A Case of Femoral Neck Insufficiency Fracture due to Tumor-Induced Osteomalacia

Tumor-induced osteomalacia (TIO) is a rare skeletal disease caused by hypersecretion of fibroblast growth factor 23 (FGF-23) from neoplasms of mesenchymal origin; patients with TIO present with insufficiency fractures, progressive bone pain, and delayed fracture unions. Herein, we report the case of...

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Main Authors: Yu Inoue, Tomoaki Fukui, Keisuke Oe, Shinya Hayashi, Teruya Kawamoto, Ryosuke Kuroda, Takahiro Niikura
Format: Article
Language:English
Published: Wiley 2021-01-01
Series:Case Reports in Orthopedics
Online Access:http://dx.doi.org/10.1155/2021/6668006
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author Yu Inoue
Tomoaki Fukui
Keisuke Oe
Shinya Hayashi
Teruya Kawamoto
Ryosuke Kuroda
Takahiro Niikura
author_facet Yu Inoue
Tomoaki Fukui
Keisuke Oe
Shinya Hayashi
Teruya Kawamoto
Ryosuke Kuroda
Takahiro Niikura
author_sort Yu Inoue
collection DOAJ
description Tumor-induced osteomalacia (TIO) is a rare skeletal disease caused by hypersecretion of fibroblast growth factor 23 (FGF-23) from neoplasms of mesenchymal origin; patients with TIO present with insufficiency fractures, progressive bone pain, and delayed fracture unions. Herein, we report the case of a 48-year-old man with an insufficiency fracture in his left femoral neck associated with TIO. The causative tumor located in the patient’s maxillary sinus had been resected; however, complete resection was impossible due to the location of the tumor. Therefore, the patient’s osteomalacia persisted, and he experienced a left femoral neck fracture in the absence of severe trauma. Because delayed fracture union was anticipated in this patient, we performed an internal fixation using an implant with a lateral plate for angular stability and multiple screws for rotational stability. Although fracture union took 15 months, the patient’s postoperative course was uneventful, and he could walk without any symptoms or assistance at his most recent follow-up 30 months after surgery. In TIO, hypersecretion of FGF-23 leads to increased renal excretion of phosphorus, increased bone resorption of calcium and phosphorus, decreased osteoblastic bone mineralization, and decreased gastrointestinal absorption of calcium and phosphorus, leading to insufficiency fractures and delayed fracture unions. Diagnosis of TIO is often delayed due to its rarity and vague symptoms. Total resection of the causative tumor is the optimal treatment; however, in cases wherein complete tumor resection is impossible, drug therapy may be insufficient, and the underlying TIO pathology, including bone fragility, may persist. Early diagnosis of TIO is important for preventing insufficiency fractures; however, when fractures are unavoidable, the surgical treatment of femoral neck fractures in patients with osteomalacia should account for a longer time frame for complete fracture union and therefore utilize implants with sufficient stability.
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spelling doaj-art-888f3dfe8f2b4403a4723439e591ea312025-08-20T02:09:17ZengWileyCase Reports in Orthopedics2090-67492090-67572021-01-01202110.1155/2021/66680066668006A Case of Femoral Neck Insufficiency Fracture due to Tumor-Induced OsteomalaciaYu Inoue0Tomoaki Fukui1Keisuke Oe2Shinya Hayashi3Teruya Kawamoto4Ryosuke Kuroda5Takahiro Niikura6Department of Orthopaedic Surgery, Kobe University Graduate School of Medicine, 7-5-1, Kusunoki-cho, Chuo-ku, Kobe 650-0017, JapanDepartment of Orthopaedic Surgery, Kobe University Graduate School of Medicine, 7-5-1, Kusunoki-cho, Chuo-ku, Kobe 650-0017, JapanDepartment of Orthopaedic Surgery, Kobe University Graduate School of Medicine, 7-5-1, Kusunoki-cho, Chuo-ku, Kobe 650-0017, JapanDepartment of Orthopaedic Surgery, Kobe University Graduate School of Medicine, 7-5-1, Kusunoki-cho, Chuo-ku, Kobe 650-0017, JapanDepartment of Orthopaedic Surgery, Kobe University Graduate School of Medicine, 7-5-1, Kusunoki-cho, Chuo-ku, Kobe 650-0017, JapanDepartment of Orthopaedic Surgery, Kobe University Graduate School of Medicine, 7-5-1, Kusunoki-cho, Chuo-ku, Kobe 650-0017, JapanDepartment of Orthopaedic Surgery, Kobe University Graduate School of Medicine, 7-5-1, Kusunoki-cho, Chuo-ku, Kobe 650-0017, JapanTumor-induced osteomalacia (TIO) is a rare skeletal disease caused by hypersecretion of fibroblast growth factor 23 (FGF-23) from neoplasms of mesenchymal origin; patients with TIO present with insufficiency fractures, progressive bone pain, and delayed fracture unions. Herein, we report the case of a 48-year-old man with an insufficiency fracture in his left femoral neck associated with TIO. The causative tumor located in the patient’s maxillary sinus had been resected; however, complete resection was impossible due to the location of the tumor. Therefore, the patient’s osteomalacia persisted, and he experienced a left femoral neck fracture in the absence of severe trauma. Because delayed fracture union was anticipated in this patient, we performed an internal fixation using an implant with a lateral plate for angular stability and multiple screws for rotational stability. Although fracture union took 15 months, the patient’s postoperative course was uneventful, and he could walk without any symptoms or assistance at his most recent follow-up 30 months after surgery. In TIO, hypersecretion of FGF-23 leads to increased renal excretion of phosphorus, increased bone resorption of calcium and phosphorus, decreased osteoblastic bone mineralization, and decreased gastrointestinal absorption of calcium and phosphorus, leading to insufficiency fractures and delayed fracture unions. Diagnosis of TIO is often delayed due to its rarity and vague symptoms. Total resection of the causative tumor is the optimal treatment; however, in cases wherein complete tumor resection is impossible, drug therapy may be insufficient, and the underlying TIO pathology, including bone fragility, may persist. Early diagnosis of TIO is important for preventing insufficiency fractures; however, when fractures are unavoidable, the surgical treatment of femoral neck fractures in patients with osteomalacia should account for a longer time frame for complete fracture union and therefore utilize implants with sufficient stability.http://dx.doi.org/10.1155/2021/6668006
spellingShingle Yu Inoue
Tomoaki Fukui
Keisuke Oe
Shinya Hayashi
Teruya Kawamoto
Ryosuke Kuroda
Takahiro Niikura
A Case of Femoral Neck Insufficiency Fracture due to Tumor-Induced Osteomalacia
Case Reports in Orthopedics
title A Case of Femoral Neck Insufficiency Fracture due to Tumor-Induced Osteomalacia
title_full A Case of Femoral Neck Insufficiency Fracture due to Tumor-Induced Osteomalacia
title_fullStr A Case of Femoral Neck Insufficiency Fracture due to Tumor-Induced Osteomalacia
title_full_unstemmed A Case of Femoral Neck Insufficiency Fracture due to Tumor-Induced Osteomalacia
title_short A Case of Femoral Neck Insufficiency Fracture due to Tumor-Induced Osteomalacia
title_sort case of femoral neck insufficiency fracture due to tumor induced osteomalacia
url http://dx.doi.org/10.1155/2021/6668006
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