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Palindromic Rheumatoid Arthritis-An Unorthodox Presentation of Whipple’s Disease

Received: 28 February 2016    Accepted: 6 March 2016    Published: 21 March 2016
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Abstract

Whipple's disease is a rare infection of the gastrointestinal tract caused by the actinomycete Tropheryma whippelii. It most commonly presents with arthralgia, abdominal pain, diarrhoea and weight loss. Invasion of the bacterium through the gastrointestinal mucosa leads to small intestinal villus blunting and malabsorption. Diagnosis is made by histological examination of small bowel biopsies. We report the case of a 78 year old gentleman who had a two year history of fleeting joint pain and multiple hospital admissions for varying symptoms. He was treated with antibiotics for a chest infection with improvement only to present again after 1 year with anorexia, fatigue, blackish loose stools and epigastric pain. Microscopic examination of duodenal biopsies showed a stunted villous architecture and expansion of the lamina propria by foamy macrophages. These expressed CD68 and cytoplasmic contents were strongly PAS positive, consistent with a diagnosis of Whipple’s disease. He was started on intravenous ceftriaxone and a prolonged course of oral co-trimoxazole (at least 1 year) with marked clinical improvement. In retrospect, the previous year’s admissions might have also been due to Whipple’s disease but since he did not have the full course of the appropriate treatment he relapsed. Whipple's disease is a difficult diagnosis to make because of the variety of clinical symptoms and the long time span between the initial unspecific symptoms (the prodromal stage) and the full-blown clinical picture of the illness (the steady-state stage). It may be misdiagnosed with a non-infectious rheumatic illness and may be fatal if untreated.

Published in Science Journal of Clinical Medicine (Volume 5, Issue 2)
DOI 10.11648/j.sjcm.20160502.12
Page(s) 20-23
Creative Commons

This is an Open Access article, distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution and reproduction in any medium or format, provided the original work is properly cited.

Copyright

Copyright © The Author(s), 2024. Published by Science Publishing Group

Keywords

Arthritis, Endoscopy, Histopathology, Infection, Tropheryma whippelii, Whipple’s Disease

References
[1] Klochan C, Anderson TA, Rose D et al. Nearly Fatal Case of Whipple’s Disease in a Patient Mistakenly on Anti-TNF Therapy. ACG Case Reports Journal. 2013; 1(1): 25-28.
[2] Magira EE, Gounaris T, Sioula E. Whipple’s Disease: Multiple Hospital Admissions of a Man with Diarrhoea, Fever and Arthralgia. J Infect. 2005 Aug; 51(2): E35-7.
[3] Schijf LJ, Becx MC, de Bruin PC et al. Whipple’s Disease: Easily Diagnosed if Considered. Neth J Med. 2008; 66 (9): 392-5.
[4] Ratnaike RL. Whipple’s Disease. Postgrad Med J. 2000; 76: 760–766.
[5] Relman DA, Schmidt TM, MacDermott RP, et al. Identification of the uncultured bacillus of Whipple’s disease. N Engl J Med. 1992; 327: 293–301.
[6] Marth T. New Insights into Whipple’s Disease – A Rare Intestinal Inflammatory Disorder. J Dig Dis. 2009; 27(4): 494-501.
[7] Günther U1, Moos V, Offenmüller G et al. Gastrointestinal diagnosis of Whipple disease: clinical, endoscopic and histopathologic features in 191 patients. Medicine. 2015 Apr; 94(15): e714.
[8] Yee A, Paget S. Expert guide to rheumatology. Philadelphia, Pa.: American College of Physicians; 2005.
[9] Jan Bureš, Marcela Kopáčová, Tomáš Douda, et al. Whipple’s Disease: Our Own Experience and Review of the Literature. Gastroenterology Research and Practice, vol. 2013, Article ID 478349, 10 pages, 2013.
[10] Fenollar F, Puéchal X, Raoult D. Medical progress: Whipple’s disease. N Engl J Med. 2007; 356: 5566.
[11] Feurle GE, Junga N, Marth T. Efficacy of ceftriaxone or meropenem as initial therapies in Whipple's disease. Gastroenterology 2010; 138: 478-86.
[12] Fenollar F, Rolain JM, Alric L, et al. Resistance to trimethoprim-sulfamethoxazole and Tropheryma whippelii. Int J Antimicrob Agents 2009; 34: 255-9.
[13] Fenollar F, Raoult D. How should classic Whipple's disease be managed? Nat Rev Gastroenterol Hepatol 2010; 7: 246-8.
[14] Lagier JC, Fenollar F, Lepidi H, Raoult D. Failure and relapse after treatment with trimethoprim/sulfamethoxazole in classic Whipple’s disease. J Antimicrob Chemother 2010 Sep; 65(9): 2005-12.
[15] Krol CG, de Meijer PHEM. Palindromic Rheumatism: consider Whipple’s Disease. Int J Rheum Dis 2013; 16: 475-476.
Cite This Article
  • APA Style

    Stephanie Santucci, Ernest Ellul, Noel Gatt, Jonathan Cutajar, Leigh Joseph Calleja. (2016). Palindromic Rheumatoid Arthritis-An Unorthodox Presentation of Whipple’s Disease. Science Journal of Clinical Medicine, 5(2), 20-23. https://doi.org/10.11648/j.sjcm.20160502.12

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    ACS Style

    Stephanie Santucci; Ernest Ellul; Noel Gatt; Jonathan Cutajar; Leigh Joseph Calleja. Palindromic Rheumatoid Arthritis-An Unorthodox Presentation of Whipple’s Disease. Sci. J. Clin. Med. 2016, 5(2), 20-23. doi: 10.11648/j.sjcm.20160502.12

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    AMA Style

    Stephanie Santucci, Ernest Ellul, Noel Gatt, Jonathan Cutajar, Leigh Joseph Calleja. Palindromic Rheumatoid Arthritis-An Unorthodox Presentation of Whipple’s Disease. Sci J Clin Med. 2016;5(2):20-23. doi: 10.11648/j.sjcm.20160502.12

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  • @article{10.11648/j.sjcm.20160502.12,
      author = {Stephanie Santucci and Ernest Ellul and Noel Gatt and Jonathan Cutajar and Leigh Joseph Calleja},
      title = {Palindromic Rheumatoid Arthritis-An Unorthodox Presentation of Whipple’s Disease},
      journal = {Science Journal of Clinical Medicine},
      volume = {5},
      number = {2},
      pages = {20-23},
      doi = {10.11648/j.sjcm.20160502.12},
      url = {https://doi.org/10.11648/j.sjcm.20160502.12},
      eprint = {https://article.sciencepublishinggroup.com/pdf/10.11648.j.sjcm.20160502.12},
      abstract = {Whipple's disease is a rare infection of the gastrointestinal tract caused by the actinomycete Tropheryma whippelii. It most commonly presents with arthralgia, abdominal pain, diarrhoea and weight loss. Invasion of the bacterium through the gastrointestinal mucosa leads to small intestinal villus blunting and malabsorption. Diagnosis is made by histological examination of small bowel biopsies. We report the case of a 78 year old gentleman who had a two year history of fleeting joint pain and multiple hospital admissions for varying symptoms. He was treated with antibiotics for a chest infection with improvement only to present again after 1 year with anorexia, fatigue, blackish loose stools and epigastric pain. Microscopic examination of duodenal biopsies showed a stunted villous architecture and expansion of the lamina propria by foamy macrophages. These expressed CD68 and cytoplasmic contents were strongly PAS positive, consistent with a diagnosis of Whipple’s disease. He was started on intravenous ceftriaxone and a prolonged course of oral co-trimoxazole (at least 1 year) with marked clinical improvement. In retrospect, the previous year’s admissions might have also been due to Whipple’s disease but since he did not have the full course of the appropriate treatment he relapsed. Whipple's disease is a difficult diagnosis to make because of the variety of clinical symptoms and the long time span between the initial unspecific symptoms (the prodromal stage) and the full-blown clinical picture of the illness (the steady-state stage). It may be misdiagnosed with a non-infectious rheumatic illness and may be fatal if untreated.},
     year = {2016}
    }
    

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  • TY  - JOUR
    T1  - Palindromic Rheumatoid Arthritis-An Unorthodox Presentation of Whipple’s Disease
    AU  - Stephanie Santucci
    AU  - Ernest Ellul
    AU  - Noel Gatt
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    AU  - Leigh Joseph Calleja
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    JF  - Science Journal of Clinical Medicine
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    UR  - https://doi.org/10.11648/j.sjcm.20160502.12
    AB  - Whipple's disease is a rare infection of the gastrointestinal tract caused by the actinomycete Tropheryma whippelii. It most commonly presents with arthralgia, abdominal pain, diarrhoea and weight loss. Invasion of the bacterium through the gastrointestinal mucosa leads to small intestinal villus blunting and malabsorption. Diagnosis is made by histological examination of small bowel biopsies. We report the case of a 78 year old gentleman who had a two year history of fleeting joint pain and multiple hospital admissions for varying symptoms. He was treated with antibiotics for a chest infection with improvement only to present again after 1 year with anorexia, fatigue, blackish loose stools and epigastric pain. Microscopic examination of duodenal biopsies showed a stunted villous architecture and expansion of the lamina propria by foamy macrophages. These expressed CD68 and cytoplasmic contents were strongly PAS positive, consistent with a diagnosis of Whipple’s disease. He was started on intravenous ceftriaxone and a prolonged course of oral co-trimoxazole (at least 1 year) with marked clinical improvement. In retrospect, the previous year’s admissions might have also been due to Whipple’s disease but since he did not have the full course of the appropriate treatment he relapsed. Whipple's disease is a difficult diagnosis to make because of the variety of clinical symptoms and the long time span between the initial unspecific symptoms (the prodromal stage) and the full-blown clinical picture of the illness (the steady-state stage). It may be misdiagnosed with a non-infectious rheumatic illness and may be fatal if untreated.
    VL  - 5
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    ER  - 

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Author Information
  • Department of Surgery, Mater Dei Hospital, Msida, Malta

  • Department of Surgery, Mater Dei Hospital, Msida, Malta

  • Department of Pathology, Mater Dei Hospital, Msida, Malta

  • Department of Surgery, Mater Dei Hospital, Msida, Malta

  • Department of Surgery, Mater Dei Hospital, Msida, Malta

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