Surgical repair of primary aorto-jejunal fistula associated with non-specific inflammatory abdominal aortic aneurysm

Surgical repair of primary aorto-jejunal fistula associated with non-specific inflammatory abdominal aortic aneurysm

Eur J Vasc Surg 5, 355-357 (1991) Surgical Repair of Primary Aorto-jejunal Fistula Associated with Non-specific Inflammatory Abdominal Aortic Aneurys...

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Eur J Vasc Surg 5, 355-357 (1991)

Surgical Repair of Primary Aorto-jejunal Fistula Associated with Non-specific Inflammatory Abdominal Aortic Aneurysm Shinsuke Mii, Toshihiro Onohara, Kenichiro Okadome, Atsushi Fukuda and Keizo Sugimachi The Second Department of Surgery, Faculty of Medicine, Kyushu University, Fukuoka 812, Japan A 28-year-old man with aorto-jejunalfistula associated with primary saccular abdominal aortic aneurysm (AAA) was effectively treated by resection and replacement with polytetraflouroethylene graft. The aetiology was suspected to be a non-specific aortitis, a rare cause of aorto-entericfistula. Key Words: Aorto-enteric fistula; Abdominal aortic aneurysm; Non-specific aortitis.

Introduction Sir Astley Cooper was a p p a r e n t l y t h e first to describe a o r t o - e n t e r i c fistula in 1829.1 The m o s t c o m m o n c a u s e of a b d o m i n a l aortic a n e u r y s m (AAA) w i t h p r i m a r y a o r t o - e n t e r i c fistula is atherosclerosis, z'3 b u t a fistula caused by non-specific aortitis, w h i c h is relatively freq u e n t in J a p a n , is m u c h rarer. W e t r e a t e d a y o u n g m a n w i t h a o r t o - j e j u n a l fistula associated w i t h a non-specific i n f l a m m a t o r y A A A . W e r e p o r t here t h e clinical p r e s e n t a tion a n d surgical m a n a g e m e n t of this patient.

Case Report A 2 8 - y e a r - o l d J a p a n e s e taxi driver h a d been h e a l t h y w i t h no h i s t o r y of a b d o m i n a l t r a u m a or specific infection. On 31 M a r c h 1 9 8 8 , he experienced intense a b d o m i n a l a n d l u m b a r p a i n followed by h a e m a t e m e s i s a n d m e l a e n a . A t two local clinics, upper a n d lower g a s t r o i n t e s t i n a l series were done but no haemorrhagic lesion was found. On 16 April 1 9 8 8 , a m e l a e n a a n d similar p a i n a c c o m p a n i e d by h i g h fever o c c u r r e d a n d clinical w o r k - u p at a n o t h e r hospital failed to reveal the source of t h e bleeding. A sacc u l a r a b d o m i n a l aortic a n e u r y s m (AAA), 3 x 3 x 5 c m in

Please address all correspondence to: Shinsuke Mii, The Second Department of Surgery, Faculty of Medicine, Kyushu University, Maidashi 3-11, Higashi-ku, Fukuoka 812, Japan. 0950-821X/91/030355+04 $03.00/0 © 1991 Grune & Stratton Ltd.

Fig. 1. A saccular aneurysm is evident on the left wall of the infrarenal abdominal aorta (emergency angiography), extravasation is absent.

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Fig. 2. Operative findings: a probe could be passed from the floor o[ the AAA through a tunnel to the jejunum located 1 cm distal to Treitz's ligament.

Fig. 3. Histological findings of the aneurysm and aorta (A) (H&E× 85). Chronic inflammatory cells infiltrated into the media and in the adventitia of the aneurysmal wall and the vasa vasorum was invaded by these cells (B) (EVGx 70). Elastic fibres in the media and adventitia of the aortic wall were extensively destroyed in close proximity to the AAA. Eur J Vasc Surg Vol 5, June 1991

size, w a s detected by u l t r a s o u n d a n d a n g i o g r a p h y , a n d o n 8 M a y 1988, he w e n t into shock following severe l u m b a r a n d a b d o m i n a l pain. He w a s b r o u g h t to o u r u n i v e r s i t y hospital the n e x t day. On admission, he w a s m o d e r a t e l y a n a e m i c a n d the blood p r e s s u r e w a s w i t h i n n o r m a l limits. A pulsatile t e n d e r m a s s w a s p a l p a t e d to t h e left of the umbilicus. On the n i g h t of admission, he v o m i t e d a b o u t l O 0 0 m l of fresh blood a n d w e n t into shock (systolic blood pressure 4 0 m m H g ) . Following blood transfusion, a n endoscopic e x a m i n a t i o n w a s m a d e w h i c h s h o w e d no evidence of a h a e m o r r h a g i c lesion in the o e s o p h a g u s , s t o m a c h , or t h e first or s e c o n d portions of t h e d u o d e n u m . The third p o r t i o n could n o t be visualised due to blood welling u p from t h e distal bowel. A l t h o u g h no e x t r a v a s a t i o n w a s detected o n a n g i o g r a p h y (Fig. 1), the p a t i e n t w a s t a k e n to the o p e r a t i n g room. A t l a p a r o t o m y , a n i n f l a m m a t o r y s a c c u l a r a n e u r y s m was f o u n d a d h e r e n t to the j e j u n u m . After c l a m p i n g the abd o m i n a l aorta, t h e a n e u r y s m w a s o p e n e d a n d a n aortoj e j u n a l fistula, 5 m m in d i a m e t e r w a s f o u n d I c m distal to Treitz's l i g a m e n t (Fig. 2). The a n e u r y s m w a s resected a n d r e p l a c e d w i t h a p o l y t e t r a f l u o r o e t h y l e n e (PTFE) graft a n d the j e j u n a l tear closed in two layers. After i r r i g a t i o n w i t h 7 0 0 0 m l saline c o n t a i n i n g the antibiotic, Cefmetazole, D a c r o n felt w a s w r a p p e d a r o u n d t h e s u t u r e line a n d covered w i t h o m e n t u m . The p o s t o p e r a t i v e course was satisfactory a n d t h e p a t i e n t w a s d i s c h a r g e d in good condition o n 30 M a y 1988. Cultures of the a n e u r y s m a l wall a n d t h r o m b u s in the a n e u r y s m r e v e a l e d no infections. Histology of t h e resected a n e u r y s m a n d aortic wall s h o w e d features of non-specific aortitis w i t h fibrous rep l a c e m e n t , diffuse infiltration of i n f l a m m a t o r y cells

A-E Fistula w i t h Non-specific Inflammatory AAA

accompanied by granulation and widespread destruction of the elastic fibres of the aortic m e d i a a n d a d v e n f i t i a (Fig. 3A, B). I m m u n o l o g i c a l e x a m i n a t i o n for s y p h i l i s a n d r h e u m a t i c fever were negative a n d CRP, ESR, a n d leukocytosis, w h i c h h a d been positive p r e o p e r a t i v e l y w e r e n e g a t i v e w h e n he left the hospital.

Discussion The incidence of non-specific i n f l a m m a t o r y s a c c u l a r a n e u r y s m w h i c h u s u a l l y occurs in individuals u n d e r 4 0 years of age, is curiously h i g h in Japan. G r o w t h of s u c h a n a n e u r y s m is rapid a n d it tends to r u p t u r e easily. 4's Hence, f o r m a t i o n of a p r i m a r y a o r t o - e n t e r i c fistula w i t h multiple s a c c u l a r a n e u r y s m s due to diffuse non-specific aortitis h a s b e e n reported, 6 a n d o u r p a t i e n t is a p p a r e n t l y the first to be effectively t r e a t e d w i t h surgery. The p r i m a r y cause of A A A w i t h a o r t o - e n t e r i c fistula is atherosclerosis. 2'3 Regardless of t h e cause, the condition h a s a h i g h m o r b i d i t y a n d e m e r g e n c y s u r g e r y h a s to be u n d e r t a k e n . However, m o r e t h a n a w e e k often passes from t h e first h a e m o r r h a g e to surgical i n t e r v e n tion, or d e a t h in 29 to 4 6 % of patients. 2'7'8 A n a c c u r a t e p r e o p e r a t i v e diagnosis is difficult especially in p a t i e n t s w i t h no h i s t o r y of aortic r e c o n s t r u c t i o n , b e c a u s e the t r i a d of pain, pulsatile m a s s a n d h a e m o r r h a g e , suggestive of such a fistula, is rarely p r e s e n t 3 a n d v a r i o u s e x a m i n a tions r e v e a l n o p e r t i n e n t information. 3,9,10 In o u r patient, 41 days p a s s e d from the first h a e m o r r h a g e to e m e r g e n c y surgery, a n d a n a c c u r a t e diagnosis could n o t be c o n firmed despite the presence of the triad. Thus, t h e key to a n early diagnosis is a h i g h index of suspicion. Surgical t r e a t m e n t is t h e o n l y h o p e of saving t h e patient. Other w o r k e r s suggested a n e u r y s m e c t o m y w i t h graft r e p l a c e m e n t following r e p a i r of the gut, T M a n d to p r e v e n t r e f o r m a t i o n of t h e fistula, a d e q u a t e p r o t e c t i o n b e t w e e n t h e g u t a n d the graft u s i n g a u t o l o g o u s tissues or a prosthetic cuff, a n d precise reperitonealisation. 9' 10 The r a t e of r e c u r r e n c e in the presence of aortitis is high, 4 h e n c e p r e v e n t i o n is essential. In o u r patient, the aetiology of A A A w a s obscure. Judging from his medical history, age, l a b o r a t o r y d a t a a n d histological findings, atherosclerosis, infection a n d t r a u m a were r u l e d out. Sterpetti et al. r e p o r t e d c o n g e n i t a l a b d o m i n a l a n e u r y s m s in t h e y o u n g , w h i c h resemble t h a t of o u r patient. 12 They were classified into t w o groups:

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t y p e 1 c o n g e n i t a l A A A , in w h i c h t h e r e is a g e n e r a l i s e d d i s o r d e r of arterial tissue a n d u s u a l l y a n e u r y s m s are p r e s e n t in o t h e r areas; a n d t y p e 2 c o n g e n i t a l A A A , in w h i c h t h e r e is a Iocalised defect oT the abcFominaI aorta, w i t h o u t a n e u r y s m s in o t h e r areas. 12 Our case w o u l d seem to belong to type 2. A c c o r d i n g to U r a y a m a a n d Sakai, most of these u n k n o w n origin A A A s were due to Beh~et's disease b e c a u s e t h e y o c c u r r e d in y o u n g males a n d a n a n e u r y s m is s o m e t i m e s the initial lesion in this condition, s W h i l e A A A w a s the o n l y evident a b n o r m a l finding in o u r patient, a careful follow-up to rule o u t the initial stage of Beh~et's disease is being made.

Acknowledgement

We thank M. Ohara for critical comments.

References 1 COOPERA: The Lectures of Sir Astley Cooper on the Principles and Practice of Surgery with Additional Notes and Cases by F. Tyrell, 5th edition. Philadelphia: Barrington and Haswell, 1939. 2 RECKLESSJPD, McCoLL I, TAYLORGW. Aorto-enteric fistulae: An uncommon complication of abdominal aortic aneurysms. Br ] Surg 1972 ; 59:458-459. 3 SWEEN~VMS, GADACZTR. Primary aortoduodenal fistula: manifestation, diagnosis, and treatment. Surgery 1984; 96: 492-497. 4 KATSUMURAT, FUIIWASAK, YAMANEM, et al. Surgical treatment of non-specific inflammatory aneurysm. (abstract) ] ]pn Coil Angiol 1981;21:319 325. 5 URAYAMAA, SAKAIB. Behqet's disease and aneurysm. (abstract) ] ]pn Coll Angiol 1975 ; 15 : 19-22. 6 MUKHOPADHAYAYAK, CHOPRAP. Diffuse non-specific aortitis with multiple saccular aneurysms and aorto-enteric fistula. Br Heart ] 1985; 54:102-104. 7 VOYLES WR, MORETZ WH. Rupture of aortic aneurysms into gastrointestinal tract. Surgery 1958 ;43 : 666-671. 8 MOLLESUPCL, STRANDL. Gastrointestinal hemorrhage due to fistula from aortic aneurysm. Acta Chir Scand 1987; 153 : 631-632. 9 BUSUTTILRW, REESW, BAKERJD, WILSONSE. Pathogenesis of aortoduodenal fistula: experimental and clinical correlates. Surgery 1979;85:1-13. 10 BENHAMOUG, DURONli. Aorto-digestive fistula. Int Surg 1982 ;67: 307-310. 11 PFEIFFERRB JR. Successful repair of three primary aortoduodenal fistulae. Arch Surg 1982;117:1098-1099. 12 STERPETTIAV, HUNTERWJ, SCHULTZRD. Congenital abdominal aortic aneurysms in the young; case report and review of the literature. J Vasc Surg 1988; 7: 763-769. Accepted 6 June 1989

Eur J Vasc Surg Vol 5, June 1991