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

doi:10.12671/jkfs.2017.30.3.146

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Case Report
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.; Journal of the Korean Fracture Society 2017;30(3):146-150.
DOI: https://doi.org/10.12671/jkfs.2017.30.3.146
Published online: July 21, 2017

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: May 2, 2017 • Revised: May 27, 2017 • Accepted: May 27, 2017

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.

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Abstract
NOTES
REFERENCES

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.
Key words: Oncogenic osteomalacia, Insufficiency fractures, Hypophosphatemia

None.

REFERENCES

1. 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;16:33-46.
2. Kumar R, Folpe AL, Mullan BP. Tumor-induced osteomalacia. Transl Endocrinol Metab, 2015;7:1871.
3. 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;471:3618-3625.
4. Jan de Beur SM. Tumor-induced osteomalacia. JAMA, 2005;294:1260-1267.
5. Jiang Y, Xia WB, Xing XP, et al. Tumor-induced osteomalacia: an important cause of adult-onset hypophosphatemic osteomalacia in China: report of 39 cases and review of the literature. J Bone Miner Res, 2012;27:1967-1975.
6. Chong WH, Andreopoulou P, Chen CC, et al. Tumor localization and biochemical response to cure in tumor-induced osteomalacia. J Bone Miner Res, 2013;28:1386-1398.
7. Zhang J, Zhu Z, Zhong D, et al. 68Ga DOTATATE PET/CT is an accurate imaging modality in the detection of culprit tumors causing osteomalacia. Clin Nucl Med, 2015;40:642-646.
8. Andreopoulou P, Dumitrescu CE, Kelly MH, et al. Selective venous catheterization for the localization of phosphaturic mesenchymal tumors. J Bone Miner Res, 2011;26:1295-1302.
9. Umphrey LG, Whitaker MD, Bosch EP, Cook CB. Clinical and bone density outcomes of tumor-induced osteomalacia after treatment. Endocr Pract, 2007;13:458-462.

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.

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Oncogenic Osteomalacia with Multiple Insufficiency Fractures: A Case Report

J Korean Fract Soc. 2017;30(3):146-150. Published online July 31, 2017

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

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Oncogenic Osteomalacia with Multiple Insufficiency Fractures: A Case Report

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.

Fig. 1

Fig. 2

Fig. 3

Fig. 4

Fig. 5

Fig. 6

Oncogenic Osteomalacia with Multiple Insufficiency Fractures: A Case Report

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

Table 1 Laboratory Findings in Oncogenic Osteomalcia