Suppr超能文献

伴有淋巴结肿大的不明原因发热的罕见病因——复发性多软骨炎:一例报告

An unusual cause of fever of unknown origin with enlarged lymph nodes-relapsing polychondritis: A case report.

作者信息

Liu Wei, Jiang Hongli, Jing Han, Mao Bing

机构信息

Division of Respiratory Medicine, Department of Integrated Traditional Chinese and Western Medicine, West China Hospital, Sichuan University, Chengdu, Sichuan, People's Republic of China.

出版信息

Medicine (Baltimore). 2017 Nov;96(46):e8734. doi: 10.1097/MD.0000000000008734.

Abstract

INTRODUCTION

Fever of unknown origin (FUO) is a common initial presentation leading to a diagnostic challenge.

PATIENT CONCERNS

A 3-month history of moderate-to-high fever was reported in an otherwise healthy 54-year-old man. Enhanced computed tomography (CT) scans of his chest showed a remarkable progressive enlargement of bilateral cervical, supraclavicular, hilar, and mediastinal lymph nodes within 2 weeks. Bronchofibroscopy manifested obvious luminal stenosis with swelling, thick pale mucosa, and disappearing of structures of trachea cricoid cartilage, followed by a 18F-fluorodeoxyglucose positron-emission tomography-computed tomography (18F-FDG PET/CT) with intense symmetric FDG uptake in larynx, tracheobronchial tree, and hilar, mediastinal, and axillary lymph nodes being demonstrated.

DIAGNOSIS

A diagnosis of relapsing polychondritis (RP) was finally reached.

INTERVENTIONS

The patient received methylprednisolone 40 mg daily with a gradual tapering in a 4-month follow-up.

OUTCOMES

The patient experienced no relapse of fever and lymph nodes enlargement in the 4-month follow-up.

LESSONS

Even though long-term fever with multiple lymphadenectasis usually lead to a diagnosis of lymphoma, the bronchoscopic features and evidence from 18F-FDG PET/CT in this case were much more approximate to RP, indicating an importance of a sensible differential diagnosis of RP in patients who present with nonspecific features such as FUO and lymph nodes enlargement. Keeping a high index of clinical suspicion in these patients can help recognize uncommon of RP and promote diagnosis and treatment. Our case highlights the significance of 18F-FDG PET/CT in helping reaching the diagnosis of RP in this condition. This report provides new data regarding the diagnostic difficulties of this rare type of autoimmune disease, and further investigations are needed as cases accumulate.

摘要

引言

不明原因发热(FUO)是一种常见的初始表现,会带来诊断挑战。

患者情况

一名54岁健康男性报告有3个月的中度至高热病史。他胸部的增强计算机断层扫描(CT)显示,双侧颈部、锁骨上、肺门和纵隔淋巴结在2周内显著进行性肿大。支气管纤维镜检查显示管腔明显狭窄,伴有肿胀、苍白增厚的黏膜,气管环状软骨结构消失,随后进行的18F-氟脱氧葡萄糖正电子发射断层扫描-计算机断层扫描(18F-FDG PET/CT)显示喉部、气管支气管树以及肺门、纵隔和腋窝淋巴结有强烈对称的FDG摄取。

诊断

最终诊断为复发性多软骨炎(RP)。

干预措施

患者每天接受40毫克甲泼尼龙治疗,并在4个月的随访中逐渐减量。

结果

患者在4个月的随访中未出现发热复发和淋巴结肿大。

经验教训

尽管长期发热伴多处淋巴结肿大通常会导致淋巴瘤的诊断,但该病例的支气管镜特征和18F-FDG PET/CT的证据更符合RP,这表明对于出现FUO和淋巴结肿大等非特异性特征的患者,合理鉴别诊断RP很重要。对这些患者保持高度的临床怀疑有助于识别罕见的RP,并促进诊断和治疗。我们的病例突出了18F-FDG PET/CT在帮助诊断这种情况下的RP的重要性。本报告提供了关于这种罕见自身免疫性疾病诊断困难的新数据,随着病例积累,需要进一步研究。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bdf9/5704863/291332b227f9/medi-96-e8734-g001.jpg

相似文献

1
An unusual cause of fever of unknown origin with enlarged lymph nodes-relapsing polychondritis: A case report.
Medicine (Baltimore). 2017 Nov;96(46):e8734. doi: 10.1097/MD.0000000000008734.
2
18F-FDG PET/CT imaging of relapsing polychondritis: A case report.
Medicine (Baltimore). 2016 Aug;95(33):e4496. doi: 10.1097/MD.0000000000004496.
3
Incidental Case of Relapsing Polychondritis Detected by 18F-FDG PET/CT.
Clin Nucl Med. 2018 Jan;43(1):25-27. doi: 10.1097/RLU.0000000000001890.
4
18F-FDG PET/CT is an ideal imaging modality for the early diagnosis of relapsing polychondritis: A case report.
Medicine (Baltimore). 2017 Jul;96(30):e7503. doi: 10.1097/MD.0000000000007503.
7
18F-FDG PET/CT Imaging of Vasculitis Complicated With Relapsing Polychondritis.
Clin Nucl Med. 2020 Jul;45(7):e327-e328. doi: 10.1097/RLU.0000000000003060.
9
Relapsing Polychondritis: The Oto-Rhino-Tracheobronchial Triad on 18 F-FDG PET/CT.
Clin Nucl Med. 2023 Aug 1;48(8):699-701. doi: 10.1097/RLU.0000000000004698. Epub 2023 Jun 3.
10
Fluorodeoxyglucose PET in relapsing polychondritis.
N Engl J Med. 2008 Jan 31;358(5):536-7. doi: 10.1056/NEJMc072254.

引用本文的文献

1
Craniopharyngioma as a Cause of Fever of Unknown Origin.
Mater Sociomed. 2023;35(2):162-165. doi: 10.5455/msm.2023.35.162-165.
2
[F]FDG PET/CT for identifying the causes of fever of unknown origin (FUO).
Am J Nucl Med Mol Imaging. 2024 Apr 25;14(2):87-96. doi: 10.62347/OQQC6007. eCollection 2024.
3
Retrospective analysis of 1,641 cases of classic fever of unknown origin.
Ann Transl Med. 2020 Jun;8(11):690. doi: 10.21037/atm-20-3875.

本文引用的文献

1
[Paniculitis as manifestation of prolonged febrile syndrome: Case report].
Rev Chil Pediatr. 2017 Jun;88(3):398-403. doi: 10.4067/S0370-41062017000300014.
2
18F-FDG PET/CT imaging of relapsing polychondritis: A case report.
Medicine (Baltimore). 2016 Aug;95(33):e4496. doi: 10.1097/MD.0000000000004496.
3
Clinical and prognostic characteristics of 158 cases of relapsing polychondritis in China and review of the literature.
Rheumatol Int. 2016 Jul;36(7):1003-9. doi: 10.1007/s00296-016-3449-8. Epub 2016 Mar 7.
4
Relapsing polychondritis.
Joint Bone Spine. 2014 Mar;81(2):118-24. doi: 10.1016/j.jbspin.2014.01.001. Epub 2014 Feb 18.
5
Relapsing polychondritis: An autoimmune disease with many faces.
Autoimmun Rev. 2010 Jun;9(8):540-6. doi: 10.1016/j.autrev.2010.02.016. Epub 2010 Mar 6.
6
Relapsing polychondritis: prevalence of expiratory CT airway abnormalities.
Radiology. 2006 Aug;240(2):565-73. doi: 10.1148/radiol.2401050562. Epub 2006 Jun 26.
7
Relapsing polychondritis.
Best Pract Res Clin Rheumatol. 2004 Oct;18(5):723-38. doi: 10.1016/j.berh.2004.05.012.
8
Relapsing polychondritis: reversible airway obstruction is not always asthma.
Mayo Clin Proc. 2004 Mar;79(3):407-9. doi: 10.4065/79.3.407.
9
Relapsing polychondritis.
Curr Opin Rheumatol. 2004 Jan;16(1):56-61. doi: 10.1097/00002281-200401000-00011.
10
Fever of unexplained origin: report on 100 cases.
Medicine (Baltimore). 1961 Feb;40:1-30. doi: 10.1097/00005792-196102000-00001.

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验