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J Neurol Surg Rep 2015; 76(02): e233-e238
DOI: 10.1055/s-0035-1562852
Case Report
Georg Thieme Verlag KG Stuttgart · New York

Sinonasal Phosphaturic Mesenchymal Tumor: A Rare and Misinterpreted Entity

Demetri Arnaoutakis
1   Department of Otolaryngology-Head and Neck Surgery, University of Texas Southwestern Medical Center, Dallas, Texas, United States
,
Iman Naseri
2   Department of Otolaryngology-Head and Neck Surgery, University of Florida College of Medicine, Jacksonville, Florida, United States
› Author Affiliations
Further Information

Publication History

13 April 2015

28 June 2015

Publication Date:
21 August 2015 (online)

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Abstract

Objectives Oncogenic osteomalacia is a paraneoplastic syndrome in which the tumor secretes a peptide-like hormone, fibroblast growth factor, resulting in urinary loss of phosphates.

Methods We present the case of a 50-year-old woman with a benign phosphaturic mesenchymal tumor (PMT) involving the ethmoid sinus with obstruction of the ostiomeatal complex causing unilateral nasal airway obstruction.

Results The tumor was initially thought to be an esthesioneuroblastoma based on primary pathology interpretation and on clinical and radiographic appearance. However, a benign PMT was later confirmed by further testing.

Conclusion The tumor was removed entirely by the endoscopic transnasal approach, leading to a full resolution of symptoms.

Keywords

phosphaturic mesenchymal tumor - skull base resection - endoscopic sinus surgery
 

  • References

  • 1 Shimada T, Mizutani S, Muto T , et al. Cloning and characterization of FGF23 as a causative factor of tumor-induced osteomalacia. Proc Natl Acad Sci U S A 2001; 98 (11) 6500-6505
    CrossrefPubMedGoogle Scholar
  • 2 Sundaram M, McCarthy EF. Oncogenic osteomalacia. Skeletal Radiol 2000; 29 (3) 117-124
    CrossrefPubMedGoogle Scholar
  • 3 Reisacher WR, Pincus RL. Pathologic quiz case 1. Pneumocystic lymphadenitis. Arch Otolaryngol Head Neck Surg 1997; 123 (8) 880 , 882–883
    CrossrefPubMedGoogle Scholar
  • 4 McCance 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 (1) 33-46
    PubMedGoogle Scholar
  • 5 Prader V. Rickets following bone tumor. [in German]. Helv Paediatr Acta 1959; 14: 554-565
    PubMedGoogle Scholar
  • 6 Linsey M, Smith W, Yamauchi H, Bernstein L. Nasopharyngeal angiofibroma presenting as adult osteomalacia: case report and review of the literature. Laryngoscope 1983; 93 (10) 1328-1331
    PubMedGoogle Scholar
  • 7 Weidner N, Santa Cruz D. Phosphaturic mesenchymal tumors. A polymorphous group causing osteomalacia or rickets. Cancer 1987; 59 (8) 1442-1454
    CrossrefPubMedGoogle Scholar
  • 8 Papotti M, Foschini MP, Isaia G, Rizzi G, Betts CM, Eusebi V. Hypophosphatemic oncogenic osteomalacia: report of three new cases. Tumori 1988; 74 (5) 599-607
    PubMedGoogle Scholar
  • 9 Gonzalez-Compta X, Mañós-Pujol M, Foglia-Fernandez M , et al. Oncogenic osteomalacia: case report and review of head and neck associated tumours. J Laryngol Otol 1998; 112 (4) 389-392
    PubMedGoogle Scholar
  • 10 Kawai Y, Morimoto S, Sakaguchi K , et al. Oncogenic osteomalacia secondary to nasal tumor with decreased urinary excretion of cAMP. J Bone Miner Metab 2001; 19 (1) 61-64
    CrossrefPubMedGoogle Scholar
  • 11 Ungari C, Rocchi G, Rinna C, Agrillo A, Lattanzi A, Pagnoni M. Hypophosphaturic mesenchymal tumor of the ethmoid associated with oncogenic osteomalacia. J Craniofac Surg 2004; 15 (3) 523-527
    CrossrefPubMedGoogle Scholar
  • 12 Inokuchi G, Tanimoto H, Ishida H , et al. A paranasal tumor associated with tumor-induced osteomalacia. Laryngoscope 2006; 116 (10) 1930-1933
    CrossrefPubMedGoogle Scholar
  • 13 Koriyama N, Nishimoto K, Kodama T , et al. Oncogenic osteomalacia in a case with a maxillary sinus mesenchymal tumor. Am J Med Sci 2006; 332 (3) 142-147
    CrossrefPubMedGoogle Scholar
  • 14 Pedrazzoli M, Colletti G, Ferrari M, Rossetti G, Moneghini L, Autelitano L. Mesenchymal phosphaturic neoplasm in the maxillary sinus: a case report. Int J Oral Maxillofac Surg 2010; 39 (10) 1027-1032
    CrossrefPubMedGoogle Scholar
  • 15 Shelekhova KV, Kazakov DV, Michal M. Sinonasal phosphaturic mesenchymal tumor (mixed connective tissue variant): report of 2 cases. Am J Surg Pathol 2010; 34 (4) 596-597
    PubMedGoogle Scholar
  • 16 Peterson NR, Summerlin DJ, Cordes SR. Multiple phosphaturic mesenchymal tumors associated with oncogenic osteomalacia: case report and review of the literature. Ear Nose Throat J 2010; 89 (6) E11-E15
    PubMedGoogle Scholar
  • 17 Parshwanath HA, Kulkarni PR, Rao R, Joshi SK, Patil P. Phosphaturic mesenchymal tumor of ethmoid sinus. Indian J Pathol Microbiol 2010; 53 (2) 384-385
    CrossrefPubMedGoogle Scholar
  • 18 Komínek P, Stárek I, Geierová M, Matoušek P, Zeleník K. Phosphaturic mesenchymal tumour of the sinonasal area: case report and review of the literature. Head Neck Oncol 2011; 3: 16
    CrossrefPubMedGoogle Scholar
  • 19 Guglielmi G, Bisceglia M, Scillitani A, Folpe AL. Oncogenic osteomalacia due to phosphaturic mesenchymal tumor of the craniofacial sinuses. Clin Cases Miner Bone Metab 2011; 8 (2) 45-49
    PubMedGoogle Scholar
  • 20 Battoo AJ, Salih S, Unnikrishnan AG , et al. Oncogenic osteomalacia from nasal cavity giant cell tumor. Head Neck 2012; 34 (3) 454-457
    CrossrefPubMedGoogle Scholar
  • 21 Deep NL, Cain RB, McCullough AE, Hoxworth JM, Lal D. Sinonasal phosphaturic mesenchymal tumor: case report and systematic review. Allergy Rhinol (Providence) 2014; 5 (3) 162-167
    CrossrefPubMedGoogle Scholar
  • 22 Okamiya T, Takahashi K, Kamada H , et al. Oncogenic osteomalacia caused by an occult paranasal sinus tumor. Auris Nasus Larynx 2015; 42 (2) 167-169
    CrossrefPubMedGoogle Scholar
  • 23 Olefsky J, Kempson R, Jones H, Reaven G. “Tertiary” hyperparathyroidism and apparent “cure” of vitamin-D-resistant rickets after removal of an ossifying mesenchymal tumor of the pharynx. N Engl J Med 1972; 286 (14) 740-745
    CrossrefPubMedGoogle Scholar
  • 24 Shenker Y, Grekin RJ. Oncogenic osteomalacia. Isr J Med Sci 1984; 20 (8) 739-741
    PubMedGoogle Scholar
  • 25 Harvey JN, Gray C, Belchetz PE. Oncogenous osteomalacia and malignancy. Clin Endocrinol (Oxf) 1992; 37 (4) 379-382
    CrossrefPubMedGoogle Scholar
  • 26 Yang IM, Park YK, Hyun YJ , et al. Oncogenic osteomalacia caused by a phosphaturic mesenchymal tumor of the oral cavity: a case report. Korean J Intern Med 1997; 12 (1) 89-95
    CrossrefPubMedGoogle Scholar
  • 27 Reyes-Múgica M, Arnsmeier SL, Backeljauw PF, Persing J, Ellis B, Carpenter TO. Phosphaturic mesenchymal tumor-induced rickets. Pediatr Dev Pathol 2000; 3 (1) 61-69
    CrossrefPubMedGoogle Scholar
  • 28 Dupond JL, Mahammedi H, Prié D , et al. Oncogenic osteomalacia: diagnostic importance of fibroblast growth factor 23 and F-18 fluorodeoxyglucose PET/CT scan for the diagnosis and follow-up in one case. Bone 2005; 36 (3) 375-378
    CrossrefPubMedGoogle Scholar
  • 29 Woo VL, Landesberg R, Imel EA , et al. Phosphaturic mesenchymal tumor, mixed connective tissue variant, of the mandible: report of a case and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2009; 108 (6) 925-932
    CrossrefPubMedGoogle Scholar
  • 30 Kaylie DM, Jackson CG, Gardner EK. Oncogenic osteomalacia caused by phosphaturic mesenchymal tumor of the temporal bone. Otolaryngol Head Neck Surg 2006; 135 (4) 653-654
    CrossrefPubMedGoogle Scholar
  • 31 Uramoto N, Furukawa M, Yoshizaki T. Malignant phosphaturic mesenchymal tumor, mixed connective tissue variant of the tongue. Auris Nasus Larynx 2009; 36 (1) 104-105
    CrossrefPubMedGoogle Scholar
  • 32 Yun KI, Kim DH, Pyo SW. A phosphaturic mesenchymal tumor of the floor of the mouth with oncogenic osteomalacia: report of a case. J Oral Maxillofac Surg 2009; 67 (2) 402-405
    CrossrefPubMedGoogle Scholar
  • 33 Savage CR, Zimmer LA. Oncogenic osteomalacia from pterygopalatine fossa mass. J Laryngol Otol 2009; 123 (9) 1052-1054
    CrossrefPubMedGoogle Scholar
  • 34 Mori Y, Ogasawara T, Motoi T , et al. Tumor-induced osteomalacia associated with a maxillofacial tumor producing fibroblast growth factor 23: report of a case and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2010; 109 (3) e57-e63
    CrossrefPubMedGoogle Scholar
  • 35 Sidell D, Lai C, Bhuta S, Barnes L, Chhetri DK. Malignant phosphaturic mesenchymal tumor of the larynx. Laryngoscope 2011; 121 (9) 1860-1863
    PubMedGoogle Scholar
  • 36 Syed MI, Chatzimichalis M, Rössle M, Huber AM. Recurrent phosphaturic mesenchymal tumour of the temporal bone causing deafness and facial nerve palsy. J Laryngol Otol 2012; 126 (7) 721-724
    CrossrefPubMedGoogle Scholar
  • 37 Luo L, Low N, Vandervord J. Mandibular phosphaturic mesenchymal tumor-mixed connective tissue variant in a young girl. Cleft Palate Craniofac J 2013; 50 (6) 751-753
    CrossrefPubMedGoogle Scholar
  • 38 Schapira D, Ben Izhak O, Nachtigal A , et al. Tumor-induced osteomalacia. Semin Arthritis Rheum 1995; 25 (1) 35-46
    CrossrefPubMedGoogle Scholar
  • 39 Ruppe MD, Jan de Beur SM, Disorders of phosphate homeostasis. In: Rosen CJ. , ed. Primer of the metabolic bone diseases and disorders of mineral metabolism. 6th ed. Washington, DC: American Society for Bone and Mineral Research; 2006
    Google Scholar
  • 40 Dulguerov P, Calcaterra T. Esthesioneuroblastoma: the UCLA experience 1970-1990. Laryngoscope 1992; 102 (8) 843-849
    CrossrefPubMedGoogle Scholar
  • 41 Folpe AL, Fanburg-Smith JC, Billings SD , 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 (1) 1-30
    CrossrefPubMedGoogle Scholar