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Adult-onset Still's disease complicated by macrophage activation syndrome during pregnancy: a case-based review. | LitMetric

Adult-onset Still's disease complicated by macrophage activation syndrome during pregnancy: a case-based review.

Clin Rheumatol

Department of Internal Medicine, Keck School of Medicine, University of Southern California, Los Angeles, USA.

Published: November 2023

AI Article Synopsis

Article 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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Source
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC10587217PMC
http://dx.doi.org/10.1007/s10067-023-06664-yDOI Listing

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