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Maximova N, Marcuzzi A, Del Rizzo I, et al. Standard treatment-refractory cytomegalovirus encephalitis unmasked by immune reconstitution inflammatory syndrome and successfully treated with virus-specific hyperimmune globulin. Clin Transl Immunology. 2020;9(11):e1201doi: 10.1002/cti2.1201.
Maximova, N., Marcuzzi, A., Del Rizzo, I., Zanon, D., Maestro, A., Barbi, E., & Sala, R. (2020). Standard treatment-refractory cytomegalovirus encephalitis unmasked by immune reconstitution inflammatory syndrome and successfully treated with virus-specific hyperimmune globulin. Clinical & translational immunology, 9(11), e1201. https://doi.org/10.1002/cti2.1201
Maximova, Natalia, et al. "Standard treatment-refractory cytomegalovirus encephalitis unmasked by immune reconstitution inflammatory syndrome and successfully treated with virus-specific hyperimmune globulin." Clinical & translational immunology vol. 9,11 (2020): e1201. doi: https://doi.org/10.1002/cti2.1201
Maximova N, Marcuzzi A, Del Rizzo I, Zanon D, Maestro A, Barbi E, Sala R. Standard treatment-refractory cytomegalovirus encephalitis unmasked by immune reconstitution inflammatory syndrome and successfully treated with virus-specific hyperimmune globulin. Clin Transl Immunology. 2020 Nov 17;9(11):e1201. doi: 10.1002/cti2.1201. eCollection 2020. PMID: 33235734; PMCID: PMC7670254.
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Clin Transl Immunology. 2020 Nov 17;9(11):e1201. doi: 10.1002/cti2.1201. eCollection 2020.

Standard treatment-refractory cytomegalovirus encephalitis unmasked by immune reconstitution inflammatory syndrome and successfully treated with virus-specific hyperimmune globulin.

Clinical & translational immunology

Natalia Maximova, Annalisa Marcuzzi, Irene Del Rizzo, Davide Zanon, Alessandra Maestro, Egidio Barbi, Roberto Sala

Affiliations

  1. Institute for Maternal and Child Health - IRCCS Burlo Garofolo Trieste Italy.
  2. University of Ferrara Ferrara Italy.
  3. University of Trieste Trieste Italy.
  4. University of Parma Parma Italy.

PMID: 33235734 PMCID: PMC7670254 DOI: 10.1002/cti2.1201

Abstract

OBJECTIVES: Cytomegalovirus (CMV)-related encephalitis is a rare but potentially life-threatening complication of CMV infection in immunocompromised patients. The high mortality rate is associated with deficient immune system reconstitution after hematopoietic stem cell transplant (HSCT) and poor bioavailability of antiviral drugs in cerebrospinal fluid (CSF). CMV-related central nervous system (CNS) infection may occur with aspecific symptoms, without evidence of either blood viral load or magnetic resonance imaging (MRI) signs of encephalitis.

METHODS: Here, we describe a 10-year-old girl who underwent an allogeneic HSCT and subsequently developed CMV encephalitis. Because of the absence of CMV antigen in the blood, the diagnosis of encephalitis was proposed only after a delay, following the onset of immune reconstitution inflammatory syndrome (IRIS). Two months of combined dual antiviral therapy with ganciclovir and foscarnet proved ineffective against CMV and caused significant bone marrow and renal toxicity. To avoid further toxicity, the girl was given daily treatment with CMV-hyperimmune globulins alone.

RESULTS: After three weeks, the CSF viral load dropped significantly and was undetectable within three more weeks. In the meantime, the renal impairment resolved, and there was a complete bone marrow recovery.

CONCLUSION: We suggest that this patient succeeded in achieving CMV CSF clearance with high dose of CMV-hyperimmune globulin, given alone, because of the ability of immunoglobulins to penetrate the blood-brain barrier (BBB).

© 2020 The Authors. Clinical & Translational Immunology published by John Wiley & Sons Australia, Ltd on behalf of Australian and New Zealand Society for Immunology, Inc.

Keywords: CMV‐hyperimmune globulin; IL‐6; cerebrospinal fluid CMV antibodies; cytomegalovirus (CMV)‐related encephalitis; immune reconstitution inflammatory syndrome (IRIS)

Conflict of interest statement

The authors have declared that no conflict of interest exists.

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