Microscopic polyangiitis complicated with ileal involvement detected by double-balloon endoscopy: a case report

BMC Gastroenterology

Tetsuro Inokuma²  Yukihiro Imai⁴  Hajime Yoshimura³  Yoshitaka Tamaki³  Yuichiro Ono¹  Satoko Inoue²  Masashi Fukushima² 

[1] Department of Hematology and Clinical Immunology, Kobe City Medical Center General Hospital, 2-1-1 Minatojimaminamimachi, Chuo-ku, Kobe, Hyogo 650-0047, Japan;Department of Gastroenterology, Kobe City Medical Center General Hospital, 2-1-1 Minatojimaminamimachi, Chuo-ku, Kobe, Hyogo 650-0047, Japan;Department of Neurology, Kobe City Medical Center General Hospital, 2-1-1 Minatojimaminamimachi, Chuo-ku, Kobe, Hyogo 650-0047, Japan;Department of Pathology, Kobe City Medical Center General Hospital, 2-1-1 Minatojimaminamimachi, Chuo-ku, Kobe, Hyogo 50-0047, Japan

关键词: ANCA-associated vasculitides;    Small intestinal involvement;    Double-balloon endoscopy;    Microscopic polyangiitis;   

Others  :  858145
DOI  :  10.1186/1471-230X-13-42

 received in 2012-07-11, accepted in 2013-02-28,  发布年份 2013

【 摘 要 】

Background

Microscopic polyangiitis is characterized by pauci-immune, necrotizing small-vessel vasculitis and an anti-neutrophil cytoplasmic antibody-associated vasculitis. Although gastrointestinal involvement in microscopic polyangiitis is not rare, endoscopic observation of it is extremely rare. To the best of our knowledge, this is the first case report of small intestinal involvement in microscopic polyangiitis detected and followed up by double-balloon endoscopy.

Case presentation

A 70-year-old Japanese woman was transferred to our hospital for close examination of suspected small intestinal lymphoma. Retrograde double-balloon endoscopy revealed various forms of ulcers with redness and edema in the ileum. Histological findings suggested ischemic changes. Because mononeuritis multiplex and a fever spike appeared later, vasculitis was suspected. The perinuclear anti-neutrophil cytoplasmic antibody titer was elevated. Nerve biopsy results suggested vasculitis. From these findings, microscopic polyangiitis was diagnosed. It was suggested that microscopic polyangiitis caused the intestinal involvement. Intravenous pulse cyclophosphamide and oral predonisolone were started. After treatment, perinuclear anti-neutrophil cytoplasmic antibodies decreased to the normal range. Retrograde double-balloon endoscopy after treatment showed ulcer scars and no ulcer.

Conclusion

The cause of gastrointestinal involvement in microscopic polyangiitis is ischemia due to vasculitis. It is difficult to diagnose small-vessel vasculitis by endoscopic biopsy. Although histological evidence of microscopic polyangiitis is important, the treatment should not be delayed by repeating the biopsy, because such delay can result in adverse sequela.

This case report shows that microscopic polyangiitis should be considered as a differential diagnosis when small intestinal changes like those in the present case are observed by endoscopy.

【 授权许可】

   
2013 Fukushima et al; licensee BioMed Central Ltd.

【 图 表 】

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