Rad Sevil Ghaffarzadeh, Bahrami Amir, Houshyar Jalil, Farrin Nazila, Najafipour Farzad, Amirazad Halimeh
Endocrine Research Center, Tabriz University of Medical Sciences, Tabriz, Iran.
Nutrition Research Center, Tabriz University of Medical Sciences, Tabriz, Iran.
Bone Rep. 2024 Sep 10;22:101804. doi: 10.1016/j.bonr.2024.101804. eCollection 2024 Sep.
Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome defined by severe hypophosphatemia, bone loss, fractures, and muscle weakness. Identifying of the tumor site is often difficult. The primary treatment for Tumor-induced osteomalacia (TIO) is currently surgical resection. Removing the primary tumor is the most definitive treatment for this disease.
Here we describe the case of a 32-year-old man who exhibited sever muscle weakness and pain that had continued for three years. The patient has three sisters and one brother, all of whom are completely healthy and free of bone and muscle problems.Laboratory data indicate low serum phosphorus, normal serum and urine calcium level, besides raised alkaline phosphatase level. Due to elevated phosphorus levels in the urine and the lack of an alternative source for phosphorus excretion, along with the absence of short stature, bone deformities, and a negative family history that might suggest the potential for Tumor-induced osteomalacia (TIO), an octreotide scan was performed to the localized the tumor site. The scan, corroborated by CT and MRI scans, displayed absorption in the right maxillary sinus. Surgical excision of the lesion confirmed it to be a central giant cell granuloma.
Following surgery and without receiving any other treatment, the patient's phosphorus levels and clinical condition improved compared to before the surgical treatment. Subsequently, the symptoms of muscle weakness and skeletal pain significantly diminished, and the patient regained the ability to move.
Tumor enucleation was conducted, and the pathological examination of the maxillary sinus lesion unveiled a central Giant cell granuloma. The patient had clinical and laboratory improvement after surgery. This finding confirmed our diagnosis of a paraneoplastic hypophosphatemia associated with a giant cell granuloma.
肿瘤诱导的骨软化症(TIO)是一种罕见的副肿瘤综合征,其特征为严重低磷血症、骨质流失、骨折和肌肉无力。确定肿瘤部位通常很困难。肿瘤诱导的骨软化症(TIO)的主要治疗方法目前是手术切除。切除原发肿瘤是治疗该疾病最有效的方法。
在此,我们描述了一名32岁男性的病例,该患者表现出严重的肌肉无力和疼痛,持续了三年。患者有三个姐妹和一个兄弟,他们都完全健康,没有骨骼和肌肉问题。实验室数据显示血清磷水平低,血清和尿钙水平正常,碱性磷酸酶水平升高。由于尿磷水平升高且缺乏磷排泄的替代来源,同时没有可能提示肿瘤诱导的骨软化症(TIO)的身材矮小、骨骼畸形和家族史阴性,因此进行了奥曲肽扫描以定位肿瘤部位。该扫描结果经CT和MRI扫描证实,显示右上颌窦有吸收。病变的手术切除证实其为中央巨细胞肉芽肿。
手术后且未接受任何其他治疗,患者的磷水平和临床状况较手术治疗前有所改善。随后,肌肉无力和骨骼疼痛的症状明显减轻,患者恢复了活动能力。
进行了肿瘤摘除术,上颌窦病变的病理检查显示为中央巨细胞肉芽肿。患者术后临床和实验室检查结果有所改善。这一发现证实了我们对与巨细胞肉芽肿相关的副肿瘤性低磷血症的诊断。