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惠普尔病:误诊为结节病,继发三尖瓣心内膜炎和肺栓塞——病例报告

Whipple's disease: misdiagnosed as sarcoidosis with further tricuspid valve endocarditis and pulmonary embolism - a case report.

作者信息

Berent Robert, Auer Johann, Lassnig Elisabeth, von Duvillard Serge P, Crouse Stephen F, Tuppy Herwig, Eber Bernd

机构信息

Center for Cardiovascular Rehabilitation, Stifterstrasse 11, Bad Schallerbach, 4701, Austria.

出版信息

BMJ Case Rep. 2009;2009. doi: 10.1136/bcr.07.2008.0441. Epub 2009 Jun 1.

Abstract

GH Whipple described a 36-year-old physician in 1907 with gradual loss of weight and strength, stools consisting chiefly of neutral fat and fatty acids, indefinite abdominal signs and a peculiar multiple arthritis. The patient died of this progressive illness. Whipple called it intestinal lipodystrophy since he observed accumulation of large masses of neutral fats and fatty acids in the lymph spaces. It was renamed Whipple's disease in 1949. An infectious aetiology was suspected as early as Whipple's initial report. However, successful treatment with antibiotics was not reported until 1952, which resulted in dramatic clinical responses. The cause is now known to be Tropheryma whipplei. Light and electron microscopy of infected tissue identified a gram-positive, non-acid-fast, periodic acid-Schiff (PAS) positive bacillus with a characteristic trilamellar plasma membrane resembling that of gram-negative bacteria. Whipple's disease is extremely rare. It is a systemic infectious disorder affecting mostly middle-aged white men. The clinical presentation is often non-specific, which may make its diagnosis difficult. The four cardinal clinical manifestations are arthralgias, weight loss, diarrhoea and abdominal pain. The frequently vague articular symptoms can precede the diagnosis of Whipple's disease by an average of 6-8 years. Lymph nodes and other tissues may present diagnostic problems, since the changes in routinely stained sections may mimic those of sarcoidosis. The detection of PAS-positive histiocytes in the small intestine remains the mainstay of the diagnosis, although Whipple's disease without gastrointestinal involvement is described. We illustrate a case in which, retrospectively, the clinical presentation would have been typical for Whipple's disease. However, the clinical presentation and the histological examinations of lymph nodes, liver biopsies and ascites initially were misinterpreted as sarcoidosis with consecutive immunosuppressive therapy and progressive worsening of the patient's health presenting at least as sepsis with endocarditis.

摘要

1907年,GH·惠普尔描述了一位36岁的内科医生,该患者体重和体力逐渐下降,粪便主要由中性脂肪和脂肪酸组成,有不明确的腹部体征以及一种特殊的多发性关节炎。患者死于这种进行性疾病。惠普尔将其称为肠道脂肪代谢障碍,因为他观察到在淋巴间隙中有大量中性脂肪和脂肪酸积聚。1949年该病被重新命名为惠普尔病。早在惠普尔最初报告时就怀疑有感染性病因。然而,直到1952年才报告使用抗生素治疗成功,且取得了显著的临床反应。现在已知病因是惠普尔氏放线杆菌。对感染组织进行光镜和电镜检查发现一种革兰氏阳性、抗酸染色阴性、过碘酸希夫(PAS)染色阳性的杆菌,其具有特征性的三层质膜,类似于革兰氏阴性菌。惠普尔病极其罕见。它是一种全身性感染性疾病,主要影响中年白人男性。临床表现通常不具有特异性,这可能使其诊断困难。四个主要临床表现为关节痛、体重减轻、腹泻和腹痛。常见的模糊关节症状平均可在惠普尔病诊断前6 - 8年出现。淋巴结和其他组织可能会带来诊断问题,因为常规染色切片中的变化可能类似于结节病。小肠中PAS阳性组织细胞的检测仍然是诊断的主要依据,尽管也有不伴有胃肠道受累的惠普尔病的描述。我们举例说明一个病例,回顾来看,其临床表现本应是惠普尔病的典型表现。然而,临床表现以及淋巴结、肝脏活检和腹水的组织学检查最初被误诊为结节病,并连续进行了免疫抑制治疗,导致患者健康状况逐渐恶化,至少出现了败血症合并心内膜炎。

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