Postpartum Disseminated Herpes Simplex Virus Type 1 Infection With Hemophagocytic Lymphohistiocytosis and Fulminant Neonatal Herpes Infection

Mette Ratzer Freytag; Sofie Eg Jørgensen; Michelle Mølgaard Thomsen; Ali Al-Mousawi; Alon Schneider Hait; David Olagnier; Jakob T Bay; Marie Helleberg; Trine H Mogensen

doi:10.1093/infdis/jiab290

Postpartum Disseminated Herpes Simplex Virus Type 1 Infection With Hemophagocytic Lymphohistiocytosis and Fulminant Neonatal Herpes Infection

Mette Ratzer Freytag, Sofie Eg Jørgensen, Michelle Mølgaard Thomsen, Ali Al-Mousawi, Alon Schneider Hait, David Olagnier, Jakob T Bay, Marie Helleberg, Trine H Mogensen^*

^*Corresponding author for this work

Research output: Contribution to journal/Conference contribution in journal/Contribution to newspaper › Journal article › Research › peer-review

5 Citations (Scopus)

Abstract

The present study describes a 19-year-old woman with systemic herpes simplex virus (HSV)-1 infection and hemophagocytic lymphohistiocytosis (HLH) postpartum, and a fatal course of neonatal herpesvirus infection. Functional investigation of cells from the mother demonstrated significantly impaired induction of antiviral interferons and cytokines in the context of normal activation of the transcription factors NF-κB and IRF3. Whole-exome sequencing did not reveal any functionally validated genetic variants. We suggest that the functionally impaired antiviral responses, potentially caused by a variant in CASP8 or other variants in noncoding regions of the genome, contributed to the unusually severe disease course observed in two generations.

Original language	English
Journal	The Journal of Infectious Diseases
Volume	225
Issue	1
Pages (from-to)	157-162
Number of pages	6
ISSN	0022-1899
DOIs	https://doi.org/10.1093/infdis/jiab290
Publication status	Published - 1 Jan 2022

Keywords

hemophagocytic lymphohistiocytosis
herpes simplex virus
innate immunity
interferon
neonatal herpes

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10.1093/infdis/jiab290

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Freytag, M. R., Jørgensen, S. E., Thomsen, M. M., Al-Mousawi, A., Hait, A. S., Olagnier, D., Bay, J. T., Helleberg, M., & Mogensen, T. H. (2022). Postpartum Disseminated Herpes Simplex Virus Type 1 Infection With Hemophagocytic Lymphohistiocytosis and Fulminant Neonatal Herpes Infection. The Journal of Infectious Diseases, 225(1), 157-162. https://doi.org/10.1093/infdis/jiab290

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abstract = "The present study describes a 19-year-old woman with systemic herpes simplex virus (HSV)-1 infection and hemophagocytic lymphohistiocytosis (HLH) postpartum, and a fatal course of neonatal herpesvirus infection. Functional investigation of cells from the mother demonstrated significantly impaired induction of antiviral interferons and cytokines in the context of normal activation of the transcription factors NF-κB and IRF3. Whole-exome sequencing did not reveal any functionally validated genetic variants. We suggest that the functionally impaired antiviral responses, potentially caused by a variant in CASP8 or other variants in noncoding regions of the genome, contributed to the unusually severe disease course observed in two generations.",

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author = "Freytag, {Mette Ratzer} and J{\o}rgensen, {Sofie Eg} and Thomsen, {Michelle M{\o}lgaard} and Ali Al-Mousawi and Hait, {Alon Schneider} and David Olagnier and Bay, {Jakob T} and Marie Helleberg and Mogensen, {Trine H}",

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doi = "10.1093/infdis/jiab290",

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Freytag, MR, Jørgensen, SE , Thomsen, MM, Al-Mousawi, A, Hait, AS, Olagnier, D, Bay, JT, Helleberg, M & Mogensen, TH 2022, 'Postpartum Disseminated Herpes Simplex Virus Type 1 Infection With Hemophagocytic Lymphohistiocytosis and Fulminant Neonatal Herpes Infection', The Journal of Infectious Diseases, vol. 225, no. 1, pp. 157-162. https://doi.org/10.1093/infdis/jiab290

Postpartum Disseminated Herpes Simplex Virus Type 1 Infection With Hemophagocytic Lymphohistiocytosis and Fulminant Neonatal Herpes Infection. / Freytag, Mette Ratzer; Jørgensen, Sofie Eg ; Thomsen, Michelle Mølgaard et al.
In: The Journal of Infectious Diseases, Vol. 225, No. 1, 01.01.2022, p. 157-162.

Research output: Contribution to journal/Conference contribution in journal/Contribution to newspaper › Journal article › Research › peer-review

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AU - Freytag, Mette Ratzer

AU - Jørgensen, Sofie Eg

AU - Thomsen, Michelle Mølgaard

AU - Al-Mousawi, Ali

AU - Hait, Alon Schneider

AU - Olagnier, David

AU - Bay, Jakob T

AU - Helleberg, Marie

AU - Mogensen, Trine H

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AB - The present study describes a 19-year-old woman with systemic herpes simplex virus (HSV)-1 infection and hemophagocytic lymphohistiocytosis (HLH) postpartum, and a fatal course of neonatal herpesvirus infection. Functional investigation of cells from the mother demonstrated significantly impaired induction of antiviral interferons and cytokines in the context of normal activation of the transcription factors NF-κB and IRF3. Whole-exome sequencing did not reveal any functionally validated genetic variants. We suggest that the functionally impaired antiviral responses, potentially caused by a variant in CASP8 or other variants in noncoding regions of the genome, contributed to the unusually severe disease course observed in two generations.

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Postpartum Disseminated Herpes Simplex Virus Type 1 Infection With Hemophagocytic Lymphohistiocytosis and Fulminant Neonatal Herpes Infection

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