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Whipple's disease diagnosed during anti-tumor necrosis factor alpha treatment: two case reports and review of the literature.

Ramos JM, Pasquau F, Galipienso N, Valero B, Navarro A, Martinez A, Rosas J, Gutiérrez A, Sanchez-Martínez R - J Med Case Rep (2015)

Bottom Line: The first patient was a 58-year-old white man with psoriatic spondylarthritis, who was treated with adalimumab, etanercept, infliximab, tocilizumab and golimumab.The second was a 73-year-old white man with rheumatoid arthritis, who received treatment with infliximab, then etanercept and rituximab.Whipple's disease should be suspected in all patients diagnosed with chronic inflammatory rheumatism, partially controlled or not controlled by treatment with tumor necrosis factor alpha blockers, whose condition worsens after treatment.

View Article: PubMed Central - PubMed

Affiliation: Department of Internal Medicine, Hospital General Universitario de Alicante, c/ Pintor Baeza, 12, 03010, Alicante, Spain. jramosrincon@yahoo.es.

ABSTRACT

Introduction: Whipple's disease is a rare infectious disease caused by Tropheryma whipplei with protean clinical manifestations. This infection may mimic chronic inflammatory rheumatisms.

Case presentation: We report two cases of Whipple's disease diagnosed in the context of an inflammatory disease with anti-tumor necrosis factor alpha failure. The first patient was a 58-year-old white man with psoriatic spondylarthritis, who was treated with adalimumab, etanercept, infliximab, tocilizumab and golimumab. The second was a 73-year-old white man with rheumatoid arthritis, who received treatment with infliximab, then etanercept and rituximab.

Conclusions: Whipple's disease should be suspected in all patients diagnosed with chronic inflammatory rheumatism, partially controlled or not controlled by treatment with tumor necrosis factor alpha blockers, whose condition worsens after treatment.

No MeSH data available.


Related in: MedlinePlus

Endoscopy. White lesions compatible with diffuse intestinal lymphangiectasia
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Fig1: Endoscopy. White lesions compatible with diffuse intestinal lymphangiectasia

Mentions: A 58-year-old white man with inflammatory back pain and large and small joint arthritis had been diagnosed with psoriatic spondylarthritis 9 years ago. Our patient had been treated with adalimumab for 4 months, after that with etarnecept for 8 months, then infliximab for 2 months, tocilizumab for 21 months and golimumab for 1 month, to treat the pain in his back and neck with the consequent difficulty in bending, and arthritis of his knee and interphalangeal joint arthritis. Our patient was admitted to the hospital with abdominal septic shock. A computed tomography (CT) scan showed multiple retroperitoneal lymph nodes. The colonoscopy result was normal and the biopsy result was normal. Three months later, he was admitted again with a fever and heart failure, which was interpreted as a side effect of the golimumab treatment. One year after that, he was admitted to the hospital with abdominal pain, diarrhea and weight loss progressing to a severe wasting syndrome. At this time, he was being treated with 5mg prednisone plus hydroxicloroquine and methotrexate (MTX). Abnormal laboratory test results included a white blood cell (WBC) count of 14,630/mm3, a hemoglobin level of 9.6g/dL and an erythrocyte sedimentation rate (ESR) of 58mm/h. A CT scan showed multiple lymph nodes. Endoscopy showed diffuse intestinal lymphangiectasia (Fig. 1). A duodenal biopsy showed distortion of the villous architecture with abundant macrophages, and bacilliform intracytoplasmic structures that stained positive with PAS with diastase digestion compatible with WD. A PCR assay for detecting T. whipplei was not done. Intravenous ceftriaxone (2g daily for 2 weeks) was commenced followed by trimethoprim and sulphamethoxazole with improved symptoms after 3 weeks; treatment was continued for 18 months. One year later, a new gastroscopy with duodenal biopsy was done. It did not show intestinal lymphangiectasia. A PCR assay result for T. whipplei was negative. There were no relapses after 19 months.Fig. 1


Whipple's disease diagnosed during anti-tumor necrosis factor alpha treatment: two case reports and review of the literature.

Ramos JM, Pasquau F, Galipienso N, Valero B, Navarro A, Martinez A, Rosas J, Gutiérrez A, Sanchez-Martínez R - J Med Case Rep (2015)

Endoscopy. White lesions compatible with diffuse intestinal lymphangiectasia
© Copyright Policy - open-access
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC4522104&req=5

Fig1: Endoscopy. White lesions compatible with diffuse intestinal lymphangiectasia
Mentions: A 58-year-old white man with inflammatory back pain and large and small joint arthritis had been diagnosed with psoriatic spondylarthritis 9 years ago. Our patient had been treated with adalimumab for 4 months, after that with etarnecept for 8 months, then infliximab for 2 months, tocilizumab for 21 months and golimumab for 1 month, to treat the pain in his back and neck with the consequent difficulty in bending, and arthritis of his knee and interphalangeal joint arthritis. Our patient was admitted to the hospital with abdominal septic shock. A computed tomography (CT) scan showed multiple retroperitoneal lymph nodes. The colonoscopy result was normal and the biopsy result was normal. Three months later, he was admitted again with a fever and heart failure, which was interpreted as a side effect of the golimumab treatment. One year after that, he was admitted to the hospital with abdominal pain, diarrhea and weight loss progressing to a severe wasting syndrome. At this time, he was being treated with 5mg prednisone plus hydroxicloroquine and methotrexate (MTX). Abnormal laboratory test results included a white blood cell (WBC) count of 14,630/mm3, a hemoglobin level of 9.6g/dL and an erythrocyte sedimentation rate (ESR) of 58mm/h. A CT scan showed multiple lymph nodes. Endoscopy showed diffuse intestinal lymphangiectasia (Fig. 1). A duodenal biopsy showed distortion of the villous architecture with abundant macrophages, and bacilliform intracytoplasmic structures that stained positive with PAS with diastase digestion compatible with WD. A PCR assay for detecting T. whipplei was not done. Intravenous ceftriaxone (2g daily for 2 weeks) was commenced followed by trimethoprim and sulphamethoxazole with improved symptoms after 3 weeks; treatment was continued for 18 months. One year later, a new gastroscopy with duodenal biopsy was done. It did not show intestinal lymphangiectasia. A PCR assay result for T. whipplei was negative. There were no relapses after 19 months.Fig. 1

Bottom Line: The first patient was a 58-year-old white man with psoriatic spondylarthritis, who was treated with adalimumab, etanercept, infliximab, tocilizumab and golimumab.The second was a 73-year-old white man with rheumatoid arthritis, who received treatment with infliximab, then etanercept and rituximab.Whipple's disease should be suspected in all patients diagnosed with chronic inflammatory rheumatism, partially controlled or not controlled by treatment with tumor necrosis factor alpha blockers, whose condition worsens after treatment.

View Article: PubMed Central - PubMed

Affiliation: Department of Internal Medicine, Hospital General Universitario de Alicante, c/ Pintor Baeza, 12, 03010, Alicante, Spain. jramosrincon@yahoo.es.

ABSTRACT

Introduction: Whipple's disease is a rare infectious disease caused by Tropheryma whipplei with protean clinical manifestations. This infection may mimic chronic inflammatory rheumatisms.

Case presentation: We report two cases of Whipple's disease diagnosed in the context of an inflammatory disease with anti-tumor necrosis factor alpha failure. The first patient was a 58-year-old white man with psoriatic spondylarthritis, who was treated with adalimumab, etanercept, infliximab, tocilizumab and golimumab. The second was a 73-year-old white man with rheumatoid arthritis, who received treatment with infliximab, then etanercept and rituximab.

Conclusions: Whipple's disease should be suspected in all patients diagnosed with chronic inflammatory rheumatism, partially controlled or not controlled by treatment with tumor necrosis factor alpha blockers, whose condition worsens after treatment.

No MeSH data available.


Related in: MedlinePlus