Case Report
Copyright ©The Author(s) 2024. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Jun 6, 2024; 12(16): 2837-2841
Published online Jun 6, 2024. doi: 10.12998/wjcc.v12.i16.2837
Managing adult-onset Still's disease in pregnancy: A case report
Ji-Hyoun Kang
Ji-Hyoun Kang, Division of Rheumatology, Department of Internal Medicine, Chonnam National University Medical School & Hospital, Gwangju 61469, South Korea
Author contributions: Kang JH designed the research study, performed the research, and analyzed the data and wrote the manuscript; Kang JH has read and approved the final manuscript.
Informed consent statement: This report was prepared with the written consent of the patient.
Conflict-of-interest statement: All the authors report no relevant conflicts of interest for this article.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: https://creativecommons.org/licenses/by-nc/4.0/
Corresponding author: Ji-Hyoun Kang, MD, PhD, Assistant Professor, Division of Rheumatology, Department of Internal Medicine, Chonnam National University Medical School & Hospital, 42 Jebong-ro, Dong-gu, Gwangju 61469, South Korea. romi918@naver.com
Received: December 13, 2023
Revised: March 7, 2024
Accepted: April 10, 2024
Published online: June 6, 2024
Processing time: 167 Days and 21.6 Hours
Abstract
BACKGROUND

Adult-onset Still’s disease (AOSD) is a rare systemic inflammatory disorder characterized by fever, arthritis, skin rash, and systemic symptoms. The etiology of AOSD is unknown; however, it is thought to be related to immune dysregulation. Although a rare disease, AOSD can significantly impact reproductive health, particularly during pregnancy. This case study assesses the implications of pregnancy in a patient with AOSD, as well as the potential for heredity of the disease. Neonatal hemophagocytic lympho-histiocytosis (HLH) is a rare and life-threatening disorder characterized by hyperinflammation and uncontrolled activation of immune cells, leading to multiple organ dysfunction. This case report aimed to introduce neonatal HLH from a mother with AOSD.

CASE SUMMARY

This case study presents a 29-year-old female with AOSD who became pregnant and gave birth to a premature infant who was diagnosed with neonatal HLH. AOSD can significantly impact pregnancy and childbirth, as it may become more severe during pregnancy, with an increased risk of fetal loss and preterm birth. The management of AOSD during pregnancy involves the use of nonsteroidal anti-inflammatory drugs and glucocorticoids, as well as immunosuppressive agents in severe cases. However, the use of immunosuppressive agents during pregnancy may be associated with potential risks to the fetus. The hereditary implications of AOSD are unclear; however, available evidence suggests that genetic factors may play a role in the disease development.

CONCLUSION

AOSD can have significant implications for pregnancy and childbirth, including an increased risk of fetal loss and preterm birth. Neonatal HLH, a complication of AOSD in pregnancy, requires prompt diagnosis and management. Women with AOSD who are considering pregnancy should discuss their options with their healthcare provider and develop a management plan that addresses the potential risks to both mother and fetus.

Keywords: Neonate, Hemophagocytic lympho-histiocytosis, Adult-onset Still’s disease, Pregnancy, Case report

Core Tip: This case study explores the intricate challenges faced by a 29-year-old woman with adult-onset Still’s disease (AOSD) during pregnancy, resulting in the premature birth of an infant diagnosed with neonatal hemophagocytic lympho-histiocytosis. Pregnancy exacerbates the risks associated with AOSD, including heightened chances of fetal loss and preterm birth. The management of AOSD during pregnancy involves utilizing nonsteroidal anti-inflammatory drugs, glucocorticoids, and, in severe cases, immunosuppressive agents despite potential fetal risks such as low birth weight and preterm birth, highlighting the delicate balance required in addressing the complexities of AOSD during gestation.