■在本文中,我们介绍了一例罕见的肿瘤诱导的骨软化症(TIO),并对这种罕见疾病进行了文献复习。
■报告1例孤立性蝶窦TIO。此外,本文对蝶窦和其他鼻窦TIO的临床特点进行了回顾和总结。
■一名35岁的肌肉无力和下背部疼痛的男子来到神经内科。没有发现明显的神经系统疾病;然而,四肢的磁共振成像意外显示腋窝有肿瘤。骨显像显示可疑骨转移。低磷血症被忽视。有趣的是,2-脱氧-2-[氟-18]氟-d-葡萄糖正电子发射断层扫描/计算机断层扫描(18F-FDGPET/CT)检测到腋下的肿瘤和蝶窦的肿瘤,但只有蝶窦肿瘤在68-镓1,4,7,10-四氮杂环十二烷-1,4,7,10-四乙酸奥曲叶酸(Ga-68DOTATATE)PET/CT中有生长抑素受体(SSTR)表达。蝶窦肿瘤被证明是磷性间充质肿瘤(PMT),手术后磷酸盐水平恢复正常。文献复习仅显示17例发生在蝶窦的TIOs,平均年龄43.3±13.7岁。只有3例蝶窦的TIO没有侵入鼻腔或其他鼻旁窦,可以确定为孤立的蝶窦疾病。我们比较了蝶骨TIO与非蝶骨鼻窦TIO的临床特征,发现蝶窦TIOs组的1,25-二羟基维生素D浓度远高于非蝶窦TIOs组。共对153例鼻窦TIOs进行了回顾。筛窦是主要部位(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例患者(43.1%)显示肿瘤侵入一个以上的窦。大多数肿瘤(69.3%)通过病理诊断为PMT,其次是血管外皮细胞瘤(14.3%)。免疫染色有助于这些肿瘤的鉴别诊断;然而,为了获得更好的准确性,需要更大的样本量。
■鼻窦的TIO,尤其是蝶窦,是罕见的。此外,孤立性蝶窦疾病易误诊。当骨软化症的临床表现不典型时,将它与蝶窦疾病联系起来更加困难。因此,蝶窦的TIO需要进一步探索。
In this paper, we present a rare case of tumor-induced osteomalacia (TIO) and a literature review of this rare disease.
A 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.
A 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.
TIO 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.