Regression of fibrous dysplasia in response to denosumab therapy: A report of two cases

被引：10
作者：
Meier, Maartje E. [1 ]
van der Bruggen, Wouter [2 ,3 ]
van de Sande, Michiel A. J. [1 ]
Appelman-Dijkstra, Natasha M. [4 ]
机构：
[1] Leiden Univ, Med Ctr, Ctr Bone Qual, Dept Orthoped Surg, Albinusdreef 2, NL-2333 ZA Leiden, Netherlands
[2] Leiden Univ, Med Ctr, Dept Radiol, Nucl Med Sect, Albinusdreef 2, NL-2333 ZA Leiden, Netherlands
[3] Slingeland Hosp, Dept Nucl Med, Kruisbergseweg 25, NL-7009 BL Doetinchem, Netherlands
[4] Leiden Univ, Med Ctr, Ctr Bone Qual, Dept Internal Med,Div Endocrinol, Albinusdreef 2, NL-2333 ZA Leiden, Netherlands
来源：
BONE REPORTS | 2021年 / 14卷
关键词：
Fibrous dysplasia; McCune Albright syndrome; Denosumab; Antiresorptive; Nuclear imaging; MCCUNE-ALBRIGHT SYNDROME; INTRAVENOUS PAMIDRONATE; BONE; SCANS; TUMOR;
D O I：
10.1016/j.bonr.2021.101058
中图分类号：
R5 [内科学];
学科分类号：
1002 ; 100201 ;
摘要：
We present two patients with fibrous dysplasia who showed a decrease in lesional size and activity after denosumab therapy. Both patients also experienced a reduction in pain and bone turnover markers, which had not been accomplished during previous bisphosphonate therapy. These cases highlight the potential of denosumab to decrease lesional size in fibrous dysplasia. This finding has been reported in mice, but not in humans. Denosumab may be considered when bisphosphonates are not tolerated or not effective (enough), or in severe cases as neoadjuvant therapy to improve surgical possibilities and outcome. In addition, these results show that Na[F-18]F PET-CT is suitable for detecting change in each fibrous dysplasia lesion distinctively.
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页数：7
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共 20 条
  • [1] Fibrous Dysplasia/McCune-Albright Syndrome: A Rare, Mosaic Disease of Gαs Activation
    Boyce, Alison M.
    Collins, Michael T.
    [J]. ENDOCRINE REVIEWS, 2020, 41 (02) : 345 - 370
  • [2] Denosumab treatment for fibrous dysplasia
    Boyce, Alison M.
    Chong, William H.
    Yao, Jack
    Gafni, Rachel I.
    Kelly, Marilyn H.
    Chamberlain, Christine E.
    Bassim, Carol
    Cherman, Natasha
    Ellsworth, Michelle
    Kasa-Vubu, Josephine Z.
    Farley, Frances A.
    Molinolo, Alfredo A.
    Bhattacharyya, Nisan
    Collins, Michael T.
    [J]. JOURNAL OF BONE AND MINERAL RESEARCH, 2012, 27 (07) : 1462 - 1470
  • [3] Long-term effects of intravenous pamidronate in fibrous dysplasia of bone
    Chapurlat, RD
    Delmas, PD
    Liens, D
    Meunier, PJ
    [J]. JOURNAL OF BONE AND MINERAL RESEARCH, 1997, 12 (10) : 1746 - 1752
  • [4] Treatment of fibrous dysplasia of bone with intravenous pamidronate: long-term effectiveness and evaluation of predictors of response to treatment
    Chapurlat, RD
    Hugueny, P
    Delmas, PD
    Meunier, PJ
    [J]. BONE, 2004, 35 (01) : 235 - 242
  • [5] Safety and efficacy of denosumab for adults and skeletally mature adolescents with giant cell tumour of bone: interim analysis of an open-label, parallel-group, phase 2 study
    Chawla, Sant
    Henshaw, Robert
    Seeger, Leanne
    Choy, Edwin
    Blay, Jean-Yves
    Ferrari, Stefano
    Kroep, Judith
    Grimer, Robert
    Reichardt, Peter
    Rutkowski, Piotr
    Schuetze, Scott
    Skubitz, Keith
    Staddon, Arthur
    Thomas, David
    Qian, Yi
    Jacobs, Ira
    [J]. LANCET ONCOLOGY, 2013, 14 (09) : 901 - 908
  • [6] An instrument to measure skeletal burden and predict functional outcome in fibrous dysplasia of bone
    Collins, MT
    Kushner, H
    Reynolds, JC
    Chebli, C
    Kelly, MH
    Gupta, A
    Brillante, B
    Leet, AI
    Riminucci, M
    Robey, PG
    Bianco, P
    Wientroub, S
    Chen, CC
    [J]. JOURNAL OF BONE AND MINERAL RESEARCH, 2005, 20 (02) : 219 - 226
  • [7] Renal phosphate wasting in fibrous dysplasia of bone is part of a generalized renal tubular dysfunction similar to that seen in tumor-induced osteomalacia
    Collins, MT
    Chebli, C
    Jones, J
    Kushner, H
    Consugar, M
    Rinaldo, P
    Wientroub, S
    Bianco, P
    Robey, PG
    [J]. JOURNAL OF BONE AND MINERAL RESEARCH, 2001, 16 (05) : 806 - 813
  • [8] Activation of RANK/RANKL/OPG Pathway Is Involved in the Pathophysiology of Fibrous Dysplasia and Associated With Disease Burden
    de Castro, Luis F.
    Burke, Andrea B.
    Wang, Howard D.
    Tsai, Jeffrey
    Florenzano, Pablo
    Pan, Kristen S.
    Bhattacharyya, Nisan
    Boyce, Alison M.
    Gafni, Rachel I.
    Molinolo, Alfredo A.
    Robey, Pamela G.
    Collins, Michael T.
    [J]. JOURNAL OF BONE AND MINERAL RESEARCH, 2019, 34 (02) : 290 - 294
  • [9] Fibrous Dysplasia of Bone and McCune-Albright Syndrome: A Bench to Bedside Review
    Hartley, Iris
    Zhadina, Maria
    Collins, Micheal T.
    Boyce, Alison M.
    [J]. CALCIFIED TISSUE INTERNATIONAL, 2019, 104 (05) : 517 - 529
  • [10] Best practice management guidelines for fibrous dysplasia/McCune-Albright syndrome: a consensus statement from the FD/MAS international consortium
    Javaid, Muhammad Kassim
    Boyce, Alison
    Appelman-Dijkstra, Natasha
    Ong, Juling
    Defabianis, Patrizia
    Offiah, Amaka
    Arunde, Paul
    Shaw, Nick
    Dal Pos, Valter
    Underhil, Ann
    Portero, Deanna
    Heral, Lisa
    Heegaard, Anne-Marie
    Masi, Laura
    Monsell, Fergal
    Stanton, Robert
    Dijkstra, Pieter Durk Sander
    Brandi, Maria Luisa
    Chapurlat, Roland
    Hamdy, Neveen Agnes Therese
    Collins, Michael Terrence
    [J]. ORPHANET JOURNAL OF RARE DISEASES, 2019, 14 (1)
12