Tumor-induced osteomalacia: experience from three tertiary care centers in India.

FGF23 hypophosphatemia phosphaturic mesenchymal tumor tumor-induced osteomalacia

Journal

Endocrine connections
ISSN: 2049-3614
Titre abrégé: Endocr Connect
Pays: England
ID NLM: 101598413

Informations de publication

Date de publication:
Mar 2019
Historique:
received: 24 01 2019
accepted: 28 01 2019
pubmed: 7 2 2019
medline: 7 2 2019
entrez: 7 2 2019
Statut: ppublish

Résumé

Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome characterized by recalcitrant hypophosphatemia. Reports from the Indian subcontinent are scarce, with most being single center experiences involving few patients. Herein, we conducted a retrospective analysis of 30 patients of TIO diagnosed at three tertiary care hospitals in India. Patients with persistent hypophosphatemia (despite correction of hypovitaminosis D), normocalcemia, elevated alkaline phosphatase, low TmP/GFR and elevated or 'inappropriately normal' FGF23 levels were labeled as having TIO. They were sequentially subjected to functional followed by anatomical imaging. Patients with a well-localized tumor underwent excision; others were put on phosphorous and calcitriol supplementation. The mean age at presentation was 39.6 years with female:male ratio of 3:2. Bone pain (83.3%) and proximal myopathy (70%) were the chief complaints; 40% of cases had fractures. The mean delay in diagnosis was 3.8 years. Tumors were clinically detectable in four patients (13.3%). The mean serum phosphate was 0.50 mmol/L with a median serum FGF23 level of 518 RU/mL. Somatostatin receptor-based scintigraphy was found to be superior to FDG-PET in tumor localization. Lower extremities were the most common site of the tumor (72%). Tumor size was positively correlated with serum FGF23 levels. Twenty-two patients underwent tumor resection and 16 of them had phosphaturic mesenchymal tumors. Surgical excision led to cure in 72.7% of patients whereas disease persistence and disease recurrence were seen in 18.2% and 9.1% of cases, respectively. At the last follow-up, serum phosphate in the surgically treated group was significantly higher than in the medically managed group.

Identifiants

pubmed: 30726771
doi: 10.1530/EC-18-0552
pii: EC-18-0552.R1
pmc: PMC6410764
doi:
pii:

Types de publication

Journal Article

Langues

eng

Pagination

266-276

Références

Endocr Pract. 2011 Mar-Apr;17(2):177-84
pubmed: 20713341
Skeletal Radiol. 2015 Dec;44(12):1825-31
pubmed: 26341245
Endocrinol Diabetes Metab Case Rep. 2015;2015:150025
pubmed: 26155363
Bone Rep. 2017 Sep 20;7:90-97
pubmed: 29021995
Lancet. 1975 Aug 16;2(7929):309-10
pubmed: 50513
J Nucl Med. 2010 Mar;51(3):353-9
pubmed: 20150249
N Engl J Med. 2003 Apr 24;348(17):1656-63
pubmed: 12711740
J Bone Miner Res. 2012 Sep;27(9):1967-75
pubmed: 22532501
Clin Kidney J. 2014 Apr;7(2):186-9
pubmed: 25852869
Endokrynol Pol. 2018;69(2):205-210
pubmed: 29442350
J Clin Endocrinol Metab. 2010 Feb;95(2):578-85
pubmed: 19965919
J Clin Endocrinol Metab. 2012 Oct;97(10):3421-5
pubmed: 22837186
J Clin Invest. 2003 Sep;112(5):785-94
pubmed: 12952927
BMC Musculoskelet Disord. 2015 Feb 26;16:43
pubmed: 25879543
Am J Med. 1984 Sep;77(3):501-12
pubmed: 6548080
Ultrastruct Pathol. 1991 Jul-Oct;15(4-5):317-33
pubmed: 1755097
J Clin Endocrinol Metab. 2014 Feb;99(2):395-8
pubmed: 24297794
Pediatr Nephrol. 2008 Aug;23(8):1203-10
pubmed: 18288501
Nutrients. 2014 Feb 21;6(2):729-75
pubmed: 24566435
Ann Clin Biochem. 2014 Mar;51(Pt 2):203-27
pubmed: 24269946
Am J Surg Pathol. 2004 Jan;28(1):1-30
pubmed: 14707860
Clin Calcium. 2014 Aug;24(8):1217-22
pubmed: 25065874
N Engl J Med. 2001 Dec 27;345(26):1883-8
pubmed: 11756579
Clin Nucl Med. 2015 Jan;40(1):e6-e10
pubmed: 24999675
J Bone Miner Res. 2017 Aug;32(8):1667-1671
pubmed: 28459498
J Clin Endocrinol Metab. 2006 Jun;91(6):2055-61
pubmed: 16551733
Bone. 2004 Feb;34(2):303-19
pubmed: 14962809
Nat Rev Dis Primers. 2017 Jul 13;3:17044
pubmed: 28703220
J Clin Endocrinol Metab. 2005 Feb;90(2):1012-20
pubmed: 15562028
Indian J Anaesth. 2017 Jul;61(7):590-593
pubmed: 28794533
Pflugers Arch. 2007 Jul;454(4):615-23
pubmed: 17333246
Clin Endocrinol (Oxf). 2014 Jul;81(1):31-7
pubmed: 24528172
BMC Musculoskelet Disord. 2017 Feb 13;18(1):79
pubmed: 28193220
J Clin Endocrinol Metab. 2014 Sep;99(9):3049-54
pubmed: 24960541
Endocrinol Nutr. 2016 Apr;63(4):181-6
pubmed: 26718193
BMC Musculoskelet Disord. 2017 Sep 21;18(1):403
pubmed: 28934935
N Engl J Med. 2002 May 30;346(22):1748-9; author reply 1748-9
pubmed: 12037159
J Clin Endocrinol Metab. 2013 Nov;98(11):4273-8
pubmed: 23956343
Hong Kong Med J. 2014 Aug;20(4):350.e1-2
pubmed: 25104013
J Bone Miner Res. 2007 Jun;22(6):931-7
pubmed: 17352646
N Engl J Med. 2007 Jul 26;357(4):422-4
pubmed: 17652663
J Am Soc Nephrol. 2007 Jun;18(6):1637-47
pubmed: 17494882
Int J Radiat Oncol Biol Phys. 2003 Sep 1;57(1):158-65
pubmed: 12909228
Curr Rheumatol Rep. 2015 Jun;17(6):512
pubmed: 25900190
J Bone Miner Metab. 2017 May;35(3):298-307
pubmed: 27085966
J Clin Endocrinol Metab. 2016 Feb;101(2):390-3
pubmed: 26691889
Head Neck Pathol. 2016 Jun;10(2):192-200
pubmed: 26577211
J Bone Miner Res. 2006 Aug;21(8):1187-96
pubmed: 16869716
Mol Cell Endocrinol. 2011 Dec 5;347(1-2):17-24
pubmed: 21914460
J Clin Endocrinol Metab. 2013 Nov;98(11):4267-72
pubmed: 24014621
J Bone Miner Res. 2013 Jun;28(6):1386-98
pubmed: 23362135
J Clin Endocrinol Metab. 2016 Oct;101(10):3575-3581
pubmed: 27533306
Curr Opin Nephrol Hypertens. 2002 Jul;11(4):423-30
pubmed: 12105393
Presse Med. 2011 Mar;40(3):309-13
pubmed: 21131163
World J Surg Oncol. 2016 Jan 08;14(1):4
pubmed: 26744291
Endocr Relat Cancer. 2011 Jun 08;18(3):R53-77
pubmed: 21490240

Auteurs

Rimesh Pal (R)

Department of Endocrinology, Post Graduate Institute of Medical Education and Research, Chandigarh, India.

Sanjay Kumar Bhadada (SK)

Department of Endocrinology, Post Graduate Institute of Medical Education and Research, Chandigarh, India.

Awesh Singhare (A)

Department of Endocrinology, P D Hinduja Hospital and Medical Research Centre, Mumbai, India.

Anil Bhansali (A)

Department of Endocrinology, Post Graduate Institute of Medical Education and Research, Chandigarh, India.

Sadishkumar Kamalanathan (S)

Department of Endocrinology, Jawaharlal Institute of Post Graduate Medical Education and Research, Pondicherry, India.

Manoj Chadha (M)

Department of Endocrinology, P D Hinduja Hospital and Medical Research Centre, Mumbai, India.

Phulrenu Chauhan (P)

Department of Endocrinology, P D Hinduja Hospital and Medical Research Centre, Mumbai, India.

Ashwani Sood (A)

Department of Nuclear Medicine, Post Graduate Institute of Medical Education and Research, Chandigarh, India.

Vandana Dhiman (V)

Department of Endocrinology, Post Graduate Institute of Medical Education and Research, Chandigarh, India.

Dinesh Chandra Sharma (DC)

Division of Endocrinology, Rabindranath Tagore Medical College, Udaipur, India.

Uma Nahar Saikia (UN)

Department of Histopathology, Post Graduate Institute of Medical Education and Research, Chandigarh, India.

Debajyoti Chatterjee (D)

Department of Histopathology, Post Graduate Institute of Medical Education and Research, Chandigarh, India.

Vikas Agashe (V)

Department of Orthopaedics, P D Hinduja Hospital and Medical Research Centre, Mumbai, India.

Classifications MeSH