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

Inoue, Yu Fukui, Tomoaki Ooe, Keisuke Hayashi, Shinya Kawamoto, Teruya Kuroda, Ryosuke Niikura, Takahiro 神戸大学

2021.10.06

概要

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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参考文献

Figure 6: Radiographs taken 15 months after surgery revealing that

the fracture achieved bone union.

with a side plate for angular stability and multiple screws for

rotational stability. We suspected that the use of an implant

with these characteristics ultimately led to a successful bone

union without failure of internal fixation; however, bony

fusion did take 15 months to occur.

Delayed diagnosis of TIO can lead to weakness of the

bony architecture due to prolonged inhibition of bone

matrix mineralization and resultant insufficiency fractures.

For early diagnosis, monitoring serum alkaline phosphatase

and phosphate levels as well as screening for muscle weakness, joint pain, and gait disturbance is crucial and can prevent additional serious complications. When insufficiency

fractures are detected in young people, clinicians should

consider the possibility of bone metabolism disorders,

including osteomalacia and, if necessary, make an early

referral to an endocrinologist.

In conclusion, we successfully completed the surgical

treatment of a femoral neck insufficiency fracture in a

patient with TIO whose causative tumor could not be

completely removed. Based on our experience with this case,

we strongly believe that rigid osteosynthesis and careful

postoperative follow-up are important for achieving successful outcomes in such cases.

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