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Record W2325914097 · doi:10.3899/jrheum.141261

Dissecting the Heterogeneity of Macrophage Activation Syndrome Complicating Systemic Juvenile Idiopathic Arthritis

2015· article· en· W2325914097 on OpenAlexvenueno aff
Francesca Minoia, Sergio Davì, AnnaCarin Horne, Francesca Bovis, Erkan Demirkaya, Jonathan Akikusa, Nuray Aktay Ayaz, Sulaiman M. Al‐Mayouf, Patrizia Barone, Bianca Bica, Isabel Bolt, Luciana Breda, Carmen De Cunto, Sandra Enciso, Romina Gallizzi, Thomas A. Griffin, Teresa Hennon, Gerd Horneff, Michael Jeng, Ageza M. Kapovic, Jeffrey M. Lipton, Silvia Magni‐Manzoni, Ingrīda Rumba-Rozenfelde, Cláudia Saad Magalhães, Wafaa Mohamed Saad Sewairi, Kimo C. Stine, Olga Vougiouka, Lehn K. Weaver, Zane Dāvidsone, Maka Ioseliani, Bianca Lattanzi, Hasan Tezer, Antonella Buoncompagni, Paolo Picco, Nicolino Ruperto, Alberto Martini, Randy Q. Cron, Angelo Ravelli

Bibliographic record

VenueThe Journal of Rheumatology · 2015
Typearticle
Languageen
FieldMedicine
TopicAutoimmune and Inflammatory Disorders Research
Canadian institutionsnot available
Fundersnot available
KeywordsMedicineHemophagocytosisMacrophage activation syndromeEtoposideHepatosplenomegalyRheumatologyArthritisInternal medicineHemophagocytic lymphohistiocytosisPediatricsBone marrowChemotherapy

Abstract

fetched live from OpenAlex

OBJECTIVE: To seek insights into the heterogeneity of macrophage activation syndrome (MAS) complicating systemic juvenile idiopathic arthritis (sJIA) through the analysis of a large patient sample collected in a multinational survey. METHODS: International pediatric rheumatologists and hemato-oncologists entered their patient data, collected retrospectively, in a Web-based database. The demographic, clinical, laboratory, histopathologic, therapeutic, and outcome data were analyzed in relation to (1) geographic location of caring hospital, (2) subspecialty of attending physician, (3) demonstration of hemophagocytosis, and (4) severity of clinical course. RESULTS: A total of 362 patients were included by 95 investigators from 33 countries. Demographic, clinical, laboratory, and histopathologic features were comparable among patients seen in diverse geographic areas or by different pediatric specialists. Patients seen in North America were given biologics more frequently. Patients entered by pediatric hemato-oncologists were treated more commonly with biologics and etoposide, whereas patients seen by pediatric rheumatologists more frequently received cyclosporine. Patients with demonstration of hemophagocytosis had shorter duration of sJIA at MAS onset, higher prevalence of hepatosplenomegaly, lower levels of platelets and fibrinogen, and were more frequently administered cyclosporine, intravenous immunoglobulin (IVIG), and etoposide. Patients with severe course were older, had longer duration of sJIA at MAS onset, had more full-blown clinical picture, and were more commonly given cyclosporine, IVIG, and etoposide. CONCLUSION: The clinical spectrum of MAS is comparable across patients seen in different geographic settings or by diverse pediatric subspecialists. There was a disparity in the therapeutic choices among physicians that underscores the need to establish uniform therapeutic protocols.

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)0.003
metaresearch head score (Gemma)0.001
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.711
Threshold uncertainty score0.220

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0030.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.001
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 head0.035
GPT teacher head0.302
Teacher spread0.267 · 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 designObservational
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

Citations75
Published2015
Admission routes1
Has abstractyes

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