Evidence map›Paper›PMID 36906275›Full record

ArticleTransplantation and cellular therapy2023

Immune Effector Cell-Associated Hemophagocytic Lymphohistiocytosis-Like Syndrome.

Melissa R Hines, Tristan E Knight, Kevin O McNerney, Mark B Leick, Tania Jain, Sairah Ahmed, Matthew J Frigault, Joshua A Hill, Michael D Jain, William T Johnson and 21 more

Erratum issued 3 registry-linked trialsOpen access · hybridAbstract read
In one paragraph

Article in Transplantation and cellular therapy, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. It is linked to 3 registered trials, which are not on this map. Cited by 219 papers, 7 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
219citing papers in PubMed, 7 pooled it
80.2field-weighted citation impact, top 1% 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.

NCT06072989 phase1recruitingnot on this map

A Single Arm, Open Label Study to Evaluate the Safety, Tolerability, Pharmacokinetics, and Antitumor Activity of B4T2-001 CAR-T in Patients With Advanced Solid Tumors

TypeinterventionalSponsorShanghai East HospitalRan2023 to 2027Enrolled24ConditionsAdvanced Solid TumorArmsB4T2-001 autologous CAR-T
NCT06703216 phase1 / phase2withdrawnnot on this mapstarted 2026, after this paper: background citation

Pilot Study of Pre-emptive Anakinra for the Prevention of Severe Cytokine Release Syndrome in Children and Young Adults With B-Acute Lymphoblastic Leukemia Receiving Chimeric Antigen Receptor (CAR) T Cells

TypeinterventionalSponsorKevin McNerneyRan2026 to 2030Enrolled0ConditionsB-Acute Lymphoblastic Leukemia, CAR-T Cell Therapy, Cytokine Release Syndrome, Immune Effector Cell Associated Neurotoxicity SyndromeArmsAnakinra (Kineret®)
NCT06879340 phase1recruitingnot on this mapstarted 2025, after this paper: background citation

A Phase 1 Multicenter, Open Label Trial Evaluating the Safety and Efficacy of DuoCAR20.19.22-D95 in Adult Patients With Relapsed or Refractory B-cell Malignancies

TypeinterventionalSponsorUniversity of Kansas Medical CenterRan2025 to 2040Enrolled54ConditionsB-Cell Non-Hodgkin Lymphoma, B-cell Acute Lymphoblastic LeukemiaArmsDuoCAR20.19.22-D95, Fludarabine (Conditional therapy), Cyclophosphamide (Conditional therapy)
3 · Its place in the literature

Who cites it

219 citing papers in PubMed, 7 syntheses or guidelines pooled it, 276 citations in OpenAlex.

  1. Guideline
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  14. Cardiotoxicity of T cell immunotherapies.Nature reviews. Cardiology · 2026
    Review
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  16. Article
  17. Article
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  19. Allogeneic CD70-Targeted Chimeric Antigen Receptor T-Cell Therapy for Advanced Renal Cell Carcinoma: Results From the Phase I TRAVERSE Trial.Journal of clinical oncology : official journal of the American Society of Clinical Oncology · 2026
    Article
  20. Article

159 more citing papers are in PubMed but not listed here.

4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

31 authors at 18 institutions in 2 countries.

Melissa R HinesDepartment of Pediatric Medicine, Division of Critical Care, St. Jude Children's Research Hospital, Memphis, Tennessee.
Tristan E KnightPediatric Hematology and Oncology, Seattle Children's Hospital and the University of Washington School of Medicine, Seattle, Washington.
Kevin O McNerneyCancer and Blood Disorders Institute, Johns Hopkins All Children's Hospital, St. Petersburg, Florida.
Mark B LeickCellular Immunotherapy Program and Blood and Marrow Transplant Program, Cancer Center, Massachusetts General Hospital, Boston, Massachusetts.
Tania JainDivision of Hematological Malignancies and Bone Marrow Transplantation, Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University, Baltimore, Maryland.
Sairah AhmedDepartments of Lymphoma and Myeloma and Stem Cell Transplantation, The University of Texas MD Anderson Cancer Center, Houston, Texas.
Matthew J FrigaultCellular Immunotherapy Program and Blood and Marrow Transplant Program, Cancer Center, Massachusetts General Hospital, Boston, Massachusetts.
Joshua A HillVaccine and Infectious Diseases Division, Fred Hutchinson Cancer Center, Seattle, Washington.
Michael D JainMoffitt Cancer Center, Tampa, Florida.
William T JohnsonDepartment of Medicine, Cellular Therapy Service, Memorial Sloan Kettering Cancer Center, New York, New York.
Yi LinDivision Hematology-Oncology and Blood and Marrow Transplantation Program, Mayo Clinic, Rochester, Minnesota.
Kris M MahadeoPediatric Transplantation and Cellular Therapy, Duke University, Durham, North Carolina.
Gabriela M MaronDepartment of Infectious Diseases, St. Jude Children's Research Hospital, Memphis, Tennessee, and Department of Pediatrics, University of Tennessee Health Science Center College of Medicine, Memphis, Tennessee.
Rebecca A MarshUniversity of Cincinnati, and Division of Bone Marrow Transplantation and Immune Deficiency, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.
Sattva S NeelapuDepartments of Lymphoma and Myeloma and Stem Cell Transplantation, The University of Texas MD Anderson Cancer Center, Houston, Texas.
Sarah NikiforowDivision of Hematologic Oncology, Department of Medical Oncology, Dana-Farber Cancer Institute, Harvard Medical School, Boston, Massachusetts.
Amanda K OmbrelloInflammatory Disease Section, National Human Genome Research Institute, National Institutes of Health, Bethesda, Maryland.
Nirav N ShahBone Marrow Transplant and Cellular Therapy Program, Division of Hematology and Oncology, Medical College of Wisconsin, Milwaukee, Wisconsin.
Aimee C TalleurDepartment of Bone Marrow Transplantation and Cellular Therapy, St. Jude Children's Research Hospital, Memphis, Tennessee and Department of Pediatrics, University of Tennessee Health Science Center College of Medicine, Memphis, Tennessee.
David TuricekPediatric Oncology Branch, Center for Cancer Research, National Cancer Institute, National Institutes of Health, Bethesda, Maryland.
Anant VatsayanDivision of Blood and Marrow Transplantation, Children's National Health System, Washington, District of Columbia.
Sandy W WongUCSF Health Division of Hematology and Oncology and Helen Diller Family Comprehensive Cancer Center, University of California, San Francisco, California.
Marcela V MausCellular Immunotherapy Program and Blood and Marrow Transplant Program, Cancer Center, Massachusetts General Hospital, Boston, Massachusetts.
Krishna V KomanduriUCSF Health Division of Hematology and Oncology and Helen Diller Family Comprehensive Cancer Center, University of California, San Francisco, California.
Nancy BerlinerBrigham and Women's Hospital, Boston, Massachusetts.
Jan-Inge HenterDivision of Pediatric Oncology and Surgery, Department of Women's and Children's Health, Karolinska Institute, and Department of Paediatric Oncology, Astrid Lindgren Children's Hospital, Karolinska University Hospital, Stockholm, Sweden.
Miguel-Angel PeralesDepartment of Medicine, Adult Bone Marrow Transplant Service, Memorial Sloan Kettering Cancer Center, New York, New York.
Noelle V FreyDivision of Hematology-Oncology, Abramson Cancer Center and Perelman School of Medicine at the University of Pennsylvania, Philadelphia, Pennsylvania.
David T TeacheyDivision of Oncology, The Children's Hospital of Philadelphia, Philadelphia, Pennsylvania.
Matthew J FrankDivision of Blood and Marrow Transplantation and Cellular Therapy, Stanford Cancer Institute, Stanford University School of Medicine, Stanford, California.
Nirali N ShahPediatric Oncology Branch, Center for Cancer Research, National Cancer Institute, National Institutes of Health, Bethesda, Maryland. Electronic address: Nirali.Shah@nih.gov.
National Institutes of Health · USMassachusetts General Hospital · USSt. Jude Children's Research Hospital · USJohns Hopkins University · USMemorial Sloan Kettering Cancer Center · USThe University of Texas MD Anderson Cancer Center · USUCSF Helen Diller Family Comprehensive Cancer Center · USBrigham and Women's Hospital · USChildren's National · USCincinnati Children's Hospital Medical Center · USDuke University · USFred Hutch Cancer Center · USHarvard University · USKarolinska University Hospital · SEMayo Clinic · USMoffitt Cancer Center · USSeattle Children's Hospital · USUniversity of Pennsylvania · US

Funding

X-RAY CRYSTALLOGRAPHYP30CA008748 · NCI · SLOAN-KETTERING INSTITUTE FOR CANCER RES · PI Hongbiao Carl Lekaye · 1985 to 2026
$347.4M
Project 4: Targeting Resistance to T-Cell Directed Therapy in Multiple MyelomaP50CA186781 · NCI · MAYO CLINIC ARIZONA · PI Peter Leif Bergsagel · 2015 to 2026
$25.5M
Immunotherapeutic approaches to treat pediatric hematologic malignanciesZIABC011823 · NCI · DIVISION OF BASIC SCIENCES - NCI · PI SHAH, NIRALI · 2019 to 2025
$6.1M
Intramural NIH HHS ZIA BC011823NCI NIH HHS P30 CA008748NCI NIH HHS P50 CA186781
6 · The paper itself

Abstract

T cell-mediated hyperinflammatory responses, such as cytokine release syndrome (CRS) and immune effector cell-associated neurotoxicity syndrome (ICANS), are now well-established toxicities of chimeric antigen receptor (CAR) T cell therapy. As the field of CAR T cells advances, however, there is increasing recognition that hemophagocytic lymphohistiocytosis (HLH)-like toxicities following CAR T cell infusion are occurring broadly across patient populations and CAR T cell constructs. Importantly, these HLH-like toxicities are often not as directly associated with CRS and/or its severity as initially described. This emergent toxicity, however ill-defined, is associated with life-threatening complications, creating an urgent need for improved identification and optimal management. With the goal of improving patient outcomes and formulating a framework to characterize and study this HLH-like syndrome, we established an American Society for Transplantation and Cellular Therapy panel composed of experts in primary and secondary HLH, pediatric and adult HLH, infectious disease, rheumatology and hematology, oncology, and cellular therapy. Through this effort, we provide an overview of the underlying biology of classical primary and secondary HLH, explore its relationship with similar manifestations following CAR T cell infusions, and propose the term "immune effector cell-associated HLH-like syndrome (IEC-HS)" to describe this emergent toxicity. We also delineate a framework for identifying IEC-HS and put forward a grading schema that can be used to assess severity and facilitate cross-trial comparisons. Additionally, given the critical need to optimize outcomes for patients experiencing IEC-HS, we provide insight into potential treatment approaches and strategies to optimize supportive care and delineate alternate etiologies that should be considered in a patient presenting with IEC-HS. By collectively defining IEC-HS as a hyperinflammatory toxicity, we can now embark on further study of the pathophysiology underlying this toxicity profile and make strides toward a more comprehensive assessment and treatment approach.

Indexed as

Lymphohistiocytosis, HemophagocyticNeurotoxicity SyndromesAdultChildCytokine Release SyndromeHumansImmunotherapy, AdoptiveT-LymphocytesUnited Statescell-associated neurotoxicityChimeric antigen receptor T cellHemophagocytic lymphohistiocytosisMacrophage activation syndrome

Identifiers

PMID36906275
PMCPMC10330221
OpenAlexW4323659812

What OpenQuestion holds

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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.