Table_1_Sphenoid sinus is a rare site for tumor-induced osteomalacia: A case report and literature review.docx

BackgroundIn this paper, we present a rare case of tumor-induced osteomalacia (TIO) and a literature review of this rare disease.MethodsA case of TIO of the isolated sphenoid sinus was reported. Furthermore, the clinical features of TIO in the sphenoid sinus and other sinonasal sinuses were also reviewed and summarized.ResultsA 35-year-old man with muscle weakness and lower back pain came to the Department of Neurology. No obvious neurological disease was found; however, magnetic resonance imaging of the extremities accidentally showed a tumor in the axilla. Bone scintigraphy showed suspicious bone metastasis. Hypophosphatemia was neglected. Interestingly, 2-deoxy-2-[fluorine-18]fluoro-d-glucose positron emission tomography/computed tomography (18F-FDG PET/CT) detected a tumor in the axilla and another in the sphenoid sinus, but only the tumor in the sphenoid sinus had somatostatin receptor (SSTR) expression in 68-gallium 1,4,7,10-tetraazacyclododecane-1,4,7,10-tetraacetic acid octreotate (Ga-68 DOTATATE) PET/CT. The sphenoid sinus tumor was proven to be a phosphaturic mesenchymal tumor (PMT), and the phosphate levels returned to normal after surgery. The literature review showed only 17 cases of TIOs that occurred in the sphenoid sinus, with an average age of 43.3 ± 13.7 years. Only three cases of TIOs in the sphenoid sinus did not invade the nasal cavity or other paranasal sinuses, which could be identified as isolated sphenoid sinus diseases. We compared the clinical features of sphenoid TIOs with those of non-sphenoid sinonasal TIOs, and it was found that the concentration of 1,25-dihydroxy vitamin D in the group with sphenoid TIOs was much higher than that in the group with non-sphenoid sinonasal TIOs. A total of 153 cases of TIOs in the sinonasal sinus were reviewed. The ethmoid sinus was found to be the major site (64.7%), followed by the nasal cavity (50.3%), maxillary sinus (19.0%), frontal sinus (16.4%), and sphenoid sinus (11.8%). There were 66 patients (43.1%) who showed tumors invading more than one sinus. Most of the tumors (69.3%) were diagnosed as PMTs by pathology, followed by hemangiopericytoma (14.3%). Immunostaining was beneficial in the differential diagnosis of these tumors; however, larger sample sizes are needed for better accuracy.ConclusionTIO in the sinonasal sinus, especially in the sphenoid sinus, is rare. Moreover, isolated sphenoid sinus disease can be easily misdiagnosed. When the clinical manifestation of osteomalacia is atypical, associating it with sphenoid sinus disease is even more difficult. Thus, TIO in the sphenoid sinus needs further exploration.

背景：本研究报告了一例罕见的肿瘤诱导性骨软化症（TIO）病例，并对其相关文献进行了综述。方法：报道了一例孤立性蝶窦TIO病例，并对蝶窦和其他鼻窦TIO的临床特征进行了回顾和总结。结果：一名35岁男性因肌无力及下背部疼痛就诊于神经科。未发现明显的神经系统疾病；然而，四肢的磁共振成像偶然发现腋窝处有肿瘤。骨显像显示可疑的骨转移。低磷血症被忽视。有趣的是，2-脱氧-2-[氟-18]氟代-D-葡萄糖正电子发射断层扫描/计算机断层扫描（18F-FDG PET/CT）检测到腋窝和蝶窦处的肿瘤，但只有蝶窦处的肿瘤在68-锝-1,4,7,10-四氮杂环十二烷-1,4,7,10-四乙酸辛酸酯（Ga-68 DOTATATE）PET/CT中表达生长抑素受体（SSTR）。经证实，蝶窦肿瘤为磷尿性间充质肿瘤（PMT），术后磷水平恢复正常。文献综述显示，蝶窦TIO病例仅有17例，平均年龄为43.3±13.7岁。蝶窦TIO病例中，仅3例未侵犯鼻腔或其他副鼻窦，可被识别为孤立性蝶窦疾病。我们将蝶窦TIO的临床特征与非蝶窦鼻窦TIO的临床特征进行了比较，发现蝶窦TIO组的1,25-二羟基维生素D浓度显著高于非蝶窦鼻窦TIO组。共回顾了鼻窦TIO病例153例。筛窦被认为是主要部位（64.7%），其次是鼻腔（50.3%）、上颌窦（19.0%）、额窦（16.4%）和蝶窦（11.8%）。有66名患者（43.1%）表现出肿瘤侵犯多个窦。其中大部分肿瘤（69.3%）经病理诊断为磷尿性间充质肿瘤（PMT），其次是血管外皮瘤（14.3%）。免疫组化有助于这些肿瘤的鉴别诊断；然而，需要更大的样本量以提高准确性。结论：鼻窦TIO，尤其是在蝶窦，是一种罕见疾病。此外，孤立性蝶窦疾病易于误诊。当骨软化症的临床表现不典型时，将其与蝶窦疾病关联更加困难。因此，蝶窦TIO需要进一步研究。