Aneurysmal bone cyst: results of an off label treatment with Denosumab.
Adolescent
Adult
Bone Cysts, Aneurysmal
/ diagnostic imaging
Child
Denosumab
/ administration & dosage
Dose-Response Relationship, Drug
Drug Administration Schedule
Female
Humans
Hypercalcemia
/ chemically induced
Male
Off-Label Use
Recurrence
Retrospective Studies
Secondary Prevention
/ methods
Tomography, X-Ray Computed
Treatment Outcome
Young Adult
Aneurysmal bone cyst
Denosumab
Prognosis
Recurrence
Journal
BMC musculoskeletal disorders
ISSN: 1471-2474
Titre abrégé: BMC Musculoskelet Disord
Pays: England
ID NLM: 100968565
Informations de publication
Date de publication:
20 Oct 2019
20 Oct 2019
Historique:
received:
13
05
2019
accepted:
27
09
2019
entrez:
22
10
2019
pubmed:
22
10
2019
medline:
25
2
2020
Statut:
epublish
Résumé
The treatment of aneurysmal bone cysts (ABCs) has evolved and less invasive methods have been tried. Denosumab is a monoclonal antibody which inhibits osteoclasts. It has been shown to be effective in giant cell tumour of bone (GCT) of bone and hence promises some effect also in ABC. We report on 6 patients treated with Denosumab and compare our results to the cases already published. Data of 6 patients with ABCs and patients whose treatment included Denosumab were retrospectively analyzed. Denosumab was used at a dose of 120 mg on days 1, 8, 15 and 29, and every 4 weeks thereafter. In some of these patients the dose was reduced at the end of the treatment. Clinical and radiological responses were evaluated. In 4 female and 2 male patients with a mean age of 17 years (range: 6-30 years) the lesions were located in the sacrum (2), in distal radius, distal femur, talus and pelvis. One of the sacral lesions healed after 12 months and has stayed stable for 3 years since. The second patient received 2 years of therapy with recalcification, but recurred 1 year later and is under renewed therapy. The pelvic lesion improved but recurred. This patient has a 13-years history of intermittent therapy including surgery, two pregnancies and remains in a stable situation. The lesion of the talus did not improve with Denosumab after surgery and was complicated by destruction of the ankle joint with osteoarthritis. Recurrent lesions of the distal femur and the distal radius, previously treated by curettage and bone grafting healed under Denosumab and have remained stable for 2 and 3 years, respectively. One case of severe hypercalcemia was observed in a 7-year old child 6 months after discontinuation of Denosumab. Denosumab provides a treatment option for ABCs in anatomically critical locations. Adjuvant application might reduce the rate of local recurrence. In young patients, severe rebound hypercalcemia months after discontinuation of Denosumab may occur.
Sections du résumé
BACKGROUND
BACKGROUND
The treatment of aneurysmal bone cysts (ABCs) has evolved and less invasive methods have been tried. Denosumab is a monoclonal antibody which inhibits osteoclasts. It has been shown to be effective in giant cell tumour of bone (GCT) of bone and hence promises some effect also in ABC. We report on 6 patients treated with Denosumab and compare our results to the cases already published.
METHODS
METHODS
Data of 6 patients with ABCs and patients whose treatment included Denosumab were retrospectively analyzed. Denosumab was used at a dose of 120 mg on days 1, 8, 15 and 29, and every 4 weeks thereafter. In some of these patients the dose was reduced at the end of the treatment. Clinical and radiological responses were evaluated.
RESULTS
RESULTS
In 4 female and 2 male patients with a mean age of 17 years (range: 6-30 years) the lesions were located in the sacrum (2), in distal radius, distal femur, talus and pelvis. One of the sacral lesions healed after 12 months and has stayed stable for 3 years since. The second patient received 2 years of therapy with recalcification, but recurred 1 year later and is under renewed therapy. The pelvic lesion improved but recurred. This patient has a 13-years history of intermittent therapy including surgery, two pregnancies and remains in a stable situation. The lesion of the talus did not improve with Denosumab after surgery and was complicated by destruction of the ankle joint with osteoarthritis. Recurrent lesions of the distal femur and the distal radius, previously treated by curettage and bone grafting healed under Denosumab and have remained stable for 2 and 3 years, respectively. One case of severe hypercalcemia was observed in a 7-year old child 6 months after discontinuation of Denosumab.
CONCLUSION
CONCLUSIONS
Denosumab provides a treatment option for ABCs in anatomically critical locations. Adjuvant application might reduce the rate of local recurrence. In young patients, severe rebound hypercalcemia months after discontinuation of Denosumab may occur.
Identifiants
pubmed: 31630689
doi: 10.1186/s12891-019-2855-y
pii: 10.1186/s12891-019-2855-y
pmc: PMC6802343
doi:
Substances chimiques
Denosumab
4EQZ6YO2HI
Types de publication
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
456Références
Eur Rev Med Pharmacol Sci. 2016 Sep;20(17):3692-5
pubmed: 27649673
J Bone Miner Res. 2004 Jul;19(7):1059-66
pubmed: 15176987
Spine (Phila Pa 1976). 2015 Nov;40(22):E1201-4
pubmed: 26730528
Spine (Phila Pa 1976). 2017 Aug 1;42(15):1130-1138
pubmed: 28009753
Orthopedics. 2017 Jul 1;40(4):204-210
pubmed: 28732103
Eur Spine J. 2018 Jul;27(Suppl 3):494-500
pubmed: 29470716
Oncogene. 2010 Jun 24;29(25):3619-29
pubmed: 20418905
Mod Pathol. 2000 Nov;13(11):1206-10
pubmed: 11106078
Bone. 2019 Mar;120:482-486
pubmed: 30572144
World J Surg Oncol. 2014 Jan 20;12:17
pubmed: 24438319
Curr Opin Oncol. 2012 Jul;24(4):397-403
pubmed: 22581354
AJR Am J Roentgenol. 1995 Mar;164(3):573-80
pubmed: 7863874
J Musculoskelet Neuronal Interact. 2012 Sep;12(3):183-8
pubmed: 22947550
Cancer. 1992 Jun 15;69(12):2921-31
pubmed: 1591685
J Bone Oncol. 2019 Aug 06;18:100255
pubmed: 31463187
Radiology. 2004 Aug;232(2):522-6
pubmed: 15215542
J Pediatr Orthop. 2016 Mar;36(2):205-12
pubmed: 25985367
Orthop Traumatol Surg Res. 2016 Apr;102(2):213-6
pubmed: 26874446
Arch Orthop Trauma Surg. 2019 Oct;139(10):1339-1349
pubmed: 30877429
Clin Orthop Relat Res. 2014 Jun;472(6):1901-10
pubmed: 24532435
Transl Res. 2014 Aug;164(2):139-48
pubmed: 24726460
Genes Chromosomes Cancer. 1999 Nov;26(3):265-6
pubmed: 10502326
Clin Orthop Relat Res. 1973 Nov-Dec;(97):52-63
pubmed: 4590226
Tumour Biol. 2015 Feb;36(2):495-501
pubmed: 25618600
Pediatr Blood Cancer. 2018 Feb;65(2):null
pubmed: 29068565
Cancer. 1988 Jun 1;61(11):2291-304
pubmed: 2835141
Tumori. 2018 Oct;104(5):344-351
pubmed: 30086700
Eur Spine J. 2013 Jun;22(6):1417-22
pubmed: 23455951
J Bone Miner Res. 2012 Jul;27(7):1462-70
pubmed: 22431375
Genes Chromosomes Cancer. 2019 Feb;58(2):88-99
pubmed: 30582658
Spine (Phila Pa 1976). 2016 Jun;41(11):E654-60
pubmed: 27244112
Int Orthop. 2017 Aug;41(8):1685-1692
pubmed: 27933423
Head Neck. 2018 Dec;40(12):E107-E113
pubmed: 30475414
Curr Rev Musculoskelet Med. 2016 Dec;9(4):435-444
pubmed: 27778155
Curr Osteoporos Rep. 2017 Aug;15(4):283-292
pubmed: 28643220
Pediatr Blood Cancer. 2015 Jun;62(6):1078-80
pubmed: 25556556
Case Rep Orthop. 2017;2017:9125493
pubmed: 29181213
Pathology. 2011 Jun;43(4):318-21
pubmed: 21532526
J Clin Endocrinol Metab. 2014 Mar;99(3):891-7
pubmed: 24423331
J Biol Chem. 2010 Nov 19;285(47):37111-20
pubmed: 20864534
Case Rep Oncol Med. 2018 Dec 9;2018:1574343
pubmed: 30631617
J Oral Pathol Med. 2017 May;46(5):321-326
pubmed: 27778394
Lancet Oncol. 2010 Mar;11(3):275-80
pubmed: 20149736
Clin Orthop Relat Res. 2010 Jun;468(6):1649-59
pubmed: 19851815