Denosumab
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Various toxicities following an off-label use: 4 case reports A retrospective case series involving four male paediatric patients aged 3–17 years developed joint pain, hypocalcaemia or hypercalcemia during off-label treatment with denosumab for Noonan syndrome-like multiple giant cell lesions (NL/MGCLs). Additionally, all patients developed a rebound effect in the form of hypercalcaemia following discontinuation of denosumab [not all outcomes stated]. Case 1: A 3-year-old male patient, who was diagnosed with NL/MGCLs, started receiving off-label treatment with SC denosumab 25mg every 4 weeks in April 2015. Subsequently, NL/MGCLs remarkably decreased. Hence, treatment with denosumab was stopped after eight doses in December 2015. Two months after discontinuation of denosumab, he refused to walk, and was diagnosed with hypercalcaemia. Hence, he had received treatment with furosemide, calcitonin, hyperhydration, and then pamidronate, which resulted in the resolution of hypercalcaemia. However, he again developed hypercalcemia after 2 weeks, which led to hospitalisation. He was treated with pamidronate, hyperhydration and calcitonin. Subsequently, his serum calcium level became normal, and he was discharged from hospital. After 2 years of stable disease, MRI revealed enlargement of NL/MGCLs. Hence, he started receiving SC denosumab 36mg monthly in April 2018, along with calcium supplementation. Follow-up MRI showed stabilisation of NL/MGCLs. Hence, denosumab was stopped in August 2019. However, he developed rebound hypercalcaemia. Hence, he was hospitalised and received treatment with pamidronate and hyperhydration. Case 2: A 17-year-old male, who was diagnosed with NL/MGCLs, had undergone two surgical resections. Due to recurrent NL/ MGCLs (in July 2016), he started receiving off-label treatment with SC denosumab injection on days 0, 7, 14, and 28 days, followed by every 4 weeks for 1 year. Concurrently, he also received elemental calcium. After 2 months of denosumab treatment, he developed symptomatic hypocalcemia in November 2016. Hence, he was treated with calcium and colecalciferol supplementation. However, because of recurrent hypocalcemia, he had received calcitriol, which resulted in the stabilisation of calcium level. After one year of denosumab treatment, dosage frequency was extended to every 3 months. However, he developed symptomatic hypercalcemia with elevated carboxy-terminal collagen telopeptide (CTX), indicative of the rebound hypercalcemia. He also had unbearable shoulder and knee pain, along with mild elbow pain. Subsequently, he received denosumab, and subsequently, his pain was resolved within 3 days. For discontinuation of denosumab without any rebound effect (2.5 months after the last of denosumab), he started receiving alendronic acid [alendronate]. Twenty-four weeks after the last dose of denosumab, his calcium level remained normal. Case 3: An 8-year-old male patient, who was diagnosed with NL/MGCLs, started receiving off-label treatment with SC denosumab 35mg every 4 weeks in May 2017. C
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