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服用L-色氨酸后出现伴有筋膜炎和间质性肌炎的嗜酸性粒细胞增多性肌痛综合征

[Eosinophilia-myalgia syndrome with fasciitis and interstitial myositis after L-tryptophan administration].

作者信息

Bartz-Bazzanella P, Genth E, Pollmann H J, Schröder J M, Völker A

机构信息

Rheumaklinik, Rheinisch-Westfälischen Technischen Hochschule Aachen.

出版信息

Z Rheumatol. 1992 Jan-Feb;51(1):3-13.

PMID:1574934
Abstract

We describe a 53-year-old women with eosinophilia-myalgia syndrome who suddenly developed severe persistent myalgias of her arms, legs, back, and shoulder after a 5-month period of daily L-tryptophan ingestion, associated with fever, progressive stenocardia and left-sided congestive heart failure. Laboratory tests showed a leukocytosis of 11.2/nl with 3.14/nl eosinophils and an elevated erythrocyte sedimentation rate. There was a marked, predominantly proximal sclerosis of her arms, legs and trunk with a brownish discoloration. The skin of her arms and legs appeared dimpled (peau d'orange). Findings of the electrophysiological examinations were consistent with sensory neuropathy and myositis. Remarkable fasciitis and interstitial myositis were present in a biopsy specimen (from skin to muscle) taken from her thigh. However, eosinophilic infiltrates were rare. Angiography revealed an apical obstructive cardiomyopathy. In this paper, we describe the clinical findings, the course over 2 years, as well as the therapeutic management. Furthermore, the most important differential diagnoses are discussed and the literature is reviewed with special attention given to more recent pathogenic insights into this newly recognized multisystem disease.

摘要

我们描述了一名患有嗜酸性粒细胞增多性肌痛综合征的53岁女性,在每日摄入L-色氨酸5个月后,突然出现手臂、腿部、背部和肩部严重持续性肌痛,并伴有发热、进行性心绞痛和左侧充血性心力衰竭。实验室检查显示白细胞计数为11.2/nl,嗜酸性粒细胞计数为3.14/nl,红细胞沉降率升高。她的手臂、腿部和躯干出现明显的、主要为近端的硬化,并伴有褐色色素沉着。她手臂和腿部的皮肤出现橘皮样改变。电生理检查结果与感觉神经病变和肌炎一致。从她大腿获取的活检标本(从皮肤到肌肉)显示存在明显的筋膜炎和间质性肌炎。然而,嗜酸性粒细胞浸润很少见。血管造影显示心尖部梗阻性心肌病。在本文中,我们描述了临床发现、2年的病程以及治疗处理。此外,还讨论了最重要的鉴别诊断,并对文献进行了综述,特别关注了对这种新认识的多系统疾病的最新发病机制见解。

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