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粒细胞集落刺激因子治疗无效的非关节型费尔蒂综合征

Non-Articular Felty Syndrome Refractory to Granulocyte Colony-Stimulating Factor Therapy.

作者信息

Saeed Hasham, Ejikeme Chidinma, Tucktuck Marina, Jawed Qirat, Kessler William

机构信息

Internal Medicine, Rutgers Health/Trinitas Regional Medical Center, Elizabeth, USA.

Internal Medicine, St. George's University Medical School, Elizabeth, USA.

出版信息

Cureus. 2021 Aug 15;13(8):e17206. doi: 10.7759/cureus.17206. eCollection 2021 Aug.

DOI:10.7759/cureus.17206
PMID:34540433
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC8440238/
Abstract

Felty syndrome (FS), an uncommon manifestation seen in patients with rheumatoid arthritis (RA), usually presents as a triad of erosive arthritis, splenomegaly, and neutropenia. It is extremely rare for RA to present as FS or develop after initially presenting as neutropenia and splenomegaly. In this report, we describe a case of a 55-year-old woman who initially presented with fever and vaginal pain. Her sepsis workup revealed genital herpes in the setting of leukopenia, with an incidental finding of splenomegaly on imaging. The patient was managed with filgrastim and valacyclovir. Two weeks later, she presented again with pleuritic chest pain and worsening leukopenia. This led to an extensive workup by the hematology team to diagnose and confirm the diagnosis of FS. We also engage in a review of the existing literature of such cases and emphasize the importance of serological testing for RA in patients with leukopenia and splenomegaly, even in the absence of joint symptoms or prior diagnosis of RA. The management should be guided towards treating the infection, correcting the neutropenia, and treating the underlying chronic disease.

摘要

费尔蒂综合征(FS)是类风湿关节炎(RA)患者中一种不常见的表现,通常表现为侵蚀性关节炎、脾肿大和中性粒细胞减少三联征。类风湿关节炎以费尔蒂综合征形式出现或在最初表现为中性粒细胞减少和脾肿大后发展为费尔蒂综合征极为罕见。在本报告中,我们描述了一例55岁女性患者,她最初表现为发热和阴道疼痛。她的败血症检查显示在白细胞减少的情况下患有生殖器疱疹,影像学检查偶然发现脾肿大。患者接受了非格司亭和伐昔洛韦治疗。两周后,她再次出现胸膜炎性胸痛和白细胞减少加重。这促使血液学团队进行了广泛检查以诊断并确诊费尔蒂综合征。我们还对这类病例的现有文献进行了综述,并强调即使在没有关节症状或既往无类风湿关节炎诊断的白细胞减少和脾肿大患者中,进行类风湿关节炎血清学检测的重要性。治疗应针对治疗感染、纠正中性粒细胞减少以及治疗潜在的慢性疾病。

相似文献

1
Non-Articular Felty Syndrome Refractory to Granulocyte Colony-Stimulating Factor Therapy.粒细胞集落刺激因子治疗无效的非关节型费尔蒂综合征
Cureus. 2021 Aug 15;13(8):e17206. doi: 10.7759/cureus.17206. eCollection 2021 Aug.
2
Neutropaenia and splenomegaly without arthritis: think rheumatoid arthritis.无关节炎的中性粒细胞减少和脾肿大:考虑类风湿关节炎。
BMJ Case Rep. 2018 Jul 11;2018:bcr-2018-225359. doi: 10.1136/bcr-2018-225359.
3
Felty syndrome: a case report.费尔蒂综合征:一例报告。
J Med Case Rep. 2021 May 27;15(1):273. doi: 10.1186/s13256-021-02802-9.
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Felty Syndrome费尔蒂综合征
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Severe Neutropenia Complicated with Necrotizing Fasciitis Unveils a Diagnosis of Rheumatoid Arthritis: A Case Report.严重中性粒细胞减少症合并坏死性筋膜炎揭示类风湿关节炎诊断:一例报告
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Infective Endocarditis-Like Presentation of Felty Syndrome: A Case Report.费尔蒂综合征的感染性心内膜炎样表现:一例报告
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Rapidly Progressive Felty Syndrome After Sudden Discontinuation of Methotrexate: A Case Report and Review of Literature.甲氨蝶呤突然停药后迅速进展的费尔蒂综合征:一例报告及文献复习
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The Diagnosis Felt(y) Right: A Case Report of Felty Syndrome With Limited Articular Involvement.诊断“感觉”正确:1例关节受累有限的费尔蒂综合征病例报告
Cureus. 2022 Apr 29;14(4):e24593. doi: 10.7759/cureus.24593. eCollection 2022 Apr.
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A case of filgrastim-induced neutrophilic dermatosis of the dorsal hands in a patient with Felty Syndrome.一例 Felty 综合征患者因使用非格司亭导致的手背嗜中性粒细胞皮肤病。
Dermatol Online J. 2023 Dec 15;29(6). doi: 10.5070/D329662999.

引用本文的文献

1
Atypical presentation of Felty syndrome: Successful management with rituximab therapy-A case report and review of literature.费尔蒂综合征的非典型表现:利妥昔单抗治疗成功——病例报告及文献综述
Clin Case Rep. 2024 Aug 18;12(8):e9339. doi: 10.1002/ccr3.9339. eCollection 2024 Aug.

本文引用的文献

1
Neutropaenia and splenomegaly without arthritis: think rheumatoid arthritis.无关节炎的中性粒细胞减少和脾肿大:考虑类风湿关节炎。
BMJ Case Rep. 2018 Jul 11;2018:bcr-2018-225359. doi: 10.1136/bcr-2018-225359.
2
Felty's Syndrome, Insights and Updates.费尔蒂综合征:见解与进展
Open Rheumatol J. 2014 Dec 31;8:129-36. doi: 10.2174/1874312901408010129. eCollection 2014.
3
A beneficial long-term and consistent response to rituximab in the treatment of refractory neutropenia and arthritis in a patient with Felty syndrome.利妥昔单抗治疗费尔蒂综合征患者难治性中性粒细胞减少症和关节炎取得长期持续有益反应。
J Clin Rheumatol. 2014 Oct;20(7):398. doi: 10.1097/RHU.0000000000000175.
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Biological agents in the management of Felty's syndrome: a systematic review.生物制剂在费尔蒂综合征治疗中的应用:系统评价。
Semin Arthritis Rheum. 2012 Apr;41(5):658-68. doi: 10.1016/j.semarthrit.2011.08.008. Epub 2011 Nov 25.
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The spectrum of large granular lymphocyte leukemia and Felty's syndrome.大颗粒淋巴细胞白血病和费尔蒂综合征的谱。
Curr Opin Hematol. 2011 Jul;18(4):254-9. doi: 10.1097/MOH.0b013e32834760fb.
6
Management of autoimmune neutropenia in Felty's syndrome and systemic lupus erythematosus.费尔蒂综合征和系统性红斑狼疮中自身免疫性中性粒细胞减少症的治疗。
Autoimmun Rev. 2011 May;10(7):432-7. doi: 10.1016/j.autrev.2011.01.006. Epub 2011 Jan 18.
7
Felty's syndrome as an initial presentation of rheumatoid arthritis: a case report.费尔蒂综合征作为类风湿关节炎的首发表现:一例病例报告。
Cases J. 2009 Nov 18;2:206. doi: 10.1186/1757-1626-2-206.
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Felty's syndrome.费尔蒂综合征。
Best Pract Res Clin Rheumatol. 2004 Oct;18(5):631-45. doi: 10.1016/j.berh.2004.05.002.
9
Autoantibodies against granulocyte colony-stimulating factor in Felty's syndrome and neutropenic systemic lupus erythematosus.费尔蒂综合征和中性粒细胞减少性系统性红斑狼疮中抗粒细胞集落刺激因子自身抗体
Arthritis Rheum. 2002 Sep;46(9):2384-91. doi: 10.1002/art.10497.
10
Treatment of severe neutropenia due to Felty's syndrome or systemic lupus erythematosus with granulocyte colony-stimulating factor.用粒细胞集落刺激因子治疗费尔蒂综合征或系统性红斑狼疮所致的严重中性粒细胞减少症。
Semin Arthritis Rheum. 1999 Oct;29(2):82-99. doi: 10.1016/s0049-0172(99)80040-7.