Whipple's disease in a man of North African descent : case report and brief review of the literature.

IF 1.5 4区 医学 Q2 Medicine Acta Gastro-Enterologica Belgica Pub Date : 2019-01-01
M Lenfant, J Callemeyn, H Alaerts, W Meersseman, W Van Moerkercke
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Abstract

A 62-year-old man of North African descent presented with weight loss in the past year and diarrhea for three weeks. His medical history included erosive rheumatoid arthritis, treated with methotrexate and adalimumab. Histological examination of a duodenal biopsy showed foamy macrophages in the lamina propria, with PAS-positive cytoplasmatic inclusions. These findings are compatible with Whipple's disease, a rare chronic infectious disease caused by Tropheryma whipplei, an opportunistic bacterium. It is typically seen in middle-aged Caucasian men and the immunocompromised host. The classical presentation of Whipple's disease consists of intermittent migratory arthralgia, followed by intestinal symptoms which typically occur six to seven years later. The clinical image can be very variable, and this complicates the diagnostic process. PAS-staining and PCR are the diagnostic cornerstones. In our case, treatment consisted of a prolonged cure of antibiotics: intravenous ceftriaxone for two weeks, followed by an oral maintenance therapy of doxycycline and hydroxychloroquine for at least one year. A therapeutic dilemma arose as continued anti-TNF blockade was necessary to maintain remission of the rheumatoid arthritis. Lifelong follow-up is necessary because relapse is possible.

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一名北非裔男子的惠普尔病:病例报告及文献综述。
一名62岁的北非裔男子在过去一年中出现体重下降和腹泻三周。病史包括糜烂性类风湿关节炎,曾用甲氨蝶呤和阿达木单抗治疗。十二指肠活检组织学检查显示固有层有泡沫状巨噬细胞,伴有pas阳性的胞浆包涵体。这些发现与惠普尔病相一致,惠普尔病是一种罕见的慢性传染病,由一种机会性细菌惠普尔Tropheryma whipplei引起。它通常见于中年高加索男性和免疫功能低下的宿主。惠普尔氏病的典型表现包括间歇性迁移性关节痛,随后通常在6至7年后出现肠道症状。临床图像可能非常多变,这使诊断过程复杂化。pas染色和PCR是诊断的基础。在我们的病例中,治疗包括延长抗生素治疗:静脉注射头孢曲松两周,然后口服强力霉素和羟氯喹维持治疗至少一年。由于持续的抗tnf阻断是维持类风湿关节炎缓解所必需的,因此出现了治疗困境。终身随访是必要的,因为复发是可能的。
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来源期刊
Acta Gastro-Enterologica Belgica
Acta Gastro-Enterologica Belgica 医学-胃肠肝病学
CiteScore
2.80
自引率
20.00%
发文量
0
审稿时长
>12 weeks
期刊介绍: The Journal Acta Gastro-Enterologica Belgica principally publishes peer-reviewed original manuscripts, reviews, letters to editors, book reviews and guidelines in the field of clinical Gastroenterology and Hepatology, including digestive oncology, digestive pathology, as well as nutrition. Pure animal or in vitro work will not be considered for publication in the Journal. Translational research papers (including sections of animal or in vitro work) are considered by the Journal if they have a clear relationship to or relevance for clinical hepato-gastroenterology (screening, disease mechanisms and/or new therapies). Case reports and clinical images will be accepted if they represent an important contribution to the description, the pathogenesis or the treatment of a specific gastroenterology or liver problem. The language of the Journal is English. Papers from any country will be considered for publication. Manuscripts submitted to the Journal should not have been published previously (in English or any other language), nor should they be under consideration for publication elsewhere. Unsolicited papers are peer-reviewed before it is decided whether they should be accepted, rejected, or returned for revision. Manuscripts that do not meet the presentation criteria (as indicated below) will be returned to the authors. Papers that go too far beyond the scope of the journal will be also returned to the authors by the editorial board generally within 2 weeks. The Journal reserves the right to edit the language of papers accepted for publication for clarity and correctness, and to make formal changes to ensure compliance with AGEB’s style. Authors have the opportunity to review such changes in the proofs.
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