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The phosphaturic mesenchymal tumor: why is definitive diagnosis and curative surgery often delayed?

Publication ,  Journal Article
Ledford, CK; Zelenski, NA; Cardona, DM; Brigman, BE; Eward, WC
Published in: Clin Orthop Relat Res
November 2013

BACKGROUND: Tumor-induced osteomalacia is a paraneoplastic syndrome resulting in renal phosphate wasting and decreased bone mineralization. Phosphaturic mesenchymal tumors represent a rare etiology of tumor-induced osteomalacia. Nonspecific symptoms of fatigue, bone pain, and musculoskeletal weakness make the diagnosis elusive and lead to a delay in surgical treatment. QUESTIONS/PURPOSES: In this case series, the following three questions were asked: (1) How do the clinical presentation and features of phosphaturic mesenchymal tumors delay the diagnosis? (2) What is the clinical course after surgical treatment of phosphaturic mesenchymal tumors? (3) How frequently do phosphaturic mesenchymal tumors recur and are there factors associated with recurrence? METHODS: This study retrospectively reviewed the cases of five adults diagnosed and treated for phosphaturic mesenchymal tumors. Patients were identified through an internal orthopaedic oncology database with clinical, surgical, and histologic data obtained through a systematic chart review. RESULTS: Five patients presented with a long-standing history of osteomalacia, generalized fatigue, pain, and weakness before the diagnosis was reached at an average of 7.2 years (range, 2-12 years) after initial symptom onset. The diagnosis appeared to be delayed owing to the cryptic medical presentation, difficulty in locating tumor by imaging, and confirming histologic appearance. Two patients treated with wide surgical resection did not experience recurrence compared with three patients who did show recurrent signs and symptoms after marginal excision. A postoperative increase in fibroblast-derived growth factor-23 was associated with recurrent disease. CONCLUSIONS: Although uncommon, the diagnosis of phosphaturic mesenchymal tumor should be considered in any patient who presents with hypophosphaturic osteomalacia and no other physiologic cause. Definitive treatment is early, wide surgical resection.

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Published In

Clin Orthop Relat Res

DOI

EISSN

1528-1132

Publication Date

November 2013

Volume

471

Issue

11

Start / End Page

3618 / 3625

Location

United States

Related Subject Headings

  • Treatment Outcome
  • Time-to-Treatment
  • Time Factors
  • Risk Factors
  • Retrospective Studies
  • Predictive Value of Tests
  • Phosphates
  • Paraneoplastic Syndromes
  • Osteomalacia
  • Orthopedics
 

Citation

APA
Chicago
ICMJE
MLA
NLM
Ledford, C. K., Zelenski, N. A., Cardona, D. M., Brigman, B. E., & Eward, W. C. (2013). The phosphaturic mesenchymal tumor: why is definitive diagnosis and curative surgery often delayed? Clin Orthop Relat Res, 471(11), 3618–3625. https://doi.org/10.1007/s11999-013-3178-1
Ledford, Cameron K., Nicole A. Zelenski, Diana M. Cardona, Brian E. Brigman, and William C. Eward. “The phosphaturic mesenchymal tumor: why is definitive diagnosis and curative surgery often delayed?Clin Orthop Relat Res 471, no. 11 (November 2013): 3618–25. https://doi.org/10.1007/s11999-013-3178-1.
Ledford CK, Zelenski NA, Cardona DM, Brigman BE, Eward WC. The phosphaturic mesenchymal tumor: why is definitive diagnosis and curative surgery often delayed? Clin Orthop Relat Res. 2013 Nov;471(11):3618–25.
Ledford, Cameron K., et al. “The phosphaturic mesenchymal tumor: why is definitive diagnosis and curative surgery often delayed?Clin Orthop Relat Res, vol. 471, no. 11, Nov. 2013, pp. 3618–25. Pubmed, doi:10.1007/s11999-013-3178-1.
Ledford CK, Zelenski NA, Cardona DM, Brigman BE, Eward WC. The phosphaturic mesenchymal tumor: why is definitive diagnosis and curative surgery often delayed? Clin Orthop Relat Res. 2013 Nov;471(11):3618–3625.
Journal cover image

Published In

Clin Orthop Relat Res

DOI

EISSN

1528-1132

Publication Date

November 2013

Volume

471

Issue

11

Start / End Page

3618 / 3625

Location

United States

Related Subject Headings

  • Treatment Outcome
  • Time-to-Treatment
  • Time Factors
  • Risk Factors
  • Retrospective Studies
  • Predictive Value of Tests
  • Phosphates
  • Paraneoplastic Syndromes
  • Osteomalacia
  • Orthopedics