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Bangladesh is a Dengue-prone country. Dengue hemorrhagic fever (DHF) is a severe form of Dengue characterized by plasma leakage, thrombocytopenia, and hemoconcentration. Without prompt management, it can progress to complications like Dengue Shock Syndrome and Expanded Dengue Syndrome, and can lead to significant morbidity and mortality. We report the case of an 18-year-old male with a history of seizures who presented with DHF. Imaging revealed peri-cholecystic edema, mild ascites, and a right-sided pleural effusion, confirming plasma leakage. In addition, while examining the patient, we found hypomelanotic macules and a Shagreen patch along with a history of seizure and learning disorder that suggest the diagnosis of Tuberous Sclerosis Complex (TSC). However, the patient recovered completely from DHF without complications, with timely clinical attention and appropriate management. The patient was referred to a specialist for further management of TSC. This case highlights the rare coexistence of DHF with previously unrecognized TSC; however, such a coexistence is not being suggested. This case underscores the importance of thorough neurological and dermatological evaluation in patients presenting with acute Dengue infection. • This case report presents a unique and rare combination of an acute tropical infection, Dengue, and a rare genetic disorder, Tuberous Sclerosis. • This article will guide clinicians to early diagnose Tuberous Sclerosis Complex (TSC) based on the neurocutaneous signs and symptoms. • It will assist healthcare providers in identifying challenges associated with managing Dengue infections and in developing clinical vigilance for handling Dengue and Severe Dengue infections, particularly in patients with underlying comorbidities like TSC. • More case reports and data are needed to clarify the interaction between Dengue and genetic disorders.