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一名感染人类嗜T淋巴细胞病毒1型（HTLV-1）的干燥综合征患者出现类TAFRO症状。

TAFRO-like Symptoms in a Sjögren's Syndrome Patient with HTLV-1 Infection.

作者信息

Masuda Takuya, Suzuki Takeshi, Ohshima Miho, Suzuki Akitake, Minemura Nobuyoshi, Nakajima Hiroyoshi

机构信息

Department General Internal Medicine, Mitsui Memorial Hospital, Japan.

Department of Rheumatology, Mitsui Memorial Hospital, Japan.

出版信息

Intern Med. 2025 Apr 1;64(7):1107-1112. doi: 10.2169/internalmedicine.4124-24. Epub 2024 Aug 28.

DOI:10.2169/internalmedicine.4124-24
PMID:39198166
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12021513/
Abstract

A 58-year-old woman was admitted to our hospital with anasarca and generalized lymphadenopathy. Laboratory data showed serum creatinine 1.48 mg/dL, C-Reactive Protein 2.38 mg/dL, platelet 102,000/μL, and anti-SS-A antibodies. A lymph node biopsy revealed a regressed germinal center and hypervascularization in the interfollicular area. The patient was diagnosed with TAFRO-like symptoms occurring with Sjögren's syndrome and human T-cell leukemia virus type 1 (HTLV-1) infection, and 80 mg of methylprednisolone and tocilizumab 8 mg/kg biweekly were initiated. Her body weight decreased from 59.4 to 41 kg, and pleural effusion disappeared 8 weeks later. This case suggests that TAFRO-like symptoms may occur in patients with Sjögren's syndrome with HTLV-1 infection.

摘要

一名58岁女性因全身水肿和全身淋巴结肿大入住我院。实验室检查数据显示血清肌酐1.48mg/dL、C反应蛋白2.38mg/dL、血小板102,000/μL，以及抗SS - A抗体。淋巴结活检显示生发中心消退，滤泡间区域血管增生。该患者被诊断为干燥综合征合并人类T细胞白血病病毒1型（HTLV - 1）感染出现类TAFRO症状，开始使用80mg甲泼尼龙和每两周8mg/kg的托珠单抗治疗。她的体重从59.4kg降至41kg，8周后胸腔积液消失。该病例提示干燥综合征合并HTLV - 1感染的患者可能出现类TAFRO症状。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ddd8/12021513/adf4ef0683a3/1349-7235-64-1107-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ddd8/12021513/6f5b61c873c5/1349-7235-64-1107-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ddd8/12021513/3f242f44a7d7/1349-7235-64-1107-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ddd8/12021513/3703caa24ed5/1349-7235-64-1107-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ddd8/12021513/adf4ef0683a3/1349-7235-64-1107-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ddd8/12021513/6f5b61c873c5/1349-7235-64-1107-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ddd8/12021513/3f242f44a7d7/1349-7235-64-1107-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ddd8/12021513/3703caa24ed5/1349-7235-64-1107-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ddd8/12021513/adf4ef0683a3/1349-7235-64-1107-g004.jpg

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本文引用的文献

1
TAFRO syndrome is associated with anti-SSA/Ro60 antibodies, in contrast to idiopathic castleman disease.与特发性Castleman病相反，TAFRO综合征与抗SSA/Ro60抗体相关。
Sci Rep. 2024 Feb 5;14(1):2889. doi: 10.1038/s41598-024-53413-5.
2
Lower prevalence of anti-HTLV-1 as expected by previous models among first-time blood donors in Japan.在日本首次献血者中，抗人类嗜T淋巴细胞病毒1型（anti-HTLV-1）的流行率低于先前模型预期。
J Med Virol. 2023 Mar;95(3):e28606. doi: 10.1002/jmv.28606.
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TAFRO syndrome: A severe manifestation of Sjogren's syndrome? A systematic review.
TAFRO 综合征：干燥综合征的严重表现？系统综述。
Autoimmun Rev. 2022 Aug;21(8):103137. doi: 10.1016/j.autrev.2022.103137. Epub 2022 Jul 6.
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Does HTLV-1 Infection Show Phenotypes Found in Sjögren's Syndrome?HTLV-1 感染是否表现出干燥综合征中发现的表型？
Viruses. 2022 Jan 6;14(1):100. doi: 10.3390/v14010100.
5
Validated international definition of the thrombocytopenia, anasarca, fever, reticulin fibrosis, renal insufficiency, and organomegaly clinical subtype (TAFRO) of idiopathic multicentric Castleman disease.特发性多中心 Castleman 病伴血小板减少、浮肿、发热、网状纤维纤维化、肾功能不全和器官肿大（TAFRO）临床亚型的验证性国际定义。
Am J Hematol. 2021 Oct 1;96(10):1241-1252. doi: 10.1002/ajh.26292. Epub 2021 Jul 28.
6
Tocilizumab has no direct effect on cell lines infected with human T-cell leukemia virus type 1.托珠单抗对感染1型人类T细胞白血病病毒的细胞系无直接作用。
J Int Med Res. 2021 Mar;49(3):3000605211002083. doi: 10.1177/03000605211002083.
7
Sequential change in serum VEGF levels in a case of tocilizumab-resistant TAFRO syndrome treated effectively with rituximab.托珠单抗耐药 TAFRO 综合征一例血清 VEGF 水平的序贯变化，利妥昔单抗治疗有效。
Mod Rheumatol Case Rep. 2021 Jan;5(1):145-151. doi: 10.1080/24725625.2020.1789304. Epub 2020 Jul 15.
8
Optimal treatments for TAFRO syndrome: a retrospective surveillance study in Japan.TAFRO 综合征的最佳治疗方法：日本的一项回顾性监测研究。
Int J Hematol. 2021 Jan;113(1):73-80. doi: 10.1007/s12185-020-03008-3. Epub 2020 Sep 24.
9
2019 Updated diagnostic criteria and disease severity classification for TAFRO syndrome.2019年TAFRO综合征更新的诊断标准和疾病严重程度分类。
Int J Hematol. 2020 Jan;111(1):155-158. doi: 10.1007/s12185-019-02780-1. Epub 2019 Nov 28.
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Association between HTLV-1 infection and adverse health outcomes: a systematic review and meta-analysis of epidemiological studies.HTLV-1 感染与不良健康结局的关联：流行病学研究的系统评价和荟萃分析。
Lancet Infect Dis. 2020 Jan;20(1):133-143. doi: 10.1016/S1473-3099(19)30402-5. Epub 2019 Oct 21.