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一名慢性血液透析患者出现严重头痛及左眼视力减退,结果发现是蝶窦棕色瘤。

Severe Headache and Deterioration of Vision in Left Eye in a Chronic Hemodialysis Patient Revealing a Brown Tumor of Sphenoid Sinus.

作者信息

Esselmani Hicham, Aimrane Abdelmohcine, Chatoui Hicham, El Hiba Omar, Najimi Mustapha, Merzouki Mohamed

机构信息

Biological Engineering Laboratory, Faculty of Sciences and Techniques, Sultan Moulay Slimane University, Beni Mellal 23000, Morocco.

Anthropogenetics, Biotechnologies and Health Laboratory, Nutritional Physiopathologies, Neurosciences and Toxicology Team, Faculty of Sciences, Chouaib Doukkali University, El Jadida 24000, Morocco.

出版信息

Neurol Int. 2025 Jan 30;17(2):22. doi: 10.3390/neurolint17020022.

DOI:10.3390/neurolint17020022
PMID:39997653
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11857977/
Abstract

BACKGROUND/OBJECTIVES: Brown tumors are rare bone lesions associated with hyperparathyroidism, particularly secondary hyperparathyroidism (SHPT), in chronic renal failure. While brown tumors commonly affect bones rich in marrow, the involvement of the sphenoid sinus is extremely rare and can present with neurological symptoms. This study reports a case of a sphenoid sinus brown tumor in a patient on hemodialysis, highlighting its clinical presentation and diagnostic challenges.

METHODS

A 31-year-old woman undergoing chronic hemodialysis presented with a severe headache, diplopia, and progressive vision loss in her left eye. Laboratory tests revealed hypercalcemia, hypophosphatemia, and elevated parathyroid hormone (PTH) levels, consistent with SHPT. The diagnosis was confirmed through a clinical examination and magnetic resonance imaging (MRI).

RESULTS

The clinical examination confirmed decreased visual acuity in the left eye. The laboratory results revealed serum calcium of 15.5 mg/dL, phosphate of 1.0 mg/dL, and PTH of 2000 pg/mL, consistent with SHPT. The imaging studies identified a brown tumor in the sphenoid sinus exerting a mass effect on adjacent structures. This case underscores the rarity of brown tumors in this location, with very few similar reports in the literature.

CONCLUSIONS

Although rare, brown tumors should be considered in patients with SHPT who present with neurological symptoms or cranial lesions. An early diagnosis through biochemical and imaging studies is crucial to prevent severe complications. The management involves treating the underlying hyperparathyroidism, with surgical intervention indicated in cases of neural compression.

摘要

背景/目的:棕色瘤是一种罕见的骨病变,与慢性肾衰竭中的甲状旁腺功能亢进,尤其是继发性甲状旁腺功能亢进(SHPT)相关。虽然棕色瘤通常影响富含骨髓的骨骼,但蝶窦受累极为罕见,且可出现神经症状。本研究报告了一例血液透析患者的蝶窦棕色瘤病例,突出了其临床表现和诊断挑战。

方法

一名31岁接受慢性血液透析的女性出现严重头痛、复视和左眼渐进性视力丧失。实验室检查显示高钙血症、低磷血症和甲状旁腺激素(PTH)水平升高,符合SHPT。通过临床检查和磁共振成像(MRI)确诊。

结果

临床检查证实左眼视力下降。实验室结果显示血清钙为15.5mg/dL,磷为1.0mg/dL,PTH为2000pg/mL,符合SHPT。影像学研究发现蝶窦有一个棕色瘤,对相邻结构产生占位效应。该病例强调了该部位棕色瘤的罕见性,文献中类似报道极少。

结论

尽管罕见,但对于出现神经症状或颅骨病变的SHPT患者应考虑棕色瘤。通过生化和影像学研究进行早期诊断对于预防严重并发症至关重要。治疗包括治疗潜在的甲状旁腺功能亢进,在神经受压的情况下需进行手术干预。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5798/11857977/5ca451a9321e/neurolint-17-00022-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5798/11857977/5ca451a9321e/neurolint-17-00022-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5798/11857977/5ca451a9321e/neurolint-17-00022-g001.jpg

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本文引用的文献

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Brown Tumors: The Hidden Face of Primary and Renal Hyperparathyroidism Amid Real-Life Settings.棕色瘤:现实环境中原发性和肾性甲状旁腺功能亢进的隐匿面貌。
J Clin Med. 2024 Jun 29;13(13):3847. doi: 10.3390/jcm13133847.
2
Brown tumors: Retrospective analysis of 26 cases.棕色瘤:26 例回顾性分析。
Arch Orthop Trauma Surg. 2024 Jul;144(7):2927-2934. doi: 10.1007/s00402-024-05372-9. Epub 2024 May 25.
3
Parathyroid Imaging: Past, Present, and Future.甲状旁腺成像:过去、现在和未来。
Front Endocrinol (Lausanne). 2022 Feb 25;12:760419. doi: 10.3389/fendo.2021.760419. eCollection 2021.
4
Mandibular Brown Tumor as a Result of Secondary Hyperparathyroidism: A Case Report with 5 Years Follow-Up and Review of the Literature.下颌骨棕色瘤继发甲状旁腺功能亢进症 1 例报告及文献复习并随访 5 年
Int J Environ Res Public Health. 2021 Jul 9;18(14):7370. doi: 10.3390/ijerph18147370.
5
Osteitis fibrosa cystica von Recklinghausen.冯雷克林霍增氏骨纤维性囊肿
J Nephrol. 2021 Jun;34(3):925-926. doi: 10.1007/s40620-020-00961-4. Epub 2021 Feb 8.
6
Disorders of Calcium and Phosphorus Metabolism and the Proteomics/Metabolomics-Based Research.钙磷代谢紊乱与基于蛋白质组学/代谢组学的研究
Front Cell Dev Biol. 2020 Sep 10;8:576110. doi: 10.3389/fcell.2020.576110. eCollection 2020.
7
A Systematic Review of Sellar and Parasellar Brown Tumors: An Analysis of Clinical, Diagnostic, and Management Profiles.鞍区及鞍旁棕色瘤的系统评价:临床、诊断和治疗特征分析。
World Neurosurg. 2019 Dec;132:e423-e429. doi: 10.1016/j.wneu.2019.08.126. Epub 2019 Aug 27.
8
Clinical Guidelines and PTH Measurement: Does Assay Generation Matter?临床指南和甲状旁腺激素(PTH)测定:检测方法的建立是否重要?
Endocr Rev. 2019 Dec 1;40(6):1468-1480. doi: 10.1210/er.2018-00220.
9
A case of adolescent giant parathyroid adenoma presenting multiple osteolytic fractures and postoperative hungry bone syndrome.一例青少年巨大甲状旁腺腺瘤伴多发溶骨性骨折及术后饥饿骨综合征
Clin Case Rep. 2015 Oct;3(10):835-40. doi: 10.1002/ccr3.360. Epub 2015 Sep 2.
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Brown tumor of the cervical spines: a case report with literature review.颈椎棕色瘤:一例病例报告并文献复习
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