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To describe extreme hypercalcemia as the presenting feature of acute lymphoblastic leukemia in an 8-yr-old girl and the combined use of pamidronate and calcitonin for its treatment.Case report.Pediatric intensive care unit.An 8-yr-old girl with 20.0 mg/dL serum calcium (reference range, 8.8–10.4 mg/dL) and 2.66 mmol/L ionized calcium (reference range, 1.13–1.32 mmol/L).Intravenous pamidronate and subcutaneous calcitonin.Our patient presented with nausea, vomiting, lethargy, weight loss, fatigue, and weakness but, remarkably, did not exhibit electrocardiographic changes. Initial treatment with hydration at 8 mL·kg−1·hr−1 and furosemide was ineffective. A single dose of 1 mg/kg intravenous pamidronate given over 24 hrs complemented by three doses of 5 units/kg subcutaneous calcitonin over 36 hrs lowered serum calcium to a normal range within 3 days. Side effects noted were hypocalcemia, hypomagnesemia, and hypophosphatemia. They were most pronounced 7–9 days after treatment, stabilized with supplementation, and returned to acceptable ranges by 1 month without need for ongoing electrolyte supplements. A renal computed tomographic scan did not show nephrocalcinosis. The patient remained free from recurrence of hypercalcemia 6 wks after initiating chemotherapy for acute lymphoblastic leukemia.Extreme hypercalcemia can be a presenting feature of acute lymphoblastic leukemia, but it may not result in life-threatening organ dysfunction. Combined treatment with pamidronate and calcitonin should be considered for treating hypercalcemia that does not respond to conventional therapy with hydration and furosemide.