Tumour-induced osteomalacia due to an intra-abdominal mesenchymal tumour

A 50-year-man presented with debilitating lower-limb proximal muscle weakness and hip pain since 3 years. Investigations (serum calcium (8.9 mg/dL), serum phosphorus (1.5 mg/dL), serum albumin (40 g/L), parathyroid hormone (116 pg/mL (12.30 pmol/L)), 25(OH)D3 (25.2 ng/mL (63 nmol/L)) 1,25(OH)2 D3 (1...

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Bibliographic Details
Published in:BMJ case reports 2019-12, Vol.12 (12), p.e231728
Main Authors: Krishnappa, Brijesh, Jadhav, Swati Ramteke, Lila, Anurag R, Bandgar, Tushar R
Format: Article
Language:English
Subjects:
Abdomen
Abdominal Neoplasms - complications
Abdominal Neoplasms - diagnosis
Abdominal Neoplasms - diagnostic imaging
Abdominal Neoplasms - pathology
Bones
calcium and bone
Case reports
endocrine cancer
Fibroblasts
Growth factors
Humans
Localization
Male
Middle Aged
Neuroendocrine tumors
Osteomalacia - diagnostic imaging
Osteomalacia - etiology
Osteomalacia - pathology
Patients
Positron Emission Tomography Computed Tomography
Positron-Emission Tomography
radiology
Rare Disease
Soft Tissue Neoplasms - complications
Soft Tissue Neoplasms - diagnosis
Soft Tissue Neoplasms - diagnostic imaging
Soft Tissue Neoplasms - pathology
Surgery
Tomography
Tumors
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Summary:A 50-year-man presented with debilitating lower-limb proximal muscle weakness and hip pain since 3 years. Investigations (serum calcium (8.9 mg/dL), serum phosphorus (1.5 mg/dL), serum albumin (40 g/L), parathyroid hormone (116 pg/mL (12.30 pmol/L)), 25(OH)D3 (25.2 ng/mL (63 nmol/L)) 1,25(OH)2 D3 (19 pg/mL (45.60 pmol/L)), tubular reabsorption of phosphate of 0.22 and elevated serum fibroblast growth factor 23 (FGF23) (387.7 RU/mL)) were consistent with tumour-induced osteomalacia (TIO). Localisation studies (68Ga DOTATATE positron emission tomography (PET)/CT and 18FDG-PET/CT) did not reveal any lesion. Re-evaluation after 2 and 5 years with 68Ga-DOTANOC PET/CT showed 2×1.4 cm progressively increasing rounded soft tissue enhancing mass close to splenic hilum (SUV max: 26.4). Tumour was resected by laparotomy. Both FGF23 (120 RU/mL on day 3) and serum phosphorus (2.5 mg/dL on day 10) normalised with significant clinical improvement after surgery. Histopathology revealed phosphaturic mesenchymal tumour. Here, we report the first case of intra-abdominal mesenchymal tumour causing TIO diagnosed by serial functional imaging.
ISSN:1757-790X
1757-790X
DOI:10.1136/bcr-2019-231728