{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,28]],"date-time":"2026-09-28T21:15:47Z","timestamp":1790630147281,"version":"4.1.0"},"reference-count":35,"publisher":"Oxford University Press (OUP)","issue":"4","license":[{"start":{"date-parts":[[2023,4,20]],"date-time":"2023-04-20T00:00:00Z","timestamp":1681948800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/pages\/standard-publication-reuse-rights"}],"funder":[{"name":"Centre de R\u00e9f\u00e9rence D\u00e9ficits Immunitaires H\u00e9r\u00e9ditaires"},{"DOI":"10.13039\/501100003762","name":"Institut Pasteur","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100003762","id-type":"DOI","asserted-by":"publisher"}]},{"name":"Necker-Enfants malades University Hospital"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2023,8,22]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Metagenomic next-generation sequencing (mNGS) was used to assess patients with primary or secondary immune deficiencies (PIDs and SIDs) who presented with immunopathological conditions related to immunodysregulation.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Thirty patients with PIDs or SIDs who presented with symptoms related to immunodysregulation and 59 asymptomatic patients with similar PIDs or SIDs were enrolled. mNGS was performed on organ biopsy. Specific Aichi virus (AiV) reverse-transcription polymerase chain reaction (RT-PCR) was used to confirm AiV infection and screen the other patients. In situ hybridization (ISH) assay was done on AiV-infected organs to identify infected cells. Virus genotype was determined by phylogenetic analysis.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>AiV sequences were detected using mNGS in tissue samples of 5 patients and by RT-PCR in peripheral samples of another patient, all of whom presented with PID and long-lasting multiorgan involvement, including hepatitis, splenomegaly, and nephritis in 4 patients. CD8+ T-cell infiltration was a hallmark of the disease. RT-PCR detected intermittent low viral loads in urine and plasma from infected patients but not from uninfected patients. Viral detection stopped after immune reconstitution obtained by hematopoietic stem cell transplantation. ISH demonstrated the presence of AiV RNA in hepatocytes (n = 1) and spleen tissue (n = 2). AiV belonged to genotype A (n = 2) or B (n = 3).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>The similarity of the clinical presentation, the detection of AiV in a subgroup of patients suffering from immunodysregulation, the absence of AiV in asymptomatic patients, the detection of viral genome in infected organs by ISH, and the reversibility of symptoms after treatment argue for AiV causality.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1093\/cid\/ciad237","type":"journal-article","created":{"date-parts":[[2023,4,20]],"date-time":"2023-04-20T08:29:14Z","timestamp":1681979354000},"page":"620-628","source":"Crossref","is-referenced-by-count":19,"title":["Chronic Aichi Virus Infection As a Cause of Long-Lasting Multiorgan Involvement in Patients With Primary Immune Deficiencies"],"prefix":"10.1093","volume":"77","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-3631-184X","authenticated-orcid":false,"given":"Jacques","family":"Fourgeaud","sequence":"first","affiliation":[{"name":"Universit\u00e9 Paris Cit\u00e9, F\u00e9d\u00e9ration pour l\u2019\u00c9tude et \u00e9valuation des Th\u00e9rapeutiques intra-Ut\u00e9rines , Paris , France"},{"name":"Microbiology Department, AP-HP, H\u00f4pital Necker Paris , France"},{"name":"Pathogen Discovery Laboratory, Institut Pasteur, Universit\u00e9 Paris Cit\u00e9 , Paris , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mathilde M","family":"Lecuit","sequence":"additional","affiliation":[{"name":"Pediatric Hematology Immunology and Rheumatology Unit, AP-HP, H\u00f4pital Necker Paris , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Philippe","family":"P\u00e9rot","sequence":"additional","affiliation":[{"name":"Pathogen Discovery Laboratory, Institut Pasteur, 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France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mael","family":"Bessaud","sequence":"additional","affiliation":[{"name":"Laboratoire signalisation antivirale, Institut Pasteur, Universit\u00e9 Paris Cit\u00e9 , Paris , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Capucine","family":"Picard","sequence":"additional","affiliation":[{"name":"Laboratory of Lymphocyte Activation and Susceptibility to EBV Infection, Universit\u00e9 Paris Cit\u00e9, Inserm, Institut Imagine Paris , France"},{"name":"Study Center for Primary Immunodeficiencies, Necker-Children's hospital, APHP Paris , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"\u00c9ric","family":"Jeziorski","sequence":"additional","affiliation":[{"name":"Pediatric Hematology Immunology Unit, Centre Hospitalier Universitaire de Montpellier , Montpellier , 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