ReviewClinical rheumatology2023
Adult-onset Still's disease complicated by macrophage activation syndrome during pregnancy: a case-based review.
Review in Clinical rheumatology, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.
What it found
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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.
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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.
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Who cites it
6 citing papers in PubMed, 7 citations in OpenAlex.
- Still's disease in pregnancy complicated by anakinra-related hepatotoxicity and rescued by intravenous immunoglobulin: a case-based review.Rheumatology international · 2026Review
- A stepwise diagnostic framework for recurrent unexplained fever in adults: an evidence-based review with focus on Latin America.Clinics (Sao Paulo, Brazil) · 2026Review
- Article
- [Musculoskeletal Complications in Rheumatic Diseases].Journal of the Korean Society of Radiology · 2026Review
- Macrophage activation syndrome-associated adult onset Still disease treatment: a scoping review of case reports and case series.Proceedings (Baylor University. Medical Center) · 2025Article
- Pregnancy-Associated Onset of Adult-Onset Still's Disease.AJP reports · 2024Review
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
2 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Adult-onset Still's disease is a rare, systemic inflammatory rheumatic disease characterized by recurrent fevers, arthritis, and an evanescent rash. One of the most serious hematologic derangements that can be seen with adult-onset Still's disease is macrophage activation syndrome. Macrophage activation syndrome is characterized by activation of lymphocytes, resulting in a cytokine storm and hemophagocytosis in the bone marrow, along with multi-organ failure. Adult-onset Still's disease with macrophage activation syndrome first presenting during pregnancy is exceptionally rare; here, we report two unique cases of such a presentation and review the pertinent literature. Both of our cases presented critically ill with end-organ failure, and responded to immunosuppression; fetal demise was present in one and an emergency caesarean section with a viable fetus was performed in the other patient. Maternal outcomes were favorable in both cases and both patients did well long-term with systemic therapy. Systemic immunosuppression, particularly anti-IL1 therapy, may be considered as treatment for this rare and life-threatening condition when presenting during pregnancy.
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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.