Ludwig’s angina is a rapidly spreading soft-tissue infection and commonly occurs following odontogenic infection. A 30-year-old male presented to the emergency department, 7 days after the extraction of molar teeth with a sudden onset of mandibular swelling. He was diagnosed with Ludwig’s angina with empyema thoracis and external carotid artery (ECA) pseudoaneurysm. He was successfully managed with video-assisted thoracoscopic surgery-guided drainage and endovascular embolization of ECA pseudoaneurysm. We share our experience of challenges faced during the management of unusual presentation of complicated Ludwig’s angina.

Hemophagocytic lymphohistiocytosis (HLH) is a hyperinflammatory syndrome caused by macrophages and cytotoxic T cells with aberrant activation. The primary (genetic) form, which is caused by mutations that affect lymphocyte cytotoxicity and immune regulation, is most prevalent in children, whereas the secondary (acquired) form is prevalent in adults. Secondary HLH is commonly caused by infections or cancers, but it can also be caused by autoimmune disorders, in which case it is known as macrophage activation syndrome (MAS; or MAS-HLH). A 25-year-old female presented with a high-grade fever that lasted for two weeks. His laboratory results revealed pancytopenia, neutropenia, hypertriglyceridemia, hypofibrinogenemia, and hyperferritinemia. Based on the clinical presentation and laboratory findings, a provisional diagnosis of HLH has been made. A HLH protocol was utilized to treat the patient. During the course of hospitalization, systemic lupus erythematosus (SLE) was identified as the underlying cause. She improved dramatically after receiving an immunosuppressive regimen of etoposide, cyclosporine, and dexamethasone according to HLH protocol-2004 with individualized modifications. The clinician should be aware that HLH may be the initial manifestation of underlying SLE. Early diagnosis and aggressive, individualized treatment are the key to improving outcomes.

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