Marcodomenico Mazza, Gaetano Paride Arcidiacono, Ilda Hoxhaj, Virginia Padoan, Giulia Tasca, Marta Burei, Stefania Sella, Paolo Simioni, Sandro Giannini, Simone Mocellin
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引用次数: 0
Abstract
Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome characterized by hypophosphatemia, bone mineralization disorders with increased risk of fragility fractures, muscle pain, and progressive weakness. TIO has been associated with increased production of the phosphaturic hormone Fibroblast Growth Factor 23 (FGF23) usually by mesenchymal tumors of soft tissue or bone (Phosphaturic Mesenchymal Tumors-PMTs). In rare cases TIO may be observed in association with other malignancies. We report the case of a 66-year-old woman with an occasional diagnosis of both a PMT and an ovarian cancer during the evaluation of TIO. We also systematically review the literature to discover possible correlations between osteomalacia, FGF23 production, and ovarian cancer. Four studies were eligible for the analysis. Two case reports described an association between TIO development and ovarian cancer, whereas the two case-control studies hypothesized a possible correlation between FGF/FGF receptor axis and cancer development. Although it does not provide conclusive evidence regarding the association between TIO and ovarian cancer, this case report highlights the possibility that in the diagnostic workup of suspected TIO, both FGF23-secreting tumors distinct from PMT and tumors unrelated to the clinical presentation of TIO could be identified. This information is important for guiding successful tumor staging and determining the necessity for surgical intervention and/or eventual adjuvant therapy.
肿瘤诱发骨软化症(TIO)是一种罕见的副肿瘤综合征,其特征是低磷血症、骨矿化障碍,并伴有脆性骨折风险增加、肌肉疼痛和进行性虚弱。TIO 与成纤维细胞生长因子 23(FGF23)这种磷酸化激素的分泌增加有关,这种激素通常由软组织或骨间充质肿瘤(磷酸化间充质肿瘤-PMTs)产生。在极少数情况下,TIO 可能与其他恶性肿瘤同时出现。我们报告了一例 66 岁女性的病例,她在评估 TIO 时偶尔被诊断为 PMT 和卵巢癌。我们还系统地回顾了相关文献,以发现骨质疏松症、FGF23的产生与卵巢癌之间可能存在的关联。有四项研究符合分析条件。两篇病例报告描述了 TIO 的发展与卵巢癌之间的关联,而两篇病例对照研究则假设 FGF/FGF 受体轴与癌症发展之间可能存在关联。本病例报告虽然没有提供 TIO 与卵巢癌之间相关性的确凿证据,但强调了在疑似 TIO 的诊断工作中,既可能发现与 PMT 不同的分泌 FGF23 的肿瘤,也可能发现与 TIO 临床表现无关的肿瘤。这些信息对于指导肿瘤成功分期、确定手术干预和/或最终辅助治疗的必要性非常重要。