{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,14]],"date-time":"2026-05-14T05:11:21Z","timestamp":1778735481519,"version":"3.51.4"},"reference-count":27,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2026,5,14]],"date-time":"2026-05-14T00:00:00Z","timestamp":1778716800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Med."],"abstract":"<jats:p>\n                    Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome characterized by renal phosphate wasting, hypophosphatemia, and impaired bone mineralization, primarily caused by phosphaturic mesenchymal tumors (PMTs). Due to the small size and non-specific clinical presentation, PMTs are often difficult to localize, resulting in delayed diagnosis. This study presents a case of a 54-year-old man who experienced a 2-year history of progressive polyarthralgia affecting the knees, hips, and lower back, which exacerbated over the past 3\u202fmonths. Laboratory tests revealed hypophosphatemia, elevated alkaline phosphatase (ALP) levels, and increased parathyroid hormone (PTH) levels. Moreover, a dual-energy X-ray absorptiometry (DXA) scan indicated low bone mineral density (BMD), while a whole-body bone scintigraphy revealed multiple rib pathological fractures, indicative of osteomalacia. Although serum fibroblast growth factor 23 (FGF23) was not measured, the clinical and biochemical profiles were highly consistent with tumor-induced osteomalacia (TIO). Subsequent Gallium-68 1,4,7,10-tetraazacyclododecane 1,4,7,10-tetraacetic acid tyrosine-3-octreotate positron emission tomography\/computed tomography (\n                    <jats:sup>68<\/jats:sup>\n                    Ga-DOTA-TATE) positron emission tomography\/computed tomography (PET\/CT) confirmed a hypermetabolic lesion in the left temporalis muscle. The lesion was completely resected, and histopathological examination confirmed the diagnosis of PMT, with positive immunostaining for special AT-rich sequence-binding protein 2 (SATB2), neural cell adhesion molecule (CD56), and somatostatin receptor type 2 (SSTR2). No tumor recurrence was observed during follow-up at 6\u202fmonths after surgery. Clinicians should consider occult PMTs in patients with unexplained hypophosphatemia, bone pain, or pathological fractures, and employ functional imaging modalities such as\n                    <jats:sup>68<\/jats:sup>\n                    Ga-DOTA-TATE PET\/CT for early tumor localization and potential cure through resection.\n                  <\/jats:p>","DOI":"10.3389\/fmed.2026.1807111","type":"journal-article","created":{"date-parts":[[2026,5,14]],"date-time":"2026-05-14T04:31:40Z","timestamp":1778733100000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":0,"title":["Phosphaturic mesenchymal tumor in the temporalis muscle causing tumor-induced osteomalacia: a case report and literature review"],"prefix":"10.3389","volume":"13","author":[{"given":"Bo","family":"Gao","sequence":"first","affiliation":[{"name":"Department of Prosthodontics, Second Affiliated Hospital of Zhejiang University School of Medicine","place":["Zhejiang, Hangzhou, China"]},{"name":"Stomatology Hospital, School of Stomatology, Zhejiang University School of Medicine, Zhejiang Provincial Clinical Research Center for Oral Diseases, Key Laboratory of Oral Biomedical Research of Zhejiang Province, Cancer Center of Zhejiang University, Engineering Research Center of Oral Biomaterials and Devices of Zhejiang Province","place":["Zhejiang, Hangzhou, China"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Chao","family":"Wang","sequence":"additional","affiliation":[{"name":"Department of Radiology, Second Affiliated Hospital, Zhejiang University School of Medicine","place":["Zhejiang, Hangzhou, China"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Xiaoming","family":"Xu","sequence":"additional","affiliation":[{"name":"Department of Pathology, Second Affiliated Hospital, Zhejiang University School of Medicine","place":["Zhejiang, Hangzhou, China"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Xiuxiu","family":"Lai","sequence":"additional","affiliation":[{"name":"Geriatric Medicine Center, Department of Geriatric Medicine, Zhejiang Provincial People\u2019s Hospital, Affiliated People\u2019s Hospital, Hangzhou Medical College","place":["Zhejiang, Hangzhou, China"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Xiaohong","family":"Xu","sequence":"additional","affiliation":[{"name":"Department of Cardiology, Zhejiang Hospital","place":["Zhejiang, Hangzhou, China"]}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1965","published-online":{"date-parts":[[2026,5,14]]},"reference":[{"key":"ref1","doi-asserted-by":"publisher","first-page":"101834","DOI":"10.1016\/j.beem.2023.101834","article-title":"Tumor-induced Osteomalacia: an overview","volume":"38","author":"Jadhav","year":"2024","journal-title":"Best Pract Res Clin Endocrinol Metab"},{"key":"ref2","doi-asserted-by":"publisher","first-page":"e3428","DOI":"10.1210\/clinem\/dgac253","article-title":"Tumor-induced Osteomalacia: a systematic review and individual patient's data analysis","volume":"107","author":"Rendina","year":"2022","journal-title":"J Clin Endocrinol Metab"},{"key":"ref3","doi-asserted-by":"publisher","first-page":"101772","DOI":"10.1016\/j.bonr.2024.101772","article-title":"Tumor-induced Osteomalacia: a systematic literature review","volume":"21","author":"Alvarez-Rivas","year":"2024","journal-title":"Bone Rep"},{"key":"ref4","doi-asserted-by":"publisher","first-page":"17044","DOI":"10.1038\/nrdp.2017.44","article-title":"Tumour-induced Osteomalacia","volume":"3","author":"Minisola","year":"2017","journal-title":"Nat Rev Dis Primers"},{"key":"ref5","doi-asserted-by":"publisher","first-page":"305","DOI":"10.1007\/s13193-021-01449-8","article-title":"Metastatic malignant Phosphaturic mesenchymal tumor of mandibular alveolus: a rare case report and review of literature","volume":"13","author":"Thakur","year":"2022","journal-title":"Indian J Surg Oncol"},{"key":"ref6","doi-asserted-by":"publisher","first-page":"6500","DOI":"10.1073\/pnas.101545198","article-title":"Cloning and characterization of Fgf23 as a causative factor of tumor-induced Osteomalacia","volume":"98","author":"Shimada","year":"2001","journal-title":"Proc Natl Acad Sci USA"},{"key":"ref7","doi-asserted-by":"publisher","first-page":"1173","DOI":"10.1007\/s40618-018-0849-5","article-title":"Phosphaturic mesenchymal Tumors: what an endocrinologist should know","volume":"41","author":"Boland","year":"2018","journal-title":"J Endocrinol Investig"},{"key":"ref8","doi-asserted-by":"publisher","first-page":"4957","DOI":"10.1210\/jc.2002-021105","article-title":"Increased circulatory level of biologically active full-length Fgf-23 in patients with Hypophosphatemic rickets\/Osteomalacia","volume":"87","author":"Yamazaki","year":"2002","journal-title":"J Clin Endocrinol Metab"},{"key":"ref9","doi-asserted-by":"publisher","first-page":"3350","DOI":"10.21037\/apm-20-1466","article-title":"Diagnostic value of 68ga-Dota-Tate pet\/Ct imaging for tumor-induced Osteomalacia","volume":"9","author":"Zhang","year":"2020","journal-title":"Ann Palliat Med"},{"key":"ref10","doi-asserted-by":"publisher","first-page":"675","DOI":"10.1507\/endocrj.EJ16-0587","article-title":"The diagnostic dilemma of tumor induced Osteomalacia: a retrospective analysis of 144 cases","volume":"64","author":"Feng","year":"2017","journal-title":"Endocr J"},{"key":"ref11","doi-asserted-by":"publisher","first-page":"1919","DOI":"10.1007\/s00198-025-07527-9","article-title":"Postoperative outcome and clinical Management of Tumor-Induced Osteomalacia: a single-Center retrospective cohort study on 117 patients","volume":"36","author":"Shan","year":"2025","journal-title":"Osteoporos Int"},{"key":"ref12","doi-asserted-by":"publisher","first-page":"367","DOI":"10.1007\/s00223-022-01005-8","article-title":"Tumor-induced Osteomalacia: a systematic clinical review of 895 cases","volume":"111","author":"Bosman","year":"2022","journal-title":"Calcif Tissue Int"},{"key":"ref13","doi-asserted-by":"publisher","first-page":"1330","DOI":"10.1530\/EC-19-0341","article-title":"Tumor induced Osteomalacia in head and neck region: single Center experience and systematic review","volume":"8","author":"Shah","year":"2019","journal-title":"Endocr Connect"},{"key":"ref14","doi-asserted-by":"publisher","first-page":"677","DOI":"10.1186\/s12891-021-04558-1","article-title":"Malignant Phosphaturic mesenchymal tumor-ossifying fibroma-like subtype: a case report and literature review","volume":"22","author":"Qin","year":"2021","journal-title":"BMC Musculoskelet Disord"},{"key":"ref15","doi-asserted-by":"publisher","first-page":"281","DOI":"10.1007\/s00424-022-02668-w","article-title":"The regulation of Fgf23 under physiological and pathophysiological conditions","volume":"474","author":"Rausch","year":"2022","journal-title":"Pflugers Arch"},{"key":"ref16","doi-asserted-by":"publisher","first-page":"429","DOI":"10.1359\/Jbmr.0301264","article-title":"Fgf-23 is a potent regulator of vitamin D metabolism and phosphate homeostasis","volume":"19","author":"Shimada","year":"2004","journal-title":"J Bone Miner Res"},{"key":"ref17","doi-asserted-by":"publisher","first-page":"1819","DOI":"10.1038\/s41598-021-81491-2","article-title":"Usefulness of (68)ga-Dotatoc pet\/Ct to localize the culprit tumor inducing Osteomalacia","volume":"11","author":"Lee","year":"2021","journal-title":"Sci Rep"},{"key":"ref18","doi-asserted-by":"publisher","first-page":"73","DOI":"10.2967\/jnmt.121.263776","article-title":"False-positive (68)ga-Dotatate pet\/Ct findings in hereditary hypophosphatemia-Osteomalacia mimicking culprit lesions of tumor-induced Osteomalacia","volume":"51","author":"Parghane","year":"2023","journal-title":"J Nucl Med Technol"},{"key":"ref19","doi-asserted-by":"publisher","first-page":"1386","DOI":"10.1002\/jbmr.1881","article-title":"Tumor localization and biochemical response to cure in tumor-induced Osteomalacia","volume":"28","author":"Chong","year":"2013","journal-title":"J Bone Miner Res"},{"key":"ref20","doi-asserted-by":"publisher","first-page":"bvac181","DOI":"10.1210\/jendso\/bvac181","article-title":"Utility of multimodality approach including systemic Fgf23 venous sampling in localizing phosphaturic mesenchymal tumors","volume":"7","author":"Kato","year":"2022","journal-title":"J Endocr Soc"},{"key":"ref21","doi-asserted-by":"publisher","first-page":"3979","DOI":"10.1210\/jc.2004-0406","article-title":"Venous sampling for fibroblast growth factor-23 confirms preoperative diagnosis of tumor-induced osteomalacia","volume":"89","author":"Takeuchi","year":"2004","journal-title":"J Clin Endocrinol Metab"},{"key":"ref22","doi-asserted-by":"publisher","first-page":"390","DOI":"10.1111\/j.1365-2796.2010.02262.x","article-title":"Clinical utility of systemic venous sampling of Fgf23 for identifying tumours responsible for tumour-induced Osteomalacia","volume":"268","author":"Ito","year":"2010","journal-title":"J Intern Med"},{"key":"ref23","doi-asserted-by":"publisher","first-page":"323","DOI":"10.1210\/endrev\/bnac026","article-title":"Tumor-induced osteomalacia: a comprehensive review","volume":"44","author":"Minisola","year":"2023","journal-title":"Endocr Rev"},{"key":"ref24","doi-asserted-by":"publisher","first-page":"1383","DOI":"10.1007\/s00198-020-05369-1","article-title":"Long-term bone mineral density changes after surgical cure of patients with tumor-induced osteomalacia","volume":"31","author":"Colangelo","year":"2020","journal-title":"Osteoporos Int"},{"key":"ref25","doi-asserted-by":"publisher","first-page":"1264","DOI":"10.1097\/CM9.0000000000001448","article-title":"Consensus on clinical Management of Tumor-Induced Osteomalacia","volume":"134","author":"Jiang","year":"2021","journal-title":"Chin Med J"},{"key":"ref26","doi-asserted-by":"publisher","first-page":"262","DOI":"10.1002\/jbmr.4184","article-title":"Interim analysis of a phase 2 open-label trial assessing Burosumab efficacy and safety in patients with tumor-induced Osteomalacia","volume":"36","author":"Imanishi","year":"2021","journal-title":"J Bone Miner Res"},{"key":"ref27","doi-asserted-by":"publisher","first-page":"81","DOI":"10.1016\/j.jbspin.2019.07.012","article-title":"Burosumab in tumor-induced osteomalacia: a case report","volume":"87","author":"Day","year":"2020","journal-title":"Joint Bone Spine"}],"container-title":["Frontiers in Medicine"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2026.1807111\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,5,14]],"date-time":"2026-05-14T04:31:54Z","timestamp":1778733114000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2026.1807111\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,5,14]]},"references-count":27,"alternative-id":["10.3389\/fmed.2026.1807111"],"URL":"https:\/\/doi.org\/10.3389\/fmed.2026.1807111","relation":{},"ISSN":["2296-858X"],"issn-type":[{"value":"2296-858X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,5,14]]},"article-number":"1807111"}}