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肿瘤相关性骨软化症合并术后骨吸收增加:病例报告。

Tumor-induced osteomalacia combined with increased bone resorption postoperatively: A case report.

机构信息

Endocrinology Department, First Affiliated Hospital of Third Military Medical University (Army Medical University), Chongqing, China.

出版信息

Medicine (Baltimore). 2023 Jul 7;102(27):e34217. doi: 10.1097/MD.0000000000034217.

Abstract

RATIONALE

Rare tumor-induced osteomalacia (TIO) usually resulted in bone pain, fragility fractures and muscle weakness in clinical, which is caused by the reduced phosphate reabsorption, thus impaired mineralization of the bone matrix and free energy transfer. The specific problems in postsurgical patients are obscure although surgical removal of the tumor is the only definitive treatment. Here, we documented a female TIO patient who suffered more severe bone pain and muscle spasms post-operation. Further, we presented and discussed our explanation for the unexpected symptoms.

PATIENT CONCERNS

The main symptoms were whole-body pain and muscle weakness. The patient also presented with osteoporosis and multiple fractures.

DIAGNOSIS

Elevated serum fibroblast growth factor 23 (FGF23) level and hypophosphatemia indicated the diagnosis of TIO. Positron emission tomography (PET)/computed tomography (CT) with 68 Ga-DOTATATE located the tumor in the dorsolateral part of the left foot. Histopathological examinations confirmed the diagnosis.

INTERVENTIONS

The tumor was surgically removed immediately after the diagnosis of TIO and localization of the tumor. Postoperatively, calcium carbonate supplement treatment was continued.

OUTCOMES

Two days after surgery, the serum FGF23 level was decreased to the normal range. Five days after surgery, N-terminal propeptide of type I procollagen and β-CrossLaps (β-CTx) had a remarkable increase. A month after surgery, the patient N-terminal propeptide of type I procollagen and β-CTx levels were decreased obviously, and serum FGF23, phosphate and 24h urinary phosphate were in the normal range.

LESSONS

We report a female patient who presented with osteoporosis and fractures. She was found with an elevation of FGF23 and diagnosis with TIO after PET/CT scanning. After surgically removing the tumor, the patient experienced more severe bone pain and muscle spasms. Active bone remodeling might be the reason for the symptoms. Further study will reveal the specific mechanism for this abnormal bone metabolism.

摘要

背景

罕见的肿瘤诱导性骨软化症(TIO)通常会导致患者出现骨痛、脆性骨折和肌肉无力等临床症状,这是由于磷酸盐重吸收减少,从而导致骨基质矿化和自由能传递受损所致。尽管手术切除肿瘤是唯一的根治方法,但术后患者的具体问题仍不清楚。在此,我们记录了一例 TIO 女性患者,其在手术后出现更严重的骨痛和肌肉痉挛。此外,我们提出并讨论了对这些意外症状的解释。

病例描述

主要症状为全身疼痛和肌肉无力。该患者还患有骨质疏松症和多处骨折。

诊断

血清成纤维细胞生长因子 23(FGF23)水平升高和低磷血症提示 TIO 诊断。68Ga-DOTATATE 正电子发射断层扫描(PET)/计算机断层扫描(CT)定位肿瘤位于左足背外侧。组织病理学检查证实了诊断。

干预措施

在诊断 TIO 并定位肿瘤后,立即进行手术切除肿瘤。术后继续进行碳酸钙补充治疗。

结果

术后 2 天,血清 FGF23 水平降至正常范围。术后 5 天,I 型前胶原 N 端前肽和β-CrossLaps(β-CTX)显著增加。术后 1 个月,患者 I 型前胶原 N 端前肽和β-CTX 水平明显降低,血清 FGF23、磷和 24 小时尿磷均恢复正常。

教训

我们报告了一例女性患者,表现为骨质疏松症和骨折。经 PET/CT 扫描发现其 FGF23 升高,诊断为 TIO。肿瘤切除后,患者出现更严重的骨痛和肌肉痉挛。活跃的骨重塑可能是导致这些症状的原因。进一步的研究将揭示这种异常骨代谢的具体机制。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f2be/10328624/69eb8d51ace5/medi-102-e34217-g001.jpg

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