Oncogenic Osteomalacia with Multiple Insufficiency Fractures: A Case Report

Young-Chang Park; Joon-Oh Seo; Kyu-Hyun Yang

doi:10.12671/jkfs.2017.30.3.146

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Park, Seo, and Yang: Oncogenic Osteomalacia with Multiple Insufficiency Fractures: A Case Report

Case Report

Journal of the Korean Fracture Society 2017; 30(3): 146-150.

Published online: 21 July 2017

DOI: https://doi.org/10.12671/jkfs.2017.30.3.146

Oncogenic Osteomalacia with Multiple Insufficiency Fractures: A Case Report

Young-Chang Park, M.D.

, Joon-Oh Seo, M.D.

, Kyu-Hyun Yang, M.D., Ph.D.

Department of Orthopaedic Surgery, Gangnam Severance Hospital, Yonsei University College of Medicine, Seoul, Korea.

Correspondence to: Kyu-Hyun Yang, M.D., Ph.D. Department of Orthopaedic Surgery, Gangnam Severance Hospital, Yonsei University College of Medicine, 211 Eonju-ro, Gangnam-gu, Seoul 06273, Korea. Tel: +82-2-2019-3414, Fax: +82-2-573-5393, kyang@yuhs.ac

Received 2 May 2017 Revised 27 May 2017 Accepted 27 May 2017

(open-access):

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

Abstract

Oncogenic osteomalacia is a rare paraneoplastic syndrome, characterized by hypophosphatemia, renal phosphate wasting, osteomalacia, and multiple insufficiency fractures, as a result of the tumor. A wide excision of the causative tumor is considered as the treatment of choice, following which, a dramatic recovery is expected. Authors report a case in which the symptoms and bone mineral density were dramatically recovered after an excision of the causative tumor around the tibialis posterior muscle in oncogenic osteomalacia.

Keywords: Oncogenic osteomalacia, Insufficiency fractures, Hypophosphatemia

Figures and Tables

Fig. 1

Spine simple x-ray lateral view shows that vertebral height of T10, 11, 12, L1, and 2 decreased slightly (A) and magnetic resonance imaging T1-weighted enhanced image shows multiple compression fracture (B).

Fig. 2

Whole body bone scan shows multifocal bony uptakes in the bilateral ribs, pelvis, femoral neck, knee, and ankle. RT: right, LT: left.

Fig. 3

Insufficiency fracture of the femoral neck. A lucent line (arrow) is called a Looser's zone or pseudofracture.

Fig. 4

(A, B) Tumor localization. Causative tumor (arrows) was found on the left lower leg by Octreoscan. (C) Magnetic resonance imaging T2-weighted image shows 4.5×2.0×2.0-cm-sized soft tissue tumor located at the tibialis posterior muscle belly. Tumor had a round, well-defined margin with heterogeneous signal intensity. RT: right.

Fig. 5

Intraoperative image. Tumor was excised completely.

Fig. 6

Bone mineral density (BMD) change. Compared to the preoperative status, BMD and Z-score at 1 year and six months follow-up after surgery showed good recovery. BMC: bone mineral content.

Table 1

Laboratory Findings in Oncogenic Osteomalcia

Variable	Oncogenic osteomalacia	Case				Reference range
Variable	Oncogenic osteomalacia	Initial visit	Preoperative	POD 1 day	POD 1 week^†	Reference range
Phosphorus (mg/dl)	↓	1.7^*	1.9^*	3.5	3.7	2.9-4.6
25(OH)D (ng/ml)	NL or ↓	11.1^*	12.1^*	-	16.5^*	30-100
1,25(OH)₂D (pg/ml)	↓ or NL	-	18.79	-	278.62^*	19.6-54.3
Alkaline phosphatase (IU/L)	↑	172^*	215^*	201^*	220^*	44-99
PTH (pg/ml)	NL or ↑	118.3^*	48	-	-	15-65
Calcium (mg/dl)	NL	8.8	8.9	8.6	8.8	8.5-10.1
FGF-23 (RU/ml)	↑	-	608^*	-	72	<180

^*Abnormal laboratory findings. ^†Serum phosphate and FGF-23 levels were normalized 1 week after excision of phosphaturic mesenchymal tumor. POD: postoperative day, PTH: parathyroid hormone, FGF-23: fibroblast growth factor-23, NL: normal.

Notes

^{Financial support} None.

^{Conflict of interests} None.

References

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ORCID iDs

Young-Chang Park
https://orcid.org/0000-0003-3726-4707

Joon-Oh Seo
https://orcid.org/0000-0002-2070-3438

Kyu-Hyun Yang
https://orcid.org/0000-0001-7183-588X

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