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Record W4410612415 · doi:10.1016/j.ero.2025.04.004

Early management of hemophagocytic lymphohistiocytosis/macrophage activation syndrome: a systematic literature review

2025· article· en· W4410612415 on OpenAlexaff
Dimitri Poddighe, Bita Shakoory, Ashley V. Geerlinks, Marta Wilejto, Kate F. Kernan, Melissa Hines, Micol Romano, David Piskin, Ovgu Kul Cınar, Jonathan Park, Angelo Ravelli, Raphaela Goldbach‐Mansky, Fabrizio De Benedetti, Rebecca Marsh, Scott Canna, Erkan Demirkaya

Bibliographic record

VenueEULAR Rheumatology Open · 2025
Typearticle
Languageen
FieldMedicine
TopicAutoimmune and Inflammatory Disorders Research
Canadian institutionsWestern University
FundersNational Institute of Allergy and Infectious DiseasesEuropean League Against RheumatismRheumatology Research FoundationAmerican College of Rheumatology Research and Education Foundation
KeywordsHemophagocytic lymphohistiocytosisMacrophage activation syndromeMedicineHemophagocytosisHistiocytosisIntensive care medicineImmunologyPathologyDisease

Abstract

fetched live from OpenAlex

Objectives A systematic literature review was undertaken to summarise and analyse the available evidence on the initial hemophagocytic lymphohistiocytosis and macrophage activation syndrome (HLH/MAS) treatment according to the distinctive pathologic background and/or triggering factors. Methods An electronic search in the main medical databases (PubMed, EMBASE, and Cochrane Library) was conducted, in order to select the studies providing information related to the HLH/MAS therapy. To further maximise the evidence derived from this systematic review, we focused our attention on clinical studies that included and described >10 HLH/MAS cases. Results This literature search led to identify 18,020 articles. After the screening by title, removal of duplicated records, and full-text assessment of the relevant articles, the final search output consisted of 167 articles that provided enough information on the medical management of patients with HLH/MAS In total, 77 clinical studies (including >10 patients) describing the early treatment of HLH/MAS have been included in the present analysis. Conclusions Primary HLH is typically managed according to the HLH-94 or HLH-2004 protocols, with the main aim of bringing the patient to a hematopoietic stem cell transplant (HSCT). Secondary forms of HLH/MAS can arise in 3 main clinical contexts, namely rheumatic disorders, oncological diseases, and previously healthy patients developing specific and/or severe infections. Accordingly, different and variable therapeutic approaches have been applied to these 3 categories of secondary HLH/MAS. Despite the survival improvements achieved in the last decades in patients with HLH/MAS, there is a strong need for prospective and controlled studies focused on each specific group of HLH/MAS.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

How this classification was reachedexpand

Full frame distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.001
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Systematic review · Consensus signal: Systematic review
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.412
Threshold uncertainty score0.690

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0000.001
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0010.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0000.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.012
GPT teacher head0.313
Teacher spread0.301 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it

Classification

machine, unvalidated

Machine predicted; a candidate call from one teacher head, not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designSystematic review
Domainnot available
GenreEmpirical

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

Quick stats

Citations3
Published2025
Admission routes1
Has abstractyes

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