Tumor-Induced Osteomalacia Localized and Excised After Pregnancy.
AACE Clin Case Rep
; 7(6): 363-366, 2021.
Article
em En
| MEDLINE
| ID: mdl-34765732
OBJECTIVE: Tumor-induced osteomalacia (TIO) is a rare osteomalacia characterized by paraneoplastic secretion of fibroblast growth factor 23. Concomitant occurrence of TIO during pregnancy is rarer still. Our objective was to report a young patient with debilitating fractures diagnosed with TIO who became pregnant and subsequently had her tumor localized by gallium-68 (Ga-68) DOTATATE positron emission tomography/magnetic resonance imaging (PET/MRI). CASE REPORT: A 28 year-old woman with a 2-year history of stress fractures was found to have the following: (1) alkaline phosphatase level, 220 (reference range, 30-95) U/L; (2) phosphorus level, 2.1 (2.5-5.0) mg/dL; (3) 1,25-dihydroxyvitamin D3 level, <8 (18-72) pg/mL; (4) 24-hour urine phosphorus level, 0.5 (0.3-1.3) g; and (5) fibroblast growth factor 23 levels, 1241 (reference range, <180) RU/mL. The patient became pregnant, and at term, a cesarean delivery was performed. Ga-68 DOTATATE PET/MRI showed a 9-mm intracortical mass in the right fibular head and right femoral and bilateral calcaneal stress fractures. The fibular lesion was resected; pathology showed a 1.5-cm lesion with positive fibroblast growth factor receptor 1 staining. DISCUSSION: This patient with TIO had an uneventful pregnancy and delivery. TIO is typically caused by benign mesenchymal tumors. Ga-68 DOTATATE PET/computed tomography has been used for localizing tumors causing TIO, yet MRI has superior contrast resolution over computed tomography. Therefore, it is not surprising that Ga-68 PET/MRI successfully localized this patient's tumor to the intracortical space of the fibular head and distinguished it from insufficiency fractures. CONCLUSION: To our knowledge, this is the first report of phosphate treatment in a pregnant patient with TIO and the first report of a tumor-inducing TIO being localized by Ga-68 DOTATATE PET/MRI.
1,25[OH]2D3, 1,25-dihydroxyvitamin D3; ALP, alkaline phosphatase; DOTATATE; FGF23, fibroblast growth factor 23; Ga-68, gallium-68; MRI, magnetic resonance imaging; PET, positron emission tomography; PTH, parathyroid hormone; SUV, standard uptake value; TIO, tumor-induced osteomalacia; TmP/GFR, tubular maximum reabsorption of phosphate per unit of glomerular filtration; fibroblast growth factor 23; hypophosphatemia; iFGF23, intact fibroblast growth factor 23; pregnancy; renal phosphate wasting; tumor-induced osteomalacia
Texto completo:
1
Coleções:
01-internacional
Base de dados:
MEDLINE
Idioma:
En
Revista:
AACE Clin Case Rep
Ano de publicação:
2021
Tipo de documento:
Article