{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,13]],"date-time":"2026-02-13T14:09:17Z","timestamp":1770991757700,"version":"3.50.1"},"reference-count":18,"publisher":"Wiley","issue":"8","license":[{"start":{"date-parts":[[2021,5,4]],"date-time":"2021-05-04T00:00:00Z","timestamp":1620086400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Journal of Medical Virology"],"published-print":{"date-parts":[[2021,8]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:label\/><jats:p>Infections due to human herpesvirus 6 (HHV\u20106) are frequent during early childhood. Usually, they have a favorable clinical course. Conversely, HHV\u20106 congenital infections occur in about 1% of neonates and may present with more severe clinical pictures. HHV\u20106 can be found in lung tissues and bronchoalveolar lavage (BAL) samples from patients with pneumonia and in immunocompromised patients can cause mild to severe pneumonia. In neonates, the role of HHV\u20106 in the genesis of severe pneumonia is poorly defined still now. We describe a healthy infant with a late\u2010onset (15 days of life) severe interstitial pneumonia and heavy HHV\u20106 genome load, persistently detected in its BAL\u00a0fluid. The baby underwent high\u2010frequency oscillatory ventilation, hydroxychloroquine, steroids, and ganciclovir for 6 weeks and at 9 months she died. Next\u2010generation sequencing of genes known to cause neonatal respiratory insufficiency revealed the presence of a \u201cprobably pathogenetic\u201d heterozygous variant in the autosomal recessive <jats:italic>DRC1<\/jats:italic> gene, a heterozygous variant of unknown significance (VUS) in the autosomal recessive <jats:italic>RSPH9<\/jats:italic> gene, and a heterozygous VUS in the autosomal recessive <jats:italic>MUC5B<\/jats:italic> gene.\u00a0HHV\u20106 infection should be considered in the differential diagnosis of late\u2010onset severe respiratory distress in neonates and the co\u2010occurrence of genetic predisposing factors or modifiers should be tested by specific molecular techniques. The intensity of HHV\u20106 genome load in BAL fluid could be an indicator of the response to antiviral therapy.<\/jats:p><\/jats:sec>","DOI":"10.1002\/jmv.27016","type":"journal-article","created":{"date-parts":[[2021,4,15]],"date-time":"2021-04-15T00:43:28Z","timestamp":1618447408000},"page":"5182-5187","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Severe herpes virus 6 interstitial pneumonia in an infant with three variants in genes predisposing to lung disease"],"prefix":"10.1002","volume":"93","author":[{"given":"Domenico Umberto","family":"De Rose","sequence":"first","affiliation":[{"name":"Neonatal Intensive Care Unit, Department of Medical and Surgical Neonatology \u201cBambino Ges\u00f9\u201d Children's Hospital IRCCS  Rome Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9820-6557","authenticated-orcid":false,"given":"Cinzia","family":"Auriti","sequence":"additional","affiliation":[{"name":"Neonatal Intensive Care Unit, Department of Medical and Surgical Neonatology \u201cBambino Ges\u00f9\u201d Children's Hospital IRCCS  Rome Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Simona","family":"Lozzi","sequence":"additional","affiliation":[{"name":"Neonatal Intensive Care Unit, Department of Medical and Surgical Neonatology \u201cBambino Ges\u00f9\u201d Children's Hospital IRCCS  Rome Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4933-4242","authenticated-orcid":false,"given":"Luana","family":"Coltella","sequence":"additional","affiliation":[{"name":"Microbiology Unit \u201cBambino Ges\u00f9\u201d Children's Hospital IRCCS  Rome Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Livia","family":"Piccioni","sequence":"additional","affiliation":[{"name":"Microbiology Unit \u201cBambino Ges\u00f9\u201d Children's Hospital IRCCS  Rome Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sabrina","family":"Rossi","sequence":"additional","affiliation":[{"name":"Department of Pathology \u201cBambino Ges\u00f9\u201d Children's Hospital IRCCS  Rome Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Antonio","family":"Novelli","sequence":"additional","affiliation":[{"name":"Medical Genetics Laboratory \u201cBambino Ges\u00f9\u201d Children's Hospital IRCCS  Rome Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Rossella","family":"Iannotta","sequence":"additional","affiliation":[{"name":"Neonatal Intensive Care Unit \u201cPoliclinico Casilino\u201d General Hospital  Rome Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Teresa","family":"Pianini","sequence":"additional","affiliation":[{"name":"Neonatal Intensive Care Unit \u201cPoliclinico Casilino\u201d General Hospital  Rome Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Simonetta","family":"Picone","sequence":"additional","affiliation":[{"name":"Neonatal Intensive Care Unit \u201cPoliclinico Casilino\u201d General Hospital  Rome Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Piermichele","family":"Paolillo","sequence":"additional","affiliation":[{"name":"Neonatal Intensive Care Unit \u201cPoliclinico Casilino\u201d General Hospital  Rome Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ferdinando","family":"Savignoni","sequence":"additional","affiliation":[{"name":"Neonatal Intensive Care Unit, Department of Medical and Surgical Neonatology \u201cBambino Ges\u00f9\u201d Children's Hospital IRCCS  Rome Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Irma","family":"Capolupo","sequence":"additional","affiliation":[{"name":"Neonatal Intensive Care Unit, Department of Medical and Surgical Neonatology \u201cBambino Ges\u00f9\u201d Children's Hospital IRCCS  Rome Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Maria Cristina","family":"Digilio","sequence":"additional","affiliation":[{"name":"Genetics and Rare Diseases Research Division \u201cBambino Ges\u00f9\u201d Children's Hospital IRCCS  Rome Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Renato","family":"Cutrera","sequence":"additional","affiliation":[{"name":"Pediatric Pulmonology &amp; 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