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一名狼疮患者中家族性地中海热与肿瘤坏死因子受体相关周期性综合征的重叠综合征。

Overlap syndrome between Familial Mediterranean fever and tumor necrosis factor receptor-associated periodic syndrome in a lupus patient.

作者信息

Nonaka Fumiaki, Migita Kiyoshi, Iwasaki Keisuke, Shimizu Toshimasa, Kawakami Atsushi, Yasunami Michio, Eguchi Katsumi

机构信息

Department of Rheumatology, Sasebo City General Hospital.

出版信息

Tohoku J Exp Med. 2014 Jun;233(2):73-7. doi: 10.1620/tjem.233.73.

Abstract

Autoinflammatory diseases represent an expanding spectrum of genetic and non-genetic inflammatory diseases characterized by recurrent episodes of fever and systemic inflammation, affecting joints, skin and serosal surfaces. Familial Mediterranean fever (FMF) is the most common autosomal recessive hereditary autoinflammatory disease. Tumor necrosis factor receptor-associated periodic syndrome (TRAPS) is an autosomal dominant hereditary autoinflammatory disease. They share some clinical manifestations such as a periodic fever and skin rash. We present here the association of FMF with TRAPS in a systemic lupus erythematosus (SLE) patient. A 54-year-old SLE patient with recurrent attacks of fever, arthritis, and skin rashes was referred to our hospital. She had been diagnosed with lupus nephritis at 19 years old. Her lupus nephritis was controlled by steroid treatments; however, since childhood she has suffered from recurrent episodes of periodic fever, abdominal pain, arthritis, and erythematous skin rashes. An initial diagnosis of FMF was suspected based on the genetic analysis, showing the compound heterozygous L110P/E148Q mutations in the MEFV gene that is responsible for FMF. Her symptoms responded to colchicine, but the febrile attacks were not completely resolved. Therefore, genetic testing for TRAPS was performed. The results revealed a heterozygous T61I mutation in the TNFRSF1A gene that encodes tumor necrosis factor-α receptor and is responsible for TRAPS. The patient was diagnosed with overlapping FMF and TRAPS, in addition to SLE. This is the first report of SLE associated with both FMF and TRAPS.

摘要

自身炎症性疾病是一类不断扩展的遗传性和非遗传性炎症性疾病,其特征为反复发热和全身性炎症发作,可累及关节、皮肤和浆膜表面。家族性地中海热(FMF)是最常见的常染色体隐性遗传性自身炎症性疾病。肿瘤坏死因子受体相关周期性综合征(TRAPS)是一种常染色体显性遗传性自身炎症性疾病。它们有一些共同的临床表现,如周期性发热和皮疹。我们在此报告一名系统性红斑狼疮(SLE)患者中FMF与TRAPS的关联。一名54岁的SLE患者因反复发热、关节炎和皮疹发作被转诊至我院。她19岁时被诊断为狼疮性肾炎。她的狼疮性肾炎通过类固醇治疗得到控制;然而,自童年起,她就反复发作周期性发热、腹痛、关节炎和皮肤红斑。基于基因分析,初步怀疑为FMF,结果显示与FMF相关的MEFV基因存在复合杂合L110P/E148Q突变。她的症状对秋水仙碱有反应,但发热发作并未完全缓解。因此,进行了TRAPS的基因检测。结果显示,编码肿瘤坏死因子-α受体且与TRAPS相关的TNFRSF1A基因存在杂合T61I突变。该患者除SLE外,还被诊断为重叠性FMF和TRAPS。这是首例SLE与FMF和TRAPS均相关的报告。

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