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CC BY-NC-ND 4.0 · AJP Rep 2024; 14(02): e145-e155
DOI: 10.1055/a-2318-0305
Review Article

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

Nawras Zayat 
1   Division of Maternal-Fetal Medicine, Department of Obstetrics and Gynecology, Montefiore Medical Center, Albert Einstein College of Medicine, Bronx, New York
,
Avish Arora
1   Division of Maternal-Fetal Medicine, Department of Obstetrics and Gynecology, Montefiore Medical Center, Albert Einstein College of Medicine, Bronx, New York
,
Joselle O'Brien
3   Division of Maternal-Fetal Medicine, Department of Obstetrics and Gynecology, Maimonides Medical Center, Brooklyn, New York
,
Japjot Bal
1   Division of Maternal-Fetal Medicine, Department of Obstetrics and Gynecology, Montefiore Medical Center, Albert Einstein College of Medicine, Bronx, New York
,
Rebekah Sugarman
1   Division of Maternal-Fetal Medicine, Department of Obstetrics and Gynecology, Montefiore Medical Center, Albert Einstein College of Medicine, Bronx, New York
,
Leeshun Rivera
1   Division of Maternal-Fetal Medicine, Department of Obstetrics and Gynecology, Montefiore Medical Center, Albert Einstein College of Medicine, Bronx, New York
,
Amir Shamshirsaz
2   Division of Maternal-Fetal Medicine and Surgical Critical Care, Baylor College of Medicine, Houston, Texas
,
Kavita Vani
1   Division of Maternal-Fetal Medicine, Department of Obstetrics and Gynecology, Montefiore Medical Center, Albert Einstein College of Medicine, Bronx, New York
,
Dimitrios S. Mastrogiannis
1   Division of Maternal-Fetal Medicine, Department of Obstetrics and Gynecology, Montefiore Medical Center, Albert Einstein College of Medicine, Bronx, New York
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Abstract

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 most cases. Management primarily involves corticosteroids and immunosuppressive medications, balancing the disease control with potential pregnancy complications. The case report highlights the complex interplay between AOSD, hemophagocytic lymphohistiocytosis, 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 associated complications. Further research is necessary to optimize care for this rare condition in the context of pregnancy.

Keywords

adult-onset Still's disease - management of AOSD during pregnancy - rheumatology - disorders - Anakinra and pregnancy - hemophagocytic lymphohistiocytosis - pregnancy complications - macrophage activation syndrome

Source of Support

This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.


Consent Statement

Informed consent was obtained from the patient for publication of this article after the nature and possible consequences of the study had been fully explained.


Authors' Contribution

All authors have made substantial contributions to the conception, design, acquisition of data, and analysis and interpretation of the case report. Specifically, each author was involved in reviewing radiologic and pathological findings and synthesizing the article into a comprehensive report for academic dissemination. All authors contributed to drafting the manuscript and revising it critically for important intellectual content. They have given final approval of the version to be published and agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.


Data Availability

All relevant data are within the article and its supporting information files.




Publication History

Received: 15 March 2024

Accepted: 10 April 2024

Accepted Manuscript online:
02 May 2024

Article published online:
24 May 2024

© 2024. The Author(s). This is an open access article published by Thieme under the terms of the Creative Commons Attribution-NonDerivative-NonCommercial License, permitting copying and reproduction so long as the original work is given appropriate credit. Contents may not be used for commercial purposes, or adapted, remixed, transformed or built upon. (https://creativecommons.org/licenses/by-nc-nd/4.0/)

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