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• W1983136670 abstract "Wegener's granulomatosis (WG) is a systemic vasculitis of medium-sized and small blood vessels. Aortic involvement in WG is very uncommon. We present a 43-year-old patient with an aortitis with aneurysm formation as a manifestation of WG. The patient was operated on and an aortoiliac Dacron inlay graft was inserted. Postoperatively, he recovered uneventfully. Abdominal pain occurring during a WG flare may result from vasculitis of large abdominal arteries with or without aneurysmatic changes for which surgical treatment and immunosuppressive agents are indicated to prevent a possible rupture. Wegener's granulomatosis (WG) is a systemic vasculitis of medium-sized and small blood vessels. Aortic involvement in WG is very uncommon. We present a 43-year-old patient with an aortitis with aneurysm formation as a manifestation of WG. The patient was operated on and an aortoiliac Dacron inlay graft was inserted. Postoperatively, he recovered uneventfully. Abdominal pain occurring during a WG flare may result from vasculitis of large abdominal arteries with or without aneurysmatic changes for which surgical treatment and immunosuppressive agents are indicated to prevent a possible rupture. Wegener's granulomatosis (WG) is a systemic vasculitis of small and medium-sized blood vessels. WG is a vasculitis involving primarily the upper respiratory tract and kidneys. Aortic involvement in WG is very uncommon.1Minnee R.C. van den Berk G.E. Groeneveld J.O. van Dijk J. Turkcan K. Visser M.J. et al.Aortic aneurysm and orchitis due to Wegener's granulomatosis.Ann Vasc Surg. 2009; 23: 786Abstract Full Text Full Text PDF PubMed Scopus (12) Google Scholar, 2Fink A.M. Miles K.A. Wraight E.P. Indium-111 labelled leucocyte uptake in aortitis.Clin Radiol. 1994; 49: 863-866Abstract Full Text PDF PubMed Scopus (34) Google Scholar, 3Durai R. Agrawal R. Piper K. Brohi K. Wegener's granulomatosis presenting as an abdominal aortic aneurysm: a case report.Cases J. 2009; 2: 9346Crossref PubMed Scopus (11) Google Scholar, 4Arlet J.B. Le Thi Huong D. Marinho A. Cluzel P. Wechsler B. Piette J.C. Arterial aneurysms in Wegener's granulomatosis: case report and literature review.Semin Arthritis Rheum. 2008; 37: 265-268Abstract Full Text Full Text PDF PubMed Scopus (27) Google Scholar, 5Aoki N. Soma K. Owada T. Ishii H. Wegener's granulomatosis complicated by arterial aneurysm.Intern Med. 1995; 34: 790-793Crossref PubMed Scopus (30) Google Scholar It is important to recognize aortic or other large vessel involvement, due to high risk of rupture. We present a patient with abdominal aortitis with aneurysm formation as a manifestation of WG. A 43-year-old man was admitted to our hospital with a 1-week history of abdominal pain and generalized malaise. In 1999, at the age of 32 years old he had been diagnosed with WG on the basis of nasopharyngeal ulcerations and glomerulonephritis. At that time, histologic examination of biopsy of ulcerative lesion in the oronasal cavity showed necrotizing giant-cell granulomatous inflammation and a necrotizing granulomatous vasculitis. Antineutrophil cytoplasmic antibodies were negative. Except for two episodes of recurrence in 2001 and 2003, the WG was in remission with prednisolone and mycophenolic acid. On his present admission, the physical examination was normal, except for pain on palpation in the umbilical region of the abdomen. Laboratory tests showed the following: leukocyte count, 11.0 × 109/L; hemoglobin, 8.3 mmol/L; C-reactive protein, 98 mg/L; normal serum lipase; and stable serum creatinine, 1.2 mg/dL. The chest X-ray was normal. Ultrasound scan evaluation of the abdomen showed an aneurysm of the infrarenal aorta with a diameter of 3.1 cm and a length of 3.5 cm. An abdominal computed tomography (CT) scan showed thickening of the infrarenal aortic wall, surrounded by a hypodense rim (Fig 1). Because the clinical picture was highly suspect for exacerbation of systemic vasculitis, the patient was initially treated with a high dose of prednisolone. Despite addition of a high dose of morphine and ketamine, the abdominal pain increased suggesting a high risk of rupture of the aneurysm. Thus, the patient was operated on urgently. The aortic aneurysm was embedded in an inflammatory process. An aorto-bi-iliac Dacron inlay graft, soaked in rifampicin, was inserted. Cultures were negative and the histology showed a necrotizing inflammation of the aortic wall and a necrotizing granulomatous vasculitis (Fig 2).Fig 2Histology of the aortic wall. Image shows media, adventitial inflammation, and intimal proliferation with granuloma and necrotizing inflammation.View Large Image Figure ViewerDownload Hi-res image Download (PPT) Postoperatively, the patient noticed pain relief and he recovered uneventfully and was discharged at the 11th postoperative day. Treatment was continued and consisted of prednisone and mycophenolate acid. After 4 weeks and 6 months, a positron emission tomography scan showed no new signs of vascular inflammation or other signs of aneurysm. After a 1-year follow-up, surveillance CT scan showed no new sign of periaortitis or aneurysmatic abnormalities in the abdomen. WG is a vasculitis involving primarily the upper respiratory tract and kidneys. Histologic examination shows necrotizing vasculitis affecting mostly small-size vessels (eg, capillaries, venules, and arterioles). Chronic inflammation can lead to medium and large-sized arterial aneurysm formation but is a very unusual feature.5Aoki N. Soma K. Owada T. Ishii H. Wegener's granulomatosis complicated by arterial aneurysm.Intern Med. 1995; 34: 790-793Crossref PubMed Scopus (30) Google Scholar WG in medium-sized vessels is more common and show complicated outcome.4Arlet J.B. Le Thi Huong D. Marinho A. Cluzel P. Wechsler B. Piette J.C. Arterial aneurysms in Wegener's granulomatosis: case report and literature review.Semin Arthritis Rheum. 2008; 37: 265-268Abstract Full Text Full Text PDF PubMed Scopus (27) Google Scholar, 5Aoki N. Soma K. Owada T. Ishii H. Wegener's granulomatosis complicated by arterial aneurysm.Intern Med. 1995; 34: 790-793Crossref PubMed Scopus (30) Google Scholar Rupture occurred in half of the patients leading to death from abdominal bleeding in three cases.4Arlet J.B. Le Thi Huong D. Marinho A. Cluzel P. Wechsler B. Piette J.C. Arterial aneurysms in Wegener's granulomatosis: case report and literature review.Semin Arthritis Rheum. 2008; 37: 265-268Abstract Full Text Full Text PDF PubMed Scopus (27) Google Scholar, 5Aoki N. Soma K. Owada T. Ishii H. Wegener's granulomatosis complicated by arterial aneurysm.Intern Med. 1995; 34: 790-793Crossref PubMed Scopus (30) Google Scholar Aortic aneurysm as a result of WG is a very rare complication. Until now, six other cases of aneurysms located in large vessels as a result of WG are described,1Minnee R.C. van den Berk G.E. Groeneveld J.O. van Dijk J. Turkcan K. Visser M.J. et al.Aortic aneurysm and orchitis due to Wegener's granulomatosis.Ann Vasc Surg. 2009; 23: 786Abstract Full Text Full Text PDF PubMed Scopus (12) Google Scholar, 2Fink A.M. Miles K.A. Wraight E.P. Indium-111 labelled leucocyte uptake in aortitis.Clin Radiol. 1994; 49: 863-866Abstract Full Text PDF PubMed Scopus (34) Google Scholar, 3Durai R. Agrawal R. Piper K. Brohi K. Wegener's granulomatosis presenting as an abdominal aortic aneurysm: a case report.Cases J. 2009; 2: 9346Crossref PubMed Scopus (11) Google Scholar, 6Blockmans D. Baeyens H. Van Loon R. Lauwers G. Bobbaers H. Periaortitis and aortic dissection due to Wegener's granulomatosis.Clin Rheumatol. 2000; 19: 161-164Crossref PubMed Scopus (71) Google Scholar, 7Carels T. Verbeken E. Blockmans D. p-ANCA-associated periaortitis with histological proof of Wegener's granulomatosis: case report.Clin Rheumatol. 2005; 24: 83-86Crossref PubMed Scopus (40) Google Scholar, 8Chirinos J.A. Tamariz L.J. Lopes G. Del Carpio F. Zhang X. Milikowski C. et al.Large vessel involvement in ANCA-associated vasculitides: report of a case and review of literature.Clin Rheumatol. 2004; 23: 152-159Crossref PubMed Scopus (74) Google Scholar of which only five cases had acute presentation and two cases had no histologic examination performed (Table). In four cases, the patients were operated on and surgical tissue biopsies showed signs of vasculitis which indicated WG. In one case, the patient died from aortic dissection.3Durai R. Agrawal R. Piper K. Brohi K. Wegener's granulomatosis presenting as an abdominal aortic aneurysm: a case report.Cases J. 2009; 2: 9346Crossref PubMed Scopus (11) Google Scholar In one case,1Minnee R.C. van den Berk G.E. Groeneveld J.O. van Dijk J. Turkcan K. Visser M.J. et al.Aortic aneurysm and orchitis due to Wegener's granulomatosis.Ann Vasc Surg. 2009; 23: 786Abstract Full Text Full Text PDF PubMed Scopus (12) Google Scholar the patient was treated medically. This patient was diagnosed with WG assuming that the periaortitis, as seen on CT scan was caused by WG, without pathologic confirmation. In another case, no histology was obtained because the specimen was lost.3Durai R. Agrawal R. Piper K. Brohi K. Wegener's granulomatosis presenting as an abdominal aortic aneurysm: a case report.Cases J. 2009; 2: 9346Crossref PubMed Scopus (11) Google ScholarTableCases presented with aortic involvement in Wegener's granulomatosis (WG)Patient (publication)Aneurysm of the aortaDuration of complaintsTreatmentRuptureHistology53-year ♀ (Minnee et al1Minnee R.C. van den Berk G.E. Groeneveld J.O. van Dijk J. Turkcan K. Visser M.J. et al.Aortic aneurysm and orchitis due to Wegener's granulomatosis.Ann Vasc Surg. 2009; 23: 786Abstract Full Text Full Text PDF PubMed Scopus (12) Google Scholar)3.8 cm5 MonthsConservativeNoNo33-year ♂ (Durai et al3Durai R. Agrawal R. Piper K. Brohi K. Wegener's granulomatosis presenting as an abdominal aortic aneurysm: a case report.Cases J. 2009; 2: 9346Crossref PubMed Scopus (11) Google Scholar)3.2 cm3 WeeksSurgeryNoNo63-year ♂ (Carels et al7Carels T. Verbeken E. Blockmans D. p-ANCA-associated periaortitis with histological proof of Wegener's granulomatosis: case report.Clin Rheumatol. 2005; 24: 83-86Crossref PubMed Scopus (40) Google Scholar)4.0 cm1 WeekSurgeryNoYes50-year ♀ (Chirinos et al8Chirinos J.A. Tamariz L.J. Lopes G. Del Carpio F. Zhang X. Milikowski C. et al.Large vessel involvement in ANCA-associated vasculitides: report of a case and review of literature.Clin Rheumatol. 2004; 23: 152-159Crossref PubMed Scopus (74) Google Scholar)Dissection from first intercostal artery to iliac bifurcation1 WeekConservativeYesYes42-year ♂ (Blockmans et al6Blockmans D. Baeyens H. Van Loon R. Lauwers G. Bobbaers H. Periaortitis and aortic dissection due to Wegener's granulomatosis.Clin Rheumatol. 2000; 19: 161-164Crossref PubMed Scopus (71) Google Scholar)3.2 cm1 WeekSurgeryNoYes45-year ♂ (Fink et al2Fink A.M. Miles K.A. Wraight E.P. Indium-111 labelled leucocyte uptake in aortitis.Clin Radiol. 1994; 49: 863-866Abstract Full Text PDF PubMed Scopus (34) Google Scholar)No AAA1 WeekSurgeryNoYesAAA, Abdominal aortic aneurysm. Open table in a new tab AAA, Abdominal aortic aneurysm. The diagnosis of WG is based on four clinical criterions as defined by the Chapel Hill Consensus Conference in 1994. These are nasal or oral inflammation, abnormal chest radiograph (showing the presence of nodules, fixed infiltrates, or cavities), abnormal urinary sediment (microscopic hematuria), and granulomatous inflammation on biopsy.9Leavitt R.Y. Fauci A.S. Bloch D.A. Michel B.A. Hunder G.G. Arend W.P. et al.The American College of Rheumatology 1990 criteria for the classification of Wegener's granulomatosis.Arthritis Rheum. 1990; 33: 1101-1107Crossref PubMed Scopus (1558) Google Scholar Treatment is based on a combination of steroids and immunosuppressants. Our patient was diagnosed with WG based on three out of four criterions. The differential diagnosis of vasculitis with large-vessel involvement includes other vasculitides that overlap with WG, most frequently Takayasu's syndrome. Glomerulonephritis are not, however, characteristic of this disorder, and pulmonary involvement is also very rare. Giant cell arteritis of the elderly (temporal arteritis) involves not only the temporal artery but also the aorta, lungs, and kidneys. The young age of our patient, in addition to the absence of cranial symptoms or polymyalgia rheumatica, ruled out this disorder as well.5Aoki N. Soma K. Owada T. Ishii H. Wegener's granulomatosis complicated by arterial aneurysm.Intern Med. 1995; 34: 790-793Crossref PubMed Scopus (30) Google Scholar In our case, there was a high risk of rupture because the patient presented with progressively severe abdominal pain and was not responding to pain medication and immunosuppressive agents, had persistent high blood pressure (190/110 mm Hg), and combined with the CT scan, had obvious thickening of the wall of the aneurysm and the hypodense rim, so we decided to operate. In summary, abdominal pain occurring during a WG flare may result from intestinal vasculitis of large abdominal arteries and may cause symptomatic aneurysmatic dilatation for which surgical treatment and immunosuppressive agents are indicated. The authors thank Unsal Yapici, from the Department of Pathology, Academic Medical Center, for providing histology." @default.
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