Crimean-Congo hemorrhagic fever (CCHF) is a life-threatening zoonotic viral infection with high mortality rates. This deadly disease is caused by a virus belonging to the Nairovirus genus and is primarily transmitted to humans through ticks of the Hyalomma genus. The clinical manifestations of CCHF include a variety of symptoms such as high fever, headache, muscle pain, nausea, vomiting, diarrhea, and in severe cases, a tendency for widespread bleeding. This review article aims to examine plasma agmatine levels in CCHF patients. Agmatine is a naturally occurring biogenic amine in biological systems, synthesized through the decarboxylation of arginine. Known for its neuroprotective, antioxidant, and anti-inflammatory properties, agmatine draws attention for its potential therapeutic role in various pathological conditions. Within this review, the potential pathophysiological and prognostic significance of changes in agmatine levels during CCHF infection is discussed. Additionally, the relationships between agmatine and other biomarkers in the current literature are examined. Studies suggest that plasma agmatine levels in CCHF patients may vary with disease progression and that this biomolecule could be used to predict the course of the disease.