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Surgical margin for phosphaturic mesenchymal tumors in soft tissues: An analysis of the radiological histopathological correlation

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JOURNAL OF ORTHOPAEDIC SCIENCE
卷 26, 期 5, 页码 870-877

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DOI: 10.1016/j.jos.2020.07.006

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This study examined the optimal surgical margin for phosphaturic mesenchymal tumors by analyzing radiological and histopathological features. It found that tumors with irregular boundaries on imaging tend to be infiltrative and should be treated with a margin resection of at least 1 cm. Tumors with clear and regular margins can be cured by marginal resection.
Background: Phosphaturic mesenchymal tumors primarily cause tumor-induced osteomalacia, a rare paraneoplastic syndrome, and half occur in soft tissues. There are few reports about the surgical margins of these tumors. This study aimed to clarify the optimal surgical margin for phosphaturic mesenchymal tumors by analyzing radiological and histopathological features. Methods: This study included eight cases, seven primary and one recurrent, of tumor-induced osteo-malacia caused by soft-tissue phosphaturic mesenchymal tumors that were surgically treated between January 2000 and January 2019. We evaluated the radiological and histopathological features of all tu-mors and investigated the correlation of these features, the surgical margin, and recurrence of hypophosphatemia. Results: The tumors were located in superficial (n = 5) and deep (n = 3) tissues. Six of the eight tumors had a clear boundary, but five had an irregular margin. Three tumors had a hypointense rim on T2-weighted images, indicating fibrous tumor encapsulation. Histopathological analysis revealed infiltra-tive growth in six of the eight tumors, which correlated with an irregular margin seen on imaging. Although there was no recurrence in patients treated with an intended wide margin >1 cm, one of the three patients treated with marginal tumor resection experienced a recurrence of hypophosphatemia, with histopathological analysis showing infiltration of subcutaneous fat. In contrast, two tumors with clear boundaries, regular margins, and fibrous capsule seen on imaging, had no infiltrative growth and were cured by marginal resection. In one recurrent case, tumor infiltration was observed in the previous surgical scar, which was not detected on preoperative imaging. Conclusions: Soft-tissue phosphaturic mesenchymal tumors with an irregular boundary seen on imaging tend to be infiltrative, especially into subcutaneous fat, and should be treated by at least a 1-cm wide margin resection. Tumors with a fibrous capsule with clear and regular margins are cured by marginal margin resection. These findings could inform surgeons' decisions regarding the resection of soft-tissue phosphaturic mesenchymal tumors. (C) 2020 The Japanese Orthopaedic Association. Published by Elsevier B.V. All rights reserved.

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