Evidence map›Paper›PMID 31064750›Full record

ArticleBlood2019

Hydroa vacciniforme-like lymphoproliferative disorder: an EBV disease with a low risk of systemic illness in whites.

Jeffrey I Cohen, Irini Manoli, Kennichi Dowdell, Tammy A Krogmann, Deborah Tamura, Pierce Radecki, Wei Bu, Siu-Ping Turk, Kelly Liepshutz, Ronald L Hornung and 12 more

2 registry-linked trialsOpen access · bronzeAbstract read
In one paragraph

Article in Blood, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to 2 registered trials, which are not on this map. Cited by 30 papers.

0numbers the graph read from it
0cells of the map it votes in
30citing papers in PubMed
3.5field-weighted citation impact, top 6% 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.

NCT00032513 recruitingnot on this map

Genetic Studies of Chronic Active Epstein-Barr Virus Infection

TypeobservationalSponsorNational Institute of Allergy and Infectious Diseases (NIAID)Ran2002Enrolled300ConditionsChronic Active Epstein-Barr Virus
NCT00369421 recruitingnot on this map

Diagnosis and Treatment of Patients With Inborn Errors of Metabolism or Other Genetic Disorders

TypeobservationalSponsorNational Human Genome Research Institute (NHGRI)Ran1978Enrolled4,000ConditionsArterial Calcification Due to Deficiency of CD73
3 · Its place in the literature

Who cites it

30 citing papers in PubMed, 70 citations in OpenAlex.

  1. Review
  2. Article
  3. Article
  4. Article
  5. Review
  6. The Significance of the Microenvironment in T/Nk-Cell Neoplasms.International journal of molecular sciences · 2025
    Review
  7. Pediatric lymphomas: overview and diagnostic challenges.Virchows Archiv : an international journal of pathology · 2025
    Review
  8. Article
  9. Updates on Non-B-Lymphoid Neoplasms: A Review of the 5Journal of clinical practice and research · 2024
    Review
  10. EBV-Related Lymphoproliferative Diseases: A Review in Light of New Classifications.Mediterranean journal of hematology and infectious diseases · 2024
    Review
  11. Review
  12. Article
  13. Article
  14. Article
  15. New concepts in EBV-associated B, T, and NK cell lymphoproliferative disorders.Virchows Archiv : an international journal of pathology · 2023
    Review
  16. Article
  17. Review
  18. Article
  19. Article
  20. Article
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

22 authors at 3 institutions in 2 countries.

Jeffrey I CohenLaboratory of Infectious Diseases, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD.ORCID 0000-0003-0238-7176
Irini ManoliMedical Genomics and Metabolic Genetics Branch, National Human Genome Research Institute, National Institutes of Health, Bethesda, MD.ORCID 0000-0003-1543-2941
Kennichi DowdellLaboratory of Infectious Diseases, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD.
Tammy A KrogmannLaboratory of Infectious Diseases, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD.
Deborah TamuraLaboratory of Cancer Biology and Genetics, National Cancer Institute, National Institutes of Health, Bethesda, MD.
Pierce RadeckiLaboratory of Infectious Diseases, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD.
Wei BuLaboratory of Infectious Diseases, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD.
Siu-Ping TurkLaboratory of Clinical Infectious Diseases, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD.
Kelly LiepshutzLaboratory of Infectious Diseases, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD.
Ronald L HornungClinical Services Program, Leidos Biomedical Research, Frederick National Laboratory for Cancer Research, Frederick, MD.
Hiva FassihiDepartment of Photodermatology, St John's Institute of Dermatology, Guy's and St Thomas' NHS Foundation Trust, London, United Kingdom.
Robert P SarkanyDepartment of Photodermatology, St John's Institute of Dermatology, Guy's and St Thomas' NHS Foundation Trust, London, United Kingdom.
Lori L BonnycastleMedical Genomics and Metabolic Genetics Branch, National Human Genome Research Institute, National Institutes of Health, Bethesda, MD.
Peter S ChinesMedical Genomics and Metabolic Genetics Branch, National Human Genome Research Institute, National Institutes of Health, Bethesda, MD.
Amy J SwiftMedical Genomics and Metabolic Genetics Branch, National Human Genome Research Institute, National Institutes of Health, Bethesda, MD.
Timothy G MyersResearch Technologies Branch, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD.
Melissa A LevoskaLaboratory of Cancer Biology and Genetics, National Cancer Institute, National Institutes of Health, Bethesda, MD.
John J DiGiovannaLaboratory of Cancer Biology and Genetics, National Cancer Institute, National Institutes of Health, Bethesda, MD.ORCID 0000-0002-2750-2313
Francis S CollinsMedical Genomics and Metabolic Genetics Branch, National Human Genome Research Institute, National Institutes of Health, Bethesda, MD.
Kenneth H KraemerLaboratory of Cancer Biology and Genetics, National Cancer Institute, National Institutes of Health, Bethesda, MD.ORCID 0000-0002-2689-3316
Stefania PittalugaLaboratory of Pathology, Center for Cancer Research, National Cancer Institute, National Institutes of Health, Bethesda, MD.ORCID 0000-0001-7688-1439
Elaine S JaffeLaboratory of Pathology, Center for Cancer Research, National Cancer Institute, National Institutes of Health, Bethesda, MD.ORCID 0000-0003-4632-0301
National Institutes of Health · USGuy's and St Thomas' NHS Foundation Trust · GBLeidos (United States) · US

Funding

Hematopathology DiagnosisZIABC011070 · NCI · DIVISION OF BASIC SCIENCES - NCI · PI JAFFE, ELAINE · 2009 to 2025
$14.5M
Epstein-Barr Virus Associated DisordersZIAAI000913 · NIAID · NATIONAL INSTITUTE OF ALLERGY AND INFECTIOUS DISEASES · PI COHEN, JEFFREY · 2009 to 2025
$14.3M
Genetics of Moebius syndrome and other congenital facial weakness disordersZIAHG200389 · NHGRI · NATIONAL HUMAN GENOME RESEARCH INSTITUTE · PI COLLINS, FRANCIS S. · 2013 to 2024
$4.9M
CCR NIH HHS HHSN261200800001CNCI NIH HHS HHSN261200800001E
6 · The paper itself

Abstract

Patients with classic hydroa vacciniforme-like lymphoproliferative disorder (HVLPD) typically have high levels of Epstein-Barr virus (EBV) DNA in T cells and/or natural killer (NK) cells in blood and skin lesions induced by sun exposure that are infiltrated with EBV-infected lymphocytes. HVLPD is very rare in the United States and Europe but more common in Asia and South America. The disease can progress to a systemic form that may result in fatal lymphoma. We report our 11-year experience with 16 HVLPD patients from the United States and England and found that whites were less likely to develop systemic EBV disease (1/10) than nonwhites (5/6). All (10/10) of the white patients were generally in good health at last follow-up, while two-thirds (4/6) of the nonwhite patients required hematopoietic stem cell transplantation. Nonwhite patients had later age of onset of HVLPD than white patients (median age, 8 vs 5 years) and higher levels of EBV DNA (median, 1 515 000 vs 250 000 copies/ml) and more often had low numbers of NK cells (83% vs 50% of patients) and T-cell clones in the blood (83% vs 30% of patients). RNA-sequencing analysis of an HVLPD skin lesion in a white patient compared with his normal skin showed increased expression of interferon-γ and chemokines that attract T cells and NK cells. Thus, white patients with HVLPD were less likely to have systemic disease with EBV and had a much better prognosis than nonwhite patients. This trial was registered at www.clinicaltrials.gov as #NCT00369421 and #NCT00032513.

Indexed as

ChildChild, PreschoolEpstein-Barr Virus InfectionsFemaleHumansHydroa VacciniformeLymphoproliferative DisordersMaleWhite People

Identifiers

PMID31064750
PMCPMC6598378
OpenAlexW2943887480

What OpenQuestion holds

Textmetadata
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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.