Top

Molecular and Cellular Pediatrics

Published in:

Open Access 01-12-2020 | Herpes Virus | Case Report

A case of recurrent herpes simplex 2 encephalitis, VZV reactivations, and dominant partial interferon-gamma-receptor-1 deficiency supports relevance of IFNgamma for antiviral defense in humans

Authors: Julia Körholz, Nicole Richter, Jochen Schäfer, Catharina Schuetz, Joachim Roesler

Published in: Molecular and Cellular Pediatrics | Issue 1/2020

Abstract

Background

Unlike infections with mycobacteria, reports of unusual viral infections in interferon-gamma-receptor (IFNγR) deficient patients are scarce. Therefore, discussion about increased susceptibility to viral infections in these patients is ongoing.

Case presentation

We describe a 51-year-old male with dominant partial interferon-gamma-receptor-1 (IFNγR1)-deficiency and recurrent Herpes simplex 2 meningoencephalitis as well as other viral reactivations since childhood.

Conclusions

This case further confirms an enhanced risk for viral disease in IFNγR-deficient patients and a role of interferon gamma for human antiviral defense.

Aurelius E, Johansson B, Sköldenberg B, Forsgren M (1993) Encephalitis in immunocompetent patients due to herpes simplex virus type 1 or 2 as determined by type-specific polymerase chain reaction and antibody assays of cerebrospinal fluid. J Med Virol 39:179–186. https://doi.org/10.1002/jmv.1890390302CrossRefPubMed

Bax HI, Freeman AF, Anderson VL, Vesterhus P, Laerum D, Pittaluga S, Wilson WH, Holland SM (2013) B-cell lymphoma in a patient with complete interferon gamma receptor 1 deficiency. J Clin Immunol 33:1062–1066. https://doi.org/10.1007/s10875-013-9907-0CrossRefPubMedPubMedCentral

Camcioglu Y, Picard C, Lacoste V, Dupuis S, Akçakaya N, Çokura H, Kaner G, Demirkesen C, Plancoulaine S, Emile J-F, Gessain A, Casanova J-L (2004) HHV-8–associated Kaposi sarcoma in a child with IFNγR1 deficiency. J Pediatr 144:519–523. https://doi.org/10.1016/j.jpeds.2003.11.012CrossRefPubMed

Casanova JL, Ochs H (1999) Interferon-gamma receptor deficiency: an expanding clinical phenotype? J Pediatr 135:543–545. https://doi.org/10.1016/s0022-3476(99)70050-8CrossRefPubMed

Dorman SE, Picard C, Lammas D, Heyne K, van Dissel JT, Baretto R, Rosenzweig SD, Newport M, Levin M, Roesler J, Kumararatne D, Casanova J-L, Holland SM (2004) Clinical features of dominant and recessive interferon gamma receptor 1 deficiencies. Lancet Lond Engl 364:2113–2121. https://doi.org/10.1016/S0140-6736(04)17552-1CrossRef

Dorman SE, Uzel G, Roesler J, Bradley JS, Bastian J, Billman G, King S, Filie A, Schermerhorn J, Holland SM (1999) Viral infections in interferon-gamma receptor deficiency. J Pediatr 135:640–643. https://doi.org/10.1016/s0022-3476(99)70064-8CrossRefPubMedPubMedCentral

Dupuis S, Döffinger R, Picard C, Fieschi C, Altare F, Jouanguy E, Abel L, Casanova JL (2000) Human interferon-gamma-mediated immunity is a genetically controlled continuous trait that determines the outcome of mycobacterial invasion. Immunol Rev 178:129–137. https://doi.org/10.1034/j.1600-065x.2000.17810.xCrossRefPubMed

Dyer J, Greenfield M (2016) Recurrent varicella in an immunocompetent woman. Cutis 97:65–69PubMed

Fensterl V, Sen GC (2009) Interferons and viral infections. BioFactors Oxf Engl 35:14–20. https://doi.org/10.1002/biof.6CrossRef

10.

Jouanguy E, Béziat V, Mogensen TH, Casanova J-L, Tangye SG, Zhang S-Y (2020) Human inborn errors of immunity to herpes viruses. Curr Opin Immunol 62:106–122. https://doi.org/10.1016/j.coi.2020.01.004CrossRefPubMed

11.

Jouanguy E, Lamhamedi-Cherradi S, Lammas D, Dorman SE, Fondanèche MC, Dupuis S, Döffinger R, Altare F, Girdlestone J, Emile JF, Ducoulombier H, Edgar D, Clarke J, Oxelius VA, Brai M, Novelli V, Heyne K, Fischer A, Holland SM, Kumararatne DS, Schreiber RD, Casanova JL (1999) A human IFNGR1 small deletion hotspot associated with dominant susceptibility to mycobacterial infection. Nat Genet 21:370–378. https://doi.org/10.1038/7701CrossRefPubMed

12.

Kang S, Brown HM, Hwang S (2018) Direct Antiviral Mechanisms of Interferon-Gamma. Immune Netw 18. https://doi.org/10.4110/in.2018.18.e33

13.

Kennedy PGE (2005) Viral encephalitis. J Neurol 252:268–272. https://doi.org/10.1007/s00415-005-0770-7CrossRefPubMed

14.

Looker KJ, Magaret AS, Turner KME, Vickerman P, Gottlieb SL, Newman LM (2015) Global estimates of prevalent and incident herpes simplex virus type 2 Infections in 2012. PLoS ONE 10:e114989. https://doi.org/10.1371/journal.pone.0114989CrossRefPubMedPubMedCentral

15.

Mateen FJ, Miller SA, Aksamit AJ (2014) Herpes Simplex Virus 2 Encephalitis in Adults. Mayo Clin Proc 89:274–275. https://doi.org/10.1016/j.mayocp.2013.12.003CrossRefPubMed

16.

Roesler J, Hedrich C, Laass MW, Heyne K, Rösen-Wolff A (2011) Meningoencephalitis Caused By Varicella-Zoster Virus Reactivation In A Child With Dominant Partial Interferon-Gamma Receptor-1 Deficiency. Pediatr Infect Dis J 30:265–266. https://doi.org/10.1097/INF.0b013e3181f6f78aCrossRefPubMed

17.

Roizmann B, Desrosiers RC, Fleckenstein B, Lopez C, Minson AC, Studdert MJ (1992) The family Herpesviridae: an update. The Herpesvirus Study Group of the International Committee on Taxonomy of Viruses. Arch Virol 123:425–449. https://doi.org/10.1007/bf01317276CrossRefPubMed

18.

Rosain J, Kong X, Martinez-Barricarte R, Oleaga-Quintas C, Ramirez-Alejo N, Markle J, Okada S, Boisson-Dupuis S, Casanova J, Bustamante J (2019) Mendelian susceptibility to mycobacterial disease: 2014–2018 update. Immunol Cell Biol 97:360–367. https://doi.org/10.1111/imcb.12210CrossRefPubMed

19.

Spiegel R, Miron D, Lumelsky D, Horovitz Y (2010) Severe meningoencephalitis due to late reactivation of Varicella-Zoster virus in an immunocompetent child. J Child Neurol 25:87–90. https://doi.org/10.1177/0883073809336296CrossRefPubMed

20.

Wright WF, Palisoc K, Baghli S (2019) Mollaret meningitis. J Neurol Sci 396:148–149. https://doi.org/10.1016/j.jns.2018.11.018CrossRefPubMed

Title: A case of recurrent herpes simplex 2 encephalitis, VZV reactivations, and dominant partial interferon-gamma-receptor-1 deficiency supports relevance of IFNgamma for antiviral defense in humans
Authors: Julia Körholz
Nicole Richter
Jochen Schäfer
Catharina Schuetz
Joachim Roesler
Publication date: 01-12-2020
Publisher: Springer Berlin Heidelberg
Keywords: Herpes Virus
Human Alphaherpesvirus 3
Meningoencephalitis
Aciclovir
Encephalitis
Interferon
Published in: Molecular and Cellular Pediatrics / Issue 1/2020
Electronic ISSN: 2194-7791
DOI: https://doi.org/10.1186/s40348-020-00106-4

Keynote webinar | Spotlight on sleep in brain health

Springer Medicine

A case of recurrent herpes simplex 2 encephalitis, VZV reactivations, and dominant partial interferon-gamma-receptor-1 deficiency supports relevance of IFNgamma for antiviral defense in humans

Abstract

Background

Case presentation

Conclusions

Keynote webinar | Spotlight on sleep in brain health

Springer Medicine

Abstract

Background

Case presentation

Conclusions

Please log in to get access to this content

Other articles of this Issue 1/2020

Nup133 and ERα mediate the differential effects of hyperoxia-induced damage in male and female OPCs

Osteogenesis imperfecta—pathophysiology and therapeutic options

A role for the alpha-8 integrin chain (itga8) in glomerular homeostasis of the kidney

Impact of early-life diet on long-term renal health

Shaping of the nephron – a complex, vulnerable, and poorly explored backdrop for noxae impairing nephrogenesis in the fetal human kidney

Genotype-phenotype correlations in children and adolescents with nonclassical congenital adrenal hyperplasia due to 21-hydroxylase deficiency