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Tumor-Induced Osteomalacia Localized and Excised After Pregnancy

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 b...

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Autores principales: Sum, Melissa, Hoda, Syed T., Rapp, Timothy, Zan, Elcin
Formato: Online Artículo Texto
Lenguaje:English
Publicado: American Association of Clinical Endocrinology 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8573288/
https://www.ncbi.nlm.nih.gov/pubmed/34765732
http://dx.doi.org/10.1016/j.aace.2021.06.008
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author Sum, Melissa
Hoda, Syed T.
Rapp, Timothy
Zan, Elcin
author_facet Sum, Melissa
Hoda, Syed T.
Rapp, Timothy
Zan, Elcin
author_sort Sum, Melissa
collection PubMed
description 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.
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spelling pubmed-85732882021-11-10 Tumor-Induced Osteomalacia Localized and Excised After Pregnancy Sum, Melissa Hoda, Syed T. Rapp, Timothy Zan, Elcin AACE Clin Case Rep Case Report 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. American Association of Clinical Endocrinology 2021-06-18 /pmc/articles/PMC8573288/ /pubmed/34765732 http://dx.doi.org/10.1016/j.aace.2021.06.008 Text en © 2021 AACE. Published by Elsevier Inc. https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
spellingShingle Case Report
Sum, Melissa
Hoda, Syed T.
Rapp, Timothy
Zan, Elcin
Tumor-Induced Osteomalacia Localized and Excised After Pregnancy
title Tumor-Induced Osteomalacia Localized and Excised After Pregnancy
title_full Tumor-Induced Osteomalacia Localized and Excised After Pregnancy
title_fullStr Tumor-Induced Osteomalacia Localized and Excised After Pregnancy
title_full_unstemmed Tumor-Induced Osteomalacia Localized and Excised After Pregnancy
title_short Tumor-Induced Osteomalacia Localized and Excised After Pregnancy
title_sort tumor-induced osteomalacia localized and excised after pregnancy
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8573288/
https://www.ncbi.nlm.nih.gov/pubmed/34765732
http://dx.doi.org/10.1016/j.aace.2021.06.008
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AT zanelcin tumorinducedosteomalacialocalizedandexcisedafterpregnancy