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Herpes simplex encephalitis, susceptibility to, 1(IIAE1)

MedGen UID:
413772
Concept ID:
C2750180
Finding
Synonyms: ENCEPHALOPATHY, ACUTE, INFECTION-INDUCED (HERPES-SPECIFIC), SUSCEPTIBILITY TO, 1; Herpes simplex encephalitis 1; IIAE1
 
Gene (location): UNC93B1 (11q13.2)
 
Monarch Initiative: MONDO:0024563
OMIM®: 610551

Definition

Herpes simplex virus (HSV)-1 is most often associated with infection of the oral mucosa. Primary infection is most commonly asymptomatic, but it may lead to symptoms usually involving the mucosa and skin. Following replication at the infection site, HSV-1 enters the epithelial endings of sensory neurons and travels up the trigeminal cranial nerves to the trigeminal ganglia, where latent infection is established. Reactivation of HSV-1, usually in the form of herpes labialis (cold sores), may occur in 20 to 40% of the population. HSV-1 seroprevalence is high, with over 85% of adults between the ages of 20 and 40 years infected. HSV-1 rarely infects the central nervous system (CNS), resulting in herpes simplex encephalitis (HSE), with an incidence of 2 to 4 per 1,000,000 people per year. In HSE, HSV-1 invades and replicates in neurons and glial cells, where focal necrotizing infections occur, primarily affecting the temporal and subfrontal regions of the brain. Untreated, HSE is fatal in at least 70% of cases, although the mortality and morbidity have been drastically reduced with antiviral therapy. Approximately one-third of all HSE cases are due to primary infections, and 30% of all HSE cases occur in children under the age of 20 years. Among children, HSE peaks between 3 months and 3 years of age, coinciding with the time of primary infection. In a subset of children, HSE results from a series of monogenic primary immunodeficiencies that impair UNC93B1- and TLR3 (603029)-dependent production of IFNA (147660)/IFNB (147640) and IFNG (147570) in the CNS (summary by Sancho-Shimizu et al., 2007). Genetic Heterogeneity of Susceptibility to Acute Infection-Induced Encephalopathy, including Herpes Simplex Encephalitis (HSE) For other forms of susceptibility to acute infection-induced encephalopathy, see herpes-specific IIAE2 (613002), caused by mutation in the TLR3 gene (603029) on chromosome 4q35; IIAE3 (608033), caused by mutation in the RANBP2 gene (601181) on chromosome 2q12; IIAE4 (614212), caused by mutation in the CPT2 gene (600650) on chromosome 1p32; herpes-specific IIAE5 (614849), caused by mutation in the TRAF3 gene (601896) on chromosome 14q32; herpes-specific IIAE6 (614850), caused by mutation in the TICAM1 gene (607601) on chromosome 19p13; herpes-specific IIAE7 (616532), caused by mutation in the IRF3 gene (603734) on chromosome 19q13; herpes-specific IIAE8 (617900), caused by mutation in the TBK1 gene (604834) on chromosome 12q14; IIAE9 (618426), caused by mutation in the NUP214 gene (114350) on chromosome 9q34; herpes-specific IIAE10 (619396), caused by mutation in the SNORA31 (619378) on chromosome 13q14; IIAE11 (619441), caused by mutation in the DBR1 gene (607024) on chromosome 3q22; and IIAE12 (620461), caused by mutation in the RNH1 gene (173320) on chromosome 11p15. [from OMIM]

Clinical features

From HPO
Herpes simplex encephalitis
MedGen UID:
75794
Concept ID:
C0276226
Disease or Syndrome
Infection of the brain parenchyma with herpes simplex virus, resulting in inflammation of the brain parenchyma with neurologic dysfunction.
Recurrent herpes
MedGen UID:
870750
Concept ID:
C4025207
Finding
Increased susceptibility to herpesvirus, as manifested by recurrent episodes of herpesvirus.

Professional guidelines

PubMed

Chilukuri S, Rosen T
Dermatol Clin 2003 Apr;21(2):311-20. doi: 10.1016/s0733-8635(02)00093-1. PMID: 12757254
Lin WR, Wozniak MA, Esiri MM, Klenerman P, Itzhaki RF
J Neurol Neurosurg Psychiatry 2001 Jan;70(1):117-9. doi: 10.1136/jnnp.70.1.117. PMID: 11118260Free PMC Article

Recent clinical studies

Etiology

Gupta S, Dutta A, Chakraborty U, Kumar R, Das D, Ray BK
QJM 2022 Apr 20;115(4):222-227. doi: 10.1093/qjmed/hcac060. PMID: 35199176Free PMC Article
Piret J, Boivin G
Clin Microbiol Rev 2020 Mar 18;33(2) Epub 2020 Feb 12 doi: 10.1128/CMR.00105-19. PMID: 32051176Free PMC Article
Giraldo D, Wilcox DR, Longnecker R
DNA Cell Biol 2017 May;36(5):329-334. Epub 2017 Mar 9 doi: 10.1089/dna.2017.3668. PMID: 28278385Free PMC Article
Lim HK, Seppänen M, Hautala T, Ciancanelli MJ, Itan Y, Lafaille FG, Dell W, Lorenzo L, Byun M, Pauwels E, Rönnelid Y, Cai X, Boucherit S, Jouanguy E, Paetau A, Lebon P, Rozenberg F, Tardieu M, Abel L, Yildiran A, Vergison A, Roivainen R, Etzioni A, Tienari PJ, Casanova JL, Zhang SY
Neurology 2014 Nov 18;83(21):1888-97. Epub 2014 Oct 22 doi: 10.1212/WNL.0000000000000999. PMID: 25339207Free PMC Article
Zhang SY, Abel L, Casanova JL
Handb Clin Neurol 2013;112:1091-7. doi: 10.1016/B978-0-444-52910-7.00027-1. PMID: 23622315

Diagnosis

Dai Y, Idorn M, Serrero MC, Pan X, Thomsen EA, Narita R, Maimaitili M, Qian X, Iversen MB, Reinert LS, Flygaard RK, Chen M, Ding X, Zhang BC, Carter-Timofte ME, Lu Q, Jiang Z, Zhong Y, Zhang S, Da L, Zhu J, Denham M, Nissen P, Mogensen TH, Mikkelsen JG, Zhang SY, Casanova JL, Cai Y, Paludan SR
Nature 2024 Aug;632(8024):383-389. Epub 2024 Jul 24 doi: 10.1038/s41586-024-07670-z. PMID: 39048823
Gupta S, Dutta A, Chakraborty U, Kumar R, Das D, Ray BK
QJM 2022 Apr 20;115(4):222-227. doi: 10.1093/qjmed/hcac060. PMID: 35199176Free PMC Article
Piret J, Boivin G
Clin Microbiol Rev 2020 Mar 18;33(2) Epub 2020 Feb 12 doi: 10.1128/CMR.00105-19. PMID: 32051176Free PMC Article
Pérez de Diego R, Mulvey C, Casanova JL, Godovac-Zimmermann J
Expert Rev Proteomics 2014 Feb;11(1):21-9. Epub 2013 Dec 18 doi: 10.1586/14789450.2014.864954. PMID: 24351021Free PMC Article
Sancho-Shimizu V, Zhang SY, Abel L, Tardieu M, Rozenberg F, Jouanguy E, Casanova JL
Curr Opin Allergy Clin Immunol 2007 Dec;7(6):495-505. doi: 10.1097/ACI.0b013e3282f151d2. PMID: 17989525

Therapy

Gupta S, Dutta A, Chakraborty U, Kumar R, Das D, Ray BK
QJM 2022 Apr 20;115(4):222-227. doi: 10.1093/qjmed/hcac060. PMID: 35199176Free PMC Article
Piret J, Boivin G
Clin Microbiol Rev 2020 Mar 18;33(2) Epub 2020 Feb 12 doi: 10.1128/CMR.00105-19. PMID: 32051176Free PMC Article
Giraldo D, Wilcox DR, Longnecker R
DNA Cell Biol 2017 May;36(5):329-334. Epub 2017 Mar 9 doi: 10.1089/dna.2017.3668. PMID: 28278385Free PMC Article
Abel L, Plancoulaine S, Jouanguy E, Zhang SY, Mahfoufi N, Nicolas N, Sancho-Shimizu V, Alcaïs A, Guo Y, Cardon A, Boucherit S, Obach D, Clozel T, Lorenzo L, Amsallem D, Berquin P, Blanc T, Bost-Bru C, Chabrier S, Chabrol B, Cheuret E, Dulac O, Evrard P, Héron B, Lazaro L, Mancini J, Pedespan JM, Rivier F, Vallée L, Lebon P, Rozenberg F, Casanova JL, Tardieu M
J Pediatr 2010 Oct;157(4):623-9, 629.e1. Epub 2010 May 31 doi: 10.1016/j.jpeds.2010.04.020. PMID: 20553844
Chilukuri S, Rosen T
Dermatol Clin 2003 Apr;21(2):311-20. doi: 10.1016/s0733-8635(02)00093-1. PMID: 12757254

Prognosis

Lafaille FG, Harschnitz O, Lee YS, Zhang P, Hasek ML, Kerner G, Itan Y, Ewaleifoh O, Rapaport F, Carlile TM, Carter-Timofte ME, Paquet D, Dobbs K, Zimmer B, Gao D, Rojas-Duran MF, Kwart D, Rattina V, Ciancanelli MJ, McAlpine JL, Lorenzo L, Boucherit S, Rozenberg F, Halwani R, Henry B, Amenzoui N, Alsum Z, Marques L, Church JA, Al-Muhsen S, Tardieu M, Bousfiha AA, Paludan SR, Mogensen TH, Quintana-Murci L, Tessier-Lavigne M, Smith GA, Notarangelo LD, Studer L, Gilbert W, Abel L, Casanova JL, Zhang SY
Nat Med 2019 Dec;25(12):1873-1884. Epub 2019 Dec 5 doi: 10.1038/s41591-019-0672-3. PMID: 31806906Free PMC Article
Sarquiz-Martínez B, González-Bonilla CR, Santacruz-Tinoco CE, Muñoz-Medina JE, Pardavé-Alejandre HD, Barbosa-Cabrera E, Ramírez-González JE, Díaz-Quiñonez JA
Intervirology 2017;60(3):118-124. Epub 2017 Sep 28 doi: 10.1159/000480508. PMID: 28954265
Pérez de Diego R, Mulvey C, Casanova JL, Godovac-Zimmermann J
Expert Rev Proteomics 2014 Feb;11(1):21-9. Epub 2013 Dec 18 doi: 10.1586/14789450.2014.864954. PMID: 24351021Free PMC Article
Zhang SY, Abel L, Casanova JL
Handb Clin Neurol 2013;112:1091-7. doi: 10.1016/B978-0-444-52910-7.00027-1. PMID: 23622315
Jouanguy E, Zhang SY, Chapgier A, Sancho-Shimizu V, Puel A, Picard C, Boisson-Dupuis S, Abel L, Casanova JL
Biochimie 2007 Jun-Jul;89(6-7):878-83. Epub 2007 May 8 doi: 10.1016/j.biochi.2007.04.016. PMID: 17561326

Clinical prediction guides

Dai Y, Idorn M, Serrero MC, Pan X, Thomsen EA, Narita R, Maimaitili M, Qian X, Iversen MB, Reinert LS, Flygaard RK, Chen M, Ding X, Zhang BC, Carter-Timofte ME, Lu Q, Jiang Z, Zhong Y, Zhang S, Da L, Zhu J, Denham M, Nissen P, Mogensen TH, Mikkelsen JG, Zhang SY, Casanova JL, Cai Y, Paludan SR
Nature 2024 Aug;632(8024):383-389. Epub 2024 Jul 24 doi: 10.1038/s41586-024-07670-z. PMID: 39048823
Bibert S, Quinodoz M, Perriot S, Krebs FS, Jan M, Malta RC, Collinet E, Canales M, Mathias A, Faignart N, Roulet-Perez E, Meylan P, Brouillet R, Opota O, Lozano-Calderon L, Fellmann F, Guex N, Zoete V, Asner S, Rivolta C, Du Pasquier R, Bochud PY
Nat Commun 2024 May 10;15(1):3969. doi: 10.1038/s41467-024-48287-0. PMID: 38730242Free PMC Article
Gupta S, Dutta A, Chakraborty U, Kumar R, Das D, Ray BK
QJM 2022 Apr 20;115(4):222-227. doi: 10.1093/qjmed/hcac060. PMID: 35199176Free PMC Article
Li W, Luo Z, Yan CY, Wang XH, He ZJ, Ouyang SH, Yan C, Liu LF, Zhou QQ, Mu HL, Gong HB, Duan WJ, Liang L, Kurihara H, Feng D, Li YF, He RR
Theranostics 2020;10(20):9032-9049. Epub 2020 Jul 11 doi: 10.7150/thno.46921. PMID: 32802177Free PMC Article
Carr DJ, Härle P, Gebhardt BM
Exp Biol Med (Maywood) 2001 May;226(5):353-66. doi: 10.1177/153537020122600501. PMID: 11393165

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