Human pegivirus identified in severe myelitis and optic neuritis in immunocompromised patients: A pathogenic role for a forgotten virus?

Elsevier

Available online 24 October 2022

Revue NeurologiqueAbstract

The role of Human pegivirus (HPgV) in patients with encephalitis has been recently questioned. We present cases of 4 patients with similar clinical, biological, and radiological characteristics, including a past history of transplantation with long-term immunosuppression and a progressive course of severe and predominantly myelitis, associated in 3 cases with optic neuropathy causing blindness. Extensive workup was negative but analysis of the CSF by use of pan-microorganism DNA- and RNA-based shotgun metagenomics was positive for HPgV. This case series further supports the hypothesis of HPgV CNS infection and highlights the utility of metagenomic next-generation sequencing of CSF in immunocompromised patients.

Introduction

Human pegivirus (HPgV), first identified in 1996, was formerly called GB virus C/hepatitis G virus since it was mistakenly associated with viral hepatitis. This enveloped and positive-strand RNA virus of the Flaviviridae family is transmitted by exposure to infected blood, sexual exposure or by maternal–foetal transmission. HPgV has been associated with a beneficial effect in human immunodeficiency virus (HIV) infection, and studies found prolonged survival in HIV-infected individuals co-infected with HPgV compared with those without HPgV viraemia. The worldwide prevalence of HPgV viremia in blood donors is approximately 1 to 5% [1]. Notably, this prevalence increases to 36% in transplant patients [2]. HPgV is lymphotropic and replicates in the spleen and bone marrow [3]. Although HPgV has been considered nonpathogenic in humans, its role in patients with encephalitis has been recently questionned [4], [5], [6], [7]. Here, we describe 4 transplant patients with HPgV identified in CNS.

Section snippetsObservation

Clinical features are in Table 1; detailed investigations are described in Supplementary table 1.

Discussion

Our observations present several arguments linking HPgV to CNS involvement. First, the patients presented striking clinical and radiological similarities, including a progressive, often intermittent course of severe extensive myelitis, and in 3 cases a bilateral ON causing blindness, mimicking neuromyelitis optica. Second, each of the 4 individuals had a history of organ or hematopoietic stem cell transplantation and received long-term immunosuppressive treatments, leading to increased risk of

Conclusion

This case series further supports the hypothesis of HPgV CNS infection and highlights the utility of metagenomic next-generation sequencing of CSF in immunocompromised patients. It remains essential to identify additional cases in order to unequivocally prove the pathogenicity of HPgV.

Disclosure of interest

The authors declare that they have no competing interest.

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