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[一例酷似弥漫性泛细支气管炎的成人T细胞白血病合并类风湿关节炎尸检病例报告]

[An Autopsy Case Report of Adult T-cell Leukemia Accompanied by Rheumatoid Arthritis Mimicking Diffuse Panbronchiolitis].

作者信息

Shiraishi Tomoko, Ishimoto Hiroshi, Akata Kentaro, Kawanami Toshinori, Yatera Kazuhiro, Mukae Hiroshi

机构信息

Department of Respiratory Medicine, School of Medicine, University of Occupational and Environmental Health, Japan.

出版信息

J UOEH. 2017;39(1):55-61. doi: 10.7888/juoeh.39.55.

Abstract

A 50-year-old female with a history of chronic sinusitis and rheumatoid arthritis visited our department with repetitive lower respiratory tract infections of Pseudomonas aeruginosa. Her chest CT showed diffuse panbronchiolitis-like pulmonary lesions, her blood examination revealed atypical lymphocytes, and she was serologically positive for anti-human T-lymphotrophic virus type 1 (HTLV-1) antibody. Her rheumatoid arthritis had been well-controlled after biological agent treatment followed by anti-inflammatory analgesic treatment. She received long-term low-dose macrolide therapy for four years. The Pseudomonas aeruginosa gradually became multi-antibiotic-resistant. Her lower respiratory infection gradually became uncontrollable, and her adult T cell leukemia (ATL) developed to the acute phase. Due to repetitive lower respiratory tract infections and respiratory failure, however, she could not receive any treatment for ATL, and she eventually died due to the progression of the disease. An autopsy revealed an invasion of abnormal lymphocytes in multiple organs, including the lungs, which indicated that the HTLV-1 infection and the progression of ATL were the dominant factors in this patient's clinical course. There have been no diffuse panbronchiolitis-like cases accompanied by rheumatoid arthritis and HTLV-1 infection so far. Because these diseases show similar clinical features, it is difficult to discriminate between them. There are presently no appropriate criteria for the proper time of treatment of patients with pulmonary lesion-associated ATL, but further research is expected to elucidate this matter.

摘要

一名50岁女性,有慢性鼻窦炎和类风湿关节炎病史,因反复发生铜绿假单胞菌引起的下呼吸道感染前来我院就诊。她的胸部CT显示弥漫性细支气管炎样肺部病变,血液检查发现非典型淋巴细胞,血清学检测抗人类T淋巴细胞病毒1型(HTLV-1)抗体呈阳性。她的类风湿关节炎在生物制剂治疗后继以抗炎镇痛治疗,病情得到了良好控制。她接受了为期四年的长期低剂量大环内酯类药物治疗。铜绿假单胞菌逐渐出现多重耐药。她的下呼吸道感染逐渐变得难以控制,成人T细胞白血病(ATL)发展至急性期。然而,由于反复的下呼吸道感染和呼吸衰竭,她无法接受任何针对ATL的治疗,最终因病情进展而死亡。尸检显示包括肺部在内的多个器官有异常淋巴细胞浸润,这表明HTLV-1感染和ATL进展是该患者临床病程中的主要因素。迄今为止,尚无伴有类风湿关节炎和HTLV-1感染的弥漫性细支气管炎样病例。由于这些疾病表现出相似的临床特征,难以对它们进行鉴别。目前对于与肺部病变相关的ATL患者的恰当治疗时机尚无合适标准,但期望进一步的研究能阐明这一问题。

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