Case Report

Transmission electron microscopy helpfulness in Whipple's disease masked by immunosuppressant therapy for arthritis

Alessandra Loiodice

Section of Gastroenterology, Department of Emergency and Organ Transplantation, University ‘Aldo Moro’ of Bari, Bari, Italy

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Giuseppe Losurdo,

Giuseppe Losurdo

Section of Gastroenterology, Department of Emergency and Organ Transplantation, University ‘Aldo Moro’ of Bari, Bari, Italy

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Andrea Iannone,

Andrea Iannone

Section of Gastroenterology, Department of Emergency and Organ Transplantation, University ‘Aldo Moro’ of Bari, Bari, Italy

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Roberta Rossi,

Roberta Rossi

Section of Pathology, Department of Emergency and Organ Transplantation, University ‘Aldo Moro’ of Bari, Bari, Italy

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Maria Grazia Fiore,

Maria Grazia Fiore

Section of Pathology, Department of Emergency and Organ Transplantation, University ‘Aldo Moro’ of Bari, Bari, Italy

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Domenico Piscitelli,

Corresponding Author

Domenico Piscitelli

[email protected]

Section of Pathology, Department of Emergency and Organ Transplantation, University ‘Aldo Moro’ of Bari, Bari, Italy

Prof. Domenico Piscitelli, Section of Pathology, Department of Emergency and Organ Transplantation, University ‘Aldo Moro’ of Bari, Piazza Giulio Cesare, 70124 Bari, Italy. e-mail: [email protected]Search for more papers by this author

Alessandra Loiodice,

Alessandra Loiodice

Section of Gastroenterology, Department of Emergency and Organ Transplantation, University ‘Aldo Moro’ of Bari, Bari, Italy

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Giuseppe Losurdo,

Giuseppe Losurdo

Section of Gastroenterology, Department of Emergency and Organ Transplantation, University ‘Aldo Moro’ of Bari, Bari, Italy

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Andrea Iannone,

Andrea Iannone

Section of Gastroenterology, Department of Emergency and Organ Transplantation, University ‘Aldo Moro’ of Bari, Bari, Italy

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Roberta Rossi,

Roberta Rossi

Section of Pathology, Department of Emergency and Organ Transplantation, University ‘Aldo Moro’ of Bari, Bari, Italy

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Maria Grazia Fiore,

Maria Grazia Fiore

Section of Pathology, Department of Emergency and Organ Transplantation, University ‘Aldo Moro’ of Bari, Bari, Italy

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Domenico Piscitelli,

Corresponding Author

Domenico Piscitelli

[email protected]

Section of Pathology, Department of Emergency and Organ Transplantation, University ‘Aldo Moro’ of Bari, Bari, Italy

First published: 20 November 2017

https://doi.org/10.1111/apm.12782

Citations: 5

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Abstract

A 61-year-old woman received a diagnosis of undifferentiated non-erosive arthritis in 2010 and assumed methotrexate and steroids in 2014. After 1 year, she experienced watery diarrhea, vomiting, fever, weight loss, and severe hypoalbuminemia, thus being admitted into our Unit. Esophagogastroduodenoscopy showed duodenal lymphangiectasia and duodenal biopsy samples several foamy PAS-positive macrophages and villous subtotal atrophy. Transmission electron microscope demonstrated several extracellular and intracellular rod-shaped bacteria (Tropheryma whipplei). Therefore, we diagnosed Whipple's disease. Our patient assumed doxycycline/hydroxychloroquine with prompt remission of gastrointestinal symptoms. At 1 year of follow-up, she was symptom-free, histological reassessment showed almost complete mucosal healing and transmission electron microscope demonstrated bacteria breaking/disappearance. The present report demonstrates that: (i) rheumatological manifestations are common onset symptoms of Whipple's disease; (ii) immunosuppressive therapy may delay the diagnosis and worsen clinical presentation; (iii) transmission electron microscopy for specific bacteria detection/disappearance is an helpful diagnostic tool, when available.

References

1Marth T, Moos V, Müller C, Biagi F, Schneider T. Tropheryma Whipplei infection and Whipple's disease. Lancet Infect Dis 2016; 16: 12–21.
Web of Science® Google Scholar
2Lagier JC, Lepidi H, Raoult D, Fenollar F. Systemic Tropheryma Whipplei clinical presentation of 142 patients with infections diagnosed or confirmed in a reference center. Medicine 2010; 89: 337–45.
10.1097/MD.0b013e3181f204a8
PubMed Web of Science® Google Scholar
3Günther U, Moos V, Offenmüller G, Oelkers G, Heise W, Moter A, et al. Gastrointestinal diagnosis of classical Whipple disease: clinical, endoscopic, and histopathologic features in 191 patient. Medicine 2015; 94: e714.
PubMed Web of Science® Google Scholar
4Meunier M, Puechal X, Hoppé E, Soubrier M, Dieudé P, Berthelot JM, et al. Rheumatic and musculoskeletal features of Whipple disease: a report of 29 cases. J Rheumatol 2013; 40: 2061–6.
10.3899/jrheum.130328
PubMed Web of Science® Google Scholar
5Silva MT, Macedo PM, Moura Nunes JF. Ultrastructure of Bacilli and the Bacillary origin of the macrophagic inclusions in Whipple's disease. J Gen Microbiol 1985; 131: 1001–13.
CAS PubMed Web of Science® Google Scholar
6Lagier JC, Fenollar F, Lepidi H, Giorgi R, Million M, Raoult D. Treatment of classic Whipple's disease: from in vitro results to clinical outcome. J Antimicrob Chemother 2014; 69: 219–27.
10.1093/jac/dkt310
CAS PubMed Web of Science® Google Scholar
7Lagier JC, Fenollar F, Lepidi H, Raoult D. Evidence of lifetime susceptibility to Tropheryma Whipplei in patients with Whipple's disease. J Antimicrob Chemother 2011; 66: 1188–9.
10.1093/jac/dkr032
CAS PubMed Web of Science® Google Scholar
8Schneider T, Moos V, Loddenkemper C, Marth T, Fenollar F, Raoult D. Whipple's disease: new aspects of pathogenesis and Treatment. Lancet Infect Dis 2008; 8: 179–90.
10.1016/S1473-3099(08)70042-2
CAS PubMed Web of Science® Google Scholar
9Kneitz C, Suerbaum S, Beer M, Muller J, Jahns R, Tony HP. Exacerbation of Whipple's disease associated with infliximab Treatment. Scand J Rheumatol 2005; 34: 148–51.
10.1080/03009740510015230
CAS PubMed Web of Science® Google Scholar
10Mahnel R, Anja Kalt MD, Ring S, Stallmach A, Strober W, Marth T. Immunosuppressive therapy in Whipple's Disease Patients is associated with the appearance of gastrointestinal manifestation. Am J Gastroenterol 2005; 100: 1167–73.
10.1111/j.1572-0241.2005.40128.x
CAS PubMed Web of Science® Google Scholar
11Ramzan NN, Loftus E Jr, Burgart LJ, Rooney M, Batts KP, Wiesner RH, et al. Diagnosis and monitoring of Whipple disease by polymerase chain reaction. Ann Intern Med 1997; 126: 520–7.
10.7326/0003-4819-126-7-199704010-00004
CAS PubMed Web of Science® Google Scholar
12Fenollar F, Raoult D. Molecular techniques in Whipple's disease. Expert Rev Mol Diagn 2001; 1: 299–309.
10.1586/14737159.1.3.299
CAS PubMed Google Scholar
13Boulos A, Rolain JM, Raoult D. Antibiotic susceptibility of Tropheryma Whipplei in MRC5 cells. Antimicrob Agents Chemother 2004; 48: 747–52.
10.1128/AAC.48.3.747-752.2004
CAS PubMed Web of Science® Google Scholar
14Ghigo E, Capo C, Aurouze M, Tung CH, Gorvel JP, Raoult D, et al. Survival of Tropheryma Whipplei, the agent of Whipple's disease, requires phagosome acidification. Infect Immun 2002; 70: 1501–6.
10.1128/IAI.70.3.1501-1506.2002
CAS PubMed Web of Science® Google Scholar
15Moos V, Feurle GE, Schinnerling K, Geelhaar A, Friebel J, Allers K, et al. Immunopathology of immune reconstitution inflammatory syndrome in Whipple's disease. J Immunol 2013; 190: 2354–61.
10.4049/jimmunol.1202171
CAS PubMed Web of Science® Google Scholar
16Biagi F, Trotta L, Di Stefano M, Balduzzi D, Marchese A, Vattiato C, et al. Previous immunosuppressive therapy is a risk factor for immune reconstitution inflammatory syndrome in Whipple's disease. Dig Liver Dis 2012; 44: 880–2.
10.1016/j.dld.2012.05.008
PubMed Web of Science® Google Scholar
17Ierardi E, Losurdo G, Iannone A, Piscitelli D, Amoruso A, Barone M, et al. Lymphocytic duodenitis or microscopic enteritis and gluten-related conditions: what needs to be explored? Ann Gastroenterol 2017; 30: 380–92.
PubMed Web of Science® Google Scholar
18Delhom RB, Mills PR, More IAR. Electron microscopy in the long term follow up of Whipple's disease. Effects of antibiotics. Am J Surg Pathol 1981; 5: 507–16.
PubMed Web of Science® Google Scholar
19De Francesco V, Zullo A, Cristofari F, Zotti M, Stoppino G, D'Agnessa MR, et al. A juvenile, misdiagnosed case of Whipple's disease. Rheumatol Int 2009; 30: 143–4.
PubMed Web of Science® Google Scholar

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Volume126, Issue1

January 2018

Pages 92-96

Transmission electron microscopy helpfulness in Whipple's disease masked by immunosuppressant therapy for arthritis

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Transmission electron microscopy helpfulness in Whipple's disease masked by immunosuppressant therapy for arthritis

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