Journal List > Korean J Gastroenterol > v.55(5) > 1006668

TOOLS
Jeong, Kim, Lee, Shim, and Moon: A Case of Limited Wegener's Granulomatosis with Gastroenteritis and Mesenteric Lymphadenopathy
Original Article

Korean J Gastroenterol 2010;55(5):331-335.

Published online: 21 February 2010

DOI: https://doi.org/10.4166/kjg.2010.55.5.331

A Case of Limited Wegener's Granulomatosis with Gastroenteritis and Mesenteric Lymphadenopathy

Yong Geun Jeong, M.D., Tae Hyo Kim, M.D., Chang Min Lee, M.D., Ki Shik Shim, M.D., In Kyu Moon, M.D.

Departments of Internal Medicine and Institute of Health Science, Gyeongsang National University School of Medicine, Jinju, Korea

Correspondence to: Tae Hyo Kim, M.D. Department of Internal Medicine, Gyeongsang National University Hospital, 90, Chilam-dong, Jinju 660-702, Korea Tel: +82-55-750-8726, Fax: +82-55-755-9078 E-mail: kimthy@medimail.co.kr

Received 10 August 2009       Accepted 23 December 2009

Copyright © 2010 The Korean Journal of Gastroenterology

Abstract

Wegener's granulomatosis (WG) is a disease characterized by a granulomatous necrotizing vasculitis of small vessels. Although any organ systems can be involved, gastrointestinal involvement in WG is notably uncommon. We present a case of 67-year-old man who was admitted with abdominal pain and diarrhea lasting for 3 weeks. Colonoscopy and abdominal CT scan revealed vasculitis and multiple mesenteric lymphadenopathy. Jejunum and mesenteric lymph nodes biopsies confirmed limited form of WG. The present case indicates that WG might in-volve only gastrointestinal tract and the histological confirmation is important for diagnosis.

Keywords: Wegener's granulomatosis, Vasculitis, Gastrointestinal tract, Mesenteric lymphadenopathy

REFERENCES

1. Haworth SJ, Pusey CD. Severe intestinal involvement in Wegener's granulomatosis. Gut. 1984; 25:1296–1300.
crossref
2. Wilson RH, Kerr PP, McLoughlin J, Gormley M. Symptomatic colitis as the initial presentation of Wegener's granulomatosis. Br J Clin Pract. 1993; 47:315–318.
3. Ozaki S. ANCA-associated vasculitis: diagnostic and therapeutic strategy. Allergol Int. 2007; 56:87–96.
4. Winter J, Neilly JB, Stevenson RD. Gastrointestinal haemorrhage complicating Wegener's granulomatosis. Br Med J (Clin Res Ed). 1985; 291:1275–1276.
crossref
5. Coward RA, Gibbons CP, Brown CB, Raftery AT, Parsons MA, Shortland JR. Gastrointestinal haemorrhage complicating Wegener's granulomatosis. Br Med J (Clin Res Ed). 1985; 291:865–866.
crossref
6. Geraghty J, Mackay IR, Smith DC. Intestinal perforation in Wegener's granulomatosis. Gut. 1986; 27:450–451.
crossref
7. Tokuda M, Kurata N, Daikuhara H, et al. Small intestinal perforation in Wegener's granulomatosis. J Rheumatol. 1989; 16:547–549.
8. Pinkney JH, Clarke G, Fairclough PD. Gastrointestinal involvement in Wegener's granulomatosis. Gastrointest Endosc. 1991; 37:411–412.
crossref
9. Spiera RF, Filippa DA, Bains MS, Paget SA. Esophageal involvement in Wegener's granulomatosis. Arthritis Rheum. 1994; 37:1404–1407.
crossref
10. Yamauchi F, Tanabe R, Nakashima R, et al. Wegener's granulomatosis with relapsed bleeding of gastric ulcers: a case report. Tokai J Exp Clin Med. 1995; 20:233–240.
11. Chang YJ, Kerr LD. Isolated abdominal vasculitis as an atypical presentation of Wegener's granulomatosis. Am J Gastroenterol. 2000; 95:297–298.
crossref
12. Fallows GA, Hamilton SF, Taylor DS, Reddy SB. Esophageal involvement in Wegener's granulomatosis: a case report and review of the literature. Can J Gastroenterol. 2000; 14:449–451.
crossref
13. Steele C, Bohra S, Broe P, Murray FE. Acute upper gastrointestinal haemorrhage and colitis: an unusual presentation of Wegener's granulomatosis. Eur J Gastroenterol Hepatol. 2001; 13:993–995.
crossref
14. Chow FY, Hooke D, Kerr PG. Severe intestinal involvement in Wegener's granulomatosis. J Gastroenterol Hepatol. 2003; 18:749–750.
15. Akç a T, Colak T, Cağ likü lekci M, Ocal K, Aydin S. Intestinal perforation in Wegener's granulomatosis: a case report. Ulus Travma Derg. 2005; 11:348–351.
16. Strivens RL, Bateman A, Arden NK, Edwards CJ. Intestinal perforation and jejunal haemorrhage due to Wegener's granulomatosis. Clin Exp Rheumatol. 2005; 23:124.
17. Deniz K, Ozş eker HS, Balas S, Akpý nar E, Sö kmensuer C. Intestinal involvement in Wegener's granulomatosis. J Gastrointestin Liver Dis. 2007; 16:329–331.
18. Yamaguchi T, Yoshioka K, Ueda M, et al. A case of Wegener's granulomatosis associated with progressive dyspha-gia owing to esophageal involvement. Mod Rheumatol. 2007; 17:521–525.
crossref
19. Deger SM, Sahin T, Vural C, et al. Wegener's granulomatosis with massive gastrointestinal hemorrhage due to jejunal and colonic involvement: report of a case. Surg Today. 2008; 38:166–169.
crossref
20. Gutié rrez-Ravé VM, Ayerza MA. Hilar and mediastinal lymphadenopathy in the limited form of Wegener's granulomatosis. Thorax. 1991; 46:219–220.
crossref

Fig. 1.
Abdominal CT scan. It showed (A) multiple distal mesenteric lymph nodes enlargement (arrows) and (B) segmental small bowel wall thickening (arrows).
kjg-55-331f1.tif
Fig. 2.
Colonoscopic findings. It showed diffuse bowel wall edema and submucosal hemorrhages.
kjg-55-331f2.tif
Fig. 3.
Microscopic findings of specimen. (A) Mesenteric lymph nodes showed vasculitis (arrow heads) with granulomatous inflammation (arrow) (H&E stain, ×100). (B) Mesenteries showed necrotizing vasculitis with small sized vessel (H&E stain, ×200).
kjg-55-331f3.tif
Fig. 4.
Post-treatment abdominal CT scan. Mesenteric lymph nodes enlargement and bowel wall thic-kenings disappeared.
kjg-55-331f4.tif
Table 1.
Summary of Previously Reported Cases of Gastrointestinal Involvement of Wegener's Granulomatosis
Author (year) 1ref. Sex/ Age C-ANCA Involved organs Gastrointestinal manifestations Biopsy site to comfirm Treatment Outcome
Haworth and Pusey (1984)1 F/46 - Lung, Kidney, eye, skin, joint Severe ileal, caecal, rectal involvement - Pd, Cyc, Aza Improved
Winter et al. (1985)4 M/37 - Lung, muscle, nerve SB perforation Muscle, SB Op, Pd, Cyc Improved
Coward et al. (1985)5 M/46 - Lung, kidney, nose, joint, eye, skin SB perforation SB Op, Pd, Cyc Improved
Geraghty et al. (1986)6 M/46 - Lung, joint, nose, skin SB perforation Nose Op, Pd, Cyc Improved
Tokuda et al. (1989)7 M/37 - Lung, kidney, nose, joint SB perforation Nose, SB Op, Pd, Cyc Improved
Pinkney et al. (1991)8 F/41 Positive Lung, joint, eye, ear SB ulceration Lung Op, Pd, Cyc Died
Wilson et al. (1993)2 M/31 Positive Nose, joint, skin Pancolitis Nose Pd, Cyc Improved
Spiera et al. (1994)9 F/54 Positive Lung, joint, oral, skin Erosive esophagitis Oral, esophagus Pd, Cyc Died
Yamauchi et al. (1995)10 M/55 Positive Lung, kidney, nose Gastric ulceration Nose Pd, Cyc Improved
Chang and Kerr (2000)11 M/47 Positive Uveitis SB arteries occlusion - Pd, Cyc Improved
Fallows et al. (2000)12 F/34 Positive Lung, kidney, nose, eye, skin, joint, ear Ulcerative esophagitis Esophagus Pd, Cyc Improved
Steele et al. (2001)13 F/34 Positive Lung, kidney, nose Esophageal, colonic ulcerations - Pd, Cyc Improved
Chow et al. (2003)14 M/46 Positive Sinusitis, kidney SB ulceration SB Op, Pd, Cyc Improved
Akç a et al. (2005)15 M/56 Positive Lung, skin, joint, lip SB perforation Lip Op, Pd, Cyc Improved
Strivens et al. (2005)16 F/56 Positive Kidney, nose, skin, ear SB perforation Skin, SB Op, Pd, Cyc Improved
Deniz et al. (2007)17 M/44 Positive Lung, nose SB perforation SB Op, Pd Improved
Yamaguchi et al. (2007)18 F/52 Positive Ear, skin Esophageal ulceration - Pd, Cyc Improved
Deger et al. (2008)19 M/34 Positive Kidney, nose, skin, joint Esophageal, gastric ulceration and SB perforation SB Op, Pd, Cyc Improved

C-ANCA, cytoplasmic antineutrophil cytoplamic antibody; Pd, predisolone; Cyc, cyclophosphamide; Aza, azathioprine; SB, small bowel; Op, operation.

TOOLS
Similar articles