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Original Article
- Sporadic Nonfamilial Hypophosphatemic Osteomalacia.
- Young Kee Shong, Joong Yeol Park, Ghi Su Kim, You Sook Cho, Goo Yeong Cho, Sang Wook Kim, Jung Sik Park, Ki Up Lee
- J Korean Endocr Soc. 1994;9(1):25-31. Published online November 6, 2019
- 1,159 View
- 27 Download
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Abstract
PDF - Chronic hypophosphatemia caused by decreased intestinal absorption or increased renal clearance, may lead to rickets or osteomalacia independently of other predisposing abnormalities. The conditions commonly associated with increased renal clearance of phosphate are X-linked hypophosphatemic rickets, tumor associated rickets/osteomalacia, RTA and Fanconi syndrome. Recently we experienced 3 men with adult-onset, histologically proven osteomalacia associated with increased renal clearance of phosphate. None of them had a family history of bone disease, tumors or other tubular defects. All of these had remarkable biochemical and clinical improvement with medical treatment such as 1, 25-dihydroxyvitamin D and phosphate supplementation. Although we did not find tumors yet, we could not rule out the possibility of tumor-associated osteomalcia since it often takes several years to make a diagnosis because of small size, benign nature and unusual location of tumors. Thus, careful long-term follow up for tumor occurrence will be maintained in these patients with sporadic nonfamilial hypophosphatemic osteomalacia.
Review Article
- Bone Metabolism
- Diagnostic Modalities for FGF23-Producing Tumors in Patients with Tumor-Induced Osteomalacia
- Seiji Fukumoto
- Endocrinol Metab. 2014;29(2):136-143. Published online June 26, 2014
- DOI: https://doi.org/10.3803/EnM.2014.29.2.136
- 4,658 View
- 53 Download
- 25 Web of Science
- 28 Crossref
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Abstract
PDF
PubReader Fibroblast growth factor 23 (FGF23) is a hormone that is produced by osteocytes and regulates phosphate and vitamin D metabolism through binding to the Klotho-FGF receptor complex. Excessive actions of FGF23 cause several kinds of hypophosphatemic rickets/osteomalacia. Tumor-induced rickets/osteomalacia (TIO) is a paraneoplastic syndrome caused by overproduction of FGF23 from the responsible tumors. Because TIO is cured by complete resection of the causative tumors, it is of great clinical importance to locate these tumors. Several imaging methods including skeletal survey by magnetic resonance imaging and octreotide scintigraphy have been used to identify the tumors that cause TIO. However, none of these imaging studies indicate that the detected tumors are actually producing FGF23. Recently, systemic venous sampling was conducted for locating FGF23-producing tumor in suspected patients with TIO and demonstrated that this test might be beneficial to a subset of patient. Further studies with more patients are necessary to establish the clinical utility of venous sampling in patients with TIO.
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Citations
Citations to this article as recorded by
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Ejigayehu G. Abate, Victor Bernet, Cherise Cortese, Hillary W. Garner
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Anke H. Hautmann, Matthias G. Hautmann, Oliver Kölbl, Wolfgang Herr, Martin Fleck
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Meghan Higley, Brooke Beckett, Sandra Schmahmann, Elizabeth Dacey, Erik Foss
Skeletal Radiology.2015; 44(12): 1825. CrossRef - Articles in 'Endocrinology and Metabolism' in 2014
Won-Young Lee
Endocrinology and Metabolism.2015; 30(1): 47. CrossRef
- Progression of phosphaturic mesenchymal tumor in the femoral bone during treatment with burosumab: A case report
Case Reports
- A Case of Sporadic Nonfamilial Hypophosphatemic Osteomalacia.
- Jun Goo Kang, Dong Sun Kim, Chan Bum Choi, Tae Jong Kim, Jong Pyo Kim, Chang Beom Lee, Yong Soo Park, You Hern Ahn, Tae Wha Kim, Sang Cheol Bae, Chan Gum Park
- J Korean Endocr Soc. 2002;17(4):610-616. Published online August 1, 2002
- 1,028 View
- 20 Download
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Abstract
PDF
- Acquired hypophosphatemic rickets, or osteomalacia, requires the recognition of the typical clinical and radiological features of osteomalacia in association with hypophosphatemia, which is caused by the decrease in intestinal absorption or impaired renal tubular phosphate reabsorption. The latter form may either be hereditary or acquired. Acquired hypophosphatemic osteomalacia includes oncogenic osteomalacia, neurofibromatosis, fibrous dysplasia, renal tubular acidosis and sporadic nonfamilial hypophosphatemic osteomalacia. A 33-year-old man presented with bone pain, progressive severe muscle weakness and a height loss of more than 10 cm over a 5 year period. The familiy history was negative for bone disease or other renal tubular defects. He was found to have hypophosphatemia, impaired phosphate reabsorption, normocalcemia, normal vitamin D metabolite levels, normal PTH and elevated alkaline phophatase. A bone biopsy showed thickened unmineralized osteoid compared to pelvic bone in control cases. Clinical symptoms, such as bone pain and muscle weakness, were improved after supplementation of oral phosphorus and calcitriol, although the serum phosphorus level did not normalize.
- A Case of Osteosarcoma induced Oncogenic Osteomalacia Detected by MRI.
- Sung Kil Lim, Young Duk Song, Hyun Chul Lee, Kap Bum Huh, Ki Hyun Park, Kyung Rae Kim, Soon Won Hong, Jae Hyun Nam, Jong In Yook, Byung Joo Choi, Mi Kyung Kim, Kyu Ho Shin
- J Korean Endocr Soc. 1999;14(2):401-409. Published online January 1, 2001
- 1,098 View
- 22 Download
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Abstract
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- Oncogenic osteomalacia is a syndrome characterized by phosphaturia, hypophosphatemia, decreased 1,25-dihydroxyvitamin D level and specific signs and symptoms of osteomalacia. It is associated with the presence of neoplasm originated from mesenchyme. Until now, less than 100 cases of oncogenic osteomalacia have been reported. The pathophysiology of oncogenic osteomalacia has not been fully understood, but it has been suggested that a certain substance released by tumor may inhibit not only la-hydroxylase activity and reduce 1,25-dihydroxyvitamin D level in part, but directly inhibit reabsorption of phosphate. And then, reduced phosphaturia, hypophosphatemia and eventually osteomalacia develop. We report a case of osteosarcoma induced oncogenic osteomalacia detected by MRI in 59 year old woman.
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