Abstract
Purpose
Bone metastases (BM) and skeletal-related events (SRE) are frequent in patients with malignant pheochromocytoma and paraganglioma (PPM) and the best modality of prevention unknown. The role of interventional radiology (IR) techniques for the prevention of SRE in the multidisciplinary management of malignant PPM has not been evaluated in that setting.
Methods
Single referral center retrospective review of all patients with malignant PPM with BM from 2000 to 2016. The primary endpoint was the time to first serious SRE (TTSRE). At time of inclusion, patients with high bone tumor burden disease were defined as those having more than five BM with the biggest exceeding 2 cm (Group A) and patients with moderate bone tumor burden disease were defined as those having five or less BM or no BM exceeding 2 cm (Group B).
Results
A total of 28 patients were included in this study. Thirteen were treated by IR techniques for prevention of first serious SRE. After a median follow-up of 48.2 months, the median TTSRE was not reached in patients treated by IR techniques and was 26.0 months in patients without IR procedures (p = .058). When comparing patients in group B, TTSRE was significantly higher in patients treated by IR (10 patients) when compared to patients without IR procedures (12 patients) (p = .021).
Conclusions
IR techniques may help to delay the occurrence of first serious SRE in patients with malignant PPM with moderate bone tumor burden disease. Prospective studies are expected to confirm these results.




Similar content being viewed by others
Abbreviations
- BM:
-
Bone metastasis
- SRE:
-
Skeletal-related event
- PPM:
-
Paragangliomas and pheochromocytomas
- IR:
-
Interventional radiology
- TTSRE:
-
Time to first serious SRE
- FDG:
-
18F-fluorodeoxyglucose
- PET:
-
Positron emission tomography
- CT:
-
Computed tomography
- DFI:
-
Disease-free interval
- LFU:
-
Last follow-up
- MIBG:
-
Metaiodobenzylguanidine
- PV:
-
Percutaneous vertebroplasty
- PO:
-
Percutaneous osteosynthesis
References
H. Chen, R.S. Sippel, M.S. O’Dorisio, A.I. Vinik, R.V. Lloyd, K. Pacak, and North American Neuroendocrine Tumor Society (NANETS), The North American Neuroendocrine Tumor Society consensus guideline for the diagnosis and management of neuroendocrine tumors: pheochromocytoma, paraganglioma, and medullary thyroid cancer. Pancreas 39, 775 (2010)
L. Amar, M. Fassnacht, A.-P. Gimenez-Roqueplo, A. Januszewicz, A. Prejbisz, H. Timmers, P.-F. Plouin, Long-term postoperative follow-up in patients with apparently benign pheochromocytoma and paraganglioma. Horm. Metab. Res. 44, 385–9 (2012)
G. Eisenhofer, S.R. Bornstein, F.M. Brouwers, N.-K.V. Cheung, P.L. Dahia, R.R. de Krijger, T.J. Giordano, L.A. Greene, D.S. Goldstein, H. Lehnert, W.M. Manger, J.M. Maris, H.P.H. Neumann, K. Pacak, B.L. Shulkin, D.I. Smith, A.S. Tischler, W.F. Young, Malignant pheochromocytoma: current status and initiatives for future progress. Endocr. Relat. Cancer 11, 423 (2004)
L. Amar, E. Baudin, N. Burnichon, S. Peyrard, S. Silvera, J. Bertherat, X. Bertagna, M. Schlumberger, X. Jeunemaitre, A.-P. Gimenez-Roqueplo, P.-F. Plouin, Succinate dehydrogenase B gene mutations predict survival in patients with malignant pheochromocytomas or paragangliomas. J. Clin. Endocrinol. Metab. 92, 3822 (2007)
M. Ayala-Ramirez, J.L. Palmer, M.-C. Hofmann, M. de la Cruz, B.S. Moon, S.G. Waguespack, M.A. Habra, C. Jimenez, Bone metastases and skeletal-related events in patients with malignant pheochromocytoma and sympathetic paraganglioma. J. Clin. Endocrinol. Metab. 98, 1492 (2013)
H.J.L.M. Timmers, C.C. Chen, J.A. Carrasquillo, M. Whatley, A. Ling, G. Eisenhofer, K.S. King, J.U. Rao, R.A. Wesley, K.T. Adams, K. Pacak, Staging and functional characterization of pheochromocytoma and paraganglioma by 18F-fluorodeoxyglucose (18F-FDG) positron emission tomography. J. Natl. Cancer Inst. 104, 700 (2012)
S. Hescot, S. Leboulleux, L. Amar, D. Vezzosi, I. Borget, C. Bournaud-Salinas, C. de la Fouchardiere, R. Libé, C. Do Cao, P. Niccoli, A. Tabarin, I. Raingeard, C. Chougnet, S. Giraud, A.-P. Gimenez-Roqueplo, J. Young, F. Borson-Chazot, J. Bertherat, J.-L. Wemeau, X. Bertagna, P.-F. Plouin, M. Schlumberger, E. Baudin, and French group of Endocrine and Adrenal tumors (Groupe des Tumeurs Endocrines-REseau NAtional des Tumeurs ENdocrines and COrtico-MEdullo Tumeurs Endocrines networks), One-year progression-free survival of therapy-naive patients with malignant pheochromocytoma and paraganglioma. J. Clin. Endocrinol. Metab. 98, 4006 (2013)
J.H. Lee, F. Barich, L.H. Karnell, R.A. Robinson, W.K. Zhen, B.J. Gantz, H.T. Hoffman, American College of Surgeons Commission on Cancer, and American Cancer Society, National Cancer Data Base report on malignant paragangliomas of the head and neck. Cancer 94, 730 (2002)
F. Deschamps, G. Farouil, T. de Baere, Percutaneous ablation of bone tumors. Diagn. Interv. Imaging 95, 659 (2014)
F. Deschamps, T. de Baere, A. Hakime, E. Pearson, G. Farouil, C. Teriitehau, L. Tselikas, Percutaneous osteosynthesis in the pelvis in cancer patients. Eur. Radiol. 26, 1631 (2016)
F. Deschamps, T. de Baere, Cementoplasty of bone metastases. Diagn. Interv. Imaging 93, 685 (2012)
F. Deschamps, G. Farouil, N. Ternes, A. Gaudin, A. Hakime, L. Tselikas, C. Teriitehau, E. Baudin, A. Auperin, T. de Baere, Thermal ablation techniques: a curative treatment of bone metastases in selected patients? Eur. Radiol. 24, 1971 (2014)
U.S. Department of Health and Human Services. National Institutes of Health. (National Cancer Institute: Common Terminology Criteria for Adverse Events (CTCAE) Version 4. 03, 2010), https://evs.nci.nih.gov/ftp1/CTCAE/CTCAE_4.03_2010-06-14_QuickReference_5x7.pdf. Accessed 7 Sept 2017
Y. Kanda, Investigation of the freely available easy-to-use software ‘EZR’ for medical statistics. Bone Marrow Transplant. 48, 452 (2013)
K. Van Loon, L. Zhang, J. Keiser, C. Carrasco, K. Glass, M.-T. Ramirez, S. Bobiak, E.K. Nakakura, A.P. Venook, M.H. Shah, E.K. Bergsland, Bone metastases and skeletal-related events from neuroendocrine tumors. Endocr. Connect. 4, 9 (2015)
M.O. Baerlocher, W.E. Saad, S. Dariushnia, J.D. Barr, J.K. McGraw, B. Nikolic; and Society of Interventional Radiology Standards of Practice Committee, Quality improvement guidelines for percutaneous vertebroplasty. J. Vasc. Interv. Radiol. 25, 165 (2014)
A. Cotten, N. Boutry, B. Cortet, R. Assaker, X. Demondion, D. Leblond, P. Chastanet, B. Duquesnoy, H. Deramond, Percutaneous vertebroplasty: state of the art. Radiographics 18, 311 (1998)
D.A. Palma, J.K. Salama, S.S. Lo, S. Senan, T. Treasure, R. Govindan, R. Weichselbaum, The oligometastatic state—separating truth from wishful thinking. Nat. Rev. Clin. Oncol. 11, 549 (2014)
Z. Wang, D. Qiao, Y. Lu, D. Curtis, X. Wen, Y. Yao, H. Zhao, Systematic literature review and network meta-analysis comparing bone-targeted agents for the prevention of skeletal-related events in cancer patients with bone metastasis. Oncologist 20, 440 (2015)
O. Sartor, R. Coleman, S. Nilsson, D. Heinrich, S.I. Helle, J.M. O’Sullivan, S.D. Fosså, A. Chodacki, P. Wiechno, J. Logue, A. Widmark, D.C. Johannessen, P. Hoskin, N.D. James, A. Solberg, I. Syndikus, N.J. Vogelzang, C.G. O’Bryan-Tear, M. Shan, Ø.S. Bruland, C. Parker, Effect of radium-223 dichloride on symptomatic skeletal events in patients with castration-resistant prostate cancer and bone metastases: results from a phase 3, double-blind, randomised trial. Lancet Oncol. 15, 738 (2014).
Author information
Authors and Affiliations
Corresponding author
Ethics declarations
Conflict of interest
The authors declare that they have no conflict of interest.
Rights and permissions
About this article
Cite this article
Gravel, G., Leboulleux, S., Tselikas, L. et al. Prevention of serious skeletal-related events by interventional radiology techniques in patients with malignant paraganglioma and pheochromocytoma. Endocrine 59, 547–554 (2018). https://doi.org/10.1007/s12020-017-1515-y
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s12020-017-1515-y