Surgical Treatment of Tumor-Induced Osteomalacia With Causative Tumors Affecting the Long Bones.
- 1Department of Orthopaedic Surgery, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing, China.
- 2Department of Endocrinology, National Health Commission Key Laboratory of Endocrinology, State Key Laboratory of Complex Severe and Rare Diseases, Translational Medicine Center, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China.
- 3Institute of Orthopaedic & Musculoskeletal Science, University College London, Royal National Orthopaedic Hospital, London, UK.
Abstract
Background
The causative tumor for tumor-induced osteomalacia (TIO) locating in the long bone is hidden and has diverse locations, resulting in great challenges to orthopedists. This study aimed to investigate and summarize the clinical features and surgical outcomes of TIO patients with culprit tumors involving the long bones to enhance clinical understanding and improve orthopedic surgical management of this rare disease.Methods
A retrospective analysis was performed on the clinical data of TIO patients with causative tumors located in the long bones who underwent surgery by the Orthopedic Bone and Soft Tissue Tumor Sub-specialty Group at our hospital between January 2013 and January 2023. This article described the clinical symptoms, location of causative tumors, tumor sizes, and other clinical features of TIO patients, and analyzed the trend of changes in blood phosphorus levels before and after surgery and pathological results. At the same time, clinical surgical experience and strategies for TIO patients with culprit tumors located in long bones were summarized.Results
All 49 patients included in the study had been pathologically confirmed and met clinical diagnostic criteria for TIO. All patients experienced bone pain (49/49), often accompanied by limited mobility, fatigue, difficulty turning over, and difficulty sitting up or walking. Importantly, 65.3% (32/49) experienced height loss during disease progression. A total of 65 operations were performed on these 49 patients, with culprit tumors located in the humerus (five cases), femur (33 cases), tibia (seven cases), fibula (three cases), and foot (one case). Twenty-nine cases involved skeletal lesions, seven cases affecting the periosteum (outside-in), while 13 showed both bone and soft tissue involvement. Following the final orthopedic procedure, serum phosphorus levels increased in 45 patients, and the median follow-up duration was 45 months (interquartile range, 28-61 months; range, 2-215 months). Nine patients showed no remission after the first orthopedic surgery, and five experienced recurrence. One patient was diagnosed with a malignant culprit tumor.Conclusion
Surgical planning for TIO with long bone involvement should account for the anatomical location and extent of the causative tumor. Currently, no standardized surgical strategy exists for this rare condition. Recurrence, non-remission, or malignant transformation remains possible due to incomplete resection or local relapse. Based on the single center experience of this case series, we summarized the surgical classification and feasible surgical strategies for TIO's orthopedic surgery with causative tumors located in the long bones. The actual clinical application effect of this classification system needs further clinical data verification.
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BibTeX
@article{liu2026surgical,
title = {{Surgical Treatment of Tumor-Induced Osteomalacia With Causative Tumors Affecting the Long Bones.}},
author = {Shuzhong Liu and Jinyi Xing and An Song and Mingjing Zhang and Jin Jin and Xi Zhou and Yong Liu and Weibo Xia},
journal = {Orthopaedic surgery},
year = {2026},
month = oct,
doi = {10.1111/os.70449},
url = {https://europepmc.org/article/MED/42893843},
note = {CC BY-NC-ND},
}
RIS
TY - JOUR
TI - Surgical Treatment of Tumor-Induced Osteomalacia With Causative Tumors Affecting the Long Bones.
AU - Shuzhong Liu
AU - Jinyi Xing
AU - An Song
AU - Mingjing Zhang
AU - Jin Jin
AU - Xi Zhou
AU - Yong Liu
AU - Weibo Xia
PY - 2026
DA - 2026/10/18
JO - Orthopaedic surgery
DO - 10.1111/os.70449
UR - https://europepmc.org/article/MED/42893843
AB - Background The causative tumor for tumor-induced osteomalacia (TIO) locating in the long bone is hidden and has diverse locations, resulting in great challenges to orthopedists. This study aimed to investigate and summarize the clinical features and surgical outcomes of TIO patients with culprit tumors involving the long bones to enhance clinical understanding and improve orthopedic surgical management of this rare disease.Methods A retrospective analysis was performed on the clinical data of TIO patients with causative tumors located in the long bones who underwent surgery by the Orthopedic Bone and Soft Tissue Tumor Sub-specialty Group at our hospital between January 2013 and January 2023. This article described the clinical symptoms, location of causative tumors, tumor sizes, and other clinical features of TIO patients, and analyzed the trend of changes in blood phosphorus levels before and after surgery and pathological results. At the same time, clinical surgical experience and strategies for TIO patients with culprit tumors located in long bones were summarized.Results All 49 patients included in the study had been pathologically confirmed and met clinical diagnostic criteria for TIO. All patients experienced bone pain (49/49), often accompanied by limited mobility, fatigue, difficulty turning over, and difficulty sitting up or walking. Importantly, 65.3% (32/49) experienced height loss during disease progression. A total of 65 operations were performed on these 49 patients, with culprit tumors located in the humerus (five cases), femur (33 cases), tibia (seven cases), fibula (three cases), and foot (one case). Twenty-nine cases involved skeletal lesions, seven cases affecting the periosteum (outside-in), while 13 showed both bone and soft tissue involvement. Following the final orthopedic procedure, serum phosphorus levels increased in 45 patients, and the median follow-up duration was 45 months (interquartile range, 28-61 months; range, 2-215 months). Nine patients showed no remission after the first orthopedic surgery, and five experienced recurrence. One patient was diagnosed with a malignant culprit tumor.Conclusion Surgical planning for TIO with long bone involvement should account for the anatomical location and extent of the causative tumor. Currently, no standardized surgical strategy exists for this rare condition. Recurrence, non-remission, or malignant transformation remains possible due to incomplete resection or local relapse. Based on the single center experience of this case series, we summarized the surgical classification and feasible surgical strategies for TIO's orthopedic surgery with causative tumors located in the long bones. The actual clinical application effect of this classification system needs further clinical data verification.
SN - 1757-7853
SN - 1757-7861
ER -