Background: L-Asparaginase (L-Asp) may induce hypertriglyceridemia; however, this has been mainly observed among pediatric patients. Treatment for L-Asp-induced hypertriglyceridemia is not standardized, ranging from fasting and diet restriction to the invasive plasmapheresis procedure. Case Report: We describe a 53-year-old male patient who presented with L-Asp-induced severe hypertriglyceridemia. He was receiving L-Asp as part of his chemotherapy regimen for natural killer T-cell lymphoma. After the 20th dose, his serum triglyceride level was 3,552 mg/dl, with a total cholesterol of 418 mg/dl. Despite the high triglyceride, the patient did not present with acute pancreatitis symptoms. Treatment comprising fasting, fenofibrate, and omega-3 fatty acids was initiated. Triglyceride levels dropped rapidly to 1,000 mg/dl within 2 days, and to 268 mg/dl after 10 days. The chemotherapy regimen was subsequently switched to exclude L-Asp. Conclusion: L-Asp-induced severe hypertriglyceridemia may occur in adults and may be conservatively managed with fasting, fibrates, and omega-3 fatty acids. Plasmapheresis or continuous insulin infusion may be used for symptomatic patients with high triglyceride levels. Lipidlowering agents should be continued for patients previously treated for hyperlipidemia. Regular monitoring of lipid levels for patients receiving L-Asp is important, especially for those with a prior history of dyslipidemia. Re-challenge with L-Asp can be undertaken on an individual basis.

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