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与肿瘤性骨软化症相关的椎体病变的临床特征和手术结果:16 例患者报告及文献复习。

Clinical characteristics and surgical outcomes of vertebral lesions associated with tumor-induced osteomalacia: report of 16 patients and review of the literature.

机构信息

Department of Endocrinology, Key Laboratory of Endocrinology, National Commission of Health, Peking Union Medical College Hospital, Chinese Academy of Medical Science, No. 1 Shuaifuyuan, Wangfujing Street, Dongcheng District, Beijing, 100730, China.

Department of Orthopedic Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China.

出版信息

Osteoporos Int. 2024 Nov;35(11):1951-1962. doi: 10.1007/s00198-024-07178-2. Epub 2024 Aug 2.

Abstract

UNLABELLED

Vertebral tumors in patients with tumor-induced osteomalacia (TIO) have a low diagnostic rate and poor postoperative outcomes. The application of  Ga-DOTATATE-PET/CT significantly increased the detection rate. Compared with tumor curettage, segmental resection was recommended as the preferred surgical type due to its high recovery rate.

PURPOSE

Tumor-induced osteomalacia (TIO) is an acquired hypophosphatemic osteomalacia, and surgery is the first-line therapy. Most TIO tumors are found in the bones of the appendicular skeleton, cranium, and paranasal sinuses but rarely in the vertebrae. Tumor curettage and segmental resection are the two main surgical options for vertebral TIO patients. However, research on the clinical characteristics and surgical prognosis of vertebral TIO patients is rare. In the present study, for the first time, we investigated the clinical characteristics of 16 vertebral TIO patients and compared the surgical outcomes of patients who underwent surgery via two different surgical methods.

METHODS

This was a retrospective cohort study. In this study, we included 16 adult TIO patients with lesions in vertebrae from Peking Union Medical College Hospital (PUMCH), all of whom underwent surgery. Baseline laboratory data were collected through medical records review. Technetium-99 m octreotide scintigraphy (Tc-OCT) and gallium-DOTA-TATE-positron emission tomography/computed tomography ( Ga-DOTATATE-PET/CT) were conducted at the Department of Nuclear Medicine of PUMCH. The tumor histopathology was confirmed by a senior pathologist at our center.

RESULTS

Vertebral TIO patients had lower serum phosphorus and TmP/GFR and higher serum alkaline phosphatase (ALP), serum parathyroid hormone (PTH), and serum C-terminal cross-linked telopeptide of type I collagen (β-CTX) levels than the normal range. The sensitivity of  Ga‒DOTATATE PET/CT was 100%, significantly greater than that of Tc-OCT (40%). After comparing the outcomes between the two surgical methods, we found that the recovery rate after segmental resection (62.5%) was greater than that after tumor curettage (12.5%). In the thoracic and sacral vertebrae, segmental resection surgery had a good prognosis.

CONCLUSION

Ga-DOTATATE PET/CT could serve as the first diagnostic tool in patients with vertebral TIO, and segmental resection could be used as the preferred surgery. This study would raise awareness of the clinical features and management of these rare vertebral TIO patients.

摘要

目的

肿瘤诱导性骨软化症(TIO)是一种获得性低磷性骨软化症,手术是一线治疗方法。大多数 TIO 肿瘤发生在四肢骨骼、颅骨和副鼻窦,但很少发生在椎骨。肿瘤刮除术和节段切除术是治疗椎骨 TIO 患者的两种主要手术选择。然而,关于椎骨 TIO 患者的临床特征和手术预后的研究很少。在本研究中,我们首次调查了 16 例椎骨 TIO 患者的临床特征,并比较了两种不同手术方法治疗的患者的手术结果。

方法

这是一项回顾性队列研究。在这项研究中,我们纳入了 16 例来自北京协和医院(PUMCH)的椎骨 TIO 成年患者,所有患者均接受了手术。通过病历回顾收集基线实验室数据。核医学科进行锝-99m 奥曲肽闪烁扫描(Tc-OCT)和镓-DOTA-TATE-正电子发射断层扫描/计算机断层扫描(Ga-DOTATATE-PET/CT)。肿瘤组织病理学由我们中心的一位资深病理学家证实。

结果

椎骨 TIO 患者的血清磷和 TmP/GFR 较低,而血清碱性磷酸酶(ALP)、血清甲状旁腺激素(PTH)和血清 I 型胶原 C 端交联肽(β-CTX)水平较高。Ga-DOTATATE-PET/CT 的灵敏度为 100%,明显高于 Tc-OCT(40%)。比较两种手术方法的结果后,我们发现节段切除术(62.5%)的恢复率大于肿瘤刮除术(12.5%)。在胸腰椎,节段切除术有良好的预后。

结论

Ga-DOTATATE-PET/CT 可作为椎骨 TIO 患者的首选诊断工具,节段切除术可作为首选手术方法。本研究将提高对这些罕见椎骨 TIO 患者的临床特征和治疗的认识。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1fff/11499524/51f1ee31a382/198_2024_7178_Fig1_HTML.jpg

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