• Da Hyun Chung, Kyu-Yup Lee, Ji-Yoon Kim, and Da Jung Jung.
• Department of Otorhinolaryngology-Head and Neck Surgery, Kyungpook National University School of Medicine, Daegu, Korea.
• Medicine (Baltimore). 2024 Jun 21; 103 (25): e38616e38616.

IntroductionHemophagocytic lymphohistiocytosis (HLH) is a potentially life-threatening syndrome for which early recognition and treatment are essential for improving outcomes. HLH is characterized by uncontrolled immune activation leading to fever, cytopenias, hepatosplenomegaly, coagulation abnormalities, and elevated typical markers. This condition can be genetic or secondary, with the latter often triggered by infections. Here, we present a unique case of HLH secondary to acute otitis media (AOM), a common ear infection.Patient ConcernsWe describe a 4-year-old boy who initially presented with a high fever and otalgia, later diagnosed with bilateral AOM. Despite antibiotic treatment, his condition deteriorated.DiagnosisThe patient fulfilled diagnostic criteria for HLH.InterventionsAggressive treatment by using combination therapy with immunoglobulins, intravenous steroids (dexamethasone), cyclosporine, and etoposide was performed.OutcomesAfter 1 month of treatment, improvement in the otologic symptoms was observed, and hematological findings gradually improved and normalized.LessionsThe link between AOM and HLH may be associated with inflammatory responses and immunological mechanisms, highlighting the importance of considering HLH in severe infection cases. This case emphasizes the need for prompt diagnosis and management, especially in secondary HLH scenarios, to improve patient outcomes. It is imperative to be aware of the potential correlation between these 2 conditions, and healthcare professionals should consider the likelihood of HLH.Copyright © 2024 the Author(s). Published by Wolters Kluwer Health, Inc.

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