Evidence map›Paper›PMID 36638922›Full record

ArticleThe Journal of allergy and clinical immunology2023

Late-onset enteric virus infection associated with hepatitis (EVAH) in transplanted SCID patients.

Quentin Riller, Jacques Fourgeaud, Julie Bruneau, Suk See De Ravin, Grace Smith, Mathieu Fusaro, Samy Meriem, Aude Magerus, Marine Luka, Ghaith Abdessalem and 48 more

Registry-linked trialOpen access · bronzeAbstract read
In one paragraph

Article in The Journal of allergy and clinical immunology, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT06659588 (Study of Populations at Risk of Developing Chronic Hepatitis Linked to Chronic Enteric Virus Infection in Patients With Primary Immunodeficiency and Secondary Humoral Deficiency), which is not on this map. Cited by 5 papers.

0numbers the graph read from it
0cells of the map it votes in
5citing papers in PubMed
2.9field-weighted citation impact, top 9% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

NCT06659588 recruitingnot on this mapstarted 2024, after this paper: background citation

Study of Populations at Risk of Developing Chronic Hepatitis Linked to Chronic Enteric Virus Infection in Patients With Primary Immunodeficiency and Secondary Humoral Deficiency

TypeobservationalSponsorAssistance Publique - Hôpitaux de ParisRan2024 to 2026Enrolled120ConditionsHumoral Primary Immunodeficiencies (PIDs), Secondary Form of Humoral Immunodeficiencies, Combined Immunodeficiency (CID), Severe Combined Immunodeficiency (SCID)ArmsPlasma, urine and stool collection
3 · Its place in the literature

Who cites it

5 citing papers in PubMed, 15 citations in OpenAlex.

  1. Article
  2. Lymphedema in patients with X-linked severe combined immunodeficiency.The journal of allergy and clinical immunology. Global · 2026
    Article
  3. Review
  4. Article
  5. Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

58 authors at 14 institutions in 4 countries.

Quentin RillerUniversity of Paris Cité, Paris, France; Laboratory of Immunogenetics of Pediatric Autoimmune Diseases, Imagine Institute, INSERM UMR 1163, Paris, France.
Jacques FourgeaudUniversity of Paris Cité, Paris, France; Microbiology Department, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France; Pathogen Discovery Laboratory, Institut Pasteur, Université de Paris, Paris, France; Prise en Charge des Anomalies Congénitales et leur Traitement, Unit 7328, Imagine Institute, University of Paris Cité, Paris, France.
Julie BruneauUniversity of Paris Cité, Paris, France; Pathology Department, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France; Imagine Institute, INSERM UMR 1163, Laboratory of Molecular Mechanisms of Hematologic Disorders and Therapeutic Implications, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.
Suk See De RavinLaboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, Md.
Grace SmithCenter for Cancer Research, National Cancer Institute, National Institutes of Health, Bethesda, Md.
Mathieu FusaroStudy Center for Primary Immunodeficiencies, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.
Samy MeriemLaboratory of Biostatistics, University of Paris Cité, Paris, France.
Aude MagerusUniversity of Paris Cité, Paris, France; Laboratory of Immunogenetics of Pediatric Autoimmune Diseases, Imagine Institute, INSERM UMR 1163, Paris, France.
Marine LukaLabtech Single-Cell@Imagine, Imagine Institute, INSERM UMR 1163, Paris, France.
Ghaith AbdessalemLabtech Single-Cell@Imagine, Imagine Institute, INSERM UMR 1163, Paris, France.
Ludovic LhermitteUniversity of Paris Cité, Paris, France; Laboratory of Onco-Haematology, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France; the Institut Necker-Enfants Malades (INEM), INSERM UMR 1151, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.
Anne JametUniversity of Paris Cité, Paris, France; Microbiology Department, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France; the Institut Necker-Enfants Malades (INEM), INSERM UMR 1151, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.
Emmanuelle SixUniversity of Paris Cité, Paris, France; Laboratory of Human Lympho-Hematopoiesis, Imagine Institute, INSERM UMR 1163, Paris, France.
Alessandra MagnaniDepartment of Biotherapy, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.
Martin CastellePediatric Hematology-Immunology and Rheumatology Unit, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.
Romain LévyUniversity of Paris Cité, Paris, France; Pediatric Hematology-Immunology and Rheumatology Unit, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France; Laboratory of Human Genetics of Infectious Diseases, Necker Branch, INSERM, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.
Mathilde M LecuitPediatric Hematology-Immunology and Rheumatology Unit, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.
Benjamin FournierUniversity of Paris Cité, Paris, France; Pediatric Hematology-Immunology and Rheumatology Unit, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.
Sarah WinterUniversity of Paris Cité, Paris, France; Pediatric Hematology-Immunology and Rheumatology Unit, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.
Michaela SemeraroUniversity of Paris Cité, Paris, France; Clinical Investigation Center, Clinical Research Unit, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.
Graziella PintoPediatric Endocrinology, Gynecology, Diabetology, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.
Hanène AbidUniversity of Paris Cité, Paris, France; Microbiology Department, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.
Nizar MahlaouiPediatric Hematology-Immunology and Rheumatology Unit, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.
Nathalie CheikhPediatric Hematology Oncology Unit, University Hospital of Besançon, Besançon, France.
Benoit FlorkinImmuno-Hémato-Rhumatologie Pédiatrique, Service de Pédiatrie, CHR Citadelle, Liege, Belgium.
Pierre FrangeUniversity of Paris Cité, Paris, France; Microbiology Department, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France; Pediatric Hematology-Immunology and Rheumatology Unit, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.
Eric JeziorskiDepartment of Pediatrics, Infectious Diseases, and Immunology, University of Montpellier, CHU Montpellier, Montpellier, France.
Felipe SuarezUniversity of Paris Cité, Paris, France; Imagine Institute, INSERM UMR 1163, Laboratory of Molecular Mechanisms of Hematologic Disorders and Therapeutic Implications, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France; Hematology Department, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.
Françoise Sarrot-ReynauldService de Médecine Interne, Centre Hospitalier Universitaire Grenoble Alpes, Grenoble, France.
Dalila NouarService d'Immunologie Clinique et d'Allergologie, Centre Hospitalier Régional Universitaire, Tours, France.
Dominique DebrayPediatric Liver Unit, National Reference Center for Rare Diseases, Biliary Atresia and Genetic Cholestasis, Inflammatory Biliary Diseases and Autoimmune Hepatitis, ERN Rare Liver, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.
Florence LacailleGastroenterology-Hepatology-Nutrition Unit, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.
Capucine PicardStudy Center for Primary Immunodeficiencies, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France; Laboratory of Lymphocyte Activation and Susceptibility to EBV Infection, Imagine Institute, INSERM UMR 1163, Paris, France.
Philippe PérotPathogen Discovery Laboratory, Institut Pasteur, Université de Paris, Paris, France; OIE Collaborating Center for the Detection and Identification in Humans of Emerging Animal Pathogens, Institut Pasteur, Paris, France.
Béatrice RegnaultPathogen Discovery Laboratory, Institut Pasteur, Université de Paris, Paris, France; OIE Collaborating Center for the Detection and Identification in Humans of Emerging Animal Pathogens, Institut Pasteur, Paris, France.
Nicolas Da RochaPathogen Discovery Laboratory, Institut Pasteur, Université de Paris, Paris, France; OIE Collaborating Center for the Detection and Identification in Humans of Emerging Animal Pathogens, Institut Pasteur, Paris, France.
Camille de CevinsUniversity of Paris Cité, Paris, France; Laboratory of Inflammatory Responses and Transcriptomic Networks in Diseases, Atip-Avenir Team, Imagine Institute, INSERM UMR 1163, Paris, France; Artificial Intelligence & Deep Analytics (AIDA) Group, Data & Data Science (DDS), Sanofi R&D, Chilly-Mazarin, France.
Laure DelageUniversity of Paris Cité, Paris, France; Laboratory of Immunogenetics of Pediatric Autoimmune Diseases, Imagine Institute, INSERM UMR 1163, Paris, France.
Brieuc P PérotUniversity of Paris Cité, Paris, France; Laboratory of Inflammatory Responses and Transcriptomic Networks in Diseases, Atip-Avenir Team, Imagine Institute, INSERM UMR 1163, Paris, France.
Angélique VinitSorbonne Université, UMS037, PASS, Plateforme de Cytométrie de la Pitié-Salpêtrière CyPS, Paris, France.
Francesco CarboneUniversity of Paris Cité, Paris, France; Labtech Single-Cell@Imagine, Imagine Institute, INSERM UMR 1163, Paris, France; Laboratory of Inflammatory Responses and Transcriptomic Networks in Diseases, Atip-Avenir Team, Imagine Institute, INSERM UMR 1163, Paris, France.
Camille BrunaudUniversity of Paris Cité, Paris, France; Laboratory of Immunogenetics of Pediatric Autoimmune Diseases, Imagine Institute, INSERM UMR 1163, Paris, France.
Manon MarchaisUniversity of Paris Cité, Paris, France; Laboratory of Immunogenetics of Pediatric Autoimmune Diseases, Imagine Institute, INSERM UMR 1163, Paris, France.
Marie-Claude StolzenbergUniversity of Paris Cité, Paris, France; Laboratory of Immunogenetics of Pediatric Autoimmune Diseases, Imagine Institute, INSERM UMR 1163, Paris, France.
Vahid AsnafiUniversity of Paris Cité, Paris, France; Laboratory of Onco-Haematology, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France; the Institut Necker-Enfants Malades (INEM), INSERM UMR 1151, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.
Thierry MolinaUniversity of Paris Cité, Paris, France; Pathology Department, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.
Frédéric Rieux-LaucatUniversity of Paris Cité, Paris, France; Laboratory of Immunogenetics of Pediatric Autoimmune Diseases, Imagine Institute, INSERM UMR 1163, Paris, France.
Luigi D NotarangeloLaboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, Md.
Stefania PittalugaNational Cancer Institute, National Institutes of Health, Bethesda, Md.
Jean Philippe JaisUniversity of Paris Cité, Paris, France; Laboratory of Biostatistics, University of Paris Cité, Paris, France.
Despina MoshousUniversity of Paris Cité, Paris, France; Pediatric Hematology-Immunology and Rheumatology Unit, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France; Laboratory of Genome Dynamics in the Immune System, Equipe Labellisée Ligue contre le Cancer, Imagine Institute, INSERM UMR 1163, Paris, France.
Stephane BlancheUniversity of Paris Cité, Paris, France; Pediatric Hematology-Immunology and Rheumatology Unit, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.
Harry MalechLaboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, Md.
Marc EloitPathogen Discovery Laboratory, Institut Pasteur, Université de Paris, Paris, France; OIE Collaborating Center for the Detection and Identification in Humans of Emerging Animal Pathogens, Institut Pasteur, Paris, France; Ecole Nationale Vétérinaire d'Alfort, Maisons-Alfort, France.
Marina CavazzanaUniversity of Paris Cité, Paris, France; Laboratory of Onco-Haematology, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France; Laboratory of Human Lympho-Hematopoiesis, Imagine Institute, INSERM UMR 1163, Paris, France.
Alain FischerPediatric Hematology-Immunology and Rheumatology Unit, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France; Collège de France, Paris, France.
Mickaël M MénagerUniversity of Paris Cité, Paris, France; Labtech Single-Cell@Imagine, Imagine Institute, INSERM UMR 1163, Paris, France; Laboratory of Inflammatory Responses and Transcriptomic Networks in Diseases, Atip-Avenir Team, Imagine Institute, INSERM UMR 1163, Paris, France.
Bénédicte NevenUniversity of Paris Cité, Paris, France; Laboratory of Immunogenetics of Pediatric Autoimmune Diseases, Imagine Institute, INSERM UMR 1163, Paris, France; Pediatric Hematology-Immunology and Rheumatology Unit, Necker-Children's Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France. Electronic address: benedicte.neven@aphp.fr.
Inserm · FRAssistance Publique – Hôpitaux de Paris · FRUniversité Paris Cité · FRNational Institutes of Health · USHôpital Necker-Enfants Malades · FRInstitut Pasteur · FRCentre hospitalier régional de la Citadelle · BECentre Hospitalier Universitaire de Besançon · FRCentre Hospitalier Universitaire de Grenoble · FRCentre Hospitalier Universitaire de Tours · FRCollège de France · FRDélégation Paris 5 · FRSorbonne Université · FRUniversité de Montpellier · FR

Funding

Characterization of the cellular and molecular bases of inborn errors of immunityZIAAI001222 · NIAID · NATIONAL INSTITUTE OF ALLERGY AND INFECTIOUS DISEASES · PI NOTARANGELO, LUIGI · 2017 to 2025
$15.2M
Gene Therapy Basic Research to Treat Inherited Primary Immune DeficienciesZIAAI000644 · NIAID · NATIONAL INSTITUTE OF ALLERGY AND INFECTIOUS DISEASES · PI MALECH, HARRY L · 2009 to 2025
$12.1M
Intramural NIH HHS ZIA AI001222
6 · The paper itself

Abstract

backgroundAllogenic hematopoietic stem cell transplantation (HSCT) and gene therapy (GT) are potentially curative treatments for severe combined immunodeficiency (SCID). Late-onset posttreatment manifestations (such as persistent hepatitis) are not uncommon.

objectiveWe sought to characterize the prevalence and pathophysiology of persistent hepatitis in transplanted SCID patients (SCIDH+) and to evaluate risk factors and treatments.

methodsWe used various techniques (including pathology assessments, metagenomics, single-cell transcriptomics, and cytometry by time of flight) to perform an in-depth study of different tissues from patients in the SCIDH+ group and corresponding asymptomatic similarly transplanted SCID patients without hepatitis (SCIDH-).

resultsEleven patients developed persistent hepatitis (median of 6 years after HSCT or GT). This condition was associated with the chronic detection of enteric viruses (human Aichi virus, norovirus, and sapovirus) in liver and/or stools, which were not found in stools from the SCIDH- group (n = 12). Multiomics analysis identified an expansion of effector memory CD8

conclusionsEnteric virus infection associated with hepatitis is related to chronic enteric viral infection and immune dysregulation and is an important risk for transplanted SCID patients with defective B-cell function.

Indexed as

Enterovirus InfectionsHematopoietic Stem Cell TransplantationHepatitisSevere Combined ImmunodeficiencyVirus DiseasesCD8-Positive T-LymphocytesHumansB-cell functionCD8(+) T cellsenteric virusgene therapyinterferonSCIDtransplantation

Identifiers

PMID36638922
PMCPMC10336473
OpenAlexW4315490495

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Registered trials

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.