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:	Melissa Sum, MD, Syed T. Hoda, MD, Timothy Rapp, MD, Elcin Zan, MD
Formato:	article
Lenguaje:	EN
Publicado:	Elsevier 2021
Materias:	tumor-induced osteomalacia hypophosphatemia fibroblast growth factor 23 renal phosphate wasting pregnancy DOTATATE Diseases of the endocrine glands. Clinical endocrinology RC648-665
Acceso en línea:	https://doaj.org/article/87d9dc5c5a2243c09801ceec0e352b15
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spelling	oai:doaj.org-article:87d9dc5c5a2243c09801ceec0e352b152021-11-06T04:32:53ZTumor-Induced Osteomalacia Localized and Excised After Pregnancy2376-060510.1016/j.aace.2021.06.008https://doaj.org/article/87d9dc5c5a2243c09801ceec0e352b152021-11-01T00:00:00Zhttp://www.sciencedirect.com/science/article/pii/S237606052100081Xhttps://doaj.org/toc/2376-0605Objective: 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.Melissa Sum, MDSyed T. Hoda, MDTimothy Rapp, MDElcin Zan, MDElsevierarticletumor-induced osteomalaciahypophosphatemiafibroblast growth factor 23renal phosphate wastingpregnancyDOTATATEDiseases of the endocrine glands. Clinical endocrinologyRC648-665ENAACE Clinical Case Reports, Vol 7, Iss 6, Pp 363-366 (2021)
institution	DOAJ
collection	DOAJ
language	EN
topic	tumor-induced osteomalacia hypophosphatemia fibroblast growth factor 23 renal phosphate wasting pregnancy DOTATATE Diseases of the endocrine glands. Clinical endocrinology RC648-665
spellingShingle	tumor-induced osteomalacia hypophosphatemia fibroblast growth factor 23 renal phosphate wasting pregnancy DOTATATE Diseases of the endocrine glands. Clinical endocrinology RC648-665 Melissa Sum, MD Syed T. Hoda, MD Timothy Rapp, MD Elcin Zan, MD Tumor-Induced Osteomalacia Localized and Excised After Pregnancy
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.
format	article
author	Melissa Sum, MD Syed T. Hoda, MD Timothy Rapp, MD Elcin Zan, MD
author_facet	Melissa Sum, MD Syed T. Hoda, MD Timothy Rapp, MD Elcin Zan, MD
author_sort	Melissa Sum, MD
title	Tumor-Induced Osteomalacia Localized and Excised After Pregnancy
title_short	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_sort	tumor-induced osteomalacia localized and excised after pregnancy
publisher	Elsevier
publishDate	2021
url	https://doaj.org/article/87d9dc5c5a2243c09801ceec0e352b15
work_keys_str_mv	AT melissasummd tumorinducedosteomalacialocalizedandexcisedafterpregnancy AT syedthodamd tumorinducedosteomalacialocalizedandexcisedafterpregnancy AT timothyrappmd tumorinducedosteomalacialocalizedandexcisedafterpregnancy AT elcinzanmd tumorinducedosteomalacialocalizedandexcisedafterpregnancy
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Tumor-Induced Osteomalacia Localized and Excised After Pregnancy

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