Tumor-Induced Osteomalacia: A Systematic Clinical Review of 895 Cases.
FGF23
Fracture
Hypophosphatemia
Osteomalacia
Rickets
Tumor-induced osteomalacia
Journal
Calcified tissue international
ISSN: 1432-0827
Titre abrégé: Calcif Tissue Int
Pays: United States
ID NLM: 7905481
Informations de publication
Date de publication:
10 2022
10 2022
Historique:
received:
05
05
2022
accepted:
01
07
2022
pubmed:
21
7
2022
medline:
17
9
2022
entrez:
20
7
2022
Statut:
ppublish
Résumé
Tumor-induced osteomalacia (TIO) is a rare and largely underdiagnosed paraneoplastic condition. Previous reviews often reported incomplete data on clinical aspects, diagnosis or prognosis. The aim of this study was to present a systematic clinical review of all published cases of TIO. A search was conducted in Pubmed, Embase, Web of Science from inception until April 23rd, 2020. We selected case reports and case series of patients diagnosed with TIO, with information on tumor localization and serum phosphate concentration. Two reviewers independently extracted data on biochemical and clinical characteristics including bone involvement, tumor localization and treatment. 468 articles with 895 unique TIO cases were included. Median age was 46 years (range 9 months-90 years) and 58.3% were males. Hypophosphatemia and inappropriately low or normal 1,25-dihydroxyvitamin D levels, characteristic for TIO, were present in 98% of cases. Median tumor size was 2.7 cm (range 0.5 to 25.0 cm). Serum fibroblast growth factor 23 was related to tumor size (r = 0.344, P < 0.001). In 32% of the cases the tumor was detected by physical examination. Data on bone phenotype confirmed skeletal involvement: 62% of cases with BMD data had a T-score of the lumbar spine ≤ - 2.5 (n = 61/99) and a fracture was reported in at least 39% of all cases (n = 346/895). Diagnostic delay was longer than 2 years in more than 80% of cases. 10% were reported to be malignant at histology. In conclusion, TIO is a debilitating disease characterized by a long diagnostic delay leading to metabolic disturbances and skeletal impairment. Increasing awareness of TIO should decrease its diagnostic delay and the clinical consequences.
Identifiants
pubmed: 35857061
doi: 10.1007/s00223-022-01005-8
pii: 10.1007/s00223-022-01005-8
pmc: PMC9474374
doi:
Substances chimiques
Fibroblast Growth Factors
62031-54-3
Types de publication
Journal Article
Review
Langues
eng
Sous-ensembles de citation
IM
Pagination
367-379Informations de copyright
© 2022. The Author(s).
Références
N Engl J Med. 1994 Jun 9;330(23):1645-9
pubmed: 8177270
Am J Surg Pathol. 2004 Jan;28(1):1-30
pubmed: 14707860
Osteoporos Int. 2020 Jul;31(7):1383-1387
pubmed: 32185436
Endocr Connect. 2019 Oct;8(10):1330-1353
pubmed: 31505461
J Intern Med. 2010 Oct;268(4):390-4
pubmed: 20698924
Lancet. 1975 Aug 16;2(7929):309-10
pubmed: 50513
J Clin Pathol. 2009 Aug;62(8):760-1
pubmed: 19638551
Proc Natl Acad Sci U S A. 2001 May 22;98(11):6500-5
pubmed: 11344269
Endocr Connect. 2019 Mar;8(3):266-276
pubmed: 30726771
J Bone Miner Res. 2020 Mar;35(3):469-477
pubmed: 31643101
Bone. 2009 Oct;45(4):814-6
pubmed: 19555782
Cancer. 1987 Apr 15;59(8):1442-54
pubmed: 3545439
Joint Bone Spine. 2013 Dec;80(6):668-9
pubmed: 23583281
JBMR Plus. 2020 Dec 18;5(2):e10436
pubmed: 33615105
Am J Med. 1984 Sep;77(3):501-12
pubmed: 6548080
J Endocr Soc. 2021 Jun 02;5(9):bvab099
pubmed: 34286168
Calcif Tissue Int. 2021 Jan;108(1):128-142
pubmed: 32504138
Br Med J. 1955 May 28;1(4925):1311-3
pubmed: 14363873
Cancer. 1985 Apr 15;55(8):1691-705
pubmed: 3978561
Ann Clin Biochem. 2009 Mar;46(Pt 2):167-9
pubmed: 19151167
Indian J Pediatr. 1999 Jul-Aug;66(4):628-31
pubmed: 10798120
Intern Med. 2008;47(5):453-7
pubmed: 18310982
J Clin Invest. 2003 Sep;112(5):683-92
pubmed: 12952917
J Clin Endocrinol Metab. 2022 Jul 14;107(8):e3428-e3436
pubmed: 35468192
Br J Radiol. 2008 Apr;81(964):e110-4
pubmed: 18344267
Bone. 2008 Jun;42(6):1235-9
pubmed: 18396126
J Clin Endocrinol Metab. 2006 Jun;91(6):2055-61
pubmed: 16551733
Am J Med. 1987 Feb;82(2):333-8
pubmed: 3101497
Nat Rev Dis Primers. 2017 Jul 13;3:17044
pubmed: 28703220
J Clin Endocrinol Metab. 2005 Feb;90(2):1012-20
pubmed: 15562028
Diagnostics (Basel). 2019 Dec 18;10(1):
pubmed: 31861469
BMJ Case Rep. 2014 Nov 17;2014:
pubmed: 25406200
Rev Endocr Metab Disord. 2001 Apr;2(2):175-86
pubmed: 11705323
Rev Rhum Engl Ed. 1995 Jun;62(6):463-6
pubmed: 7552213
PLoS Med. 2021 Mar 29;18(3):e1003583
pubmed: 33780438
Joint Bone Spine. 2005 Oct;72(5):424-6
pubmed: 16112595
Kidney Int. 2006 Oct;70(8):1486-94
pubmed: 16941023
Endocr J. 2017 Jul 28;64(7):675-683
pubmed: 28450684
Q J Med. 1976 Jul;45(179):411-20
pubmed: 948543
Am J Med Sci. 1991 May;301(5):322-8
pubmed: 1902351
J Bone Miner Res. 1997 Sep;12(9):1502-11
pubmed: 9286768
JAMA. 2005 Sep 14;294(10):1260-7
pubmed: 16160135
Am J Surg Pathol. 1989 Jul;13(7):588-99
pubmed: 2544116
Calcif Tissue Int. 2009 Apr;84(4):313-23
pubmed: 19219382
J Bone Miner Res. 2012 Sep;27(9):1967-75
pubmed: 22532501
Clin Chem. 2011 Nov;57(11):1476-7
pubmed: 21914788
Pflugers Arch. 2006 Jan;451(4):579-87
pubmed: 16151791
J Pediatr. 1986 Dec;109(6):994-1000
pubmed: 3023599
Kidney Int. 2004 Jan;65(1):1-14
pubmed: 14675031
J Bone Miner Res. 2013 Jun;28(6):1386-98
pubmed: 23362135
Pediatr Radiol. 1976 Dec 15;5(2):120-2
pubmed: 189280
J Clin Endocrinol Metab. 2013 Mar;98(3):887-91
pubmed: 23393166
Eur J Nucl Med Mol Imaging. 2018 Sep;45(10):1710-1720
pubmed: 29532101
J Clin Endocrinol Metab. 2016 Oct;101(10):3575-3581
pubmed: 27533306
Endocr Relat Cancer. 2011 Jun 08;18(3):R53-77
pubmed: 21490240
J Bone Miner Res. 2021 Aug;36(8):1502-1509
pubmed: 33950560