Adult onset Still's disease and pregnancy

Carolis, Sara De; Cianci, Francesco; Sordo, Gelsomina Del; Garofalo, Serafina; Garufi, Cristina; Lanzone, Antonio; Zoli, Angelo; Gremese, Elisa

doi:10.1016/j.autrev.2019.102356

Cited by 8 publications

(2 citation statements)

References 18 publications

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“…Our literature review reveals a 63.16% recurrence rate, with postpartum assessments showing 36.36% of patients experiencing flare-ups or condition deterioration, suggesting pregnancy may act as a catalyst for disease relapse, even in previously stable patients. This observation aligns with findings by De Carolis et al, 39 further affirming the relationship between pregnancy and AOSD flare-ups. Following childbirth, our patient resumed treatment with azathioprine and, fortunately, experienced an uneventful postpartum recovery.…”

Section: Discussionsupporting

confidence: 92%

Pregnancy-Associated Onset of Adult-Onset Still's Disease

Zayat,

Arora,

O'Brien

et al. 2024

AJP Rep

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Objective: This study aims to elucidate the clinical manifestations, diagnostic challenges, and management strategies of Adult-Onset Still’s Disease (AOSD) during pregnancy, leveraging a case series overview and a detailed case report from our center. Study Design: A comprehensive review of 21 published case reports on AOSD diagnosed during pregnancy was conducted, alongside a detailed case report of a patient diagnosed and managed at our center. This study emphasizes the importance of recognizing AOSD in pregnant patients, outlines the therapeutic challenges encountered, and discusses the potential complications arising from the disease and its treatment. Results: The onset of AOSD during pregnancy predominantly occurs in the first or second trimester, with a polycyclic disease course observed in the majority of cases. Management primarily involves corticosteroids and immunosuppressive medications. The case report highlights the complex interplay between AOSD, Hemophagocytic Lymphohistiocytosis (HLH), and pregnancy, illustrating a multidisciplinary approach to management that ensured favorable maternal and fetal outcomes despite the significant challenges. Conclusion: AOSD presents unique diagnostic and therapeutic challenges during pregnancy, requiring careful consideration of maternal and fetal health. Early diagnosis, a multidisciplinary approach to care, and judicious use of immunosuppressive therapy are critical for managing AOSD flares and associate

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Section: Discussionsupporting

confidence: 92%

Pregnancy-Associated Onset of Adult-Onset Still's Disease

Zayat,

Arora,

O'Brien

et al. 2024

AJP Rep

View full text Add to dashboard Cite

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“…In addition, cases of AOSD in which the onset occurs during the perinatal period are rare. A summary of reported cases of AOSD from 1971 onwards in which the onset occurred during the perinatal period is shown in Table (13,14,(17)(18)(19)(20)(21)(22)(23)(24)(25)(26)(27)(28)(29)(30). Of the 22 patients, including the present patient, 20 (90.9%) were treated with corticosteroids.…”

Section: Discussionmentioning

confidence: 92%

Adult-onset Still's Disease during Pregnancy Treated with Tocilizumab

et al. 2022

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A 28-year-old woman exhibited a spiking fever, arthritis, and liver disfunction when she was 22 weeks pregnant. She was diagnosed with adult-onset Still's disease (AOSD). As her condition was resistant to corticosteroid therapy, tocilizumab (TCZ) was selected. The TCZ treatment was effective, and she delivered a healthy child while receiving TCZ treatment. Cases in which AOSD first arises during pregnancy are rare, and there have been no reports of TCZ treatment for AOSD being initiated during pregnancy. Although the safety of TCZ treatment during pregnancy has not been established, it may be effective against severe AOSD that develops during pregnancy.

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

Wise

Zell

2023

Clin Rheumatol

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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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Adult onset Still's disease and pregnancy

Cited by 8 publications

References 18 publications

Pregnancy-Associated Onset of Adult-Onset Still's Disease

Pregnancy-Associated Onset of Adult-Onset Still's Disease

Adult-onset Still's Disease during Pregnancy Treated with Tocilizumab

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

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