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成人骨纤维发育不良停用地诺单抗后出现反弹性高钙血症:一例报告及临床警示

Rebound Hypercalcemia After Denosumab Cessation in Adult Fibrous Dysplasia: A Case Report and Clinical Alert.

作者信息

Liu Danni, Chen Jiayi, Chen Hongmei, Hu Wenxue, Lin Jinxin

机构信息

Department of Nephrology, Guangdong Provincial People's Hospital, Guangdong Academy of Medical Sciences, Guangdong Provincial Geriatrics Institute, Southern Medical University, Guangzhou, China.

Department of Endocrinology, Guangdong Provincial People's Hospital, Guangdong Academy of Medical Sciences, Guangdong Provincial Geriatrics Institute, Southern Medical University, Guangzhou, China.

出版信息

Case Rep Endocrinol. 2025 Sep 4;2025:4553039. doi: 10.1155/crie/4553039. eCollection 2025.

DOI:10.1155/crie/4553039
PMID:40949890
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12425620/
Abstract

Fibrous dysplasia (FD) is a rare congenital bone disease. Denosumab, a monoclonal antibody targeting nuclear factor kappa-B ligand (RANKL), suppresses osteoclast activity and exhibits therapeutic potential for FD. We present the case of an adult female patient diagnosed with FD who had undergone 7 treatment cycles of denosumab (120 mg/dose, sc.) with a cumulative dose of 840 mg. After discontinuing denosumab for 7 months, the patient experienced a crisis of rebound hypercalcemia. Although, rare reports of hypercalcemia induced by discontinuation of denosumab are primarily seen in adolescents. By reporting this case, we aim to alert clinicians to the risk of rebound hypercalcemia in adult patients with FD undergoing denosumab treatment.

摘要

骨纤维发育不良(FD)是一种罕见的先天性骨病。地诺单抗是一种靶向核因子κB配体(RANKL)的单克隆抗体,可抑制破骨细胞活性,并显示出对FD的治疗潜力。我们报告了一例成年女性患者,该患者被诊断为FD,接受了7个周期的地诺单抗治疗(120mg/剂量,皮下注射),累积剂量为840mg。在停用7个月后,患者经历了反弹性高钙血症危机。虽然,停用后诱导高钙血症的罕见报告主要见于青少年。通过报告这个病例,我们旨在提醒临床医生,接受地诺单抗治疗的成年FD患者存在反弹性高钙血症的风险。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/be45/12425620/77b9fd196007/CRIE2025-4553039.005.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/be45/12425620/ef94dde789be/CRIE2025-4553039.001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/be45/12425620/e3094462de9d/CRIE2025-4553039.002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/be45/12425620/b6f0251a6f79/CRIE2025-4553039.003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/be45/12425620/b1895f8ff993/CRIE2025-4553039.004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/be45/12425620/77b9fd196007/CRIE2025-4553039.005.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/be45/12425620/ef94dde789be/CRIE2025-4553039.001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/be45/12425620/e3094462de9d/CRIE2025-4553039.002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/be45/12425620/b6f0251a6f79/CRIE2025-4553039.003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/be45/12425620/b1895f8ff993/CRIE2025-4553039.004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/be45/12425620/77b9fd196007/CRIE2025-4553039.005.jpg

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本文引用的文献

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Denosumab-Induced Rebound Hypercalcemia Treated With Bisphosphonates in a Pediatric Patient.双膦酸盐治疗小儿患者地诺单抗诱导的反弹性高钙血症
JCEM Case Rep. 2023 Oct 28;1(5):luad133. doi: 10.1210/jcemcr/luad133. eCollection 2023 Sep.
2
RANK-L inhibitor as a promising agent for refractory extensive craniofacial fibrous dysplasia: A case report.RANK-L 抑制剂治疗难治性广泛颅面骨纤维结构不良:一例报告。
Head Neck. 2024 Jan;46(1):E1-E5. doi: 10.1002/hed.27546. Epub 2023 Oct 12.
3
Denosumab use in bone fibrous dysplasia refractory to bisphosphonate: A retrospective multicentric study.
地舒单抗治疗对双膦酸盐难治性骨纤维结构不良的疗效:一项回顾性多中心研究。
Bone. 2023 Sep;174:116819. doi: 10.1016/j.bone.2023.116819. Epub 2023 Jun 8.
4
Safety and Efficacy of Denosumab for Fibrous Dysplasia of Bone.地诺单抗治疗骨纤维发育不良的安全性和有效性。
N Engl J Med. 2023 Feb 23;388(8):766-768. doi: 10.1056/NEJMc2214862.
5
Pharmacological Interventions Targeting Pain in Fibrous Dysplasia/McCune-Albright Syndrome.靶向纤维性骨发育不良/ McCune-Albright 综合征疼痛的药物干预。
Int J Mol Sci. 2023 Jan 29;24(3):2550. doi: 10.3390/ijms24032550.
6
Successful treatment with denosumab for pelvic fibrous dysplasia: A case report and review of the literature.成功使用地舒单抗治疗骨盆纤维结构不良:病例报告及文献复习。
Medicine (Baltimore). 2021 Dec 10;100(49):e28138. doi: 10.1097/MD.0000000000028138.
7
Hypercalcemia following discontinuation of denosumab therapy: A systematic review.地诺单抗治疗中断后的高钙血症:一项系统评价。
Bone Rep. 2021 Nov 13;15:101148. doi: 10.1016/j.bonr.2021.101148. eCollection 2021 Dec.
8
Mechanisms of acute hypercalcemia in pediatric patients following the interruption of Denosumab.地诺单抗中断治疗后儿科患者急性高钙血症的机制
J Endocrinol Invest. 2022 Jan;45(1):159-166. doi: 10.1007/s40618-021-01630-4. Epub 2021 Jul 3.
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Safety of therapy with and withdrawal from denosumab in fibrous dysplasia and McCune-Albright syndrome: an observational study.成骨不全症和 McCune-Albright 综合征中地舒单抗治疗和停药的安全性:一项观察性研究。
J Bone Miner Res. 2021 Sep;36(9):1729-1738. doi: 10.1002/jbmr.4380. Epub 2021 Jun 10.
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