{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,10]],"date-time":"2026-07-10T12:56:20Z","timestamp":1783688180960,"version":"3.55.0"},"reference-count":15,"publisher":"Oxford University Press (OUP)","issue":"1","license":[{"start":{"date-parts":[[2021,5,26]],"date-time":"2021-05-26T00:00:00Z","timestamp":1621987200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/journals\/pages\/open_access\/funder_policies\/chorus\/standard_publication_model"}],"funder":[{"DOI":"10.13039\/501100002739","name":"Aarhus Universitets Forskningsfond","doi-asserted-by":"publisher","award":["AUFF-E-2015-FLS-66"],"award-info":[{"award-number":["AUFF-E-2015-FLS-66"]}],"id":[{"id":"10.13039\/501100002739","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100003554","name":"Lundbeckfonden","doi-asserted-by":"publisher","award":["R268-2016\u20133927"],"award-info":[{"award-number":["R268-2016\u20133927"]}],"id":[{"id":"10.13039\/501100003554","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2022,1,5]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>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-\u03baB 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.<\/jats:p>","DOI":"10.1093\/infdis\/jiab290","type":"journal-article","created":{"date-parts":[[2021,5,21]],"date-time":"2021-05-21T19:12:05Z","timestamp":1621624325000},"page":"157-162","source":"Crossref","is-referenced-by-count":7,"title":["Postpartum Disseminated Herpes Simplex Virus Type 1 Infection With Hemophagocytic Lymphohistiocytosis and Fulminant Neonatal Herpes Infection"],"prefix":"10.1093","volume":"225","author":[{"given":"Mette Ratzer","family":"Freytag","sequence":"first","affiliation":[{"name":"Department of Infectious Diseases, Aarhus University Hospital, Aarhus, Denmark"},{"name":"Department of Biomedicine, Aarhus University, Aarhus, Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sofie Eg","family":"J\u00f8rgensen","sequence":"additional","affiliation":[{"name":"Department of Infectious Diseases, Aarhus University Hospital, Aarhus, Denmark"},{"name":"Department of Biomedicine, Aarhus University, Aarhus, Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michelle M\u00f8lgaard","family":"Thomsen","sequence":"additional","affiliation":[{"name":"Department of Infectious Diseases, Aarhus University Hospital, Aarhus, Denmark"},{"name":"Department of Biomedicine, Aarhus University, Aarhus, Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ali","family":"Al-Mousawi","sequence":"additional","affiliation":[{"name":"Department of Infectious Diseases, Aarhus University Hospital, Aarhus, Denmark"},{"name":"Department of Biomedicine, Aarhus University, Aarhus, Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alon Schneider","family":"Hait","sequence":"additional","affiliation":[{"name":"Department of Infectious Diseases, Aarhus University Hospital, Aarhus, Denmark"},{"name":"Department of Biomedicine, Aarhus University, Aarhus, Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David","family":"Olagnier","sequence":"additional","affiliation":[{"name":"Department of Biomedicine, Aarhus University, Aarhus, Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jakob T","family":"Bay","sequence":"additional","affiliation":[{"name":"Department of Clinical Immunology, Rigshospitalet, Copenhagen, Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2370-1657","authenticated-orcid":false,"given":"Marie","family":"Helleberg","sequence":"additional","affiliation":[{"name":"Department of Infectious Diseases, Rigshospitalet, Copenhagen, Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1853-9704","authenticated-orcid":false,"given":"Trine H","family":"Mogensen","sequence":"additional","affiliation":[{"name":"Department of Infectious Diseases, Aarhus University Hospital, Aarhus, Denmark"},{"name":"Department of Biomedicine, Aarhus University, Aarhus, Denmark"},{"name":"Department of Clinical Medicine, Aarhus University Hospital, Aarhus, Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2021,5,26]]},"reference":[{"key":"2022010519004578300_CIT0001","doi-asserted-by":"crossref","first-page":"106","DOI":"10.1016\/j.coi.2020.01.004","article-title":"Human inborn errors of immunity to herpes viruses.","volume":"62","author":"Jouanguy","year":"2020","journal-title":"Curr Opin Immunol"},{"key":"2022010519004578300_CIT0002","doi-asserted-by":"crossref","first-page":"47","DOI":"10.1016\/j.clp.2014.10.005","article-title":"Neonatal herpes simplex virus infection: epidemiology and treatment.","volume":"42","author":"James","year":"2015","journal-title":"Clin Perinatol"},{"key":"2022010519004578300_CIT0003","doi-asserted-by":"crossref","first-page":"1726","DOI":"10.12688\/f1000research.16157.1","article-title":"Clinical management of herpes simplex virus infections: past, present, and future.","volume":"7(F1000 Faculty Rev)","author":"Whitley","year":"2018","journal-title":"F1000Res"},{"key":"2022010519004578300_CIT0004","doi-asserted-by":"crossref","first-page":"51","DOI":"10.1093\/clinids\/22.1.51","article-title":"Disseminated herpesvirus infection during pregnancy.","volume":"22","author":"Young","year":"1996","journal-title":"Clin Infect Dis"},{"key":"2022010519004578300_CIT0005","doi-asserted-by":"crossref","first-page":"168","DOI":"10.1053\/j.semperi.2018.02.004","article-title":"Neonatal herpes simplex virus infections.","volume":"42","author":"Pinninti","year":"2018","journal-title":"Semin Perinatol"},{"key":"2022010519004578300_CIT0006","doi-asserted-by":"crossref","first-page":"436","DOI":"10.1016\/j.reprotox.2004.11.014","article-title":"Herpes simplex virus and Epstein-Barr virus infections in pregnancy: consequences of neonatal or intrauterine infection.","volume":"21","author":"Avgil","year":"2006","journal-title":"Reprod Toxicol"},{"key":"2022010519004578300_CIT0007","doi-asserted-by":"crossref","first-page":"638","DOI":"10.1016\/j.jaci.2015.07.043","article-title":"GATA2 deficiency underlying severeblastomycosis and fatal herpes simplex virus-associated hemophagocytic lymphohistiocytosis.","volume":"137","author":"Spinner","year":"2016","journal-title":"J Allergy Clin Immunol"},{"key":"2022010519004578300_CIT0008","doi-asserted-by":"crossref","first-page":"e27929","DOI":"10.1002\/pbc.27929","article-title":"Challenges in the diagnosis of hemophagocytic lymphohistiocytosis: recommendations from the North American Consortium for Histiocytosis (NACHO).","volume":"66","author":"Jordan","year":"2019","journal-title":"Pediatr Blood Cancer"},{"key":"2022010519004578300_CIT0009","doi-asserted-by":"crossref","first-page":"5794","DOI":"10.1182\/blood-2011-07-370148","article-title":"Hypomorphic mutations in PRF1, MUNC13-4, and STXBP2 are associated with adult-onset familial HLH.","volume":"118","author":"Zhang","year":"2011","journal-title":"Blood"},{"key":"2022010519004578300_CIT0010","doi-asserted-by":"crossref","first-page":"124","DOI":"10.1002\/pbc.21039","article-title":"HLH-2004: diagnostic and therapeutic guidelines for hemophagocytic lymphohistiocytosis.","volume":"48","author":"Henter","year":"2007","journal-title":"Pediatr Blood Cancer"},{"key":"2022010519004578300_CIT0011","doi-asserted-by":"crossref","first-page":"1503","DOI":"10.1016\/S0140-6736(13)61048-X","article-title":"Adult haemophagocytic syndrome.","volume":"383","author":"Ramos-Casals","year":"2014","journal-title":"Lancet"},{"key":"2022010519004578300_CIT0013","doi-asserted-by":"crossref","first-page":"bcr2016218310","DOI":"10.1136\/bcr-2016-218310","article-title":"Haemophagocytic lymphohistiocytosis associated with fulminant hepatitis and multiorgan failure following primary Epstein-Barr virus and herpes simplex virus type 1 infection.","volume":"2017","author":"Honsig","year":"2017","journal-title":"BMJ Case Rep"},{"key":"2022010519004578300_CIT0014","doi-asserted-by":"crossref","first-page":"2919","DOI":"10.2169\/internalmedicine.8490-16","article-title":"Successful treatment of herpes simplex virus (HSV)-1-associated hemophagocytic lymphohistiocytosis (HLH) with acyclovir: a case report and literature review.","volume":"56","author":"Yabushita","year":"2017","journal-title":"Intern Med"},{"key":"2022010519004578300_CIT0015","doi-asserted-by":"crossref","first-page":"395","DOI":"10.1038\/nature01063","article-title":"Pleiotropic defects in lymphocyte activation caused by caspase-8 mutations lead to human immunodeficiency.","volume":"419","author":"Chun","year":"2002","journal-title":"Nature"},{"key":"2022010519004578300_CIT0016","doi-asserted-by":"crossref","first-page":"e20200687","DOI":"10.1542\/peds.2020-0687","article-title":"Rare genetic variants in immune genes and neonatal herpes simplex viral infections.","volume":"147","author":"Cummings","year":"2021","journal-title":"Pediatrics"}],"container-title":["The Journal of Infectious Diseases"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/jid\/advance-article-pdf\/doi\/10.1093\/infdis\/jiab290\/41158565\/jiab290.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/jid\/article-pdf\/225\/1\/157\/41937272\/jiab290.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/jid\/article-pdf\/225\/1\/157\/41937272\/jiab290.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2022,1,5]],"date-time":"2022-01-05T19:06:16Z","timestamp":1641409576000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/jid\/article\/225\/1\/157\/6284333"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,5,26]]},"references-count":15,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2021,5,26]]},"published-print":{"date-parts":[[2022,1,5]]}},"URL":"https:\/\/doi.org\/10.1093\/infdis\/jiab290","relation":{},"ISSN":["0022-1899","1537-6613"],"issn-type":[{"value":"0022-1899","type":"print"},{"value":"1537-6613","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2022,1,1]]},"published":{"date-parts":[[2021,5,26]]}}}