Hemophagocytic lymphohistiocytosis (HLH) is a rapidly progressing, life-threatening syndrome characterized by excessive immune activation, often presenting as a complex cytokine storm. This hyperactive immune response can lead to multi-organ failure and systemic damage, resulting in an extremely short survival period if left untreated. Over the past decades, although HLH has garnered increasing attention from researchers, there have been few advancements in its treatment. The cytokine storm plays a crucial role in the treatment of HLH. Investigating the detailed mechanisms behind cytokine storms offers insights into targeted therapeutic approaches, potentially aiding in early intervention and improving the clinical outcome of HLH patients. To date, there is only one targeted therapy, emapalumab targeting interferon-γ, that has gained approval for primary HLH. This review aims to summarize the current treatment advances, emerging targeted therapeutics and underlying mechanisms of HLH, highlighting its newly discovered targets potentially involved in cytokine storms, which are expected to drive the development of novel treatments and offer fresh perspectives for future studies. Besides, multi-targeted combination therapy may be essential for disease control, but further trials are required to determine the optimal treatment mode for HLH.
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http://dx.doi.org/10.1186/s13045-024-01621-x | DOI Listing |
Obstet Med
January 2025
Intensive Care and Obstetric Research Group (GRICIO), Universidad de Cartagena, Cartagena, Colombia.
Objective: This report details the complexities of diagnosing and treating rapid-onset multisystemic hemophagocytic lymphohistiocytosis (HLH) during pregnancy, as evidenced by a fatal case in early pregnancy with severe hematological and obstetric complications.
Case Presentation: A 20-year-old pregnant woman in her second pregnancy presented at 8 weeks of gestation with abdominal pain, fever, and rectal bleeding. Laboratory tests revealed leukopenia, thrombocytopenia, and anemia, leading to immediate transfusion and intensive care unit admission.
Front Pediatr
January 2025
Key Laboratory of Birth Defects and Related Diseases of Women and Children of Ministry of Education (MOE), West China Institute of Women and Children's Health, Key Laboratory of Development and Diseases of Women and Children of Sichuan Province, Department of Pediatrics, Department of Pediatric Cardiology, West China Second University Hospital, Sichuan University, Chengdu, Sichuan, China.
Background: Cardiovascular involvement is a rare but severe complication of Epstein-Barr virus (EBV) infections. Patients with chronic active EBV (CAEBV) are at increased risk of developing cardiovascular complications and have a poor prognosis. Here, we report the rare case of a pediatric patient with CAEBV and EBV- hemophagocytic lymphohistiocytosis (HLH) complicated with a giant coronary artery aneurysm (CAA) and thrombosis, a giant Valsalva sinus aneurysm, and ascending aorta dilation seven years after the disease onset.
View Article and Find Full Text PDFCell Mol Biol Lett
January 2025
University Cote d'Azur, Inserm, C3M, Nice, France.
Vacuolization of hematopoietic precursors cells is a common future of several otherwise non-related clinical settings such as VEXAS, Chediak-Higashi syndrome and Danon disease. Although these disorders have a priori nothing to do with one other from a clinical point of view, all share abnormal vacuolization in different cell types including cells of the erythroid/myeloid lineage that is likely the consequence of moderate to drastic dysfunctions in the ubiquitin proteasome system and/or the endo-lysosomal pathway. Indeed, the genes affected in these three diseases UBA1, LYST or LAMP2 are known to be direct or indirect regulators of lysosome trafficking and function and/or of different modes of autophagy.
View Article and Find Full Text PDFJ Immunol Methods
January 2025
Division of Bone Marrow Transplantation and Immune Deficiency, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, United States; Department of Pediatrics, University of Cincinnati, Cincinnati, OH, United States.
Hemophagocytic lymphohistiocytosis (HLH) is a rare clinical syndrome caused by severe systemic hyperinflammation. HLH can be rapidly fatal if unrecognized or inadequately treated. It is important that clinicians are able to utilize diagnostic testing to assess for HLH and determine the underlying causes including possible inborn errors of immunity (IEI).
View Article and Find Full Text PDFOncol Lett
March 2025
Department of Breast Medical Oncology, National Center for Global Health and Medicine, Tokyo 162-8655, Japan.
Recently, the anti-programmed cell death protein 1 antibody pembrolizumab, a type of immune checkpoint inhibitor (ICI), has been used in preoperative systemic chemotherapy for hormone receptor and human epidermal growth factor 2-negative breast cancer, also known as triple-negative breast cancer (TNBC). Chemotherapy with pembrolizumab has demonstrated clinical activity in terms of pathologic complete response and event-free survival. Despite their efficacy, the current understanding of the full spectrum of side effects associated with relatively new ICIs remains incomplete.
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