Nonremission and Recurrent Tumor-Induced Osteomalacia: A Retrospective Study.
PROGNOSIS
RECURRENT
REFRACTORY
SURGERY
TUMOR-INDUCED OSTEOMALACIA
Journal
Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research
ISSN: 1523-4681
Titre abrégé: J Bone Miner Res
Pays: United States
ID NLM: 8610640
Informations de publication
Date de publication:
03 2020
03 2020
Historique:
received:
16
08
2019
revised:
14
10
2019
accepted:
17
10
2019
pubmed:
24
10
2019
medline:
29
7
2021
entrez:
24
10
2019
Statut:
ppublish
Résumé
Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome. It is curable by excision of the causative tumor. However, a few cases may persist or relapse after tumor resection. We aimed to investigate the rate of these events and related factors. We retrospectively studied TIO patients treated with surgery in a tertiary hospital. TIO was established based on a pathologic examination or the reversion of hypophosphatemia. Refractory TIO patients consisted of those with nonremission or recurrent hypophosphatemia after surgery. A total of 230 patients were confirmed as having TIO. After primary surgery, 26 (11.3%) cases persisted, and 16 (7.0%) cases recurred. The overall refractory rate was 18.3%. The median time of recurrence was 33 months. Compared with patients in the recovery group, patients in the refractory group were more likely to be female (59.5% versus 41.0%, p = .029) and have a lower serum phosphate level (0.44 ± 0.13 versus 0.50 ± 0.11 mmol/L, p = .002). The refractory rate was lowest in head/neck tumors (7.5%) and highest in spine tumors (77.8%). Regarding the tissue involved of tumor location, the refractory rate was higher in tumors involving bone than tumors involving soft tissue (32.7% versus 7.0%, p < .001). The outcomes of malignant tumors were worse than those of benign tumors (p < .001): nonremission rate, 21.4% versus 9.7%; recurrence rate, 28.6% versus 6.5%. In the multivariate regression analysis, female sex, spine tumors, bone tissue-involved tumors, malignancy, and low preoperation serum phosphorus levels were identified as risk factors for refractory outcomes. High preoperative fibroblast growth factor 23 (FGF23) levels were also associated with refractory after adjusting for involving tissue and tumor malignancy. In summary, we are the first to report the rate and clinical characteristics of refractory TIO in a large cohort. For patients with multiple risk factors, especially spine tumors, clinical practitioners should be aware of a poor surgical prognosis. © 2019 The Authors. Journal of Bone and Mineral Research published by American Society for Bone and Mineral Research.
Identifiants
pubmed: 31643101
doi: 10.1002/jbmr.3903
pmc: PMC7140180
doi:
Substances chimiques
FGF23 protein, human
0
Fibroblast Growth Factors
62031-54-3
Fibroblast Growth Factor-23
7Q7P4S7RRE
Types de publication
Journal Article
Research Support, Non-U.S. Gov't
Langues
eng
Sous-ensembles de citation
IM
Pagination
469-477Informations de copyright
© 2019 The Authors. Journal of Bone and Mineral Research published by American Society for Bone and Mineral Research.
Références
BMC Musculoskelet Disord. 2015 Feb 26;16:43
pubmed: 25879543
Am J Surg Pathol. 2004 Jan;28(1):1-30
pubmed: 14707860
Endocr J. 2015;62(9):811-6
pubmed: 26135520
J Clin Endocrinol Metab. 2006 Jun;91(6):2055-61
pubmed: 16551733
Auris Nasus Larynx. 2018 Apr;45(2):377-383
pubmed: 28579442
Nat Rev Dis Primers. 2017 Jul 13;3:17044
pubmed: 28703220
Metabolism. 2020 Feb;103S:153865
pubmed: 30664852
J Clin Endocrinol Metab. 2019 Feb 1;104(2):258-265
pubmed: 30165559
Comput Assist Surg (Abingdon). 2017 Dec;22(1):14-19
pubmed: 28081649
Mod Pathol. 2019 Feb;32(2):189-204
pubmed: 30206408
Eur Arch Otorhinolaryngol. 2019 Jun;276(6):1677-1684
pubmed: 30879192
Endocr Connect. 2019 Mar;8(3):266-276
pubmed: 30726771
J Bone Miner Metab. 2017 May;35(3):298-307
pubmed: 27085966
Head Neck Pathol. 2016 Jun;10(2):192-200
pubmed: 26577211
J Orthop Sci. 2015 Jul;20(4):765-71
pubmed: 24504987
J Bone Miner Res. 2012 Sep;27(9):1967-75
pubmed: 22532501
Eur Ann Otorhinolaryngol Head Neck Dis. 2018 Oct;135(5):365-369
pubmed: 30026073
World J Surg Oncol. 2015 May 08;13:176
pubmed: 25951872
J Bone Miner Res. 2013 Jun;28(6):1386-98
pubmed: 23362135