16 February 2023 : Case report
[In Press] Perioperative Management of Recurrent Hemophagocytic Syndrome in a Pregnant Woman: A Case Report
Challenging differential diagnosis, Management of emergency care, Patient complains / malpractice, Rare disease, Educational Purpose (only if useful for a systematic review or synthesis)Ayako Sumii1ABEF, Hirotsugu Miyoshi 1ADEF, Takahiro Kato 1BE, Sachiko Otsuki1ABE, Yousuke T. Horikawa12AE, Shiho Satomi1ABD, Noboru Saeki1BF, Yusuke Yoshida3EF, Yasuo M. Tsutsumi 1AE
Am J Case Rep In Press; DOI: 10.12659/AJCR.939369
Available online: 2023-02-16, In Press, Corrected Proof
Publication in the "In-Press" formula aims at speeding up the public availability of the pending manuscript while waiting for the final publication. The assigned DOI number is active and citable. The availability of the article in the Medline, PubMed and PMC databases as well as Web of Science will be obtained after the final publication according to the journal schedule
Hemophagocytic syndrome (HPS) is a rare syndrome characterized by abnormal activation of histiocytes and hemophagocytosis. We report the clinical management of recurrent HPS following 2 cesarean sections in the same patient.
A 33-year-old primiparous mother presented during her second trimester of pregnancy, and HPS was diagnosed based on pancytopenia, hyperferritinemia (13 170 ng/ml), and hemophagocytosis in bone marrow examination. Despite steroid therapy, her HPS did not improve. Following the delivery of a healthy premature infant, there was no improvement in HPS, and immunochemotherapy was started 4 days postoperatively. Thrombocytopenia and hyperferritinemia persisted but normalized over the next 2 months, and immunochemotherapy was discontinued after 6 months. About 1 year after chemotherapy, the patient became pregnant with her second child. At 35 weeks of gestation, recurrence of HPS was suspected, and a C-section was performed at 36 weeks of gestation. The surgery was complicated by placenta previa, and general anesthesia was initiated after successful delivery of the infant. Epidural anesthesia was not performed due to concerns for postoperative thrombocytopenia.
Interestingly, HPS was likely triggered twice by pregnancy in this patient. Although reports of HPS during pregnancy are rare, there have been reports of rapid deterioration and death. Early diagnosis and therapeutic intervention are essential.
Keywords: Cesarean Section; Lymphohistiocytosis, Hemophagocytic; Perioperative Medicine; Pregnancy
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