Department of Clinical, Internal, Anesthesiologic and Cardiovascular Sciences, Sapienza University of Rome, Viale del Policlinico 155, 00161, Rome, Italy.
Department of Molecular Medicine, Sapienza University, Rome, Italy.
Arch Osteoporos. 2023 Jul 12;18(1):94. doi: 10.1007/s11657-023-01305-y.
Tumor-induced osteomalacia (TIO) is an uncommon paraneoplastic syndrome due to the overproduction of fibroblast growth factor 23 (FGF23). It is predominantly caused by mesenchymal tumors and cured upon their complete removal. Non-surgical treatment is an alternative option but limited to specific clinical conditions.
We report a challenging case of TIO caused by a tumor involving the occipital bone. We also performed a literature review of TIO caused by tumors localized at this site, focusing on clinical findings, treatment, and outcomes.
The patient, a 62-year-old male, presented with a long-lasting history of progressive weakness. Biochemical evaluation revealed severe hypophosphatemia due to low renal tubular reabsorption of phosphate with raised intact FGF23 values. A Ga-DOTATATE PET/TC imaging showed a suspicious lesion located in the left occipital bone that MRI and selective venous catheterization confirmed to be the cause of TIO. Stereotactic gamma knife radiosurgery was carried out, but unfortunately, the patient died of acute respiratory failure. To date, only seven additional cases of TIO have been associated to tumors located in the occipital bone. Furthermore, the tumor involved the left side of the occipital bone in all these patients.
The occipital region is a difficult area to access so a multidisciplinary approach for their treatment is required. If anatomical differences could be the basis for the predilection of the left side of the occipital bone, it remains to be clarified.
肿瘤相关性骨软化症(TIO)是一种罕见的副瘤综合征,由成纤维细胞生长因子 23(FGF23)过度产生引起。它主要由间叶肿瘤引起,通过完全切除肿瘤即可治愈。非手术治疗是一种替代选择,但仅限于特定的临床情况。
我们报告了一例由枕骨肿瘤引起的 TIO 病例,该病例极具挑战性。我们还对发生在该部位的肿瘤引起的 TIO 进行了文献回顾,重点关注临床发现、治疗和结局。
患者为 62 岁男性,表现为进行性肌无力的长期病史。生化评估显示严重低磷血症,原因是肾小管对磷酸盐的重吸收减少,而完整的 FGF23 值升高。Ga-DOTATATE PET/CT 成像显示左枕骨可疑病变,MRI 和选择性静脉导管插入术证实该病变为 TIO 的病因。进行了立体定向伽玛刀放射外科手术,但不幸的是,患者死于急性呼吸衰竭。迄今为止,仅发现另外 7 例与位于枕骨的肿瘤相关的 TIO 病例。此外,所有这些患者的肿瘤均累及枕骨的左侧。
枕骨区域难以触及,因此需要多学科方法进行治疗。如果解剖差异可能是左侧枕骨易患性的基础,那么这仍有待阐明。