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BMJ Case Rep. 2023 Apr 12;16(4):e254829. doi: 10.1136/bcr-2023-254829.
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本文引用的文献

1
Sarcoidosis with marked necrosis in enlarged lymph nodes mimics mycobacterial infection: a case report.淋巴结显著坏死的结节病模拟分枝杆菌感染:病例报告。
J Med Case Rep. 2021 Apr 18;15(1):178. doi: 10.1186/s13256-021-02797-3.
2
Sarcoidosis: A Clinical Overview from Symptoms to Diagnosis.结节病:从症状到诊断的临床概述。
Cells. 2021 Mar 31;10(4):766. doi: 10.3390/cells10040766.
3
Regression of Sarcoidosis during Pregnancy: Case Report and Review of the Literature.孕期结节病的缓解:病例报告及文献综述
Mediterr J Rheumatol. 2020 Dec 28;31(4):416-420. doi: 10.31138/mjr.31.4.416. eCollection 2020 Dec.
4
Treatment of Sarcoidosis: A Multidisciplinary Approach.结节病的治疗:多学科方法。
Front Immunol. 2020 Nov 19;11:545413. doi: 10.3389/fimmu.2020.545413. eCollection 2020.
5
The incidence, comorbidity and mortality of sarcoidosis in Korea, 2008-2015: a nationwide population-based study.2008 - 2015年韩国结节病的发病率、合并症及死亡率:一项基于全国人口的研究
Sarcoidosis Vasc Diffuse Lung Dis. 2020;37(1):24-26. doi: 10.36141/svdld.v37i1.7660. Epub 2020 Mar 15.
6
Maternal and infant outcomes in sarcoidosis pregnancy: a Swedish population-based cohort study of first births.妊娠结节病母婴结局:一项瑞典基于人群的初产妇队列研究。
Respir Res. 2020 Aug 27;21(1):225. doi: 10.1186/s12931-020-01493-y.
7
Cutaneous Sarcoidosis.皮肤结节病。
Semin Respir Crit Care Med. 2020 Oct;41(5):689-699. doi: 10.1055/s-0040-1713130. Epub 2020 Jun 27.
8
Osseous manifestations of sarcoidosis.结节病的骨表现
Reumatologia. 2020;58(2):93-100. doi: 10.5114/reum.2020.95363. Epub 2020 Apr 30.
9
Histopathological Features of Subcutaneous Sarcoidosis.皮下结节性脂膜炎的组织病理学特征。
Am J Dermatopathol. 2020 Apr;42(4):233-243. doi: 10.1097/DAD.0000000000001509.
10
F-FDG PET/CT in bone sarcoidosis: an observational study.氟代脱氧葡萄糖正电子发射断层显像/计算机断层扫描在骨结节病中的应用:一项观察性研究。
Clin Rheumatol. 2020 Sep;39(9):2727-2734. doi: 10.1007/s10067-020-05022-6. Epub 2020 Mar 20.

罕见的难治性肺外结节病表现。

Rare manifestations of refractory extrapulmonary sarcoidosis.

机构信息

Faculdade de Medicina, Universidade de Coimbra, Coimbra, Portugal

Faculdade de Medicina, Universidade de Coimbra, Coimbra, Portugal.

出版信息

BMJ Case Rep. 2023 Apr 12;16(4):e254829. doi: 10.1136/bcr-2023-254829.

DOI:10.1136/bcr-2023-254829
PMID:37045548
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10105993/
Abstract

Sarcoidosis is a multisystemic disease, characterised by histopathologic presence of non-necrotising granulomas, primarily affecting the lungs (>90%). We present three cases predominantly characterised by uncommon extrapulmonary manifestations of sarcoidosis, where only one had pulmonary involvement. We describe three female patients, between the second and third decades of life, whose sarcoidosis was a diagnostic challenge due to their atypical extrapulmonary manifestations, from which we highlight: livedo reticularis and painful subcutaneous nodules with uncommon localisation, size and histology, being the first reported case of extensive subcutaneous nodules triggered by intramuscular penicillin; extensive symptomatic and refractory osseous involvement; and dispersed erythema nodosum affecting the entire body surface. All three patients required third-line treatment (antitumour necrosis factor agents) to achieve significant clinical and imagiological improvement. Through this case series, we highlight the importance of considering the rare and atypical presentations of sarcoidosis to avoid diagnostic delays and serious repercussions on the patient's prognosis.

摘要

结节病是一种多系统疾病,其特征为组织病理学上存在非坏死性肉芽肿,主要影响肺部(>90%)。我们报告了三个主要表现为罕见肺外结节病表现的病例,其中只有一个病例有肺部受累。我们描述了 3 名女性患者,年龄在第二和第三个十年之间,由于其非典型的肺外表现,结节病的诊断具有挑战性,其中突出表现为:网状青斑和疼痛性皮下结节,具有罕见的位置、大小和组织学,这是首例由肌肉内青霉素注射引起的广泛皮下结节的报告;广泛的症状性和难治性骨受累;以及广泛分布的结节性红斑累及整个体表面积。所有 3 名患者都需要三线治疗(抗肿瘤坏死因子制剂)才能获得显著的临床和影像学改善。通过这个病例系列,我们强调了考虑结节病罕见和非典型表现的重要性,以避免诊断延迟和对患者预后产生严重影响。