Introduction: L-asparaginase and corticosteroids are widely used in the chemotherapy of acute lymphoblastic leukemia (ALL). This combination is known to cause several adverse effects one of which is hypertriglyceridemia. Although the condition is asymptomatic and reversible it can lead to serious complications.Case description: This case report of 2-years 6 months old Indian male baby who was on treatment for ALL on day 41 triglycerides levels were 3324mg/dL with no significant symptoms, it was well managed, and the triglyceride levels drop down to 117mg/dL without any complications and interruption in the ongoing treatment. Conclusion:Combination therapy of L-asparaginase and steroids cause hypertriglyceridemia through various mechanisms leading to various life-threatening complications. However, there are no standard guidelines available to treat drug induced hypertriglyceridemia. Frequent monitoring of serum levels may aid in early diagnosis, prevents complications.

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