematologic disease [7][8][9][10][11]. A review of the literature indicates that our patient is the first reported case to occur in the treatment of rheumatoid arthritis, the only inflammatory arthritis for which rituximab has an approved indication.…”

Section: Discussionmentioning

confidence: 99%

Enteroviral Meningoencephalitis as a Complication of Rituximab Therapy for Rheumatoid Arthritis

Cook¹,

Ford²,

Lindholm³

et al. 2021

Cureus

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Enteroviral meningoencephalitis is a rare complication of rituximab therapy that has been described in the treatment of hematologic malignancies. We report the first case of enteroviral meningoencephalitis in a patient receiving rituximab for the treatment of rheumatoid arthritis.A 37-year-old female treated with rituximab for severe rheumatoid arthritis presented with fever, headache, confusion, and tremor. Magnetic resonance imaging (MRI) of the brain was unrevealing. Cerebrospinal fluid showed a lymphocytic pleocytosis and multiplex polymerase chain reaction (PCR) was positive for enterovirus. She was treated with intravenous immunoglobulin (IVIG) for five days and had significant improvement in symptoms.Rituximab is an anti-CD20 monoclonal antibody that induces B-cell depletion and possible hypogammaglobulinemia, putting patients at increased risk for viral infections. We present this case to highlight that patients on rituximab, regardless of the indication for use, are susceptible to severe complications of otherwise typically self-limited viral infections. Furthermore, we describe the potential use of IVIG in treating these patients.

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“…There is currently an evolving recognition of susceptibility to enterovirus among people treated with rituximab, a B cell depleting agent widely used for lymphoma, multiple sclerosis, and systemic lupus erythematosus. [41][42][43][44][45][46][47][48][49][50][51][52][53][54] The population treated with rituximab has generally had a more acute presentation of disseminated disease or meningoencephalitis compared to the slow onset seen in XLA;…”

Section: Diagnosismentioning

confidence: 99%

“…There is currently an evolving recognition of susceptibility to enterovirus among people treated with rituximab, a B cell depleting agent widely used for lymphoma, multiple sclerosis, and systemic lupus erythematosus 41–54 . The population treated with rituximab has generally had a more acute presentation of disseminated disease or meningoencephalitis compared to the slow onset seen in XLA; however, late relapses and late relapses in different organs have been observed suggesting incomplete clearance even in the setting of clinical improvement.…”

Section: Viruses Causing a Distinct Phenotype In Patients With Ieimentioning

confidence: 99%

Rubella virus chronic inflammatory disease and other unusual viral phenotypes in inborn errors of immunity

Kilich,

Perelygina,

Sullivan

2023

Immunological Reviews

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SummaryInfectious susceptibility is a component of many inborn errors of immunity. Nevertheless, antibiotic use is often used as a surrogate in history taking for infectious susceptibility, thereby disadvantaging patients who present with viral infections as their phenotype. Further complicating clinical evaluations are unusual manifestations of viral infections which may be less familiar that the typical respiratory viral infections. This review covers several unusual viral phenotypes arising in patients with inborn errors of immunity and other settings of immune compromise. In some cases, chronic infections lead to oncogenesis or tumor‐like growths and the conditions and mechanisms of viral‐induced oncogenesis will be described. This review covers enterovirus, rubella, measles, papillomavirus, and parvovirus B19. It does not cover EBV and hemophagocytic lymphohistiocytosis nor lymphomagenesis related to EBV. EBV susceptibility has been recently reviewed. Our goal is to increase awareness of the unusual manifestations of viral infections in patients with IEI and to describe treatment modalities utilized in this setting. Coincidentally, each of the discussed viral infections can have a cutaneous component and figures will serve as a reminder of the physical features of these viruses. Given the high morbidity and mortality, early recognition can only improve outcomes.

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Brainstem encephalitis caused by Coxsackie A16 virus in a rituximab-immunosuppressed patient

Cited by 6 publications

References 18 publications

A 3‐month‐old neonatal rhesus macaque HFMD model caused by coxsackievirus B1 infection and viral tissue tropism

A 3‐month‐old neonatal rhesus macaque HFMD model caused by coxsackievirus B1 infection and viral tissue tropism

Enteroviral Meningoencephalitis as a Complication of Rituximab Therapy for Rheumatoid Arthritis

Rubella virus chronic inflammatory disease and other unusual viral phenotypes in inborn errors of immunity

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