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Record W4415726476 · doi:10.14740/jmc5126

Histoplasmosis-Associated Hemophagocytic Lymphohistiocytosis in the Setting of Granulomatosis With Polyangiitis

2025· article· en· W4415726476 on OpenAlexvenueno aff
Shamoona Naqvi, Jonathan Shakesprere, Grant Wojdyla, Nolan Holley, Cara Randall, Ion Prisneac, Danish Safi

Bibliographic record

VenueJournal of Medical Cases · 2025
Typearticle
Languageen
FieldMedicine
TopicAutoimmune and Inflammatory Disorders Research
Canadian institutionsnot available
Fundersnot available
KeywordsHemophagocytic lymphohistiocytosisPancytopeniaGranulomatosis with polyangiitisMicroscopic polyangiitisSepsisCytopeniaHemophagocytosisBone marrowOrgan dysfunctionHistiocytosis

Abstract

fetched live from OpenAlex

Hemophagocytic lymphohistiocytosis (HLH) is an aggressive and potentially life-threatening syndrome of aberrant immune hyperactivation characterized by excess cytokine release due to abnormal cytotoxic T-cell and macrophage activation. Secondary, or acquired, HLH occurs in the setting of underlying infection, malignancy, or autoimmune processes and often presents with systemic symptoms and multi-organ dysfunction that can initially be misattributed to infection, leading to a delay in diagnosis and management. Histoplasmosis-associated HLH is an infrequently described manifestation of secondary HLH that can occur in the setting of immunocompromised states. A 67-year-old woman with a history of granulomatosis with polyangiitis on active mycophenolate mofetil treatment initially presented with persistent flu-like symptoms in the setting of pancytopenia and elevated liver enzymes. Despite appropriate sepsis evaluation and extensive antimicrobial treatment, she remained persistently febrile and developed acute respiratory failure. Lab work revealed severely abnormal coagulation factors, hypofibrinogenemia, hyperferritinemia, and hypertriglyceridemia concerning for underlying hematologic disease process. Bone marrow biopsy obtained showed a hypercellular marrow with histiocytosis and hemophagocytic forms as well as intracellular narrow-based budding yeast, confirming an HLH diagnosis and suggestive of disseminated histoplasmosis. She was subsequently started on a high-dose steroid taper, intravenous immunoglobulin, and anti-fungal therapy with clinical improvement and stabilization of blood counts. Post-discharge follow-up has not involved repeat hospitalizations. Diagnosing HLH requires consideration of both clinical and laboratory criteria but there remains a lack of consensus use, especially in adult patients and those with associated autoimmune disease. Furthermore, the clinical presentation of HLH can overlap significantly with sepsis or other acute illnesses, thus delaying timely diagnosis and interventions for optimal patient outcomes. Our case emphasizes the importance of early consideration and comprehensive evaluation in this setting for HLH and secondary causes, including fungal etiologies. Additional clinical reporting of this rare presentation can increase clinician recognition and highlight potential diagnostic and treatment approaches. Subsequent close clinical observation with serial examinations and biochemical marker follow-up are needed to assess response and evaluate for changes in treatment approach.

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 machine prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.003
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Case report · Consensus signal: Case report
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.002
Threshold uncertainty score0.008

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.003
Meta-epidemiology (narrow)0.0010.001
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0010.001
Science and technology studies0.0020.001
Scholarly communication0.0010.001
Open science0.0010.001
Research integrity0.0020.002
Insufficient payload (model declined to judge)0.0020.001

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 head0.014
GPT teacher head0.305
Teacher spread0.291 · 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 source (direct Gemma or distilled Codex), not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designCase report
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

Citations0
Published2025
Admission routes1
Has abstractyes

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