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Oncogenic osteomalacia from pterygopalatine fossa mass

Published online by Cambridge University Press:  02 March 2009

C R Savage
Affiliation:
Department of Otolaryngology, Head and Neck Surgery, University of Cincinnati Medical Center, Ohio, USA
L A Zimmer*
Affiliation:
Department of Otolaryngology, Head and Neck Surgery, University of Cincinnati Medical Center, Ohio, USA
*
Address for correspondence: Dr Lee A Zimmer, University of Cincinnati, Department of Otolaryngology-Head and Neck Surgery, 231 Albert Sabin Way, ML 0528, Cincinnati, OH 45267, USA. Fax: +1 513 558 4477 E-mail: lee.zimmer@uc.edu

Abstract

Introduction:

Oncogenic osteomalacia, or tumour-induced osteomalacia, is an uncommon cause of osteomalacia. It has been reported to occur in patients with hypophosphataemia due to excess renal phosphate excretion secondary to mesenchymal tumours. Occurrence of this pathological process in the head and neck is extremely rare.

Methods:

Case report and literature review.

Results:

We present a case of a 73-year-old woman with tumour-induced osteomalacia. She was initially followed by the endocrinologists for osteomalacia and pathological fractures. An indium-111 pentetreotide scan showed activity in the left pterygopalatine fossa. A mass was endoscopically resected, and the histopathological appearance was consistent with a haemangiopericytoma. Following surgery, the patient's hypophosphataemia and vitamin D deficiency corrected and her symptoms resolved.

Conclusions:

Oncogenic osteomalacia, or tumour-induced osteomalacia, is a rare entity in the head and neck. Current research is elucidating the mechanism by which phosphaturic wasting occurs. In most patients, symptoms resolve once the offending tumour is removed.

Type
Clinical Records
Copyright
Copyright © JLO (1984) Limited 2009

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Footnotes

Presented at the American Rhinologic Society Annual Meeting, 15 September 2007, Washington DC, USA.

References

1 Jan de Beur, S. Tumor-induced osteomalacia. JAMA 2005;294:1260–7Google Scholar
2 McCrance, RA. Osteomalacia with Looser's nodes (milkman's syndrome) due to a raised resistance to vitamin D acquired about the age of 15 years. Q J Med 1947;16:3346Google Scholar
3 Crouzet, J, Mimoune, H, Beraneck, L, Juan, LH. Hypophosphatemic osteomalacia with plantar neurilemoma. A review of the literature (100 cases). Rev Rhum Engl Ed 1995;62:463–6Google Scholar
4 Folpe, AL, Fanburg-Smith, JC, Billings, SD, Bisceglia, M, Bertoni, F, Cho, JY et al. Most osteomalacia-associated mesenchymal tumors are a single histopathologic entity: an analysis of 32 cases and a comprehensive review of the literature. Am J Surg Pathol 2004;28:130Google Scholar
5 Gonzalez-Compta, X, Manos-Pujol, M, Foglia-Fernandez, M, Peral, E, Condom, E, Claveguera, T et al. Oncogenic osteomalacia: case report and review of head and neck associated tumours. J Laryngol Otol 1998;112:389–92Google Scholar
6 Inokuchi, G, Tanimoto, H, Ishida, H, Sugimoto, T, Yamauchi, M, Miyauchi, A et al. A paranasal tumor associated with tumor-induced osteomalacia. Laryngoscope 2006;116:1930–3CrossRefGoogle ScholarPubMed
7 Sandhu, FA, Martuza, RL. Craniofacial hemangiopericytoma associated with oncogenic osteomalacia: case report. J Neurooncol 2000;46:241–7CrossRefGoogle ScholarPubMed
8 Fuentealba, C, Pinto, D, Ballesteros, F, Pacheco, D, Boettiger, O, Soto, N et al. Oncogenic hypophosphatemic osteomalacia associated with a nasal hemangiopericytoma. J Clin Rheumatol 2003;9:373–9Google Scholar
9 Rhee, Y, Lee, JD, Shin, KH, Lee, HC, Huh, KB, Lim, SK. Oncogenic osteomalacia associated with mesenchymal tumour detected by indium-111 octreotide scintigraphy. Clin Endo 2001;54:551–4Google Scholar
10 Jan de Beur, SM, Streeten, EA, Civelek, AC, McCarthy, EF, Uribe, L, Marx, SJ et al. Localisation of mesenchymal tumours by somatostatin receptor imaging. Lancet 2002;359:761–3Google Scholar
11 Jonsson, KB, Zahradnik, R, Larsson, T, White, KE, Sugimoto, T, Imanishi, Y et al. Fibroblast growth factor 23 in oncogenic osteomalacia and X-linked hyperphosphatemia. N Engl J Med 2003;348:1656–63CrossRefGoogle Scholar
12 Shimada, T, Mitzutani, S, Muto, T, Yoneya, T, Hino, R, Takeda, S et al. Cloning and characterization of FGF23 as a causative factor of tumor-induced osteomalacia. Proc Natl Acad Sci U S A 2001;98:6500–5Google Scholar
13 Bowe, A, Finnegan, R, Jan de Beur, SM, Cho, J, Levine, MA, Kumar, R et al. FGF23 inhibits phosphate transport in vitro and is a substrate for the PHEX endopeptidase. Biochem Biophys Res Commun 2001;284:977–81Google Scholar
14 Shimada, T, Urakawa, I, Yamazaki, Y, Hasegawa, H, Hino, R, Yoneya, T et al. FGF-23 transgenic mice demonstrate hypophosphatemic rickets with reduced expression of sodium phosphate cotransporter type IIa. Biochem Biophys Res Commun 2004;314:409–14CrossRefGoogle ScholarPubMed